Frequency and Characterization of Movement Disorders in Anti-IgLON5 Disease


Por: Gaig, C, Compta, Y, Heidbreder, A, Marti, MJ, Titulaer, MJ, Crijnen, Y, Hogl, B, Lewerenz, J, Erro, ME, Garcia-Monco, JC, Nigro, P, Tambasco, N, Patalong-Ogiewa, M, Erdler, M, Macher, S, Berger-Sieczkowski, E, Hoftberger, R, Geis, C, Hutterer, M, Milan-Tomas, A, Martin-Bastida, A, Manzanares, LL, Quintas, S, Hoglinger, GU, Mohn, N, Schoberl, F, Thaler, FS, Asioli, GM, Provini, F, Plazzi, G, Berganzo, K, Blaabjerg, M, Bruggemann, N, Farias, T, Ng, CF, Giordana, C, Herrero-San Martin, A, Huebra, L, Kotschet, K, Liendl, H, Montojo, T, Morata, C, Perez, JP, Puertas, I, Seifert-Held, T, Seitz, C, Simabukuro, MM, Tellez, N, Villacieros-Alvarez, J, Willekens, B, Sabater, L, Iranzo, A, Cano, JS, Dalmau, J, Graus, F

Publicada: 5 oct 2021
Resumen:
Background and Objectives Anti-IgLON5 disease is a recently described neurologic disease that shares features of autoimmunity and neurodegeneration. Abnormal movements appear to be frequent and important but have not been characterized and are underreported. We describe the frequency and types of movement disorders in a series of consecutive patients with this disease. Methods In this retrospective, observational study, the presence and phenomenology of movement disorders were assessed with a standardized clinical questionnaire. Available videos were centrally reviewed by 3 experts in movement disorders. Results Seventy-two patients were included. In 41 (57%), the main reason for initial consultation was difficulty walking along with one or several concurrent movement disorders. At the time of anti-IgLON5 diagnosis, 63 (87%) patients had at least 1 movement disorder with a median of 3 per patient. The most frequent abnormal movements were gait and balance disturbances (52 patients [72%]), chorea (24 [33%]), bradykinesia (20 [28%]), dystonia (19 [26%]), abnormal body postures or rigidity (18 [25%]), and tremor (15 [21%]). Other hyperkinetic movements (myoclonus, akathisia, myorhythmia, myokymia, or abdominal dyskinesias) occurred in 26 (36%) patients. The craniofacial region was one of the most frequently affected by multiple concurrent movement disorders (23 patients [32%]) including dystonia (13), myorhythmia (6), chorea (4), or myokymia (4). Considering any body region, the most frequent combination of multiple movement disorders consisted of gait instability or ataxia associated with craniofacial dyskinesias or generalized chorea observed in 31 (43%) patients. In addition to abnormal movements, 87% of patients had sleep alterations, 74% bulbar dysfunction, and 53% cognitive impairment. Fifty-five (76%) patients were treated with immunotherapy, resulting in important and sustained improvement of the movement disorders in only 7 (13%) cases. Discussion Movement disorders are a frequent and leading cause of initial neurologic consultation in patients with anti-IgLON5 disease. Although multiple types of abnormal movements can occur, the most prevalent are disorders of gait, generalized chorea, and dystonia and other dyskinesias that frequently affect craniofacial muscles. Overall, anti-IgLON5 disease should be considered in patients with multiple movement disorders, particularly if they occur in association with sleep alterations, bulbar dysfunction, or cognitive impairment.

Filiaciones:
Gaig, C:
 Inst Invest Biomed August Pi & Sunyer IDIBAPS, Neuroimmunol Program, Barcelona, Spain

 Hosp Clin Barcelona, Dept Neurol, Barcelona, Spain

 Hosp Clin Barcelona, Multidisciplinary Sleep Disorders Unit, Barcelona, Spain

Compta, Y:
 Hosp Clin Barcelona, Dept Neurol, Barcelona, Spain

 Hosp Clin Barcelona, Parkinsons Dis & Movement Disorders Unit, Barcelona, Spain

Heidbreder, A:
 Univ Hosp Muenster, Dept Neurol, Div Sleep Med & Neuromuscular Disorders, Munster, Germany

 Med Univ Innsbruck, Dept Neurol, Innsbruck, Austria

Marti, MJ:
 Hosp Clin Barcelona, Dept Neurol, Barcelona, Spain

 Hosp Clin Barcelona, Parkinsons Dis & Movement Disorders Unit, Barcelona, Spain

Titulaer, MJ:
 Erasmus MC Univ Med Ctr, Dept Neurol, Rotterdam, Netherlands

Crijnen, Y:
 Erasmus MC Univ Med Ctr, Dept Neurol, Rotterdam, Netherlands

Hogl, B:
 Med Univ Innsbruck, Dept Neurol, Innsbruck, Austria

Lewerenz, J:
 Ulm Univ, Dept Neurol, Ulm, Germany

Erro, ME:
 Complejo Hosp Navarra, Navarra Inst Hlth Res IdiSNA, Neurol Dept, Pamplona, Spain

Garcia-Monco, JC:
 Hosp Univ Basun, Neurol Dept, Bilbao, Spain

 Hosp Galdakao, Dept Neurol, Bizkaia, Spain

Nigro, P:
 Perugia Gen Hosp, Movement Disorders Ctr, Perugia, Italy

 Univ Perugia, Perugia, Italy

Tambasco, N:
 Perugia Gen Hosp, Movement Disorders Ctr, Perugia, Italy

 Univ Perugia, Perugia, Italy

Patalong-Ogiewa, M:
 Med Univ Silesia, Fac Med Sci Katowice, Dept Neurol & Neurorehabil, Katowice, Poland

Erdler, M:
 Karl Landsteiner Inst, Dept Neurol, Klin Donaustadt, Vienna, Austria

Hoftberger, R:
 Med Univ Vienna, Dept Neurol, Div Neuropathol & Neurochem, Vienna, Austria

Geis, C:
 Jena Univ Hosp, Dept Neurol, Sect Translat Neuroimmunol, Jena, Germany

Hutterer, M:
 Kepler Univ Klinikum Linz, Dept Neurol 1, Neuromed Campus, Linz, Austria

 St John God Hosp Linz, Stroke Unit, Dept Neurol, Linz, Austria

 St John God Hosp Linz, Acute Geriatr, Linz, Austria

Milan-Tomas, A:
 Clin Univ Navarra, Dept Neurol & Neurosci, Pamplona, Spain

Martin-Bastida, A:
 Clin Univ Navarra, Dept Neurol & Neurosci, Pamplona, Spain

Manzanares, LL:
 Hosp La Princesa, Dept Neurol, Madrid, Spain

Quintas, S:
 Hosp La Princesa, Dept Neurol, Madrid, Spain

Hoglinger, GU:
 Hannover Med Sch, Dept Neurol, Hannover, Germany

Mohn, N:
 Hannover Med Sch, Dept Neurol, Hannover, Germany

Schoberl, F:
 Ludwig Maximilians Univ Munchen, Dept Neurol, Univ Hosp, Munich, Germany

 Ludwig Maximilians Univ Munchen, Univ Hosp, Inst Clin Neuroimmunol, Munich, Germany

 Ludwig Maximilians Univ Munchen, Biomed Ctr, Munich, Germany

Asioli, GM:
 Alma Mater Studiorum Univ Bologna, Dept Biomed & NeuroMotor Sci DiBiNeM GMA, FP, Bologna, Italy

Provini, F:
 Alma Mater Studiorum Univ Bologna, Dept Biomed & NeuroMotor Sci DiBiNeM GMA, FP, Bologna, Italy

 IRCCS Ist Sci Neurol Bologna, Bologna, Italy

Plazzi, G:
 Univ Modena & Reggio Emilia, Dept Biomed Metab & Neural Sci, Bologna, Italy

 Inst Neurol Sci Bologna, IRCCS, Bologna, Italy

Berganzo, K:
 Cruces Univ Hosp, Neurol Dept, Baracaldo, Spain

Blaabjerg, M:
 Odense Univ Hosp, Dept Neurol, Odense, Denmark

Bruggemann, N:
 Univ Lubeck, Dept Neurol, Lubeck, Germany

 Univ Lubeck, Inst Neurogenet, Lubeck, Germany

Farias, T:
 Hosp Geral Cleriston Andrade, Dept Neurol, Med Clin, Feira De Santana, BA, Brazil

Ng, CF:
 Univ Kebangsaan Malaysia, Dept Med, Neurol Unit, Med Ctr, Kuala Lumpur, Malaysia

Giordana, C:
 CHU Nice, Dept Movement Disorders & Neurol, Nice, France

Herrero-San Martin, A:
 Hosp Univ 12 Octubre, Dept Neurol, Madrid, Spain

Huebra, L:
 Univ Fed Sao Paulo, Dept Psychobiol, Sao Paulo, SP, Brazil

Kotschet, K:
 St Vincents Hosp, Clin Neurosci, Melbourne, Vic, Australia

Liendl, H:
 LKH Murtal, Dept Neurol, Standort Knittelfeld, Austria

Montojo, T:
 Fdn Jimenez Diaz, Dept Neurol, Madrid, Spain

Morata, C:
 Hosp Univ & Politecn La Fe, Dept Neurol, Valencia, Spain

Perez, JP:
 Hosp Santa Creu & Sant Pau, Dept Neurol, Barcelona, Spain

Puertas, I:
 Hosp La Paz, Dept Neurol, Madrid, Spain

Seifert-Held, T:
 Med Univ Graz, Dept Neurol, Graz, Austria

Seitz, C:
 Mainz Univ Hosp, Dept Neurol, Mainz, Germany

Simabukuro, MM:
 Univ Sao Paulo, Dept Neurol, Med Sch, Sao Paulo, SP, Brazil

Tellez, N:
 Hosp Clin Univ, Dept Neurol, Valladolid, Spain

Villacieros-Alvarez, J:
 Hosp Univ Rey Juan Carlos, Dept Neurol, Madrid, Spain

Willekens, B:
 Antwerp Univ Hosp, Dept Neurol, Antwerp, Belgium

 Univ Antwerp, Antwerp, Belgium

Sabater, L:
 Inst Invest Biomed August Pi & Sunyer IDIBAPS, Neuroimmunol Program, Barcelona, Spain

Iranzo, A:
 Inst Invest Biomed August Pi & Sunyer IDIBAPS, Neuroimmunol Program, Barcelona, Spain

 Hosp Clin Barcelona, Dept Neurol, Barcelona, Spain

 Hosp Clin Barcelona, Multidisciplinary Sleep Disorders Unit, Barcelona, Spain

Cano, JS:
 Inst Invest Biomed August Pi & Sunyer IDIBAPS, Neuroimmunol Program, Barcelona, Spain

 Hosp Clin Barcelona, Dept Neurol, Barcelona, Spain

 Hosp Clin Barcelona, Multidisciplinary Sleep Disorders Unit, Barcelona, Spain

Dalmau, J:
 Inst Invest Biomed August Pi & Sunyer IDIBAPS, Neuroimmunol Program, Barcelona, Spain

 Hosp Clin Barcelona, Dept Neurol, Barcelona, Spain

 Inst Catalana Recerca & Estudis Avancats ICREA, Barcelona, Spain

Graus, F:
 Inst Invest Biomed August Pi & Sunyer IDIBAPS, Neuroimmunol Program, Barcelona, Spain
ISSN: 00283878
Editorial
LIPPINCOTT WILLIAMS & WILKINS, TWO COMMERCE SQ, 2001 MARKET ST, PHILADELPHIA, PA 19103 USA, USA
Tipo de documento: Article
Volumen: 97 Número: 14
Páginas: 1367-1381
WOS Id: 000711572500019
ID de PubMed: 34380749
imagen Green Submitted

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