Outcomes of Research or Clinical Trials Activity Levels Acute Flaccid Paralysis Ageing Anaerobic Threshold Anaesthesia Assistive Technology Brain Cardiorespiratory Cardiovascular Clinical Evaluation Cold Intolerance Complementary Therapies Continence Coping Styles and Strategies Cultural Context Diagnosis and Management Differential Diagnosis Drugs Dysphagia Dysphonia Epidemiology Exercise Falls Fatigue Fractures Gender Differences Immune Response Inflammation Late Effects of Polio Muscle Strength Muscular Atrophy Orthoses Pain Polio Immunisation Post-Polio Motor Unit Psychology Quality of Life Renal Complications Respiratory Complications and Management Restless Legs Syndrome Sleep Analaysis Surgery Vitality Vocational Implications

Effective More Research Required Not Effective
Category: Fatigue

Title: A 5-year longitudinal study of fatigue in patients with late-onset sequelae of poliomyelitis
Author: Tersteeg IM (1), Koopman FS, Stolwijk-Swüste JM, Beelen A, Nollet F; CARPA Study Group
Affiliation: (1) Department of Rehabilitation, Academic Medical Center, University of Amsterdam, Amsterdam, The Netherlands. [email protected]
Journal: Archives of Physical Medicine and Rehabilitation
Citation: Arch Phys Med Rehabil. 2011 Jun;92(6):899-904. doi: 10.1016/j.apmr.2011.01.005
Publication Year and Month: 2011 06

Abstract: OBJECTIVES: To study the severity and 5-year course of fatigue in patients with late-onset sequelae of poliomyelitis (LOSP) and to identify physical and psychosocial determinants of fatigue.

DESIGN: Prospective cohort study with 5 measurements over 5 years.

SETTING: University hospital.

PARTICIPANTS: Patients with LOSP (N=168); 89% of the subjects completed the study.

INTERVENTIONS: Not applicable.

MAIN OUTCOME MEASURES: Fatigue assessed with the Fatigue Severity Scale (FSS). Potential determinants were perceived physical functioning, bodily pain and mental health, extent of paresis, walking capacity, comorbidity, sleeping disorders, coping, and social support. Associations were investigated by multivariable longitudinal analysis using generalized estimating equations.

RESULTS: The mean FSS score ± SD at baseline was 5.1±1.4, which did not change significantly during the 5-year follow-up. Reduced physical functioning, increased bodily pain, reduced sleep quality, more psychologic distress, and higher task-oriented coping were independently associated with fatigue. The extent of paresis and walking capacity were strongly associated with physical functioning.

Conclusions: Fatigue is severe and persistent in patients with LOSP due to physical and psychologic factors, which has implications for counseling and treatment. In addition to the commonly applied interventions targeting physical aspects, psychologic interventions are a potential area for reducing fatigue.

Outcome of Research: Effective

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Category: Diagnosis and Management

Title: A positive turning point in life -- how persons with late effects of polio experience the influence of an interdisciplinary rehabilitation programme
Author: Larsson Lund M (1), Lexell J
Affiliation: (1) Department of Community Medicine and Rehabilitation, Occupational Therapy , Umeå University, Umeå, Sweden
Journal: Journal of Rehabilitation Medicine
Citation: J Rehabil Med. 2010 Jun;42(6):559-65. doi: 10.2340/16501977-0559
Publication Year and Month: 2010 06

Abstract: OBJECTIVE: To describe and enhance our understanding of how persons with late effects of polio experience the influence of an interdisciplinary rehabilitation programme.

PARTICIPANTS: Twelve persons with clinically verified late effects of polio who had participated in an individualized, goal-oriented, comprehensive interdisciplinary rehabilitation programme.

METHODS: Qualitative research interviews analysed using the constant comparative method of grounded theory.

RESULTS: The rehabilitation programme was experienced as a turning point in the participants' lives. Before rehabilitation they felt they were on a downward slope without control. Rehabilitation was the start of a process of change whereby they acquired new skills, which, over time, contributed to a different but good life. After approximately a year, they had a sense of control and had accepted life with late effects of polio. They had also established new habits, taken on a changed valued self and could look to the future with confidence.

Conclusions: This qualitative study has shown that persons with late effects of polio can benefit from an individualized, goal-oriented, comprehensive interdisciplinary rehabilitation programme and experience positive changes in their management of daily activities and in their view of their late effects of polio, their future and their self.

Outcome of Research: Effective

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Category: Sleep Analysis

Title: Analysis of sleep characteristics in post-polio syndrome patients
Author: Silva TM (1), Moreira GA, Quadros AA, Pradella-Hallinan M, Tufik S, Oliveira AS
Affiliation: (1) Department of Neurology, Escola Paulista de Medicina, Universidade Federal de São Paulo, São Paulo, SP, Brazil. [email protected]
Journal: Arquivos de Neuropsiquiatria
Citation: Arq Neuropsiquiatr. 2010 Aug;68(4):535-40
Publication Year and Month: 2010 08

Abstract: The main post-polio syndrome (PPS) symptoms are new weakness, new atrophy, fatigue, pain and sleep disturbances. Polysomnography is the gold standard for sleep analysis.

OBJECTIVE: To analyze sleep patterns in PPS patients.

METHOD: Sixty patients (mean age 46.8+/-11.3 years) at the Federal University of São Paulo (UNIFESP/EPM) complaining of sleep disturbances were evaluated by means of polysomnography, performed at the Sleep Institute.

RESULTS: Sleep efficiency was lower due to high sleep latency and arousal index. The apnea and hypopnea index (AHI) and the periodic limb movements (PLM) index were higher. Sleep architecture was also impaired. There were no abnormalities of oxygen saturation, carbon dioxide levels, respiratory rate or heart rate.

Conclusions: New post-polio sleep disturbances were isolated symptoms. It appears that these symptoms were not due to post-polio features, but rather, that they were due to dysfunction of the surviving motor neurons in the brainstem. Abnormal dopamine production, which is responsible for many sleep-related breathing disorders and abnormal movements, may also have been implicated in the present findings.

Outcome of Research: Effective

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Category: Activity Levels

Title: Change in physical mobility over 10 years in post-polio syndrome.
Author: Bickerstaffe, A., Beelen, A., Nollet, F.
Affiliation: Department of Rehabilitation, Academic Medical Centre, The Netherlands
Journal: Neuromuscular Disorders
Citation: Bickerstaffe, A., Beelen, A., Nollet, F. (2015) Change in physical mobility over 10 years in post-polio syndrome. Neuromuscular Disorders. 25(3):225-30
Publication Year and Month: 2015 03

Abstract: Post-polio syndrome is characterised by progressive muscle weakness and other symptoms which can limit physical mobility. We assessed the rate of decline in mobility over 10 years in relation to strength decline; and investigated potential predictors for the rate of decline of walking capacity, a measure of mobility, in 48 patients with post-polio syndrome and proven quadriceps dysfunction at baseline. Average walking capacity and self-reported physical mobility declined over 10 years, by 6 and 14%, respectively. Concomitantly people lost an average of 15% of isometric quadriceps strength. Significantly more people used walking aids offering greater support at follow-up. Notably, there was much individual variation, with 18% of participants losing a substantial amount of walking capacity (27% decline) and concomitant self-reported physical mobility (38% decline). Loss of quadriceps strength only explained a small proportion of the variance of the decline in walking capacity (R = 11%) and the rate of decline could not be predicted from baseline values for strength, walking capacity, self-reported physical mobility or basic demographics. The individual variability, yet lack of predictive factors, underscores the need for personally tailored care based on actual functional decline in patients with post-polio syndrome.

Conclusions: • The majority of post-polio patients experienced modest declines in physical mobility in 10 years.
• One-fifth of patients experienced substantial declines in walking capacity.
• The rate of decline in walking capacity could not be predicted from baseline quadriceps strength.
• These findings underscore the need for personally tailored care based on actual functional
decline.

Outcome of Research: Effective

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Category: Exercise

Title: Comparison of two 6-minute walk tests to assess walking capacity in polio survivors
Author: Merel-Anne Brehm, PhD, Suzan Verduijn, MSc, Jurgen Bon, MD, Nicoline Bredt, MSc and Frans Nollet, MD, PhD
Affiliation: The authors declare no conflicts of interest.
Journal: Journal of Rehabilitation Medicine
Citation: Merel-Anne Brehm, PhD, Suzan Verduijn, MSc, Jurgen Bon, MD, Nicoline Bredt, MSc and Frans Nollet, MD, PhD. Comparison of two 6-minute walk tests to assess walking capacity in polio survivors. J Rehabil Med 2017; 49: 00–00
Publication Year and Month: 2017 09

Abstract: Objective: To compare walking dynamics and test-retest reliability for 2 frequently applied walk tests in polio survivors: the 6-minute walk test (6MWT) to walk as far as possible; and the 6-minute walking energy cost test (WECT) at comfortable speed.

Design: Observational study.

Participants: Thirty-three polio survivors, able to walk ≥ 150 m.

Methods: On the same day participants performed a 6MWT and a WECT, which were repeated 1–3 weeks later. For each test, distance walked, heart rate and reduction in speed were assessed.

Results: The mean distance walked and mean heart rate were significantly higher in the 6MWT (441 m (standard deviation) (SD 79.7); 118 bpm (SD 19.2)) compared with the WECT (366 m (SD 67.3); 103 bpm (SD 14.3)); p < 0.001. Furthermore, during the 6MWT, patients continuously slowed down (–6%), while during the WECT speed dropped only slightly during the first 2 min, by –1.8% in total. Test-retest reliability of both tests was excellent (intraclass correlation coefficient (ICC) ≥ 0.95; lower bound 95% confidence interval (95% CI) ≥ 0.87). The smallest detectable change for the walked distance was 42 m (9.7% change from the mean) and 50 m (13.7%) on the 6MWT and WECT, respectively.

Conclusion: Both the 6MWT and the WECT are reliable to assess walking capacity in polio survivors, with slightly superior sensitivity to detect change for the 6MWT. Differences in walking dynamics confirm that the tests cannot be used interchangeably. The 6MWT is recommended for measuring maximal walking capacity and the WECT for measuring submaximal walking capacity.

Conclusions: In conclusion, this study of polio survivors with a minimum self-reported walking distance of 150 m shows that both the 6MWT and the WECT are reliable and can be used to evaluate changes in walking capacity, with the 6MWT showing slightly superior sensitivity to detect change. The study also shows a significantly higher heart rate (57%HRR on average) at the expense of a reduction in walking speed at this heart rate during the 6MWT compared with the WECT. These findings indicate distinct patterns of walking dynamics between the 6MWT and WECT, where the 6MWT is more likely a measure of maximal walking capacity (i.e. what a person can do) and the WECT of submaximal walking capacity (i.e. what a person does do). The difference in walking dynamics confirms that these tests cannot be used interchangeably, and that the choice to use either test should be tailored to the construct to be measured. Responsiveness to change in this patient population should be further investigated for both tests.

Outcome of Research: Effective

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Category: Exercise

Title: Dynamic water exercise in individuals with late poliomyelitis
Author: Willén C, Sunnerhagen KS, Grimby G
Affiliation: Department of Rehabilitation Medicine, Göteborg University, Göteborg, Sweden - [email protected]
Journal: Archives of Physical Medicine and Rehabilitation
Citation: Arch Phys Med Rehabil. 2001 Jan;82(1):66-72
Publication Year and Month: 2001 01

Abstract: OBJECTIVE: To evaluate the specific effects of general dynamic water exercise in individuals with late effects of poliomyelitis.

DESIGN: Before-after tests.

SETTING: A university hospital department.

PARTICIPANTS: Twenty-eight individuals with late effects of polio, 15 assigned to the training group (TG) and 13 to the control group (CG).

INTERVENTION: The TG completed a 40-minute general fitness training session in warm water twice weekly. Assessment instruments included the bicycle ergometer test, isokinetic muscle strength, a 30-meter walk indoors, Berg balance scale, a pain drawing, a visual analog scale, the Physical Activity Scale for the Elderly, and the Nottingham Health Profile (NHP).

MAIN OUTCOME MEASURES: Peak load, peak work load, peak oxygen uptake, peak heart rate (HR), muscle function in knee extensors and flexors, and pain dimension of the NHP.

RESULTS: The average training period was 5 months; compliance was 75% (range, 55-98). No negative effects were seen. The exercise did not influence the peak work load, peak oxygen uptake, or muscle function in knee extensors compared with the controls. However, a decreased HR at the same individual work load was seen, as well as a significantly lower distress in the dimension pain of the NHP. Qualitative aspects such as increased well-being, pain relief, and increased physical fitness were reported.

Conclusions: A program of nonswimming dynamic exercises in heated water has a positive impact on individuals with late effects of polio, with a decreased HR at exercise, less pain, and a subjective positive experience. The program was well tolerated (no adverse effects were reported) and can be recommended for this group of individuals.

Outcome of Research: Effective.

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Category: Inflammation

Title: Elevated expression of prostaglandin E2 synthetic pathway in skeletal muscle of prior polio patients
Author: Melin E (1), Lindroos E, Lundberg IE, Borg K, Korotkova M
Affiliation: (1) Department of Clinical Sciences, Karolinska Institutet Danderyds Hospital, 18288 Stockholm, Sweden. [email protected]
Journal: Journal of Rehabilitation Medicine
Citation: J Rehabil Med. 2014 Jan;46(1):67-72. doi: 10.2340/16501977-1230
Publication Year and Month: 2014 01

Abstract: OBJECTIVE: The aim of this study was to investigate signs of inflammation in muscle of patients with prior polio, since the main symptoms in these patients are muscle pain, weakness and fatigue. In the context of pain and inflammation, the prostaglandin E2 pathway is of interest. Prostaglandin E2 has many biological actions and is a mediator of inflammation and pain.

PATIENTS AND METHODS: Skeletal muscle biopsies from 8 patients with prior polio and post-polio symptoms, presenting with pain and muscular weakness, and from 6 healthy controls were studied. Immunohistochemistry, conventional microscopy, and computerized image analysis were performed.

RESULTS: There was statistically significant higher expression of enzymes of the prostaglandin E2 synthetic pathway, in muscle from patients, compared with controls. Expression of prostaglandin enzymes was mainly in scattered cells and blood vessels, and may indicate an inflammatory process of the muscle, which could be secondary to systemic inflammation.

Conclusions: This data may indicate an inflammatory process in muscle of prior polio patients. Up-regulation of the prostaglandin E2 pathway reveals a potential background to the pain experienced by these patients, and may provide opportunities for directed pharmacological and physical therapies, which could lead to better outcomes of rehabilitation interventions.

Outcome of Research: Effective

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Category: Assistive Technology, Orthoses

Title: Evaluation of gait symmetry in poliomyelitis subjects: Comparison of a conventional knee-ankle-foot orthosis and a new powered knee-ankle-foot orthosis
Author: Arazpour M (1), Ahmadi F (2), Bahramizadeh M (2), Samadian M (3), Mousavi ME (2), Bani MA (4), Hutchins SW (5)
Affiliation: (1) Department of Orthotics and Prosthetics, University of Social Welfare and Rehabilitation Sciences, Tehran, Islamic Republic of Iran Pediatric Neurorehabilitation Research Center, University of Social Welfare and Rehabilitation Sciences, Tehran, Iran; (2) Department of Orthotics and Prosthetics, University of Social Welfare and Rehabilitation Sciences, Tehran, Islamic Republic of Iran; (3) Loghman Hakim Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Islamic Republic of Iran; (4) Department of Orthotics and Prosthetics, University of Social Welfare and Rehabilitation Sciences, Tehran, Islamic Republic of Iran; (5) Institute of Health & Social Care Research (IHSCR), Faculty of Health & Social Care, University of Salford, Manchester, Salford, UK
Journal: Prosthetics and Orthotics International
Citation: Prosthet Orthot Int. 2015 Aug 12. pii: 0309364615596063
Publication Year and Month: 2015 08

Abstract: BACKGROUND: Compared to able-bodied subjects, subjects with post-polio syndrome and poliomyelitis demonstrate a preference for weight-bearing on the non-paretic limb, causing gait asymmetry.

OBJECTIVES: The purpose of this study was to evaluate the gait symmetry of the poliomyelitis subjects when ambulating with either a drop-locked knee-ankle-foot orthosis or a newly developed powered knee-ankle-foot orthosis.

STUDY DESIGN: Quasi experimental study.

METHODS: Seven subjects with poliomyelitis who routinely wore conventional knee-ankle-foot orthoses participated in this study and received training to enable them to ambulate with the powered knee-ankle-foot orthosis on level ground, prior to gait analysis.

RESULTS: There were no significant differences in the gait symmetry index of step length (p = 0.085), stance time (p = 0.082), double-limb support time (p = 0.929), or speed of walking (p = 0.325) between the two test conditions. However, using the new powered knee-ankle-foot orthosis improved the symmetry index in step width (p = 0.037), swing time (p = 0.014), stance phase percentage (p = 0.008), and knee flexion during swing phase (p ⩽ 0.001) compared to wearing the drop-locked knee-ankle-foot orthosis.

Conclusions: The use of a powered knee-ankle-foot orthosis for ambulation by poliomyelitis subjects affects gait symmetry in the base of support, swing time, stance phase percentage, and knee flexion during swing phase.

CLINICAL RELEVANCE: A new powered knee-ankle-foot orthosis can improve gait symmetry for poliomyelitis subjects by influencing step width, swing time, stance time percentage, and knee flexion during swing phase when compared to ambulating with a drop-locked knee-ankle-foot orthosis.

Outcome of Research: Effective

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Category: Diagnosis and Management

Title: Frequency and clinical manifestations of post-poliomyelitis syndrome in a Brazilian tertiary care center
Author: Quadros AA (1), Conde MT, Marin LF, Silva HC, Silva TM, Paula MB, Pereira RD, Ramos PE, Abe G, Oliveira AS
Affiliation: (1) Department of Neurology and Neurosurgery, Division of Neuromuscular Disorders, Federal University of São Paulo (Unifesp), São Paulo SP, Brazil - [email protected]
Journal: Arquivos de Neuro-psiquiatria
Citation: Arq Neuropsiquiatr. 2012 Aug;70(8):571-3
Publication Year and Month: 2012 08

Abstract: OBJECTIVE: To determine the frequency and clinical manifestations of patients with post-poliomyelitis syndrome (PPS) in a Brazilian division of neuromuscular disorders.

METHODS: A total of 167 patients with prior history of paralytic poliomyelitis was investigated for PPS, based on international diagnostic criteria. Other variables analyzed were: gender, race, age at poliomyelitis infection, age at PPS onset, and PPS symptoms.

RESULTS: One hundred and twenty-nine patients presented PPS, corresponding to 77.2% of the studied population. 62.8% were women and 37.2% were men. Mean age of patients with PPS at onset of PPS symptoms was 39.9±9.69 years. Their main clinical manifestations were: new weakness in the previously affected limbs (69%) and in the apparently not affected limbs (31%); joint pain (79.8%); fatigue (77.5%); muscle pain (76%); and cold intolerance (69.8%).

Conclusions: Most patients of our sample presented PPS. In Brazil, PPS frequency and clinical features are quite similar to those of other countries.

Outcome of Research: Effective

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Category: Orthoses

Title: Gait patterns in association with underlying impairments in polio survivors with calf muscle weakness
Author: Ploeger, H.E., Bus, S.A., Nollet, F., Brehm, M-A.
Affiliation: Nil identified
Journal:
Citation: Ploeger, H.E., Bus, S.A., Nollet, F., Brehm, M-A. Gait patterns in association with underlying impairments in polio survivors with calf muscle weakness. Gait & Posture. 2017 58:146-153. doi: 10.1016/j.gaitpost.2017.07.107.
Publication Year and Month: 2017 07

Abstract: The objective was to identify gait patterns in polio survivors with calf muscle weakness and associate them to underlying lower extremity impairments, which are expected to help in the search for an optimal orthosis.

Unilaterally affected patients underwent barefoot 3D-gait analyses. Gait pattern clusters were created based on the ankle and knee angle and ankle moment shown in midstance of the affected limb. Impairment clusters were created based on plantarflexor and knee-extensor strength, and ankle and knee joint range-of-motion. The association between gait patterns and underlying impairments were examined descriptively. The Random Forest Algorithm and regression analyses were used to predict gait patterns and parameters.

Seven gait patterns in 73 polio survivors were identified, with two dominant patterns: one with a mildly/non-deviant ankle angle, ankle moment and knee angle (n = 23), and one with a strongly deviant ankle angle and a mildly/non-deviant ankle moment and knee angle (n = 18). Gait pattern prediction from underlying impairments was 49% accurate with best prediction performance for the second dominant gait pattern (sensitivity 78% and positive predictive value 74%). The underlying impairments explained between 20 and 32% of the variance in individual gait parameters.

Polio survivors with calf muscle weakness who present a similar impairment profile do not necessarily walk the same. From physical examination alone, the gait pattern nor the individual gait parameters could be accurately predicted. The patient’s gait should therefore be measured to help in the prescription and evaluation of orthoses for these patients.

Conclusions:

Outcome of Research: Effective

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Comments (if any): There are many differences in gait patterns and it can be difficult to detect the forces and weight distribution from observation. 3D gait analysis appears to be more effective in combination with physical examination of muscle weakness in prescription of effective orthoses.

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Category: Polio Immunisation

Title: Immunogenicity of two different sequential schedules of inactivated polio vaccine followed by oral polio vaccine versus oral polio vaccine alone in healthy infants in China
Author: Li RC (1), Li CG (2), Wang HB (3), Luo HM (3), Li YP (1), Wang JF (2), Ying ZF (2), Yu WZ (3), Shu JD (4), Wen N (3), Vidor E (5)
Affiliation: (1) Guangxi Center for Disease Prevention and Control, Nanning, China; (2) National Institutes for Food and Drug Control (NIFDC), Beijing, China; (3) Chinese Center for Disease Control and Prevention, Beijing, China; (4) Sanofi Pasteur, Beijing, China; (5) Sanofi Pasteur, Lyon, France
Journal: Journal of the Pediatric Infectious Diseases Society
Citation: J Pediatric Infect Dis Soc. 2015 Apr 16. pii: piv017
Publication Year and Month: 2015 04

Abstract: BACKGROUND: Two vaccination schedules where inactivated polio vaccine (IPV) was followed by oral polio vaccine (OPV) were compared to an OPV-only schedule.

METHODS: Healthy Chinese infants received a 3-dose primary series of IPV-OPV-OPV (Group A), IPV-IPV-OPV (Group B), or OPV-OPV-OPV (Group C) at 2, 3, and 4 months of age. At pre-Dose 1, 1-month, and 14-months post-Dose 3, polio 1, 2, and 3 antibody titers were assessed by virus-neutralizing antibody assay with Sabin or wild-type strains. Adverse events were monitored.

RESULTS: Anti-polio 1, 2, and 3 titers were ≥8 (1/dil) in >99% of participants, and Group A and Group B were noninferior to Group C at 1-month post-Dose 3 as assessed by Sabin strain-based assay (SSBA). In Group A 1-month post-Dose 3, there was no geometric mean antibody titers (GMT) differences for types 1 and 3; type 2 GMTs were ≈3-fold higher by wild-type strain-based assay (WTBA) versus SSBA. For Group B, GMTs were ≈1.7- and 3.6-fold higher for types 1 and 2 via WTBA, while type 3 GMTs were similar. For Group C, GMTs were ≈6.3- and 2-fold higher for types 1 and 3 with SSBA, and type 2 GMTs were similar. Antibodies persisted in >96.6% of participants. Adverse event incidence in each group was similar.

Conclusions: A primary series of 1 or 2 IPV doses followed by 2 or 1 OPV doses was immunogenic and noninferior to an OPV-only arm. SSBA was better at detecting antibodies elicited by OPV with antibody titers correlated to the number of OPV doses (NCT01475539 - https://clinicaltrials.gov/ct2/show/study/NCT01475539).

Outcome of Research: Effective

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Category: Drugs

Title: Immunoglobulin treatment in post-polio syndrome: Identification of responders and non-responders
Author: Östlund G (1), Broman L, Werhagen L, Borg K
Affiliation: (1) Department of Rehabilitation Medicine, Danderyd University Hospital, Building 39, 3rd floor, SE-182 88 Stockholm, Sweden
Journal: Journal of Rehabilitation Medicine
Citation: J Rehabil Med. 2015 Aug 18. doi: 10.2340/16501977-1985
Publication Year and Month: 2015 08

Abstract: OBJECTIVE: To define and characterize responders and non-responders in a group of 124 patients with post-polio syndrome who received a single treatment with intravenous immunoglobulin.

DESIGN: Open trial, prospective follow-up study.

METHODS: Clinical examination and data from medical records. Short Form 36 (SF-36), Physical Activity Scale for the Elderly (PASE) and visual analogue scale (VAS) measured quality of life, physical activity and intensity of pain, respectively. Data were obtained before treatment and at 6-month follow-up.

RESULTS: Two responder groups were identified with the outcome SF-36 Vitality and 3 with Bodily pain, respectively. Forty-five percent were positive-responders, identified before treatment by reduced physical function, muscle atrophy in the lower extremities, higher levels of fatigue and pain, and a VAS pain score above 20. Negative-responders were identified by good physical function and mental health, lesser muscle atrophy in the lower extremities, and low levels of fatigue and pain.

Conclusions: Intravenous immunoglobulin is a biological intervention, and therefore it is important to be able to identify responders and non-responders. In order to maximize a positive outcome it is suggested that patients with a high level of fatigue and/or pain and reduced physical function are selected.

Outcome of Research: Effective

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Category: Drugs

Title: IVIG treatment in post-polio patients: evaluation of responders
Author: Ostlund G (1), Broman L, Werhagen L, Borg K
Affiliation: (1) Division of Rehabilitation Medicine, Department of Clinical Sciences, Karolinska Institute, Danderyd Hospital, Building 39, 3rd floor, 182 88 Stockholm, Sweden
Journal: Journal of Neurology
Citation: J Neurol. 2012 Dec;259(12):2571-8. doi: 10.1007/s00415-012-6538-y. Epub 2012 May 17
Publication Year and Month: 2012 05

Abstract: The aim of this work is to evaluate the outcome of IVIG treatment in patients with post-polio syndrome (PPS) and to identify responders. The study included 113 PPS patients who had received one IVIG treatment in an open trial, prospective follow-up study. Clinical examination was performed and clinical data were retrieved from medical records. The short form 36 (SF-36), physical activity scale for the elderly (PASE), and the visual analogue scale (VAS) were used as measurements of quality of life, physical activity, and the intensity of pain. Data before treatment and at 6-month follow-up were collected. Analysis was performed in subgroups based on demographic and medical parameters. A statistically significant increase of the SF-36 sub domains bodily pain, vitality, social function, role emotional, and the mental compound score (MCS) was found at the 6-month follow-up. A significant decrease of pain was found in patients who reported pain intensity over VAS of 20 mm, in patients younger than 65 years of age and in patients who had paresis in the lower extremities. A trend was found in patients with PPS as the only diagnosis. IVIG leads to increase of quality of life at 6-month follow-up for SF-36 regarding sub domains of bodily pain, vitality, social function, role emotional, as well as for pain. Age below 65 years, paresis in the lower extremities, and lack of concomitant disorders may be the main indicators for a future identification of responders.

Conclusions:

Outcome of Research: Effective

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Category: Post-Polio Motor Unit

Title: Loss of motor unit size and quadriceps strength over 10 years in post-polio syndrome
Author: Bickerstaffe A (1), van Dijk JP (2), Beelen A (3), Zwarts MJ (4), Nollet F (5)
Affiliation: (1) Department of Rehabilitation, Academic Medical Center (AMC), Postbus 22660, 1100 DD Amsterdam, The Netherlands. Electronic address: [email protected]; (2) Radboud University Nijmegen Medical Centre, Donders Institute for Brain, Cognition and Behaviour, Department of Neurology/Clinical Neurophysiology, Postbus 910, 6500 HB Nijmegen, The Netherlands; Epilepsy Centre Kempenhaeghe, Postbus 61, 5590 AB Heeze, The Netherlands. Electronic address: [email protected]; (3) Department of Rehabilitation, Academic Medical Center (AMC), Postbus 22660, 1100 DD Amsterdam, The Netherlands. Electronic address: [email protected]; (4) Epilepsy Centre Kempenhaeghe, Postbus 61, 5590 AB Heeze, The Netherlands. Electronic address: [email protected]; (5) Department of Rehabilitation, Academic Medical Center (AMC), Postbus 22660, 1100 DD Amsterdam, The Netherlands. Electronic address: [email protected]
Journal: Clinical Neurophysiology
Citation: Clin Neurophysiol. 2014 Jun;125(6):1255-60. doi: 10.1016/j.clinph.2013.11.003
Publication Year and Month: 2014 06

Abstract: OBJECTIVE: To investigate whether strength decline in post-polio syndrome (PPS) results from excessive distal axonal degeneration of enlarged motor units.

METHODS: We assessed changes over 10 years in isometric quadriceps strength, mean motor unit action potential (MUAP) size, root mean squared (RMS) amplitude, and level of interference (LOI) in 47 patients with PPS and 12 healthy controls, using high density surface EMG. At baseline, all patients had symptomatic quadriceps dysfunction, evidenced by transmission defects on single-fibre EMG.

RESULTS: MU size and strength declined significantly by 20% and 15%, respectively in patients with PPS. Those with the largest initial MU sizes exhibited the greatest losses of mean MU size (27%) and proportional decreases in quadriceps strength (23%). Initial strength, change in LOI and change in RMS amplitude together explained 35% of the variability in strength changes in patients. MU size of controls did not change, although they lost 29% strength.

SIGNIFICANCE: This long term follow-up study provides evidence that size diminution of enlarged MUs combined with a reduced number of active MUs contributes to the gradual strength decline in PPS.

Conclusions: MU size and strength declined concomitantly in a homogeneous cohort of patients with PPS and quadriceps dysfunction.

Outcome of Research: Effective

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Category: Polio Immunisation

Title: Next generation inactivated polio vaccine manufacturing to support post polio-eradication biosafety goals
Author: Thomassen YE (1), van 't Oever AG (1), van Oijen MG (1), Wijffels RH (2), van der Pol LA (1), Bakker WA (1)
Affiliation: (1) Institute for Translational Vaccinology (Intravacc), Bilthoven, The Netherlands; (2) Bioprocess Engineering, Wageningen University, Wageningen, The Netherlands
Journal: Public Library of Science
Citation: PLoS One. 2013 Dec 12;8(12):e83374. doi: 10.1371/journal.pone.0083374
Publication Year and Month: 2013 12

Abstract: Worldwide efforts to eradicate polio caused a tipping point in polio vaccination strategies. A switch from the oral polio vaccine, which can cause circulating and virulent vaccine derived polioviruses, to inactivated polio vaccines (IPV) is scheduled. Moreover, a manufacturing process, using attenuated virus strains instead of wild-type polioviruses, is demanded to enhance worldwide production of IPV, especially in low- and middle income countries. Therefore, development of an IPV from attenuated (Sabin) poliovirus strains (sIPV) was pursued. Starting from the current IPV production process based on wild type Salk strains, adaptations, such as lower virus cultivation temperature, were implemented. sIPV was produced at industrial scale followed by formulation of both plain and aluminium adjuvanted sIPV. The final products met the quality criteria, were immunogenic in rats, showed no toxicity in rabbits and could be released for testing in the clinic. Concluding, sIPV was developed to manufacturing scale. The technology can be transferred worldwide to support post polio-eradication biosafety goals.

Conclusions:

Outcome of Research: Effective

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Category: Diagnosis and Management

Title: Post-poliomyelitis syndrome as a possible viral disease
Author: Baj A (1), Colombo M (1), Headley JL (2), McFarlane JR (3), Liethof MA (4), Toniolo A (5)
Affiliation: (1) Laboratory of Clinical Microbiology, University of Insubria Medical School, Viale Borri 57, 21100 Varese, Italy; (2) Post-Polio Health International, Saint Louis, Missouri, USA; (3) European Polio Union, Huldenberg, Belgium; (4) Polio Australia Incorporated, Kew, Victoria, Australia; (5) Laboratory of Clinical Microbiology, University of Insubria Medical School, Viale Borri 57, 21100 Varese, Italy. Electronic address: [email protected]
Journal: International Journal of Infectious Diseases
Citation: Int J Infect Dis. 2015 May 1;35:107-116. doi: 10.1016/j.ijid.2015.04.018
Publication Year and Month: 2015 05

Abstract: This review summarizes current concepts on post-polio syndrome (PPS), a condition that may arise in polio survivors after partial or complete functional recovery followed by a prolonged interval of stable neurological function. PPS affects 15-20 million people worldwide. Epidemiological data are reported, together with the pathogenic pathways that possibly lead to the progressive degeneration and loss of neuromuscular motor units. As a consequence of PPS, polio survivors experience new weakness, generalized fatigue, atrophy of previously unaffected muscles, and a physical decline that may culminate in the loss of independent life. Emphasis is given to the possible pathogenic role of persistent poliovirus infection and chronic inflammation. These factors could contribute to the neurological and physical decline in polio survivors. A perspective is then given on novel anti-poliovirus compounds and monoclonal antibodies that have been developed to contribute to the final phases of polio eradication. These agents could also be useful for the treatment or prevention of PPS. Some of these compounds/antibodies are in early clinical development. Finally, current clinical trials for PPS are reported. In this area, the intravenous infusion of normal human immunoglobulins appears both feasible and promising.

Conclusions:

Outcome of Research: Effective.

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Category: Quality of Life

Title: Quality of life in Swedish patients with post-polio syndrome with a focus on age and sex
Author: Jung TD (1), Broman L, Stibrant-Sunnerhagen K, Gonzalez H, Borg K
Affiliation: (1) Department of Clinical Sciences, Division of Rehabilitation Medicine, Karolinska Institutet, Danderyds Hospital, Stockholm; Institute for Neuroscience and Physiology, Section for Clinical Neuroscience and Rehabilitation, Göteborg University, Göteborg, Sweden; Department of Rehabilitation Medicine, Kyungpook National University Hospital, Daegu, South Korea
Journal: International Journal of Rehabilitation Research
Citation: Int J Rehabil Res. 2014 Jun;37(2):173-9. doi: 10.1097/MRR.0000000000000052
Publication Year and Month: 2014 06

Abstract: To investigate the health-related quality of life (QOL) in Swedish patients with post-polio syndrome (PPS), with a focus on sex and age. A total of 364 patients were recruited from five Swedish post-polio clinics. Analysis was carried out using SF-36 and data were compared with those of a normal population. QOL was significantly lower in PPS patients for all eight subdomains and the two main scores (physical compound score and mental compound score) when compared with the controls. Male patients had a significantly higher QOL than female patients for all subdomains and also for mental compound score and physical compound score, a phenomenon also observed in the normal population. There was a decrease in QOL in the physical domains and an increase in vitality with age. PPS decreases health-related QOL in both sexes, more in female patients. QOL for physical domains decreases whereas vitality increases with age in both sexes.

Conclusions:

Outcome of Research: Effective

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Category: Muscle Strength

Title: Quantitative muscle ultrasound and quadriceps strength in patients with post-polio syndrome
Author: Bickerstaffe A (1), Beelen A, Zwarts MJ, Nollet F, van Dijk JP
Affiliation: (1) Department of Rehabilitation, Academic Medical Center, Postbus 22660, 1100 DD, Amsterdam, The Netherlands
Journal: Muscle & Nerve
Citation: Muscle Nerve. 2015 Jan;51(1):24-9. doi: 10.1002/mus.24272
Publication Year and Month: 2015 01

Abstract: INTRODUCTION: We investigated whether muscle ultrasound can distinguish muscles affected by post-polio syndrome (PPS) from healthy muscles and whether severity of ultrasound abnormalities is associated with muscle strength.

METHODS: Echo intensity, muscle thickness, and isometric strength of the quadriceps muscles were measured in 48 patients with PPS and 12 healthy controls.

RESULTS: Patients with PPS had significantly higher echo intensity and lower muscle thickness than healthy controls. In patients, both echo intensity and muscle thickness were associated independently with muscle strength. A combined measure of echo intensity and muscle thickness was more strongly related to muscle strength than either parameter alone.

Conclusions: Quantitative ultrasound distinguishes healthy muscles from those affected by PPS, and measures of muscle quality and quantity are associated with muscle strength. Hence, ultrasound could be a useful tool for assessing disease severity and monitoring changes resulting from disease progression or clinical intervention in patients with PPS.

Outcome of Research: Effective

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Category: Clinical Evaluation

Title: Quantitative muscle ultrasound and quadriceps strength in patients with post-polio syndrome.
Author: Bickerstaffe, A., Beelen, A., Zwarts, M.J., Nollet, F., van Dijk, J.P.
Affiliation: Department of Rehabilitation, Academic Medical Center, The Netherlands
Journal: Muscle & Nerve
Citation: Bickerstaffe, A et al (2015) Quantitative muscle ultrasound and quadriceps strength in patients with post-polio syndrome. Muscle and Nerve. 51(1):24-9
Publication Year and Month: 2015 01

Abstract: INTRODUCTION:
We investigated whether muscle ultrasound can distinguish muscles affected by post-polio syndrome (PPS) from healthy muscles and whether severity of ultrasound abnormalities is associated with muscle strength.
METHODS:
Echo intensity, muscle thickness, and isometric strength of the quadriceps muscles were measured in 48 patients with PPS and 12 healthy controls.
RESULTS:
Patients with PPS had significantly higher echo intensity and lower muscle thickness than healthy controls. In patients, both echo intensity and muscle thickness were associated independently with muscle strength. A combined measure of echo intensity and muscle thickness was more strongly related to muscle strength than either parameter alone.
CONCLUSIONS:
Quantitative ultrasound distinguishes healthy muscles from those affected by PPS, and measures of muscle quality and quantity are associated with muscle strength. Hence, ultrasound could be a useful tool for assessing disease severity and monitoring changes resulting from disease progression or clinical intervention in patients with PPS.

Conclusions:
Ultrasound can be effective to assess and monitor changes in muscle properties of a patient with post-polio syndrome.

Outcome of Research: Effective

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Category: Renal Complications

Title: Renal failure in a patient with postpolio syndrome and a normal creatinine level
Author: Leming MK (1), Breyer MJ
Affiliation: (1) Department of Emergency Medicine, Christiana Care Health System, Newark, DE 19718, USA. [email protected]
Journal: The American Journal of Emergency Medicine
Citation: Am J Emerg Med. 2012 Jan;30(1):247.e1-3. doi: 10.1016/j.ajem.2010.07.026
Publication Year and Month: 2012 01

Abstract: Patients with renal failure who are taking trimethoprim have an increased risk of developing hyperkalemia, which can cause muscle weakness. In patients with postpolio syndrome, a normal creatinine level could be abnormally high, renal failure is possible because of lack of creatinine production, and the muscle weakness from resultant hyperkalemia could be more severe because of their underlying condition. This abnormally high creatinine level has been termed from this point relative renal failure. The objective of the study was to review a case in which relative renal failure and hyperkalemia caused muscle weakness that manifested as shortness of breath and confusion with electrocardiographic changes. A dehydrated patient with relative renal failure and postpolio syndrome had taken trimethoprim-sulfamethoxazole that caused symptomatic hyperkalemia. The patient presented with muscle weakness, shortness of breath, and confusion, with her postpolio syndrome compounding the situation and likely making the muscle weakness more severe. A patient on trimethoprim with renal failure is at an increased risk of developing hyperkalemia. Patients with postpolio syndrome could have severe muscle weakness from the hyperkalemia and could have renal failure even with a normal creatinine level. This case report will remind treating physicians to evaluate such patients for hyperkalemia if they present with muscle weakness, especially if the patient has renal failure and is on trimethoprim.

Conclusions:

Outcome of Research: Effective

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Category: Drugs

Title: Response of postpoliomyelitis patients to bisphosphonate treatment
Author: Alvarez A (1), Kremer R, Weiss DR, Benedetti A, Haziza M, Trojan DA
Affiliation: (1) Department of Neurology and Neurosurgery, Montreal Neurological Institute and Hospital, McGill University Health Centre, McGill University, Montreal, QC, Canada
Journal: PM&R: The Journal of Injury, Function, and Rehabilitation
Citation: PM R. 2010 Dec;2(12):1094-103. doi: 10.1016/j.pmrj.2010.08.009.
Publication Year and Month: 2010 12

Abstract: OBJECTIVE: To evaluate (1) the rate of change of bone mineral density (BMD) at the hip in postpolio patients treated with bisphosphonates compared with the rate of change in BMD in (a) postpolio patients not treated with bisphosphonates and (b) non-postpolio patients treated with bisphosphonates; and (2) to compare the fracture rate in postpolio patients before and after treatment.

DESIGN: Retrospective chart review.

SETTING: University-affiliated hospital postpolio clinic and bone metabolism clinic.

PARTICIPANTS: Patients with at least 2 BMD assessments. We included 144 postpolio patients and 112 non-postpolio patients. For the fracture analysis, 32 postpolio patients with a history of fractures and treatment with bisphosphonates were included.

METHODS: The effect of treatment on BMD in postpolio patients was analyzed with use of a multiple linear regression model and a mixed effects model, with the rate of change in hip BMD and the change in BMD from baseline, respectively, as the dependent variables. The effect of treatment on occurrence of fractures in postpolio patients was analyzed with use of conditional logistic regression and Poisson regression.

MAIN OUTCOME MEASURES: BMD measurements at the femoral neck (g/cm²) and occurrence of fractures before and after initiation of treatment.

RESULTS: In an adjusted model, postpolio patients treated with bisphosphonates (54/144) had a greater rate of change in BMD (0.031 g/cm²/year; 95% confidence interval 0.010-0.052) compared with nontreated postpolio patients. The effect of treatment in postpolio patients was similar to that in non-postpolio patients. Evidence indicated that treated postpolio patients have a lower risk of fracture after treatment (odds ratio 0.3, P = .046; rate ratio 0.4, P = .183).

Conclusions: In this retrospective study, it was found that treatment with oral bisphosphonates significantly increases BMD at the hip in postpolio patients. The effect of bisphosphonate treatment appears to be similar in postpolio patients compared with a control group without polio. Treatment with bisphosphonates may have a protective effect on fracture risk in this population.

Outcome of Research: Effective

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Category: Restless Legs Syndrome

Title: Restless legs syndrome in patients with sequelae of poliomyelitis
Author: Kumru H (1), Portell E (2), Barrio M (2), Santamaria J (3)
Affiliation: (1) Institut Guttmann, Institut Universitari de Neurorehabilitació adscrit a la UAB, 08916 Badalona, Barcelona, Spain; Univ Autonoma de Barcelona, 08193 Bellaterra, Cerdanyola del Vallès, Spain; Fundació Institut d'Investigació en Ciències de la Salut Germans Trias i Pujol, Badalona, Barcelona, Spain; (2) Institut Guttmann, Institut Universitari de Neurorehabilitació adscrit a la UAB, 08916 Badalona, Barcelona, Spain; Univ Autonoma de Barcelona, 08193 Bellaterra, Cerdanyola del Vallès, Spain; Fundació Institut d'Investigació en Ciències de la Salut Germans Trias i Pujol, Badalona, Barcelona, Spain; (3) Hospital Clinic, Institut d'Investigació Biomèdica August Pi I Sunyer (IDIBAPS), University of Barcelona, Barcelona, Spain
Journal: Parkinsonism & Related Disorders
Citation: Parkinsonism Relat Disord. 2014 Oct;20(10):1056-8. doi: 10.1016/j.parkreldis.2014.06.014
Publication Year and Month: 2014 06

Abstract: BACKGROUND: No studies have examined the association between RLS and the sequelae of poliomyelitis (PM). We studied the frequency and severity of RLS in a group of consecutive patients with the sequelae of poliomyelitis (PM) and the effect of treatment with dopaminergic drugs.

METHODS: A diagnosis of RLS was made according to the criteria of the International RLS Study Group, and severity was assessed by the RLS rating scale. Information on sex, age, age at onset, site affected by PM, disease duration of PM, and history of post-polio syndrome (pPS) was obtained in a cohort of 52 PM patients.

RESULT: The mean age was 55.9 ± 6.5 years; 39 patients had post-polio syndrome (75%). RLS was diagnosed in 21 (40.4%) patients. Sixteen of the 21 patients (76.2%) with RLS had pPS, which was similar to the non-RLS group (74.2% patients with pPS). RLS symptoms were very severe in 5 patients, severe in 13, moderate in 2 and mild in 1. Nineteen of the 21 patients with RLS had symptoms predominantly in the more affected lower limb (90% of patients). Sixteen patients received dopaminergic agonist treatment with a significant reduction in their scores on the RLS severity scale from 28.3 ± 4.7 to 6.9 ± 7.3 (p < 0.001).

DISCUSSION: RLS occurs frequently in patients with PM, both in those with and without pPS, and responds well to treatment with dopaminergic drugs.

Conclusions:

Outcome of Research: Effective

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Category: Respiratory Complications and Management

Title: Sleep-disordered breathing in neuromuscular disease
Author: Aboussouan LS
Affiliation: Respiratory Institute, Cleveland Clinic, Cleveland, Ohio
Journal: American Journal of Respiratory and Critical Care Medicine
Citation: Am J Respir Crit Care Med. 2015 May 1;191(9):979-89. doi: 10.1164/rccm.201412-2224CI
Publication Year and Month: 2015 05

Abstract: Sleep-disordered breathing in neuromuscular diseases is due to an exaggerated reduction in lung volumes during supine sleep, a compromised physiologic adaptation to sleep, and specific features of the diseases that may promote upper airway collapse or heart failure. The normal decrease in the rib cage contribution to the tidal volume during phasic REM sleep becomes a critical vulnerability, resulting in saw-tooth oxygen desaturation possibly representing the earliest manifestation of respiratory muscle weakness. Hypoventilation can occur in REM sleep and progress into non-REM sleep, with continuous desaturation and hypercarbia. Specific characteristics of neuromuscular disorders, such as pharyngeal neuropathy or weakness, macroglossia, bulbar manifestations, or low lung volumes, predispose patients to the development of obstructive events. Central sleep-disordered breathing can occur with associated cardiomyopathy (e.g., dystrophies) or from instability in the control of breathing due to diaphragm weakness. Mitigating factors such as recruitment of accessory respiratory muscles, reduction in REM sleep, and loss of normal REM atonia in some individuals may partially protect against sleep-disordered breathing. Noninvasive ventilation, a standard-of-care management option for sleep-disordered breathing, can itself trigger specific sleep-disordered breathing events including air leaks, patient-ventilator asynchrony, central sleep apnea, and glottic closure. These events increase arousals, reduce adherence, and impair sleep architecture. Polysomnography plays an important role in addressing pitfalls in the diagnosis of sleep-disordered breathing in neuromuscular diseases, identifying sleep-disordered breathing triggered by noninvasive ventilation, and optimizing noninvasive ventilation settings.

Conclusions:

Outcome of Research: Effective.

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Category: Muscle Strength

Title: Strength, endurance, and work capacity after muscle strengthening exercise in postpolio subjects
Author: Agre JC, Rodriquez AA, Franke TM
Affiliation: Department of Rehabilitation Medicine, University of Wisconsin Medical School, Madison, USA
Journal: Archives of Physical Medicine and Rehabilitation
Citation: Arch Phys Med Rehabil. 1997 Jul; 78(7):681-6
Publication Year and Month: 1997 07

Abstract: OBJECTIVE: To determine whether a 12-week home quadriceps muscle strengthening exercise program would increase muscle strength, isometric endurance, and tension time index (TTI) in postpolio syndrome subjects without adversely affecting the surviving motor units or the muscle.

DESIGN: A longitudinal study to investigate the effect of a 12-week exercise program on neuromuscular function and electromyographic variables.

SETTING: Neuromuscular laboratory of a university hospital.

SUBJECTS: Seven subjects were recruited from a cohort of 12 subjects who had participated in a previous exercise study. All subjects had greater than antigravity strength of the quadriceps. Upon completion of a postpolio questionnaire, all acknowledged common postpolio syndrome symptoms such as new fatigue, pain, and weakness; 6 of the 7 acknowledged new strength decline.

INTERVENTION: On Mondays and Thursdays subjects performed three sets of four maximal isometric contractions of the quadriceps held for 5 seconds each. On Tuesdays and Fridays subjects performed three sets of 12 dynamic knee extension exercises with ankle weights.

MAIN OUTCOME MEASURES: Neuromuscular variables of the quadriceps muscles were measured at the beginning and completion of the exercise program and included: isokinetic peak torque (ISOKPT, at 60 degrees/sec angular velocity) and total work performed of four contractions (ISOKTW), isometric peak torque (MVC), endurance (EDUR, time subject could hold isometric contraction at 40% of the initial MVC), isometric tension time index (TTI, product of endurance time and torque at 40% of MVC), and initial and final ankle weight (WGT, kg) lifted. Electromyographic variables included: fiber density (FD), jitter (MCD), and blocking (BLK) from single fiber assessment and median macro amplitude (MACRO). Serum creatine kinase (CK) was also measured initially and at 4-week intervals throughout the study.

RESULTS: The following variables significantly (p < .05) increased: WGT by 47%, ISOKPT, 15%, ISOKTW, 15%; MVC, 36%; EDUR, 21%; TTI, 18%. The following variables did not significantly (p > .05) change: FD, MCD, BLK, MACRO, and CK.

Conclusions: This home exercise program significantly increased strength, endurance, and TTI without apparently adversely affecting the motor units or the muscle, as the EMG and CK variables did not change.

Outcome of Research: Effective

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Category: Sleep

Title: Transcranial direct current stimulation (tDCS) for sleep disturbances and fatigue in patients with post-polio syndrome
Author: Acler M (1), Bocci T, Valenti D, Turri M, Priori A, Bertolasi L
Affiliation: (1) Sezione di Neurologia, Dipartimento di Scienze Neurologiche e del Movimento, Università di Verona, Verona, Italy
Journal: Restorative Neurology and Neuroscience
Citation: Restor Neurol Neurosci. 2013;31(5):661-8. doi: 10.3233/RNN-130321
Publication Year and Month: 2013

Abstract: PURPOSE: Post-polio syndrome develops about 20-40 years after acute paralytic poliomyelitis, and manifests with progressively deteriorating muscle strength and endurance. Here, we assessed whether transcranial direct current stimulation (tDCS) improves sleep and fatigue symptoms in patients with post-polio syndrome.

METHODS: We enrolled 32 patients with a diagnosis of post-polio syndrome. tDCS (1.5 mA, 15 min) was delivered by a direct current stimulator connected to three electrodes: two anodal electrodes on the scalp over the right and left pre-motor cortex and the other above the left shoulder (cathode). 16 patients received anodal tDCS and the remainder sham tDCS. We evaluated changes induced by tDCS (daily for five days a week, for three weeks) on clinical scales (Short Form Health Survey [SF-36], Piper Fatigue Scale [PFS], Fatigue Severity Scale [FSS], 101-Point Numerical Rating [PNR-101], Hamilton Rating Scale for Depression [HRSD], Pittsburgh Sleep Quality Index [PSQI]) at baseline (T0) and three weeks later (T1).

RESULTS: At T1 SF-36 sub-items physical functioning, role physical, vitality, social functioning and role emotional improved significantly more in patients who received tDCS (p < 0.01) than in sham-treated patients. Also, PSQI scores improved more in treated patients (p < 0.05, two-way ANOVA with "stimulation" and "time" as factors: p < 0.01). tDCS-induced benefits were more pronounced in patients who were younger at primary infection (p < 0.05).

Conclusions: Anodal tDCS over the pre-motor areas for fifteen days improved sleep and fatigue symptoms in patients with post-polio syndrome. tDCS could be a non-invasive and valuable new tool for managing post-polio patients.

Outcome of Research: Effective

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There are currently 25 research papers whose outcomes are rated as “effective”.

Category: Polio Immunisation

Title: Applying the concept of peptide uniqueness to anti-polio vaccination
Author: Kanduc D (1), Fasano C (1), Capone G (1), Pesce Delfino A (2), Calabrò M (2), Polimeno L (2)
Affiliation: (1) Department of Biosciences, Biotechnologies, and Biopharmaceutics, University of Bari, 70126 Bari, Italy; (2) Department of Emergency and Organ Transplantation (DETO), University of Bari, 70124 Bari, Italy
Journal: Journal of Immunology Research
Citation: J Immunol Res. 2015;2015:541282. Epub 2015 Oct 19
Publication Year and Month: 2015 10

Abstract: BACKGROUND: Although rare, adverse events may associate with anti-poliovirus vaccination thus possibly hampering global polio eradication worldwide.

OBJECTIVE: To design peptide-based anti-polio vaccines exempt from potential cross-reactivity risks and possibly able to reduce rare potential adverse events such as the postvaccine paralytic poliomyelitis due to the tendency of the poliovirus genome to mutate.

METHODS: Proteins from poliovirus type 1, strain Mahoney, were analyzed for amino acid sequence identity to the human proteome at the pentapeptide level, searching for sequences that (1) have zero percent of identity to human proteins, (2) are potentially endowed with an immunologic potential, and (3) are highly conserved among poliovirus strains.

RESULTS: Sequence analyses produced a set of consensus epitopic peptides potentially able to generate specific anti-polio immune responses exempt from cross-reactivity with the human host.

Conclusions: Peptide sequences unique to poliovirus proteins and conserved among polio strains might help formulate a specific and universal anti-polio vaccine able to react with multiple viral strains and exempt from the burden of possible cross-reactions with human proteins. As an additional advantage, using a peptide-based vaccine instead of current anti-polio DNA vaccines would eliminate the rare post-polio poliomyelitis cases and other disabling symptoms that may appear following vaccination.

Outcome of Research: More research required

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Category: Fatigue

Title: Circadian fatigue or unrecognized restless legs syndrome? The post-polio syndrome model
Author: Romigi A, Maestri M
Affiliation: Neurophysiopathology Unit, Department of Systems Medicine, Sleep Medicine Centre, Tor Vergata University and Hospital , Rome , Italy ; IRCCS Neuromed , Pozzilli , Italy; Neurology Unit, Department of Clinical and Experimental Medicine, University of Pisa , Pisa , Italy.
Journal: Frontiers in Neurology
Citation: Front Neurol. 2014 Jul 7;5:115. doi: 10.3389/fneur.2014.00115
Publication Year and Month: 2014 07

Abstract: This paper does not have an abstract.

Conclusions:

Outcome of Research: More research required.

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Category: Drugs

Title: Effect of intravenous immunoglobulin in patients with post-polio syndrome - an uncontrolled pilot study
Author: Kaponides G, Gonzalez H, Olsson T, Borg K
Affiliation: Department of Public Health Sciences, Division of Rehabilitation Medicine, Stockholm, Sweden - [email protected]

Journal: Journal of Rehabilitation Medicine
Citation: J Rehabil Med. 2006 Mar;38(2):138-40
Publication Year and Month: 2006 03

Abstract: OBJECTIVE: To analyse changes in muscle strength, physical performance and quality of life during intravenous immunoglobulin (IVIg) treatment in patients with post-polio syndrome.

DESIGN: Open clinical trial.

PATIENTS: A total of 14 patients (6 women, 8 men; mean age 57 years, range 43-67 years) were included in the study.

INTERVENTION: Treatment with 90 g IVIg (30 g daily for 3 days).

MAIN OUTCOME: Muscle strength, measured with dynamic dynamometry, muscle function, by means of performing the 6-minute walk test, and quality of life, analysed by means of the SF-36 questionnaire, were performed before and after treatment.

RESULTS: For quality of life there was a statistically significant improvement for all but one of the 8 multi-item scales of SF-36 when comparing data before and after treatment with IVIg. The multi-item scale most improved was Vitality. There was no significant increase in muscle strength and physical performance.

Conclusions: Data indicate that IVIg may have a clinically relevant effect, with an improvement in quality of life. The effect may be due to a decrease in an inflammatory process in the central nervous system, which earlier has been reported in patients with past-polio syndrome after IVIg treatment. Since a possible placebo effect cannot be ruled out, a randomized controlled study is needed.

Outcome of Research: More research required.

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Category: Drugs

Title: Efficacy of Modafinil on fatigue and excessive daytime sleepiness associated with neurological disorders: a systematic review and meta-analysis
Author: Sheng P, Hou L, Wang X, Wang X, Huang C, Yu M, Han X, Dong Y.
Affiliation: Department of Neurosurgery, Shanghai Institute of Neurosurgery, Military Institute of Neurosurgery, Changzheng Hospital, Second Military Medical University, Shanghai, China
Journal: Public Library of Science
Citation: PLoS One. 2013 Dec 3;8(12):e81802. doi: 10.1371/journal.pone.0081802
Publication Year and Month: 2013 03

Abstract: BACKGROUND: Modafinil is a novel wake-promoting agent approved by the FDA ameliorating excessive daytime sleepiness (EDS) in three disorders: narcolepsy, shift work sleep disorder and obstructive sleep apnea. Existing trials of modafinil for fatigue and EDS associated with neurological disorders provided inconsistent results. This meta-analysis was aimed to assess drug safety and effects of modafinil on fatigue and EDS associated with neurological disorders.

METHODS: A comprehensive literature review was conducted in order to identify published studies assessing the effects of modafinil on fatigue and EDS associated with neurological disorders. Primary outcomes included fatigue and EDS. Secondary outcomes included depression and adverse effects.

FINDINGS: Ten randomized controlled trials were identified including 4 studies of Parkinson's disease (PD), 3 of multiple sclerosis (MS), 2 of traumatic brain injury (TBI) and 1 of post-polio syndrome (PPS). A total of 535 patients were enrolled. Our results suggested a therapeutic effect of modafinil on fatigue in TBI (MD -0.82 95% CI -1.54 - -0.11 p=0.02, I(2)=0%), while a beneficial effect of modafinil on fatigue was not confirmed in the pooled studies of PD or MS. Treatment results demonstrated a clear beneficial effect of modafinil on EDS in patients with PD (MD -2.45 95% CI -4.00 - -0.91 p=0.002 I(2)=14%), but not with MS and TBI. No difference was seen between modafinil and placebo treatments in patients with PPS. Modafinil seemed to have no therapeutic effect on depression. Adverse events were similar between modafinil and placebo groups except that more patients were found with insomnia and nausea in modafinil group.

Conclusions: Existing trials of modafinil for fatigue and EDS associated with PD, MS, TBI and PPS provided inconsistent results. The majority of the studies had small sample sizes. Modafinil is not yet sufficient to be recommended for these medical conditions until solid data are available.

Outcome of Research: More research required.

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Category: Respiratory Complications and Management

Title: Feasibility of lung volume recruitment in early neuromuscular weakness: a comparison between amyotrophic lateral sclerosis, myotonic dystrophy, and postpolio syndrome
Author: Kaminska M (1,2), Browman F (3), Trojan DA (4), Genge A (4), Benedetti A (2,5), Petrof BJ (6,7)
Affiliation: (1) Respiratory Division, McGill University Health Centre, 1001 Decarie Blvd, D05.2504 Montreal, Quebec, Canada H4A 3J1; (2) Respiratory Epidemiology and Clinical Research Unit, McGill University, Montreal, Quebec, Canada; (3) Department of Respiratory Therapy, Montreal Chest Institute, McGill University Health Centre, Montreal, Quebec, Canada; (4) Department of Neurology and Neurosurgery, Montreal Neurological Institute and Hospital, McGill University Health Centre, Montreal, Quebec, Canada; (5) Departments of Medicine and Epidemiology, Biostatistics & Occupational Health, McGill University Health Centre, Montreal, Quebec, Canada; (6) Meakins-Christie Laboratories, McGill University and Research Institute of the McGill University Health Centre, Montreal, Quebec, Canada; (7) Respiratory Division, McGill University Health Centre, 1001 Decarie Blvd, D05.2506 Montreal, Quebec, Canada H4A 3J1
Journal: PM&R: The Journal of Injury, Function, and Rehabilitation
Citation: PM R. 2015 Apr;1-8
Publication Year and Month: 2015 04

Abstract: BACKGROUND: Lung volume recruitment (LVR) is a cough assistance technique used in persons with neuromuscular disorders (NMDs), most typically in those requiring noninvasive ventilation (NIV). Whether it may be useful in persons with NMDs who have milder respiratory impairment is unknown.

OBJECTIVE: To assess the feasibility, impact on quality of life (QOL), and preliminary physiological effects of daily LVR in different categories of persons with NMDs who have an early stage of respiratory impairment.

DESIGN: Feasibility study.

SETTING: Academic tertiary care center.

PARTICIPANTS: Outpatients diagnosed with amyotrophic lateral sclerosis (n = 8), postpolio syndrome (n = 10), and myotonic dystrophy (n = 6) who had restrictive respiratory defects but were not yet using NIV.

METHODS: Participants were asked to perform LVR up to 4 times daily and log their LVR use in a diary. Physiological measurements and questionnaires were completed at baseline and after 3 months.

MAIN OUTCOME MEASUREMENTS: Compliance with LVR use was assessed, along with QOL and willingness to continue the treatment. Physiological measurements included forced vital capacity (FVC), lung insufflation capacity (LIC), and the LIC minus FVC difference.

RESULTS: Of the 24 recruited subjects, 7 with amyotrophic lateral sclerosis, 7 with postpolio syndrome, and 5 with myotonic dystrophy completed the study (n = 19). At baseline, mean values for FVC and spontaneous peak cough flow were 59.9% predicted and 373.1 L/min, respectively. For subjects completing the study, 74% were willing to continue long-term LVR use, and QOL scores were not adversely affected by LVR in any NMD subgroup. The LIC-FVC difference increased from baseline to follow-up by a mean of 0.243 L (P = .006) in all subjects (n = 19), suggesting a possible improvement in respiratory system mechanics.

Conclusions: In patients with NMDs who have early restrictive respiratory defects but do not yet require NIV, regular use of LVR is feasible with no negative impact on QOL over a 3-month period and may have physiological benefits. Further work is needed to determine whether early institution of LVR can improve respiratory system mechanics and help delay ventilatory failure in persons with NMDs.

Outcome of Research: More research required

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Category: Polio Immunisation

Title: Genetically Thermo-Stabilised, Immunogenic Poliovirus Empty Capsids; a Strategy for Non-replicating Vaccines
Author: Fox, H., Knowlson, S., Minor, P.D., Macadam, A.J.
Affiliation: This work was supported by the WHO Polio Research Committee (http://www.polioeradication.org/Research/Grantsandcollaboration.aspx) through I8-TSA-043 & I8-TSA-083 as well as by NIBSC (National Institute for Biological Standards and Control) through core funding received from the UK Department of Health to fund biological standards and control. The funders had no role in study design, data collection and analysis, decision to publish or preparation of the manuscript.
Journal: NEW - PUT DETAILS IN CITATION FIELD
Citation: Fox H, Knowlson S, Minor PD, Macadam AJ (2017) Genetically Thermo-Stabilised, Immunogenic Poliovirus Empty Capsids; a Strategy for Non-replicating Vaccines. PLoS Pathog13(1): e1006117. https://doi.org/10.1371/journal.ppat.1006117
Publication Year and Month: 2017 01

Abstract: While wild type polio has been nearly eradicated there will be a need to continue immunisation programmes for some time because of the possibility of re-emergence and the existence of long term excreters of poliovirus. All vaccines in current use depend on growth of virus and most of the non-replicating (inactivated) vaccines involve wild type viruses known to cause poliomyelitis. The attenuated vaccine strains involved in the eradication programme have been used to develop new inactivated vaccines as production is thought safer. However it is known that the Sabin vaccine strains are genetically unstable and can revert to a virulent transmissible form. A possible solution to the need for virus growth would be to generate empty viral capsids by recombinant technology, but hitherto such particles are so unstable as to be unusable. We report here the genetic manipulation of the virus to generate stable empty capsids for all three serotypes. The particles are shown to be extremely stable and to generate high levels of protective antibodies in animal models.

Conclusions: The final preparations studied were extremely stable compared to IPV and could conceivably give rise to a vaccine that would not require a cold chain; moreover they were more immunogenic than IPV made from the equivalent strains in the animal model used for testing IPV potency and in challenge studies in transgenic mice. It is possible that this is partly because the particles, unlike IPV, were not treated with formalin. The viruses from which they were derived had lost infectivity presumably because they were unable to uncoat by virtue of their hyperstable capsids. A suitable expression platform would be required to make this a viable vaccine production system and is the focus of one strand of current work of the Consortium, but the properties of the particles are very promising.

Outcome of Research: More research required

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Category: Complementary Therapies

Title: Hatha yoga and meditation in patients with post-polio syndrome
Author: DeMayo W, Singh B, Duryea B, Riley D
Affiliation: Southern California University of the Health Sciences, USA
Journal: Alternative Therapies in Health and Medicine
Citation: Altern Ther Health Med. 2004 Mar-Apr;10(2):24-5
Publication Year and Month: 2004 03

Abstract: This paper does not have an abstract. The following is an extract:
Conemaugh Health System has completed a preliminary outcome study evaluating the benefits of Hatha yoga and meditation in patients with post-polio syndrome (PPS). This research integrates clinical trials investigating the application of Hatha yoga with ongoing patient care and education. The results of this clinical trial will be used to develop a longitudinal data collection effort integrating research and clinical trials investigating the applications of Hatha yoga in ongoing patient care and education.

Conclusions:

Outcome of Research: More research required

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Category: Drugs

Title: Immunoglobulin g for the treatment of chronic pain: report of an expert workshop
Author: Tamburin S (1), Borg K, Caro XJ, Jann S, Clark AJ, Magrinelli F, Sobue G, Werhagen L, Zanette G, Koike H, Späth PJ, Vincent A, Goebel A
Affiliation: (1) Department of Neurological and Movement Sciences, University of Verona, Verona, Verona, Italy
Journal: Pain Medicine
Citation: Pain Med. 2014 Jul;15(7):1072-82. doi: 10.1111/pme.12319
Publication Year and Month: 2014 07

Abstract: BACKGROUND: The treatment of chronic pain is still unsatisfactory. Despite the availability of different drugs, most patients with chronic pain do not receive satisfactory pain relief or report side effects. Converging evidence implicates involvement of the immune system in the pathogenesis of different types of nociceptive and neuropathic chronic pain.

DESIGN: At a workshop in Liverpool, UK (October 2012), experts presented evidence suggesting immunological involvement in chronic pain and recent data supporting the concept that the established immune-modulating drug, polyvalent immunoglobulin G (IgG), either given intravenously (IVIg) or subcutaneously (SCIg), may reduce pain in some peripheral neuropathies and a range of other pain disorders. Workshop's attendees discussed the practicalities of using IVIg and SCIg in these disorders, including indications, cost-effectiveness, and side effects.

RESULTS: IgG may reduce pain in a range of nociceptive and neuropathic chronic pain conditions, including diabetes mellitus, Sjögren's syndrome, fibromyalgia, complex regional pain syndrome, post-polio syndrome, and pain secondary to pathological autoantibodies.

Conclusions: IgG is a promising treatment in several chronic pain conditions. IgG is a relatively safe therapeutic strategy, with uncommon and mild side effects but high costs. Randomized, controlled trials and predictive tests are needed to better support the use of IgG for refractory chronic pain.

Outcome of Research: More research required

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Category: Drugs

Title: Intravenous immunoglobulin for postpolio syndrome: a systematic review and meta-analysis
Author: Huang Y-H (1), Chen H-C (2,3), Huang K-W (4,5,6), Chen P-C (1,7), Hu C-J (1,8), Tsai C-P (5,9), Tam K-W (2,10,11,12,13,14), Kuan Y-C (1,5,8,14)
Affiliation: (1) Department of Neurology, Taipei Medical University-Shuang Ho Hospital, New Taipei City, Taiwan; (2) Center for Evidence-Based Health Care, Taipei Medical University-Shuang Ho Hospital, New Taipei City, Taiwan; (3) Department of Physical Medicine and Rehabilitation, Taipei Medical University-Shuang Ho Hospital, New Taipei City, Taiwan; (4) Department of Gastroenterology, College of Medicine, Taipei Medical University, Taipei, Taiwan; (5) Faculty of Medicine, School of Medicine, National Yang-Ming University, Taipei, Taiwan; (6) Department of Gastroenterology, Taipei Medical University-Shuang Ho Hospital, New Taipei City, Taiwan; (7) College of Medical Science and Technology, Taipei Medical University, Taipei, Taiwan; (8) Department of Neurology, School of Medicine, Taipei Medical University, Taipei, Taiwan; (9) Department of Neurology, Neurological Institute, Taipei Veterans General Hospital, Taipei, Taiwan; (10) Department of Surgery, School of Medicine, College of Medicine, Taipei Medical University, Taipei, Taiwan; (11) Graduate Institute of Clinical Medicine, College of Medicine, Taipei Medical University, Taipei, Taiwan; (12) Division of General Surgery, Department of Surgery, Taipei Medical University-Shuang Ho Hospital, Taipei, Taiwan; (13) Center for Evidence-Based Medicine, College of Medicine, Taipei Medical University, Taipei, Taiwan; (14) Taipei Medical University-Shuang Ho Hospital, 291 Zhongzheng Road, Zhonghe District, New Taipei City 23561, Taiwan
Journal: BioMed Central Neurology
Citation: BMC Neurology 2015, 15:39 doi:10.1186/s12883-015-0301-9
Publication Year and Month: 2015 03

Abstract: BACKGROUND: Postpolio syndrome (PPS) is characterized by progressive disabilities that develop decades after prior paralytic poliomyelitis. Because chronic inflammation may be the process underlying the development of PPS, immunomodulatory management, such as intravenous immunoglobulin (IVIg) administration, may be beneficial.

METHODS: We performed a systematic review and meta-analysis of published randomized controlled trials (RCTs) and prospective studies that evaluated the efficacy of IVIg in managing PPS. Electronic databases, including PubMed, EMBASE, CINAHL, and the Cochrane Central Register of Controlled Trials, were searched for articles on PPS published before December 2014. The primary outcomes were pain severity, fatigue scores, and muscle strength. The secondary outcomes were physical performance, quality of life (QoL), and cytokine expression levels.

RESULTS: We identified 3 RCTs involving 241 patients and 5 prospective studies involving 267 patients. The meta-analysis of pain severity (weighted mean difference [WMD] = −1.02, 95% confidence interval [CI] = −2.51 to 0.47), fatigue scores (WMD = 0.28, 95% CI −0.56 to 1.12), and muscle strength revealed no significant differences between the IVIg and the placebo group. Regarding QoL, the RCTs yielded controversial outcomes, with improvement in only certain domains of the Short Form 36 (SF-36). Moreover, one prospective study reported significant improvement on SF-36, particularly in patients aged younger than 65 years, those with paresis of the lower limbs, and high pain intensity.

Conclusions: The present review indicated that IVIg is unlikely to produce significant improvements in pain, fatigue, or muscle strength. Thus, routinely administering IVIg to patients with PPS is not recommended based on RCTs. However, a potential effect in younger patients with lower limbs weakness and intense pain requires confirmation from further well-structured trials.

Outcome of Research: More research required

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Category: Drugs

Title: Intravenous immunoglobulin treatment of the post-polio syndrome: sustained effects on quality of life variables and cytokine expression after one year follow up
Author: Gonzalez H (1), Khademi M (2), Borg K (1), Olsson T (2)
Affiliation: (1) Division of Rehabilitation Medicine, Department of Clinical Sciences, Karolinska Institutet, Danderyd Hospital, blg 39, fl 3, S-192 88, Stockholm, Sweden; (2) Neuroimmunology Unit, Department of Clinical Neuroscience, Karolinska Institutet, Stockholm, Sweden
Journal: Journal of Neuroinflammation
Citation: Journal of Neuroinflammation. 2012; 9: 167. doi: 10.1186/1742-2094-9-167
Publication Year and Month: 2012 07

Abstract: BACKGROUND: Expression of inflammatory cytokines in cerebrospinal fluid (CSF) has led to the hypothesis of intrathecal chronic inflammation to explain the denervation observed in post-polio syndrome (PPS). It has been shown that therapy with intravenous immunoglobulin (IVIG) improves physical performance and dampens down the inflammatory process at 6 months in PPS patients. We here examined the effects of IVIG on cytokine expression and clinical outcome one year after IVIG treatment.

METHODS: From a previous study with 135 PPS patients included, 41 patients were further evaluated before un-blinding for one year (21 placebo and 20 treated with IVIG, Xepol® 50 mg/ml), and were assessed for clinical variables by performing the Short Form-36 survey (SF-36) questionnaire assessment, the 6 minute walk distance test (6MWT) and registering pain level by Visual Analogue Scale (VAS) after IVIG treatment. A separate cohort of 37 PPS patients went through lumbar puncture (LP) at baseline and 20 patients, treated with IVIG, repeated the LP one year later. Thirty patients affected with other neurological diseases (OND) were used as control group. Inflammatory cytokines TNF, TGFβ, IFNγ, IL-23, IL-13 and IL-10 were measured in blood cells and CSF cells with RT-PCR.

RESULTS: Scores of the physical components of SF-36 were significantly higher at the one year follow up time-point in the IVIG-treated patients when compared to baseline as well as to the control subjects. Pain VAS score and 6MWT improved significantly in the IVIG-treated patients when compared with baseline Relative expression of TNF and IFN-γ in both PBMCs and CSF from PPS patients were increased compared to OND subjects at baseline (p < 0.05). One year after IVIG-treatment a decreased expression of IFN-γ and IL23 was found in CSF of PPS patients, while anti-inflammatory IL-13 was increased (p < 0.05).

Conclusions: IVIG has effects on relevant QoL variables and inflammatory cytokines up to one year in patients with PPS. This gives a basis for scheduling IVIG in upcoming trials with this therapy.

Outcome of Research: More research required.

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Category: Activity Levels, Psychology,

Title: No reduction of severe fatigue in patients with postpolio syndrome by exercise therapy or cognitive behavioral therapy: results of an RCT
Author: Koopman FS (1), Voorn EL (1), Beelen A (1), Bleijenberg G (2), de Visser M (1), Brehm MA (1), Nollet F (1)
Affiliation: (1) University of Amsterdam, Amsterdam, The Netherlands; (2) Radboud University Medical Centre, Nijmegen, The Netherlands
Journal: Neurorehabilitation & Neural Repair
Citation: Neurorehabil Neural Repair. 2015 Aug 7. pii: 1545968315600271
Publication Year and Month: 2015 08

Abstract: BACKGROUND: People with postpolio syndrome (PPS) commonly experience severe fatigue that persists over time and negatively affects functioning and health-related quality of life (HRQoL).

OBJECTIVES: To study the efficacy of exercise therapy (ET) and cognitive behavioral therapy (CBT) on reducing fatigue and improving activities and HRQoL in patients with PPS.

METHODS: We conducted a multicenter, single-blinded, randomized controlled trial. Over 4 months, severely fatigued patients with PPS received ET, CBT, or usual care (UC). The primary end point (fatigue) was assessed using the subscale fatigue severity of the Checklist Individual Strength (CIS20-F). Secondary end points included activities and HRQoL, which were assessed with the Sickness Impact Profile and the 36-Item Short-Form, respectively. End points were measured at baseline and at 4, 7, and 10 months.

RESULTS: A total of 68 patients were randomized. No differences were observed between the intervention groups and UC group for fatigue (mean differences in CIS20-F score = 1.47, 95%CI = -2.84 to 5.79, for ET versus UC; and 1.87, 95%CI = -2.24 to 5.98, for CBT versus UC), activities, or HRQoL.

Conclusions: Our results demonstrate that neither ET nor CBT were superior to UC in reducing fatigue in severely fatigued PPS patients. Further research should investigate explanations for the lack of efficacy of these 2 currently advised approaches in clinical practice, which may provide clues to improving treatment aimed at reducing fatigue in PPS.

Outcome of Research: More research required

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Category: Immune Response

Title: Normal serum levels of immune complexes in postpolio patients
Author: Melin E (1), Sohrabian A (2), Rönnelid J (2), Borg K (1)
Affiliation: (1) Division of Rehabilitation Medicine, Department of Clinical Sciences, Karolinska Institutet, Danderyd University Hospital, Stockholm, Sweden; (2) Department of Immunology, Genetics and Pathology, Uppsala University, Uppsala, Sweden
Journal: Results in Immunology
Citation: Results in Immunology. 2014; 4: 54–57. doi: 10.1016/j.rinim.2014.06.001
Publication Year and Month: 2014 06

Abstract: OBJECTIVE: The pathophysiology of the postpolio syndrome is not fully understood. Increased cytokine levels in cerebrospinal fluid and peripheral blood indicate a systemic inflammatory process. Decreased cytokine levels and the clinical effect of intravenous immunoglobulin treatment further indicate an inflammatory/immunological pathogenesis. The aim of the present study was to evaluate whether an autoimmune process follows the initial infection, by means of analyzing immune complexes.

PATIENTS AND METHODS: Circulating immune complexes were analyzed from blood samples of 20 postpolio patients and 95 healthy controls. To compensate for differences in age between patients and controls, a sub-analysis was performed using only the 30 oldest controls. Tumor necrosis factor-inducing properties of polyethylene glycol-precipitated immune complexes were compared between the postpolio patients and 10 healthy controls.

RESULTS: When comparing levels in postpolio patients to the whole control group, including the 30 oldest investigated, there were no statistically significant differences. No difference was found in tumor necrosis factor levels induced by immune complexes when comparing patients and controls.

Conclusions: There was no increase in circulating immune complex or in tumor necrosis factor-inducing effects of circulating immune complex between postpolio patients and healthy controls, indicating that the postpolio syndrome is not due to an autoimmune reaction.

Outcome of Research: More research required.

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Category: Polio Immunisation

Title: Patients with Primary Immunodeficiencies Are a Reservoir of Poliovirus and a Risk to Polio Eradication
Author: Aghamohammadi A (1), Abolhassani H (1), Kutukculer N (2), Wassilak SG (3), Pallansch MA (4), Kluglein S (5), Quinn J (6), Sutter RW (7), Wang X (8), Sanal O (9), Latysheva T (10), Ikinciogullari A (11), Bernatowska E (12), Tuzankina IA (13), Costa-Carvalho BT (14), Franco JL (15), Somech R (16), Karakoc-Aydiner E (17), Singh S (18), Bezrodnik L (19), Espinosa-Rosales FJ (20), Shcherbina A (21), Lau Y (22,23), Nonoyama S (24), Modell F (6), Modell V (6), The JMF Centers Network Investigators and Study Collaborators, Barbouche M (25), and McKinlay MA (5)
Affiliation: (1) Research Center for Immunodeficiencies, Pediatrics Center of Excellence, Children’s Medical Center, Tehran University of Medical Science, Tehran, Iran; (2) Faculty of Medicine, Department of Pediatric Immunology, Ege University, Izmir, Turkey; (3) Global Immunization Division, Centers for Disease Control and Prevention, Atlanta, GA, United States; (4) Division of Viral Diseases, Centers for Disease Control and Prevention, Atlanta, GA, United States; (5) Center for Vaccine Equity, Task Force for Global Health, Atlanta, GA, United States; (6) Jeffrey Modell Foundation, New York, NY, United States; (7) Research and Product Development, World Health Organization, Geneva, Switzerland' (8) Department of Clinical Immunology, Children’s Hospital of Fudan University, Shanghai, China; (9) Division of Immunology, Department of Pediatrics, Hacettepe University Faculty of Medicine, Ankara, Turkey; (10) Department of Allergology and Immunotherapy, Institute of Immunology, Moscow, Russia; (11) Department of Pediatric Immunology and Allergy, Ankara University School of Medicine, Ankara, Turkey; (12) Department of Clinical Immunology, The Children’s Memorial Health Institute, Warsaw, Poland; (13) Institute of Immunology and Physiology, Ural Branch of the Russian Academy of Sciences, Yekaterinburg, Russia; (14) Department of Pediatrics, Federal University of São Paulo, São Paulo, Brazil; (15) Grupo de Inmunodeficiencias Primarias, Facultad de Medicina, Departamento de Microbiología y Parasitología, Universidad de Antioquia, Medellín, Colombia; (16) Pediatric Department A and the Immunology Service, Sheba Medical Center, Tel Hashomer, Jeffrey Modell Foundation Center, Affiliated to the Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel; (17) Division of Pediatric Allergy and Immunology, Marmara Medical Faculty, Istanbul, Turkey; (18) Pediatric Allergy and Immunology Unit, Advanced Pediatrics Centre, PGIMER, Chandigarh, India; (19) Dr. Ricardo Gutierrez Hospital de Niños, Buenos Aires, Argentina; (20) Clinical Immunology and Allergy Unit, Instituto Nacional de Pediatría, Ciudad de México, Mexico; (21) Department of Clinical Immunology, Dmitry Rogachev Federal Research and Clinical Center of Pediatric Hematology, Oncology and Immunology, Moscow, Russia; (22) Department of Paediatrics and Adolescent Medicine, The University of Hong Kong, Queen Mary Hospital, Hong Kong, Hong Kong; (23) Shenzhen Primary Immunodeficiency Diagnostic and Therapeutic Laboratory, Hong Kong University-Shenzhen Hospital, Shenzhen, China; (24) Department of Pediatrics, National Defense Medical College, Saitama, Japan; (25) Department of Immunology, Institut Pasteur de Tunis, University Tunis El-Manar, Tunis, Tunisia.
Journal: Frontiers in Immunology
Citation: Front. Immunol. 8:685. doi: 10.3389/fimmu.2017.00685
Publication Year and Month: 2017 06

Abstract: Immunodeficiency-associated vaccine-derived polioviruses (iVDPVs) have been isolated from primary immunodeficiency (PID) patients exposed to oral poliovirus vaccine (OPV). Patients may excrete poliovirus strains for months or years; the excreted viruses are frequently highly divergent from the parental OPV and have been shown to be as neurovirulent as wild virus. Thus, these patients represent a potential reservoir for transmission of neurovirulent polioviruses in the post-eradication era. In support of WHO recommendations to better estimate the prevalence of poliovirus excreters among PIDs and characterize genetic evolution of these strains, 635 patients including 570 with primary antibody deficiencies and 65 combined immunodeficiencies were studied from 13 OPV-using countries. Two stool samples were collected over 4 days, tested for enterovirus, and the poliovirus positive samples were sequenced. Thirteen patients (2%) excreted polioviruses, most for less than 2 months following identification of infection. Five (0.8%) were classified as iVDPVs (only in combined immunodeficiencies and mostly poliovirus serotype 2). Non-polio enteroviruses were detected in 30 patients (4.7%). Patients with combined immunodeficiencies had increased risk of delayed poliovirus clearance compared to primary antibody deficiencies. Usually, iVDPV was detected in subjects with combined immunodeficiencies in a short period of time after OPV exposure, most for less than 6 months. Surveillance for poliovirus excretion among PID patients should be reinforced until polio eradication is certified and the use of OPV is stopped. Survival rates among PID patients are improving in lower and middle income countries, and iVDPV excreters are identified more frequently. Antivirals or enhanced immunotherapies presently in development represent the only potential means to manage the treatment of prolonged excreters and the risk they present to the polio endgame.

Conclusions: This study provides an estimate of the global iVDPV prevalence among PID patients without paralytic disease and supports expanded screening for iVDPV excretion in these patients. Although most previous studies focused on the risk of long-term iVDPV infection in antibody deficient patients, the predominance of risk in patients with combined immunodeficiencies included in the current study highlights the importance of considering this group of PID patients in any surveillance program. Reinfection with poliovirus and NPEV excretion in PID patients described elsewhere demonstrates the need for prolonged follow-up (17).

The Global Polio Eradication Initiative plans to cease use of OPV worldwide once WPV has been certified as eradicated, which will end the generation of new iVDPVs. However, there is currently no means for addressing the threat posed by existing immunodeficient persons infected with iVDPVs, either to the infected individual’s risk of paralytic disease, or to the community of a continuing source of poliovirus transmission. Antivirals represent a potential means to manage the treatment of iVDPV excreters and the risk they present to the eradication effort (32, 34). Two safe virus-specific antivirals acting by differing mechanisms are now being developed and may be used as a combination (e.g., pocapavir and V-7404). This strategy may resolve the individual’s infection, stop iVDPV excretion, and serve to eliminate the risk of poliovirus transmission in the community. Currently, pocapavir is being considered for use in poliovirus excreting PID patients on a compassionate use basis.

The potential risk posed by iVDPV excreters to the polio eradication effort indicates the immediate need to develop and implement a global iVDPV surveillance strategy. Utilizing this approach, individuals at risk of prolonged poliovirus excretion can be identified and antiviral treatment can be initiated.

Outcome of Research: More research required

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Category: Diagnosis and Management

Title: Physiotherapy for poliomyelitis: a descriptive study in the Republic of Congo
Author: Mancini S, Coldiron ME, Nicholas S, Llosa AE, Mouniaman-Nara I, Ngala J, Grais RF, Porten K
Affiliation: Epicentre, Paris, France - [email protected]
Journal: BioMedCentral Research Notes
Citation: BMC Res Notes. 2014 Oct 23;7:755. doi: 10.1186/1756-0500-7-755
Publication Year and Month: 2014 10

Abstract: BACKGROUND: A large poliomyelitis outbreak occurred in 2010 in the Republic of Congo. This paper describes the demographic and clinical characteristics of poliomyelitis cases and their outcomes following physiotherapy.

FINDINGS: Demographic and clinical data were collected on 126 individuals between November 23, 2010 and March 23, 2011. The male/female ratio was 2.5 and the median age was 19 years (IQR: 13.5-23). The most severe forms of the disease were more common in older patients, 81 of the 126 patients (64.3%) had multiple evaluations of muscle strength. Among patients with multiple evaluations, 38.1% had improved strength at final evaluation, 48.3% were stable and 13.6% had decreased strength.

Conclusions: Most acute poliomyelitis patients receiving physiotherapy had improved or stable muscle strength at their final evaluation. These descriptive results highlight the need for further research into the potential benefits of physiotherapy in polio affected patients.

Outcome of Research: More research required.

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Category: Diagnosis and Management

Title: Polio survivors perceptions of a multi-disciplinary rehabilitation programme
Author: Atwal, A., Duncan, H., Queally, C., Cedar, S.H.
Affiliation: Nil
Journal: Disability and Rehabilitation
Citation: Atwal, A., Duncan, H., Queally, C., Cedar, S.H. (2017) Polio survivors perceptions of a multi-disciplinary rehabilitation programme. Disability and Rehabilitation. DOI: 10.1080/09638288.2017.1381184
Publication Year and Month: 2017 10

Abstract: Purpose: Post-polio syndrome refers to a late complication of the poliovirus infection. Management of post-polio syndrome is complex due to the extensive symptomology. European and United Kingdom guidelines have advised the use of rehabilitation programmes to manage post-polio syndrome. There is a paucity of research in relation to the effectiveness of rehabilitation interventions. The objective of this study is to explore polio survivor’s perceptions of an in-patient multi-disciplinary rehabilitation programme.

Methods: Semi-structured interviews of community dwelling polio survivors who attended in-patient rehabilitation programme in the United Kingdom. Thematic analysis was used to describe and interpret interview data.

Results: Participants’ experiences were influenced by past experiences of polio and their self-concept. Participants generally had a positive experience and valued being with other polio survivors. Positive strategies, such as pacing and reflection changed their mind-sets into their lives after the programme, though they still faced challenges in daily living. Some participants supported others with post-polio syndrome after completing the programme.

Conclusions: The research identified that participants experienced long term positive benefits from attending a rehabilitation programme. Strategies that users found helpful that explored the effectiveness of interventions to manage polio are not cited within a Cochrane review. If we are to recognise the lived experience and service user empowerment within a model of co- production it is essential that patient preferences are evaluated and used as evidence to justify service provision. Further research is required with polio survivors to explore how best rehabilitation programmes can adopt the principles of co-production.

Implications for Rehabilitation
The patients’ expertise and lived experience must be at the centre of a rehabilitation programme.

Strategies such as pacing and reflection are perceived as important strategies to enable self-management of polio and post-polio syndrome despite the limited evidence base to support these interventions.

Polio rehabilitation programmes should not be time limited and commissioners and therapists need to ensure that follow up support is provided.

When measuring outcomes patient preferences and views must be evaluated.

Outcome of Research: More research required

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Comments (if any): This investigation supports feedback from polio survivors in Australia on the benefits of group-based rehabilitation, especially where there is an opportunity to learn and discuss their rehabilitation with fellow participants.

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Category: Quality of Life

Title: Polio survivors' perceptions of the meaning of quality of life and strategies used to promote participation in everyday activities
Author: Atwal A, Spiliotopoulou G, Coleman C, Harding K, Quirke C, Smith N, Osseiran Z, Plastow N, Wilson L
Affiliation: Division of Occupational Therapy, School of Health Sciences and Social Care, Brunel University, Uxbridge, UK
Journal: Health Expectations
Citation: Health Expect. 2014 Jan;doi:10.1111/hex.12152
Publication Year and Month: 2014 01

Abstract: INTRODUCTION: The term ‘post-polio syndrome’ (PPS) is used to describe new and late manifestations of poliomyelitis that occur later in life. Research in this area has focused upon health status rather than its effect on quality of life.

AIM: To gain an in-depth understanding of the meaning of quality of life for polio survivors and to determine the type of strategies that are used by people with PPS and the support that they consider as important to facilitate participation in everyday life activities that have an impact on their quality of life.

METHOD: Six focus groups were conducted with 51 participants from two regions in England. Data were audio-taped and analysed using thematic analysis.

RESULTS: Our research found that polio survivors used terms used to describe quality of life which could be associated with that of happiness. Our research has identified resolvable factors that influence quality of life namely inaccessible environments, attitudes of health-care professionals and societal attitudes. Polio survivors have tried alternative therapies, chiefly acupuncture and massage, and found them to be effective in enhancing their quality of life.

Conclusions: It is suggested that health-care professionals should consider factors which influence happiness and implement a person-centred approach with the views of the polio survivor being listened to. The three factors that influenced quality of life could be resolved by health-care professionals and by society. With regard to strategies used, we suggest that polio survivors should have access to the treatments that they perceive as important, although further research is required to design optimal interventions for this client group.

Outcome of Research: More research required

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Category: Pain

Title: Predictive factors and correlates for pain in postpoliomyelitis syndrome patients
Author: Vasiliadis HM, Collet JP, Shapiro S, Venturini A, Trojan DA
Affiliation: Department of Epidemiology and Biostatistics, Jewish General Hospital, McGill University, Montreal, Que, Canada
Journal: Archives of Physical Medicine and Rehabilitation
Citation: Arch Phys Med Rehabil. 2002 Aug;83(8):1109-15
Publication Year and Month: 2002 08

Abstract: OBJECTIVE: To identify predictive and associated factors for muscle and joint pain in postpoliomyelitis syndrome (PPS).

DESIGN: Cross-sectional study design.

SETTING: Postpolio clinics.

PARTICIPANTS: Baseline data on 126 PPS patients entered into a multicentered clinical trial.

INTERVENTIONS: Not applicable.

MAIN OUTCOME MEASURES: Adjusted odds ratios were computed by using logistic regression modeling. Patients with or without muscle or joint pain were compared with regard to predictors and associated factors.

RESULTS: In multivariate analyses for muscle pain, significant predictive and associated factors were female gender (P=.0006), longer duration of general fatigue (P=.019), and a lower score on the general health scale (P=.009) of the Medical Outcomes Study 36-Item Short-From Health Survey (SF-36). In multivariate analyses for joint pain, significant predictive and associated factors were female gender (P<.003), longer latency (duration of stability after polio; P=.008), younger age at interview (P<.002), greater weakness at acute polio (P<.07), weaker lower-extremity muscle strength (P<.04), and a lower SF-36 general health scale score (P<.02).

Conclusions: Women are more likely to report muscle and joint pain in PPS. Greater initial motor unit involvement and lower-extremity weakness may be additional important factors for determining joint pain. Both muscle and joint pain are associated with reductions in quality of life.

Outcome of Research: More research required

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Category: Fatigue

Title: Psychometric properties of fatigue severity and fatigue impact scales in postpolio patients
Author: Oncu J (1), Atamaz F, Durmaz B, On A
Affiliation: (1) Department of Physical Medicine and Rehabilitation, Şişli Etfal Teaching Hospital, Istanbul; Department of Physical Medicine and Rehabilitation, Medical Faculty of Ege University, Bornova-Izmir, Turkey.
Journal: International Journal of Rehabilitation Research
Citation: Int J Rehabil Res. 2013 Dec;36(4):339-45. doi: 10.1097/MRR.0b013e3283646b56
Publication Year and Month: 2013 12

Abstract: We evaluate the reliability, validity, and responsiveness of the Fatigue Severity Scale (FSS) and the Fatigue Impact Scale (FIS) and to determine whether these scales are potentially applicable for measuring fatigue in postpolio patients (PPS). After the Turkish adaptation of FSS and FIS using a forward-backward procedure, the scales were administered to 48 PPS patients without additional health problems that may induce fatigue. Reliability studies were carried out by determination of intraclass correlation coefficient and internal consistency by the Cronbach-α coefficient. Validity was tested by within-scale analyses and analyses against the external criteria including convergent validity and discriminant validity. Correlations with the Notthingham Health Profile (NHP), fatigue, pain and cramp severity (visual analog scale), and manual muscle testing were performed. Sensitivity to changes was determined by standardized response mean values. All patients completed scales, suggesting their satisfactory acceptance. Reliability studies were satisfactory, with higher Cronbach-α values and intraclass correlation coefficients than 0.80. The FSS score was correlated moderately with visual analog scale-fatigue (r=0.41) and the NHP-energy dimension (r=0.29). All FIS scores except cognitive scores were moderately related to the NHP-social isolation score (r=0.40, 0.37, and 0.43 for FIS-physical, social, and total scores, respectively). There was also a significant correlation between the FIS-physical score and the NHP-energy score (r=0.31). On the basis of the standardized response mean values, response to treatment for these two questionnaires was satisfactory (P=0.00). The Turkish versions of FSS and FIS were reliable, sensitive to clinical changes, and also well accepted by patients with PPS. Although they had somewhat satisfactory convergent validity, the absence of strong correlations did not support the validity entirely.

Conclusions:

Outcome of Research: More research required

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Category: Pain

Title: Psychosocial factors and adjustment to pain in individuals with postpolio syndrome
Author: Hirsh AT, Kupper AE, Carter GT, Jensen MP
Affiliation: Department of Rehabilitation Medicine, University of Washington School of Medicine, Seattle, Washington 98104, USA
Journal: American Journal of Physical Medicine & Rehabilitation
Citation: Am J Phys Med Rehabil. 2010 Mar;89(3):213-24. doi: 10.1097/PHM.0b013e3181c9f9a1
Publication Year and Month: 2010 03

Abstract: OBJECTIVE: The purpose of the current study was to examine the associations among measures of psychosocial factors, pain, and adjustment in persons with postpoliomyelitis syndrome.

DESIGN: A cross-sectional survey design was used. Sixty-three community-dwelling individuals with postpoliomyelitis syndrome returned completed questionnaires (71% response rate) that included measures of pain intensity and interference, psychological functioning, pain catastrophizing, social support, and pain-related beliefs and coping.

RESULTS: After controlling for demographic variables, the group of psychosocial variables accounted for an additional 23% of the variance in pain intensity. These variables explained an additional 35% and 50% of the variance in pain interference and psychological functioning, respectively, after accounting for demographic variables and pain intensity. Social support was associated with both psychological functioning and pain interference, whereas catastrophizing was most closely related to psychological functioning. Individual pain beliefs and coping strategies were variably related to the three criterion measures.

Conclusions: The overall results of the current study are consistent with a biopsychosocial framework for understanding pain and functioning in individuals with postpoliomyelitis syndrome. Although additional research is needed to clarify the nature of the relationships between individual psychosocial variables and functional indices, the findings suggest the need for a multidisciplinary approach to pain management in individuals with postpoliomyelitis syndrome.

Outcome of Research: More research required.

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Category: Drugs

Title: Pyridostigmine in postpolio syndrome: no decline in fatigue and limited functional improvement
Author: Horemans H (1), Nollet F (1), Beelen A (1), Drost G (2), Stegeman D (2), Zwarts M (2), Bussmann J (3), de Visser M (4), Lankhorst G (1)
Affiliation: (1) Department of Rehabilitation Medicine, VU University Medical Centre, Amsterdam, Netherlands; (2) Department of Clinical Neurophysiology, University Medical Centre Nijmegen, Netherlands; (3) Department of Rehabilitation Medicine, Erasmus MC, University Medical Centre Rotterdam, Netherlands; (4) Department of Neurology, Academic Medical Centre, University of Amsterdam, Netherlands
Journal: Journal of Neurology, Neurosurgery and Psychiatry
Citation: J Neurol Neurosurg Psychiatry 2003;74:1655-1661 doi:10.1136/jnnp.74.12.1655
Publication Year and Month: 2003 12

Abstract: OBJECTIVES: To investigate the effect of pyridostigmine on fatigue, physical performance, and muscle function in subjects with postpoliomyelitis syndrome.

METHODS: 67 subjects with increased fatigue and new weakness in one quadriceps muscle showing neuromuscular transmission defects, were included in a randomised, double blind, placebo controlled trial of 60 mg pyridostigmine four times a day for 14 weeks. Primary outcome was fatigue (on the "energy" category of the Nottingham health profile). Secondary outcomes included two minute walking distance and quadriceps strength and jitter. Motor unit size of the quadriceps was studied as a potential effect modifier. The primary data analysis compared the changes from baseline in the outcomes in the last week of treatment between groups.

RESULTS: 31 subjects treated with pyridostigmine and 31 subjects treated with placebo completed the trial. No significant effect of pyridostigmine was found on fatigue. The walking distance improved more in the pyridostigmine group than in the placebo group (by 7.2 m (6.0%); p<0.01). Subgroup analysis showed that a significant improvement in walking performance was only found in subjects with normal sized motor units. Quadriceps strength improved more in the pyridostigmine group than in the placebo group (by 6.7 Nm (7.2%); p = 0.15). No effect of pyridostigmine was found on jitter.

Conclusions: Pyridostigmine in the prescribed dose did not reduce fatigue in subjects with postpoliomyelitis syndrome. However, it may have a limited beneficial effect on physical performance, especially in subjects with neuromuscular transmission defects in normal sized motor units.

Outcome of Research: More research required.

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Category: Activity Levels

Title: Reproducibility of walking at self-preferred and maximal speed in patients with postpoliomyelitis syndrome
Author: Horemans HL (1), Beelen A (2), Nollet F (2), Lankhorst GJ (1)
Affiliation: (1) Department of Rehabilitation Medicine, VU University Medical Center, Amsterdam, The Netherlands; (2) Academic Medical Center, University of Amsterdam, Amsterdam, The Netherlands
Journal: Archives of Physical Medicine and Rehabilitation
Citation: Arch Phys Med Rehabil. 2004 Dec;85(12):1929-32
Publication Year and Month: 2004 12

Abstract: OBJECTIVE:
To assess the reproducibility of walking performance, heart rate, and perceived exertion at self-preferred speed and maximal walking speed in patients with the postpoliomyelitis syndrome (PPS).

DESIGN: Repeated measurement at a 3-week interval.

SETTING: University hospital.

PARTICIPANTS: Convenience sample of 65 patients with PPS.

INTERVENTIONS: Not applicable.

MAIN OUTCOME MEASURES: Walking performance: the distance walked in 2 minutes at a self-preferred speed and the time needed to walk 75 m at maximal speed, heart rate, and rating of perceived exertion (RPE) on an 11-point scale.

RESULTS: Test-retest reliability of walking performance was excellent for both tests (intraclass correlation coefficient [ICC] range, .94-.97). No systematic differences existed between test and retest. The smallest detectable change for an individual was 15% for both tests. Test-retest reliability for heart rate was good (ICC=.86) but moderate for RPE (Spearman rho range, .67-.70). The smallest detectable change for RPE was between 4 and 6 scale points. The variability in walking performance was significantly correlated with the variability in heart rate at self-preferred speed (r =.36, P <.01) but not with the variability in RPE (r =.20, P =.11).

Conclusions: Both walking tests showed good reproducibility and may be appropriate to monitor (individual) changes in walking capacity in patients with PPS. Because of its moderate reproducibility, RPE does not seem to be suitable to monitor physical exertion. The usefulness of an objective measure such as heart rate for this purpose needs further investigation.

Outcome of Research: More research required

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Category: Restless Legs Syndrome

Title: Restless legs syndrome in post-polio syndrome: a series of 10 patients with demographic, clinical and laboratorial findings
Author: Marin LF (1), Carvalho LB, Prado LB, Quadros AA, Oliveira AS, Prado GF
Affiliation: (1) Neuro-Sono Sleep Center, Universidade Federal de São Paulo, Rua Claudio Rossi 394, Sao Paulo, SP, Brazil. [email protected]
Journal: Parkinsonism & Related Disorders
Citation: Parkinsonism Relat Disord. 2011 Aug;17(7):563-4. doi: 10.1016/j.parkreldis.2011.02.011
Publication Year and Month: 2011 08

Abstract: BACKGROUND: Few studies have described the occurrence of restless legs syndrome in post-polio syndrome.

METHODS: We studied 10 consecutive patients with post-polio syndrome and symptoms of restless legs syndrome. We look at demographic, clinical and laboratorial data.

RESULTS: A remarkable finding was the concomitant onset of symptoms of both diseases, suggesting a possible underlying mechanism. Severity of restless legs symptoms was moderate to very severe.

Conclusions: Epidemiological studies with larger samples are needed to better establish the relationship and the incidence of restless legs syndrome in post-polio syndrome.

Outcome of Research: More research required

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Category: Restless Legs Syndrome

Title: Sleep disorders frequency in post-polio syndrome patients caused by periodic limb movements
Author: Araujo MA (1), Silva TM, Moreira GA, Pradella-Hallinan M, Tufik S, Oliveira AS
Affiliation: (1) Universidade Federal de São Paulo (UNIFESP/EPM), Instituto do Sono, São Paulo SP, Brazil
Journal: Arquivos de Neuro-Psiquiatria
Citation: Arq Neuropsiquiatr. 2010 Feb;68(1):35-8.
Publication Year and Month: 2010 02

Abstract: Post-polio syndrome (PPS) in individuals with polio longer than 15 years is characterized by weakness and/or muscle fatigue, deficit of deglutition and breath and periodic limb movements (PLM) during sleep. We undertook a review of 99 patients with PPS, and assessed the frequency of PLM through polysomnographic recordings at our sleep disorders unit. The total number of PLM, total time of sleep (TTS), efficiency of sleep (EfS), awaking index (AI) and apnea-hypopnea index (AHI) were analyzed. Sixteen patients presented PLM in excess of 5 for the entire night. When comparing these with the group without PLM, a correlation was found (p=0.001). Significant difference was found for the correlation of the parameters: IAH, ID, TTS and EfS when compared the two groups. There is a close relationship between PPS and PLM.

Conclusions:

Outcome of Research: More research required

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Category: Quality of Life

Title: The association between post-polio symptoms as measured by the Index of Post-Polio Sequelae and self-reported functional status
Author: Schwartz I, Gartsman I, Adler B, Friedlander Y, Manor O, Levine H, Meiner Z
Affiliation: Department of Physical Medicine and Rehabilitation, Hadassah Medical Center, Israel; The Hebrew University-Hadassah, Braun School of Public Health, Israel; Department of Physical Medicine and Rehabilitation, Hadassah Medical Center, Israel. Electronic address: [email protected]
Journal: Journal of the Neurological Sciences
Citation: J Neurol Sci. 2014 Oct 15;345(1-2):87-91. doi: 10.1016/j.jns.2014.07.012
Publication Year and Month: 2014 10

Abstract: OBJECTIVE: To evaluate the association between self-reported severity of polio sequelae and current functional status among polio survivors.

METHODS: This was a cross sectional study of 195 polio survivors attending a polio outpatient clinic at a university hospital. The main outcome measures of demographic, medical, social, and functional data were gleaned from a questionnaire adapted for the polio population. The severity of polio sequelae was evaluated with the self-reported Index of Post-Polio Sequelae (IPPS).

RESULTS: The mean age of our sample was 57.6 ± 10.5 years, 53% were men, 38% had acquired higher education and 37% were employed. We found significant correlations between the total IPPS score and independence in activity of daily living (P<0.05), the use of walking aids (P<0.005) and mobility in and out-of-doors (P<0.0001). A positive correlation was also found between the total IPPS score and subjective assessment of physical and mental health (P<0.0001).

Conclusions: Higher disability in ADL and mobility and lower perception of physical and mental health in polio survivors were associated with a higher score on the IPPS, reflecting greater severity of polio sequelae. These findings demonstrate the IPPS as a useful tool in the clinical evaluation of the polio population, however further data is needed in order to determine if this index can assess clinically significant changes over time.

Outcome of Research: More research required.

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Category: Diagnosis and Management

Title: Treatment for postpolio syndrome
Author: Koopman FS, Beelen A, Gilhus NE, de Visser M, Nollet F
Affiliation: Department of Rehabilitation, University of Amsterdam Academic Medical Center, PO Box 22660, Amsterdam, North Holland, Netherlands, 1100 DD
Journal: Cochrane Database of Systematic Reviews
Citation: Cochrane Database Syst Rev. 2015 May 18;5:CD007818
Publication Year and Month: 2015 05

Abstract: BACKGROUND: Postpolio syndrome (PPS) may affect survivors of paralytic poliomyelitis and is characterised by a complex of neuromuscular symptoms leading to a decline in physical functioning. The effectiveness of pharmacological treatment and rehabilitation management in PPS is not yet established. This is an update of a review first published in 2011.

OBJECTIVES: To systematically review the evidence from randomised and quasi-randomised controlled trials for the effect of any pharmacological or non-pharmacological treatment for PPS compared to placebo, usual care or no treatment.

SEARCH METHODS: We searched the following databases on 21 July 2014: Cochrane Neuromuscular Disease Group Specialized Register, the Cochrane Central Register of Controlled Trials (CENTRAL), MEDLINE, EMBASE, PsycINFO and CINAHL Plus. We also checked reference lists of all relevant articles, searched the Database of Abstracts of Reviews of Effects (DARE), the Health Technology Assessment (HTA) Database and trial registers and contacted investigators known to be involved in research in this area.

SELECTION CRITERIA: Randomised and quasi-randomised trials of any form of pharmacological or non-pharmacological treatment for people with PPS. The primary outcome was self perceived activity limitations and secondary outcomes were muscle strength, muscle endurance, fatigue, pain and adverse events.

DATA COLLECTION AND ANALYSIS: We used standard methodological procedures expected by The Cochrane Collaboration.

MAIN RESULTS: We included 10 pharmacological (modafinil, intravenous immunoglobulin (IVIg), pyridostigmine, lamotrigine, amantadine, prednisone) and three non-pharmacological (muscle strengthening, rehabilitation in a warm climate (that is temperature ± 25°C, dry and sunny) and a cold climate (that is temperature ± 0°C, rainy or snowy), static magnetic fields) studies with a total of 675 participants with PPS in this review. None of the included studies were completely free from any risk of bias, the most prevalent risk of bias being lack of blinding.There was moderate- and low-quality evidence that IVIg has no beneficial effect on activity limitations in the short term and long term, respectively, and inconsistency in the evidence for effectiveness on muscle strength. IVIg caused minor adverse events in a substantial proportion of the participants. Results of one trial provided very low-quality evidence that lamotrigine might be effective in reducing pain and fatigue, resulting in fewer activity limitations without generating adverse events. Data from two single trials suggested that muscle strengthening of thumb muscles (very low-quality evidence) and static magnetic fields (moderate-quality evidence) are safe and beneficial for improving muscle strength and pain, respectively, with unknown effects on activity limitations. Finally, there was evidence varying from very low quality to high quality that modafinil, pyridostigmine, amantadine, prednisone and rehabilitation in a warm or cold climate are not beneficial in PPS.

Conclusions: Due to insufficient good-quality data and lack of randomised studies, it was impossible to draw definite conclusions about the effectiveness of interventions for PPS. Results indicated that IVIg, lamotrigine, muscle strengthening exercises and static magnetic fields may be beneficial but need further investigation to clarify whether any real and meaningful effect exists.

Outcome of Research: More research required.

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Category: Diagnosis and Management

Title: Update on current and emerging treatment options for post-polio syndrome
Author: Farbu E
Affiliation: Neurocenter and National Competence Center for Movement Disorders, Stavanger University Hospital, Stavanger, Norway
Journal: Therapeutics and Clinical Risk Management
Citation: Ther Clin Risk Manag. 2010 Jul 21;6:307-13
Publication Year and Month: 2010 07

Abstract: Post-polio syndrome (PPS) refers to the clinical deterioration experienced by many polio survivors several decades after their acute illness. The symptoms are new muscle weakness, decreased muscle endurance, fatigue, muscle pain, joint pain, cold intolerance, and this typical clinical entity is reported from different parts of the world. The pathophysiology behind PPS is not fully understood, but a combination of distal degeneration of enlarged motor units caused by increased metabolic demands and the normal aging process, in addition to inflammatory mechanisms, are thought to be involved. There is no diagnostic test for PPS, and the diagnosis is based on a proper clinical workup where all other possible explanations for the new symptoms are ruled out. The basic principle of management of PPS lies in physical activity, individually tailored training programs, and lifestyle modification. Muscle weakness and muscle pain may be helped with specific training programs, in which training in warm water seems to be particularly helpful. Properly fitted orthoses can improve the biomechanical movement pattern and be energy-saving. Fatigue can be relieved with lifestyle changes, assistive devices, and training programs. Respiratory insufficiency can be controlled with noninvasive respiratory aids including biphasic positive pressure ventilators. Pharmacologic agents like prednisone, amantadine, pyridostigmine, and coenzyme Q10 are of no benefit in PPS. Intravenous immunoglobulin (IVIG) has been tried in three studies, all having positive results. IVIG could probably be a therapeutic alternative, but the potential benefit is modest, and some important questions are still unanswered, in particular to which patients this treatment is useful, the dose, and the therapeutic interval.

Conclusions:

Outcome of Research: More research required.

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Category: Continence

Title: Urologic manifestations of postpolio syndrome
Author: Johnson VY, Hubbard D, Vordermark JS
Affiliation: Not stated
Journal: Journal of Wound, Ostomy, and Continence Nursing
Citation: J Wound Ostomy Continence Nurs. 1996 Jul;23(4):218-23
Publication Year and Month: 1996 07

Abstract: To determine the character and prevalence of bladder dysfunction among persons with a history of polio, we conducted a survey of randomly selected polio survivors. The survey solicited information on the acute episode of polio and the nature of late-onset neurologic symptoms that could be attributed to postpolio syndrome. There were 242 female and 88 male respondents to the study. Symptoms attributable to postpolio syndrome were present in 87.2% of female subjects and 73.9% of male subjects. Respondents with postpolio syndrome had a significantly greater prevalence of urologic symptoms than seen among respondents without postpolio syndrome, although no dominant pattern of voiding dysfunction was noted. The early onset of erectile dysfunction was more common among male subjects with postpolio syndrome than among male subjects without postpolio syndrome. Genuine stress incontinence was seen in 36.3% of the survey population. Sixteen women with postpolio syndrome underwent surgical repair for urinary incontinence, with a success rate of 60.5%. Bladder disorders are common among persons with PPS, but further clinical and urodynamic data are necessary to define the nature and magnitude of this dysfunction.

Conclusions:

Outcome of Research: More research required

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Category: Fatigue

Title: Vitality among Swedish patients with post-polio: a physiological phenomenon
Author: Ostlund G (1), Wahlin A (2), Sunnerhagen KS (3,4), Borg K (1)
Affiliation: (1) Divison of Rehabilitation Medicine, Karolinska Institutet, Department of Clinical Sciences at Danderyd Hospital, Sweden; (2) Department of Psychology, Stockholm University, Stockholm, Sweden; (3) Institute for Neuroscience and Physiology, Section for Clinical Neuroscience and Rehabilitation, Göteborg University, Göteborg, Sweden; (4) Sunnaas Rehabilitation Hospital, Faculty of Medicine, University of Oslo, Oslo, Norway - [email protected]
Journal: Journal of Rehabilitation Medicine
Citation: J Rehabil Med. 2008 Oct;40(9):709-14. doi: 10.2340/16501977-0253
Publication Year and Month: 2008 10

Abstract: OBJECTIVE: To evaluate vitality and fatigue in post-polio patients, and the relative contributions of physiological and psychological parameters to the level of vitality.

DESIGN: Multi-centre study.

SUBJECTS: One hundred and forty-three patients with post-polio syndrome.

METHODS: Inventories of background, quality of life, fatigue and sleep quality were used. Pain was evaluated using a visual analogue scale. Descriptive statistics and correlations were used for all selected parameters. Hierarchical regression models were constructed to examine predictors of variations in vitality, pain, reduced activity and physical fatigue.

RESULTS: General fatigue accounted for 68% of the variation in vitality. Of this, 91% was accounted for by physiological indicators. After controlling for age, physiological para notmeters accounted for 56.6% and 25%, if entered before and after the psychological parameters, respectively. The impact of the psychological parameters decreased after accounting for the physiological parameters. Physical fatigue, age and sleep quality were associated with variation in pain. Body mass index, pain and sleep quality accounted for differences in reduced activity and physical fatigue.

Conclusions: Vitality in post-polio patients depends on physio-logical parameters. Mental fatigue is not a prominent predictor. Subgroups with or without fatigue, independent of age, need further study.

Outcome of Research: More research required.

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Category: Assistive Technology, Orthoses

Title: Wearable monitoring devices for assistive technology: case studies in post-polio syndrome
Author: Andreoni G (1), Mazzola M (1), Perego P (1), Standoli CE (1), Manzoni S (1), Piccini L (2), Molteni F (3)
Affiliation: (1) Design Department, Politecnico di Milano, via G. Durando 38/A, Milan 20158, Italy; (2) 6SXT-Sistemi per Telemedicina s.r.l., via M. D'Oggiono 18/A, Lecco 23900, Italy; (3) Villa Beretta Rehabilitation Center, Valduce Hospital, Via N.Sauro, 17 - 23845 Costa Masnaga (LC), Italy
Journal: Sensors
Citation: Sensors (Basel). 2014 Jan 24;14(2):2012-27. doi: 10.3390/s140202012
Publication Year and Month: 2014 01

Abstract: The correct choice and customization of an orthosis are crucial to obtain the best comfort and efficiency. This study explored the feasibility of a multivariate quantitative assessment of the functional efficiency of lower limb orthosis through a novel wearable system. Gait basographic parameters and energetic indexes were analysed during a Six-Minute Walking Test (6-MWT) through a cost-effective, non-invasive polygraph device, with a multichannel wireless transmission, that carried out electro-cardiograph (ECG); impedance-cardiograph (ICG); and lower-limb accelerations detection. Four subjects affected by Post-Polio Syndrome (PPS) were recruited. The wearable device and the semi-automatic post-processing software provided a novel set of objective data to assess the overall efficiency of the patient-orthosis system. Despite the small number of examined subjects, the results obtained with this new approach encourage the application of the method thus enlarging the dataset to validate this promising protocol and measuring system in supporting clinical decisions and out of a laboratory environment.

Conclusions:

Outcome of Research: More research required

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There are currently 29 research papers whose outcomes are rated as “more research required”.

Category: Complementary Therapies

Title: A randomized controlled trial of coenzyme Q10 for fatigue in the late-onset sequelae of poliomyelitis
Author: Peel MM (1), Cooke M (1), Lewis-Peel HJ (1), Lea RA (2), Moyle W (1)
Affiliation: (1) NHMRC Centre for Research Excellence in Nursing Interventions, Menzies Health Institute Queensland, Centre for Health Practice Innovation, Griffith University, Queensland, Australia; (2) Institute of Health and Biomedical Innovation, Queensland University of Technology, Queensland, Australia
Journal: Complementary Therapies in Medicine
Citation: Complementary Therapies in Medicine 23 (2015), pp. 789-793; DOI information: 10.1016/j.ctim.2015.09.002

Publication Year and Month: 2015 12

Abstract: OBJECTIVE: To determine if coenzyme Q10 alleviates fatigue in the late-onset sequelae of poliomyelitis.

DESIGN: Parallel-group, randomized, placebo-controlled trial.

BACKGROUND SETTING: Coenzyme Q10 has been shown to boost muscle energy metabolism in post-polio subjects but it does not promote muscle strength, endurance or function in polio survivors with post-poliomyelitis syndrome. However, the collective increased energy metabolism might contribute to a reduction in post-polio fatigue.

PARTICIPANTS: Polio survivors from the Australian post-polio networks in Queensland and New South Wales who attribute a moderate to high level of fatigue to their diagnosed late-onset sequelae of poliomyelitis. Those with fatigue-associated comorbidities of diabetes, anaemia, hypothyroidism and fibromyalgia were excluded.

METHOD: Participants were assigned (1:1), with stratification of those who use energy-saving mobility aids, to receive 100 mg coenzyme Q10 or matching placebo daily for 60 days. Participants and investigators were blinded to group allocation. Fatigue was assessed by the Multidimensional Assessment of Fatigue as the primary outcome and the Fatigue Severity Scale as secondary outcome.

RESULTS: Of 103 participants, 54 were assigned to receive coenzyme Q10 and 49 to receive the placebo. The difference in the mean score reductions between the two groups was not statistically significant for either fatigue measure. Oral supplementation with coenzyme Q10 was safe and well-tolerated.

The registration number for the clinical trial is ACTRN 12612000552886.

Conclusions: A daily dose of 100 mg coenzyme Q10 for 60 days does not alleviate the fatigue of the late-onset sequelae of poliomyelitis.

Outcome of Research: Not effective

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Category: Fatigue

Title: Brain volume and fatigue in patients with postpoliomyelitis syndrome
Author: Trojan DA (1), Narayanan S (2), Francis SJ (3), Caramanos Z (4), Robinson A (5), Cardoso M (6), Arnold DL (7)
Affiliation: (1) Department of Neurology and Neurosurgery, Montreal Neurological Institute and Hospital, McGill University Health Centre, 3801 University Street, Montreal, Quebec, Canada H3A 2B4; (2) Magnetic Resonance Spectroscopy Unit, McConnell Brain Imaging Centre, Montreal Neurological Institute and Hospital, McGill University, Montreal, Quebec, Canada; (3) Magnetic Resonance Spectroscopy Unit, McConnell Brain Imaging Centre, Montreal Neurological Institute and Hospital, McGill University, Montreal, Quebec, Canada; (4) Magnetic Resonance Spectroscopy Unit, McConnell Brain Imaging Centre, Montreal Neurological Institute and Hospital, McGill University, Montreal, Quebec, Canada; (5) Department of Neurology and Neurosurgery, Montreal Neurological Institute and Hospital, McGill University Health Centre, Montreal, Quebec, Canada; (6) Clinical Research Unit, Montreal Neurological Institute and Hospital, McGill University, Montreal, Quebec, Canada; (7) Department of Neurology and Neurosurgery, Montreal Neurological Institute and Hospital, McGill University Health Centre, Montreal, Quebec, Canada.
Journal: PM&R: The Journal of Injury, Function, and Rehabilitation
Citation: PM R. 2014 Mar;6(3):215-20. doi: 10.1016/j.pmrj.2013.09.009
Publication Year and Month: 2014 03

Abstract: BACKGROUND: Acute paralytic poliomyelitis is associated with encephalitis. Early brain inflammation may produce permanent neuronal injury with brain atrophy, which may result in symptoms such as fatigue. Brain volume has not been assessed in postpoliomyelitis syndrome (PPS).

OBJECTIVE: To determine whether brain volume is decreased compared with that in normal controls, and whether brain volume is associated with fatigue in patients with PPS.

DESIGN: A cross-sectional study.

SETTING: Tertiary university-affiliated hospital postpolio and multiple sclerosis (MS) clinics.

PARTICIPANTS: Forty-nine ambulatory patients with PPS, 28 normal controls, and 53 ambulatory patients with MS.

METHODS: We studied the brains of all study subjects with magnetic resonance imaging by using a 1.5 T Siemens Sonata machine. The subjects completed the Fatigue Severity Scale. Multivariable linear regression models were computed to evaluate the contribution of PPS and MS compared with controls to explain brain volume.

MAIN OUTCOME MEASUREMENTS: Normalized brain volume (NBV) was assessed with the automated program Structured Image Evaluation, using Normalization, of Atrophy method from the acquired magnetic resonance images. This method may miss brainstem atrophy.

RESULTS: Technically adequate NBV measurements were available for 42 patients with PPS, 27 controls, and 49 patients with MS. The mean (standard deviation) age was 60.9 ± 7.6 years for patients with PPS, 47.0 ± 14.6 years for controls, and 46.2 ± 9.4 years for patients with MS. In a multivariable model adjusted for age and gender, NBV was not significantly different in patients with PPS compared with that in controls (P = .28). As expected, when using a similar model for patients with MS, NBV was significantly decreased compared with that in controls (P = .006). There was no significant association between NBV and fatigue in subjects with PPS (Spearman ρ = 0.23; P = .19).

Conclusions: No significant whole-brain atrophy was found, and no association of brain volume with fatigue in PPS. Brain atrophy was confirmed in MS. It is possible that brainstem atrophy was not recognized by this study.

Outcome of Research: Not effective

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Category: Fatigue

Title: Cognitive functioning in post‐polio patients with and without general fatigue
Author: Ostlund G, Borg K, Wahlin A.
Affiliation: Department of Public Health Science, Karolinska Institutet, Stockholm, Sweden.
Journal: Journal of Rehabilitation Medicine
Citation: J Rehabil Med. 2005 May;37(3):147-51
Publication Year and Month: 2005 03

Abstract: OBJECTIVE AND DESIGN: This study examined 2 main hypotheses. First, whether patients with post-polio suffering from general fatigue (n=10) demonstrate cognitive deficits compared with patients with post-polio without general fatigue (n=10). Secondly, by systematically varied test order administration we examined whether such differences varied as a function of increasing cognitive load during cognitive testing.

SUBJECTS: Twenty patients diagnosed with post-polio syndrome, 10 with general fatigue and 10 without fatigue.

RESULTS: Neither of the 2 hypotheses were confirmed. The group with general fatigue reported elevated levels of depression. However, no systematic association between level of depression and cognitive performance could be detected.

Conclusions: The results of this study provide no evidence that general fatigue or cognitive load affects cognitive functioning in post-polio.

Outcome of Research: Not effective.

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There are currently 3 research papers whose outcomes are rated as “not effective”.

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