Bassetti, Claudio

Kallweit, Ulf

Tartarotti, Sonja

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LETTER TO THE EDITORSAssociation of restless legs syndrome, chronic motor tic disorderand migraine with aura: a case of a single familySonja Tartarotti • Ulf Kallweit • Claudio L. BassettiReceived: 20 September 2009 / Revised: 1 January 2010 / Accepted: 20 January 2010 / Published online: 13 February 2010 Springer-Verlag 2010Sirs,Over the past 20 years there has been a growing interest inthe pathophysiology and the possible association betweeninvoluntary movement disorders such as restless legs syn-drome (RLS) and Tourette syndrome (TS) [1, 2]. Indeed,recent findings have found support for a common geneticbackground of RLS and TS. Lespe´rance et al. [3] reported ahigh prevalence of RLS in children and adolescents withTS, pointing out that the occurrence of RLS in individualswith TS seems to be linked to maternal RLS. There is,however, little data supporting the association of thesesyndromes with migraine [4, 5]. The study by Kwak et al.[4] suggested that patients with TS show a higher co-morbidity with migraine than in the general population. Inturn, Larner [5] examined a family suffering from migrainewith aura (MA) and RLS, suggesting a genetic linkbetween the two disorders. This finding supports thehypothesis of a shared genetic aetiology of RLS andmigraine proposed in a previous publication [6]. However,to the best of our knowledge, the co-morbidity of the threedisorders, RLS, TS, and MA has not yet been reported.Here we report the case of 59-year-old woman and herimmediate family, who, at the time of examination, pre-sented with the rare combination of RLS, chronic motor ticdisorder, and migraine aura without headache. The patientpresented to the clinic with choreatiform movements of theright shoulder, as well as twitches of the face-muscles onthe same side. These were triggered by stress and agitationand brought a feeling of relief to the sensation of tensenessfelt before. These movements were never accompanied byvocalisations and thus fit the criteria of a chronic motor ticdisorder according to the International Classification ofDiseases, 10th revision (2007). Since the age of 20, she hadbeen complaining about aching legs, including paresthesia,which emerged while being in a recumbent position. Thesebecame worse in the evening and night hours and wereassociated with the urge to move. Laboratory analyses,including iron status, were normal. Therefore, conformingto the criteria of International Classification of Sleep Dis-orders, 2nd edition, idiopathic RLS was diagnosed. Duringpolysomnography, slightly increased periodic leg move-ments during sleep were found (PLMS Index 16/h). Atherapy-trial with pramipexole (up to 0.5 mg/d) for1 month aggravated involuntary motor movements and didnot lead to any improvement of RLS-symptoms, whileclonazepam (0.5 mg) in the evening had a positive effecton both RLS and motor tic disorder. Furthermore, thepatient was afflicted with visual aura without headache,meeting the criteria of The International Classification ofHeadache Disorders, 2nd edition.Family history (see Fig. 1), revealed that her father (I.1),brother (II.1), and one of two daughters (III.4) displayed allthree disorders while her sister (II.2) and the other daughter(III.5) suffered from MA and RLS, without signs of motortic disorder. Additionally, three nephews (III.1/2/3)exclusively exhibited MA.Finally, the co-occurrence of these syndromes in thisfamily suggests that there might be a pathophysiologicalrelationship between RLS, chronic motor tic disorder andmigraine. Some similar clinical characteristics of RLS andmotor tic disorder, in particular the urge to move whichprecedes it and the subsequent alleviation by motor activ-ity, indicate an implication of the basal ganglia and theS. Tartarotti  U. Kallweit (&)  C. L. BassettiDepartment of Neurology, University Hospital Zurich,Frauenklinikstrasse 26, 8091 Zurich, Switzerlande-mail: ulf.kallweit@usz.ch123J Neurol (2010) 257:1043–1044DOI 10.1007/s00415-010-5483-xlimbic network in the pathophysiology of the two disorders[7, 8]. Moreover, Barbanti et al. [9] concluded that the highprevalence of migraine in TS might be linked to a dys-function of the extrapyramidal system.Further genetic analyses are needed to confirm ourhypothesis and may lead to new therapeutic strategies.References1. Voderholzer U, Muller N, Haag C, Riemann D, Straube A (1997)Periodic limb movements during sleep are a frequent finding inGilles de la Tourette’s syndrome. J Neurol 244:521–5262. Lipinski JF, Sallee FR, Jackson C, Sethuraman G (1997)Dopamine agonist treatment of Tourette disorder in children:result of an open-label trial of pergolide. Mov Disord 12:402–4073. Lespe´rance P, Djerroud N, Diaz Anzaldua A, Rouleau GA,Chouinard S, Richer F, The Montre´al Tourette Study Group (2004)Restless legs in Tourette syndrome. Mov Disord 19:1084–10874. Kwak C, Dat Vuong K, Jankovic J (2003) Migraine headache inpatients with Tourette syndrome. Arch Neurol 60:1595–15985. Larner AJ (2007) Migraine with aura and restless legs syndrome. JHeadache Pain 8:141–1426. Sabayan B, Bagheri M, Borhani Haghighi A (2007) Possible jointorigin of restless legs syndrome (RLS) and migraine. MedHypotheses 69:64–667. Turjanski N, Lees AJ, Brooks DJ (1999) Striatal dopaminergicfunction in restless legs syndrome: 18F-dopa and 11C-raclopridePET studies. Neurology 52:932–9378. Singer HS (2000) Current issues in Tourette syndrome. MovDisord 15:1051–10639. Barbanti P, Fabbrini G (2004) Migraine and Tourette syndrome.Arch Neurol 61:606–607Fig. 1 Family tree. Striped square migraine aura without headache,RLS, motor tic disorder; dark filled square MA, RLS, motor ticdisorder; light filled square MA, RLS; cross stripped square MA1044 J Neurol (2010) 257:1043–1044123

Association of restless legs syndrome, chronic motor tic disorder and migraine with aura: a case of a single family

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Association of restless legs syndrome, chronic motor tic disorder and migraine with aura: a case of a single family

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