Treatment of MOG antibody associated disorders: results of an international survey.,Journal of Neurology

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Treatment of MOG antibody associated disorders: results of an international survey.
Journal of Neurology ( IF 6 ) Pub Date : 2020-07-04 , DOI: 10.1007/s00415-020-10026-y
D H Whittam ₁ , V Karthikeayan ₁ , E Gibbons ₁ , R Kneen ₂ , S Chandratre ₃ , O Ciccarelli ₄ , Y Hacohen _{4,

5} , J de Seze ₆ , K Deiva ₇ , R Q Hintzen ₈ , B Wildemann ₉ , S Jarius ₉ , I Kleiter _{10,

11} , K Rostasy ₁₂ , P Huppke ₁₃ , B Hemmer _{14,

15} , F Paul ₁₆ , O Aktas ₁₇ , A K Pröbstel _{18,

19} , G Arrambide ₂₀ , M Tintore ₂₀ , M P Amato _{21,

22} , M Nosadini ₂₃ , M M Mancardi ₂₄ , M Capobianco ₂₅ , Z Illes ₂₆ , A Siva ₂₇ , A Altintas ₂₈ , G Akman-Demir ₂₉ , L Pandit ₃₀ , M Apiwattankul ₃₁ , J Y Hor ₃₂ , S Viswanathan ₃₃ , W Qiu ₃₄ , H J Kim ₃₅ , I Nakashima ₃₆ , K Fujihara _{37,

38} , S Ramanathan _{39,

40} , R C Dale _{39,

40} , M Boggild ₄₁ , S Broadley ₄₂ , M A Lana-Peixoto ₄₃ , D K Sato ₄₄ , S Tenembaum ₄₅ , P Cabre ₄₆ , D M Wingerchuk ₄₇ , B G Weinshenker ₄₈ , B Greenberg ₄₉ , M Matiello ₅₀ , E C Klawiter ₅₀ , J L Bennett ₅₁ , A I Wallach ₅₂ , I Kister ₅₂ , B L Banwell ₅₃ , A Traboulsee ₅₄ , D Pohl ₅₅ , J Palace ₃ , M I Leite ₃ , M Levy ₅₀ , R Marignier ₅₆ , T Solomon _{1,

57} , M Lim _{58,

59} , S Huda ₁ , A Jacob _{1,

60}

Affiliation

Department of Neurology, The Walton Centre NHS Foundation Trust, Liverpool, UK.
Department of Paediatric Neurology, Alder Hey Children's Hospital, Liverpool, UK.
Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, Oxford, UK.
Queen Square Multiple Sclerosis Centre, UCL Institute of Neurology, London, UK.
Department of Paediatric Neurology, Great Ormond Street Hospital for Children, London, UK.
Department of Neurology and Clinical Investigation Center, CHU de Strasbourg, Hôpital de Hautepiere, Strasbourg, France.
Department of Paediatric Neurology, CHU Paris-Sud, Hôpital de Bicêtre, Le Kremlin Bicêtre, France.
Department of Neurology, MS Centre ErasMS, Erasmus MC, Rotterdam, The Netherlands.
Molecular Neuroimmunology Group, Department of Neurology, University of Heidelberg, Heidelberg, Germany.
Department of Neurology, St. Josef Hospital, Ruhr University Bochum, Bochum, Germany.
Marianne-Strauß-Klinik, Behandlungszentrum Kempfenhausen für Multiple Sklerose Kranke, Berg, Germany.
Department of Paediatric Neurology, Children's Hospital Datteln, Witten/Herdrecke University, Datteln, Germany.
Department of Paediatrics and Paediatric Neurology, University Medical Center Göttingen, Georg-August University, Göttingen, Germany.
Department of Neurology, School of Medicine, Technical University of Munich, Munich, Germany.
Munich Cluster for Systems Neurology (SyNergy), Munich, Germany.
NeuroCure Clinical Research Center, and Experimental and Clinical Research Center, Max Delbrueck Center for Molecular Medicine, Charité Universitätsmedizin Berlin, Berlin, Germany.
Department of Neurology, Heinrich-Heine-Universität Düsseldorf, 40225, Düsseldorf, Germany.
Department of Neurology and Weill Institute for Neurosciences, University of California, San Francisco, CA, USA.
Neurologic Clinic and Policlinic, Department of Medicine and Biomedicine, University Hospital Basel, University of Basel, Basel, Switzerland.
Servei de Neurologia-Neuroimmunologia, Centre d'Esclerosi Múltiple de Catalunya, (Cemcat), Vall d'Hebron Institut de Recerca, Hospital Universitari Vall d'Hebron, Universitat Autònoma de Barcelona, Barcelona, Spain.
Department NEUROFARBA, Neurosciences Section, University of Florence, Florence, Italy.
IRCCS Fondazione Don Carlo Gnocchi, Florence, Italy.
Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Padua, Italy.
Department of Medical and Surgical Neurosciences and Rehabilitation, IRCCS Giannina Gaslini Institute, Genoa, Italy.
Department of Neurology and Regional Multiple Sclerosis Centre, University Hospital San Luigi Gonzaga, Orbassano, Italy.
Department of Neurology, Odense University Hospital, Odense, Denmark.
Department of Neurology, Istanbul University Cerrahpaşa School of Medicine, Istanbul, Turkey.
Department of Neurology, School of Medicine, Koc University, Istanbul, Turkey.
Department of Neurology, Bilim University, Istanbul, Turkey.
Department of Neurology, Nitte University, Mangalore, Karnataka, India.
Neuroimmunology Unit, Department of Neurology, Prasat Neurological Institute, Ministry of Public Health, Bangkok, Thailand.
Department of Neurology, Penang General Hospital, Penang, Malaysia.
Department of Neurology, Kuala Lumpur Hospital, Ministry of Health of Malaysia, Kuala Lumpur, Malaysia.
Department of Neurology, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, Guangdong, China.
Department of Neurology, Research Institute and Hospital of National Cancer Center, Goyang, Korea.
Department of Neurology, Tohoku Medical and Pharmaceutical University, Sendai, Japan.
Department of Multiple Sclerosis Therapeutics, Fukushima Medical University School of Medicine and Southern TOHOKU Research Institute for Neuroscience, Koriyama, Japan.
MS and NMO Center, Southern TOHOKU Research Institute for Neuroscience (STRINS), Koriyama, Japan.
Sydney Medical School, University of Sydney, Sydney, NSW, Australia.
Neuroimmunology Group, Kids Neuroscience Centre, Children's Hospital At Westmead, Sydney, NSW, Australia.
Department of Neurology, Townsville Hospital, Townsville, QLD, Australia.
School of Medicine, Gold Coast Campus, Griffith University, Mount Gravatt, QLD, Australia.
CIEM MS Research Center, Federal University of Minas Gerais Medical School, Belo Horizonte, MG, Brazil.
Instituto do Cérebro do Rio Grande do Sul (InsCer), Pontifícia Universidade Católica do Rio Grande do Sul, Porto Alegre, Brazil.
Department of Neurology, National Pediatric Hospital Dr. Juan P. Garrahan, Buenos Aires, Argentina.
Department of Neurology, Pierre Zobda Quitman Hospital, Fort-de-France, France.
Department of Neurology, Mayo Clinic, Scottsdale, AZ, USA.
Department of Neurology, Mayo Clinic, Rochester, MN, USA.
Department of Neurology and Neurotherapeutics, University of Texas Southwestern Medical Center, Dallas, TX, USA.
Department of Neurology, Massachusetts General Hospital and Harvard Medical School, Boston, MA, USA.
Department of Ophthalmology, University of Colorado at Anschutz Medical Campus, Aurora, CO, USA.
Department of Neurology, NYU School of Medicine, New York, NY, USA.
Department of Neurology, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Division of Neurology, Department of Medicine, University of British Columbia, Vancouver, BC, Canada.
Division of Neurology, Children's Hospital of Eastern Ontario, Ottawa, ON, Canada.
Service de Neurologie, Hôpital Neurologique Pierre Wertheimer Hospices Civils de Lyon, Lyon, France.
National Institute for Health Research Health Protection Research Unit in Emerging and Zoonotic Infections, Institute of Infection and Global Health, University of Liverpool, Liverpool, UK.
Children's Neuroscience, Evelina London Children's Hospital, Guy's and St Thomas' NHS Foundation Trust, London, UK.
Faculty of Life Sciences and Medicine, King's College London, London, UK.
Department of Neurology, Cleveland Clinic Abu Dhabi, Abu Dhabi, United Arab Emirates.

Introduction

While monophasic and relapsing forms of myelin oligodendrocyte glycoprotein antibody associated disorders (MOGAD) are increasingly diagnosed world-wide, consensus on management is yet to be developed.

Objective

To survey the current global clinical practice of clinicians treating MOGAD.

Method

Neurologists worldwide with expertise in treating MOGAD participated in an online survey (February–April 2019).

Results

Fifty-two responses were received (response rate 60.5%) from 86 invited experts, comprising adult (78.8%, 41/52) and paediatric (21.2%, 11/52) neurologists in 22 countries. All treat acute attacks with high dose corticosteroids. If recovery is incomplete, 71.2% (37/52) proceed next to plasma exchange (PE). 45.5% (5/11) of paediatric neurologists use IV immunoglobulin (IVIg) in preference to PE. Following an acute attack, 55.8% (29/52) of respondents typically continue corticosteroids for ≥ 3 months; though less commonly when treating children. After an index event, 60% (31/51) usually start steroid-sparing maintenance therapy (MT); after ≥ 2 attacks 92.3% (48/52) would start MT. Repeat MOG antibody status is used by 52.9% (27/51) to help decide on MT initiation. Commonly used first line MTs in adults are azathioprine (30.8%, 16/52), mycophenolate mofetil (25.0%, 13/52) and rituximab (17.3%, 9/52). In children, IVIg is the preferred first line MT (54.5%; 6/11). Treatment response is monitored by MRI (53.8%; 28/52), optical coherence tomography (23.1%; 12/52) and MOG antibody titres (36.5%; 19/52). Regardless of monitoring results, 25.0% (13/52) would not stop MT.

Conclusion

Current treatment of MOGAD is highly variable, indicating a need for consensus-based treatment guidelines, while awaiting definitive clinical trials.

中文翻译：

MOG抗体相关疾病的治疗：一项国际调查的结果。

介绍

尽管在世界范围内越来越多地诊断出单相和复发性的髓磷脂少突胶质细胞糖蛋白抗体相关疾病（MOGAD），但关于治疗的共识尚未建立。

目的

调查临床医生治疗MOGAD的当前全球临床实践。

方法

全世界具有治疗MOGAD专门知识的神经科医生参加了一项在线调查（2019年2月至4月）。

结果

来自22个国家的86位受邀专家中有52位回应者（回应率为60.5％），包括成人（78.8％，41/52）和儿童（21.2％，11/52）专家。所有人都用大剂量皮质类固醇治疗急性发作。如果恢复不完全，则在血浆置换（PE）旁进行71.2％（37/52）。小儿神经科医生中有45.5％（5/11）的患者优先使用IV免疫球蛋白（IVIg）代替PE。急性发作后，有55.8％（29/52）的受访者通常继续使用皮质类固醇激素≥3个月；虽然在治疗儿童时较少见。发生指数事件后，通常有60％（31/51）的患者开始进行类固醇激素维持治疗（MT）；≥2次攻击后92.3％（48/52）将启动MT。52.9％（27/51）使用重复MOG抗体状态来帮助决定是否开始MT。成人常用的一线MT是硫唑嘌呤（30.8％，16/52），霉酚酸酯（25.0％，13/52）和利妥昔单抗（17.3％，9/52）。在儿童中，IVIg是首选的第一线MT（54.5％； 6/11）。通过MRI（53.8％; 28/52），光学相干断层扫描（23.1％; 12/52）和MOG抗体滴度（36.5％; 19/52）监测治疗反应。无论监测结果如何，25.0％（13/52）都不会阻止MT。