Parkinsonism and Related Disorders xxx (2014) 1e3
Contents lists available at ScienceDirect
Parkinsonism and Related Disorders journal homepage: www.elsevier.com/locate/parkreldis
Restless legs syndrome in patients with sequelae of poliomyelitis Hatice Kumru a, b, c, *, Enric Portell a, b, c, Manuela Barrio a, b, c, Joan Santamaria d adscrit a la UAB, 08916 Badalona, Barcelona, Spain Institut Guttmann, Institut Universitari de Neurorehabilitacio Univ Autonoma de Barcelona, 08193 Bellaterra, Cerdanyola del Vall es, Spain c Institut d'Investigacio en Ci Fundacio encies de la Salut Germans Trias i Pujol, Badalona, Barcelona, Spain d Biom Hospital Clinic, Institut d'Investigacio edica August Pi I Sunyer (IDIBAPS), University of Barcelona, Barcelona, Spain a
b
a r t i c l e i n f o
a b s t r a c t
Article history: Received 31 January 2014 Received in revised form 13 June 2014 Accepted 17 June 2014
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. © 2014 Elsevier Ltd. All rights reserved.
Keywords: Restless leg syndrome Sequelae of poliomyelitis Dopaminergic agonist
1. Introduction Restless legs syndrome (RLS), also known as WilliseEkbom disease, is a common disorder affecting up to 5e10% of the general population and is characterized by variable degrees of unpleasant sensations deep inside the legs, occurring especially at rest and during the night [1e4]. According to the standard criteria, the diagnosis of RLS may be established when the following four clinical features are met [4]: 1) an urge to move the limbs with or without sensations, 2) improvement with activity, 3) worsening at rest, and 4) worsening in the evening or night. A positive therapeutic response to dopaminergic drugs also supports the diagnosis of RLS [4,5]. Increased RLS frequency has also been reported in neurodegenerative disorders [6] and multiple sclerosis [7].
Institut Guttmann, Camí * Corresponding author. Hospital de Neurorehabilitacio Can Ruti s/n. Barcelona, 08916 Barcelona, Spain. Tel.: þ34 93 497 77 00; fax: þ34 93 497 77 15. E-mail addresses: [email protected], [email protected] (H. Kumru).
However, no studies have examined the association between RLS and the sequelae of poliomyelitis (PM). Acute poliomyelitis (PM) induces a flaccid paralysis that usually leads to a clinically evident sequel of non-progressive motor deficit in one or more limbs with decreased or absent tendon reflexes and without sensory or cognitive loss [8]. Post-polio syndrome (pPS) is a neuromuscular condition characterized by new neurological, musculoskeletal and systemic symptoms and signs including new muscle weakness and atrophy or increased limb fatigability, pain from muscles and joints [9]. Because poliomyelitis affects the anterior horn cells in the anterior part of the spinal cord (the first part of the motor unit), post-polio syndrome is a lower motor neuron disorder [9]. Although there are isolated reports of RLS occurring in pPS [10,11], no studies have described the frequency of RLS in patients following PM or in pPS. Here, we studied the frequency and severity of RLS in a group of consecutive patients with PM and evaluated the effect of treatment with dopaminergic drugs. 2. Patients and methods Consecutive patients with the sequelae of poliomyelitis (PM) were included in the study during their routine visit (by EP) to our sequelae of poliomyelitis Centre at
http://dx.doi.org/10.1016/j.parkreldis.2014.06.014 1353-8020/© 2014 Elsevier Ltd. All rights reserved.
Please cite this article in press as: Kumru H, et al., Restless legs syndrome in patients with sequelae of poliomyelitis, Parkinsonism and Related Disorders (2014), http://dx.doi.org/10.1016/j.parkreldis.2014.06.014
2
H. Kumru et al. / Parkinsonism and Related Disorders xxx (2014) 1e3
the Institut Guttmann between February 2011 and March 2013. For the diagnosis of PPS, we used the criteria given by the March of Dimes [12]. The Institutional review board and the Ethical Committee of Institut Guttmann revised and approved the protocol. For patients with the sequalae of PM admitted to our hospital we use a routine protocol which includes general blood analysis including renal and hepatic function and electromyography (EMG). Patients were admitted to the study after a neurologist (HK) established the presence of all four criteria for RLS using the four standard diagnostic criteria proposed by the International RLS Study Group [4]. When RLS symptoms occurred at least 2 times/wk, treatment with dopaminergic agonist (pramipexole 0.18 mg/1 h before the beginning of RLS symptoms) was offered to the patient. Severity of RLS symptoms was assessed with the RLS severity scale [13]. This instrument consists of 10 items assessing subjective severity of RLS-related complaints on a scale from 0 to 4, with a maximum score of 40. The RLS severity scale was repeated at two months. Six months after the initiation of treatment, we evaluated the patients to establish whether the effect of treatment was sustained. Patients were also asked whether their symptoms were symmetrical in their legs and if not in which side were they more disturbing (the affected or the less-affected or non-affected leg).
3. Data analysis We separated the PM patients into two groups, with and without RLS. Data were expressed as mean and standard deviation (SD). We used Student-t test and c2 tests for the comparison of demographic and clinical data between two groups. Student-t test was used compare the RLS rating scale before and during treatment. Statistical analyses were performed with SPSS 13.0. p value was set at
Contents lists available at ScienceDirect
Parkinsonism and Related Disorders journal homepage: www.elsevier.com/locate/parkreldis
Restless legs syndrome in patients with sequelae of poliomyelitis Hatice Kumru a, b, c, *, Enric Portell a, b, c, Manuela Barrio a, b, c, Joan Santamaria d adscrit a la UAB, 08916 Badalona, Barcelona, Spain Institut Guttmann, Institut Universitari de Neurorehabilitacio Univ Autonoma de Barcelona, 08193 Bellaterra, Cerdanyola del Vall es, Spain c Institut d'Investigacio en Ci Fundacio encies de la Salut Germans Trias i Pujol, Badalona, Barcelona, Spain d Biom Hospital Clinic, Institut d'Investigacio edica August Pi I Sunyer (IDIBAPS), University of Barcelona, Barcelona, Spain a
b
a r t i c l e i n f o
a b s t r a c t
Article history: Received 31 January 2014 Received in revised form 13 June 2014 Accepted 17 June 2014
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. © 2014 Elsevier Ltd. All rights reserved.
Keywords: Restless leg syndrome Sequelae of poliomyelitis Dopaminergic agonist
1. Introduction Restless legs syndrome (RLS), also known as WilliseEkbom disease, is a common disorder affecting up to 5e10% of the general population and is characterized by variable degrees of unpleasant sensations deep inside the legs, occurring especially at rest and during the night [1e4]. According to the standard criteria, the diagnosis of RLS may be established when the following four clinical features are met [4]: 1) an urge to move the limbs with or without sensations, 2) improvement with activity, 3) worsening at rest, and 4) worsening in the evening or night. A positive therapeutic response to dopaminergic drugs also supports the diagnosis of RLS [4,5]. Increased RLS frequency has also been reported in neurodegenerative disorders [6] and multiple sclerosis [7].
Institut Guttmann, Camí * Corresponding author. Hospital de Neurorehabilitacio Can Ruti s/n. Barcelona, 08916 Barcelona, Spain. Tel.: þ34 93 497 77 00; fax: þ34 93 497 77 15. E-mail addresses: [email protected], [email protected] (H. Kumru).
However, no studies have examined the association between RLS and the sequelae of poliomyelitis (PM). Acute poliomyelitis (PM) induces a flaccid paralysis that usually leads to a clinically evident sequel of non-progressive motor deficit in one or more limbs with decreased or absent tendon reflexes and without sensory or cognitive loss [8]. Post-polio syndrome (pPS) is a neuromuscular condition characterized by new neurological, musculoskeletal and systemic symptoms and signs including new muscle weakness and atrophy or increased limb fatigability, pain from muscles and joints [9]. Because poliomyelitis affects the anterior horn cells in the anterior part of the spinal cord (the first part of the motor unit), post-polio syndrome is a lower motor neuron disorder [9]. Although there are isolated reports of RLS occurring in pPS [10,11], no studies have described the frequency of RLS in patients following PM or in pPS. Here, we studied the frequency and severity of RLS in a group of consecutive patients with PM and evaluated the effect of treatment with dopaminergic drugs. 2. Patients and methods Consecutive patients with the sequelae of poliomyelitis (PM) were included in the study during their routine visit (by EP) to our sequelae of poliomyelitis Centre at
http://dx.doi.org/10.1016/j.parkreldis.2014.06.014 1353-8020/© 2014 Elsevier Ltd. All rights reserved.
Please cite this article in press as: Kumru H, et al., Restless legs syndrome in patients with sequelae of poliomyelitis, Parkinsonism and Related Disorders (2014), http://dx.doi.org/10.1016/j.parkreldis.2014.06.014
2
H. Kumru et al. / Parkinsonism and Related Disorders xxx (2014) 1e3
the Institut Guttmann between February 2011 and March 2013. For the diagnosis of PPS, we used the criteria given by the March of Dimes [12]. The Institutional review board and the Ethical Committee of Institut Guttmann revised and approved the protocol. For patients with the sequalae of PM admitted to our hospital we use a routine protocol which includes general blood analysis including renal and hepatic function and electromyography (EMG). Patients were admitted to the study after a neurologist (HK) established the presence of all four criteria for RLS using the four standard diagnostic criteria proposed by the International RLS Study Group [4]. When RLS symptoms occurred at least 2 times/wk, treatment with dopaminergic agonist (pramipexole 0.18 mg/1 h before the beginning of RLS symptoms) was offered to the patient. Severity of RLS symptoms was assessed with the RLS severity scale [13]. This instrument consists of 10 items assessing subjective severity of RLS-related complaints on a scale from 0 to 4, with a maximum score of 40. The RLS severity scale was repeated at two months. Six months after the initiation of treatment, we evaluated the patients to establish whether the effect of treatment was sustained. Patients were also asked whether their symptoms were symmetrical in their legs and if not in which side were they more disturbing (the affected or the less-affected or non-affected leg).
3. Data analysis We separated the PM patients into two groups, with and without RLS. Data were expressed as mean and standard deviation (SD). We used Student-t test and c2 tests for the comparison of demographic and clinical data between two groups. Student-t test was used compare the RLS rating scale before and during treatment. Statistical analyses were performed with SPSS 13.0. p value was set at
