Araştırma Makalesi
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Clinical Characteristics and Treatment Outcomes of MOGAD: A Detailed Analysis from a Single-Center Four-Year Retrospective Cohort

Yıl 2024, , 164 - 171, 30.06.2024
https://doi.org/10.31832/smj.1438799

Öz

Introduction: This study aims to delineate the prevalence, clinical characteristics, diagnostic findings, and treatment outcomes of Myelin Oligodendrocyte Glycoprotein Associated Disease (MOGAD) in a cohort over four years, providing a basis for improved diagnostic criteria and therapeutic strategies. Materials and Methods: In a retrospective cohort study at a tertiary care center, we analyzed medical records of 90 patients presenting with CNS demyelinating symptoms, focusing on those diagnosed with MOGAD based on the International MOGAD Panel criteria. Data on clinical presentation, serum Anti-MOG antibody testing, MRI, VEP scans, CSF analysis, and treatment outcomes were evaluated. Results: Among the cohort of 90 patients, 7 patients were identified with positive Anti- MOG antibodies, indicating a prevalence of 7.8%. Clinical manifestations varied widely, including optic neuritis, myelitis, and cerebral cortical encephalitis. Diagnostic findings highlighted the absence of oligoclonal bands in CSF analysis and diverse MRI lesions. Most patients responded well to immunosuppressive treatments, though relapses occurred in two cases. The study underscores the heterogeneity of MOGAD presentations and the importance of personalized treatment approaches. Conclusion: Our findings contribute to the growing understanding of MOGAD, emphasizing its distinct clinical and diagnostic features compared to other CNS demyelinating disorders. The study advocates for the integration of MOG antibody testing in clinical practice to enhance diagnostic accuracy and patient outcomes. Future research should aim at longitudinal and multicentric studies to validate our findings and further refine MOGAD management strategies.

Kaynakça

  • 1. Reindl M, Schanda K, Woodhall M, Tea F, Ramanathan S, Sagen J, et al. International multicenter examination of MOG antibody assays. Neurol Neuroimmunol Neuroinflamm. 2020;7(2):e674. doi: 10.1212/NXI.0000000000000674. Erratum in: Neurol Neuroimmunol Neuroinflamm. 2020 Mar 20;7(3): PMID: 32024795; PMCID: PMC7051197.
  • 2. Cobo-Calvo A, Ruiz A, Maillart E, Audoin B, Zephir H, Bourre B, et al. Clinical spectrum and prognostic value of CNS MOG autoimmunity in adults: The MOGADOR study. Neurology. 2018;90(21):e1858-1869. doi: 10.1212/WNL.0000000000005560.
  • 3. Jarius S, Paul F, Aktas O, Asgari N, Dale RC, de Seze J, et al. MOG encephalomyelitis: international recommendations on diagnosis and antibody testing. J Neuroinflammation. 2018;15(1):134.
  • 4. Marignier R, Hacohen Y, Cobo-Calvo A, Pröbstel AK, Aktas O, Alexopoulos H, et al. Myelin-oligodendrocyte glycoprotein antibody-associated disease. Lancet Neurol. 2021;20(9):762-72. doi: 10.1016/S1474-4422(21)00218-0. Erratum in: Lancet Neurol. 2021;20(10):e6. Erratum in: Lancet Neurol. 2022 Jan;21(1):e1.
  • 5. Hacohen Y, Absoud M, Deiva K, Hemingway C, Nytrova P, Woodhall M, et al. Myelin oligodendrocyte glycoprotein antibodies are associated with a non-MS course in children. Neurol Neuroimmunol Neuroinflamm. 2015;2(2):e81. doi: 10.1212/NXI.0000000000000081.
  • 6. Solomon AJ, Arrambide G, Brownlee WJ, Brownlee WJ, Flanagan EP, Amato MP, et al. Differential diagnosis of suspected multiple sclerosis: an updated consensus approach. Lancet Neurol. 2023;22(8):750-68. doi: 10.1016/S1474-4422(23)00148-5.
  • 7. Waters PJ, McKeon A, Leite MI, Rajasekharan S, Lennon VA, Villalobos A, et al. Serologic diagnosis of NMO: a multicenter comparison of aquaporin-4-IgG assays. Neurology. 2012;78(9):665-71; discussion 669. doi: 10.1212/WNL.0b013e318248dec1.
  • 8. López-Chiriboga AS, Majed M, Fryer J, Dubey D, McKeon A, Flanagan EP, et al. Association of MOG-IgG Serostatus With Relapse After Acute Disseminated Encephalomyelitis and Proposed Diagnostic Criteria for MOG-IgG-Associated Disorders. JAMA Neurol. 2018;75(11):1355-63. doi: 10.1001/jamaneurol.2018.1814.
  • 9. Jurynczyk M, Messina S, Woodhall MR, Raza N, Everett R, Roca-Fernandez A, et al. Clinical presentation and prognosis in MOG-antibody disease: a UK study. Brain. 2017;140(12):3128-38. doi: 10.1093/brain/awx276. Erratum in: Brain. 2018 Apr 1;141(4):e31.
  • 10. Banwell B, Bennett JL, Marignier R, Kim HJ, Brilot F, Flanagan EP, et al. Diagnosis of myelin oligodendrocyte glycoprotein antibody-associated disease: International MOGAD Panel proposed criteria. Lancet Neurol. 2023;22(3):268-82. doi: 10.1016/S1474-4422(22)00431-8. Epub 2023 Jan 24. PMID: 36706773.
  • 11. Shahriari M, Sotirchos ES, Newsome SD, Yousem DM. MOGAD: How It Differs From and Resembles Other Neuroinflammatory Disorders. AJR Am J Roentgenol. 2021;216(4):1031-9. doi: 10.2214/AJR.20.24061. 12. Sechi E, Cacciaguerra L, Chen JJ, Mariotto S, Fadda G, Dinoto A, et al. Myelin Oligodendrocyte Glycoprotein Antibody-Associated Disease (MOGAD): A Review of Clinical and MRI Features, Diagnosis, and Management. Front Neurol. 2022;13:885218. doi: 10.3389/fneur.2022.885218.
  • 13. Hegen H, Reindl M. Recent developments in MOG-IgG associated neurological disorders. Ther Adv Neurol Disord. 2020;13:1756286420945135. doi: 10.1177/1756286420945135.
  • 14. Healy S, ElHadd KT, Gibbons E, Whittam D, Griffiths MJ, Jacob A, et al. Treatment of myelin oligodendrocyte glycoprotein immunoglobulin G–associated disease. Clin Exp Neuroimmunol. 2021;12:22–41.
  • 15. Waters PJ, Komorowski L, Woodhall M, Lederer S, Majed M, Fryer J, et al. A multicenter comparison of MOG-IgG cell-based assays. Neurology. 2019;92(11):e1250-5. doi: 10.1212/WNL.0000000000007096.
  • 16. Whittam DH, Karthikeayan V, Gibbons E, Kneen R, Chandratre S, Ciccarelli O, et al. Treatment of MOG antibody associated disorders: results of an international survey. J Neurol. 2020;267(12):3565-77. doi: 10.1007/s00415-020-10026-y.
  • 17. Chen JJ, Flanagan EP, Bhatti MT, Jitprapaikulsan J, Dubey D, Lopez Chiriboga ASS, et al. Steroid-sparing maintenance immunotherapy for MOG-IgG associated disorder. Neurology. 2020;95(2):e111-20. doi: 10.1212/WNL.0000000000009758.
  • 18. Lana-Peixoto MA, Talim N. Neuromyelitis optica spectrum disorder and anti-MOG syndromes. Biomedicines. 2019;7(2):42.
  • 19. Ambrosius W, Michalak S, Kozubski W, Kalinowska A. Myelin oligodendrocyte glycoprotein antibody-associated disease: current insights into the disease pathophysiology, diagnosis, and management. Int J Mol Sci. 2020;22(1):100.

Miyelin Oligodendrosit Glikoprotein İlişkili Hastalığı (MOGAD): Dört Yıllık Retrospektif Kohort Çalışmasında Prevalans, Klinik Belirtiler ve Tedavi Yanıtları

Yıl 2024, , 164 - 171, 30.06.2024
https://doi.org/10.31832/smj.1438799

Öz

Amaç: Bu çalışma, dört yıl boyunca bir kohorttaki Miyelin Oligodendrosit Glikoprotein İlişkili Hastalığın (MOGAD) prevalansını, klinik özelliklerini, tanı bulgularını ve tedavi sonuçlarını açıklamayı ve gelişmiş tanı kriterleri ve tedavi stratejileri için bir temel sağlamayı amaçlamaktadır.
Yöntem ve Gereçler: Üçüncü basamak bir merkezde yapılan retrospektif bir kohort çalışmasında, Uluslararası MOGAD Paneli kriterlerine göre MOGAD tanısı konanlara odaklanarak, Merkezi sinir sistemi (MSS) demiyelinizan semptomlarıyla başvuran 90 hastanın tıbbi kayıtlarını analiz ettik. Klinik tablo, serum Anti-MOG antikor testi, MR, VEP taramaları, BOS analizi ve tedavi sonuçlarına ilişkin veriler değerlendirildi.
Bulgular: 90 hastadan oluşan kohort arasında, 7 hastada pozitif Anti-MOG antikorları belirlendi; bu da %7,8'lik bir prevalansa işaret etmekte idi. Klinik bulgular, optik nörit, miyelit ve serebral kortikal ensefalit dahil olmak üzere çok çeşitli idi. Tanısal bulgular, çeşitli MR lezyonları göstermekle birlikte BOS analizinde oligoklonal bantların bulunmadığını vurguladı. Çoğu hasta immünosupresif tedavilere iyi yanıt verdi, ancak iki vakada rekürrens meydana geldi. Çalışma, MOGAD sunumlarının heterojenliğini ve kişiselleştirilmiş tedavi yaklaşımlarının önemini vurgulamakta idi.
Sonuç: Bulgularımız, MOGAD'ın diğer MSS demiyelinizan hastalıklarla karşılaştırıldığında farklı klinik ve tanısal özelliklerini vurgulayarak, MOGAD'ın giderek daha iyi anlaşılmasına katkıda bulunmaktadır. Çalışma, teşhis doğruluğunu ve hasta sonuçlarını iyileştirmek için MOG antikor testinin klinik uygulamaya entegrasyonunu savunmaktadır. Gelecekteki araştırmalar, bulgularımızı doğrulamak ve MOGAD yönetim stratejilerini daha da geliştirmek için boylamsal ve çok merkezli çalışmaları amaçlamalıdır.

Etik Beyan

Considering the retrospective design of the study, ethics committee consent was waived However, strict confidentiality andanonymity of patient data were maintained throughout the study, in accordance withethical guidelines and the Declaration of Helsinki. Informed consent was obtained from all individual participants included in the study.

Destekleyen Kurum

Yok

Kaynakça

  • 1. Reindl M, Schanda K, Woodhall M, Tea F, Ramanathan S, Sagen J, et al. International multicenter examination of MOG antibody assays. Neurol Neuroimmunol Neuroinflamm. 2020;7(2):e674. doi: 10.1212/NXI.0000000000000674. Erratum in: Neurol Neuroimmunol Neuroinflamm. 2020 Mar 20;7(3): PMID: 32024795; PMCID: PMC7051197.
  • 2. Cobo-Calvo A, Ruiz A, Maillart E, Audoin B, Zephir H, Bourre B, et al. Clinical spectrum and prognostic value of CNS MOG autoimmunity in adults: The MOGADOR study. Neurology. 2018;90(21):e1858-1869. doi: 10.1212/WNL.0000000000005560.
  • 3. Jarius S, Paul F, Aktas O, Asgari N, Dale RC, de Seze J, et al. MOG encephalomyelitis: international recommendations on diagnosis and antibody testing. J Neuroinflammation. 2018;15(1):134.
  • 4. Marignier R, Hacohen Y, Cobo-Calvo A, Pröbstel AK, Aktas O, Alexopoulos H, et al. Myelin-oligodendrocyte glycoprotein antibody-associated disease. Lancet Neurol. 2021;20(9):762-72. doi: 10.1016/S1474-4422(21)00218-0. Erratum in: Lancet Neurol. 2021;20(10):e6. Erratum in: Lancet Neurol. 2022 Jan;21(1):e1.
  • 5. Hacohen Y, Absoud M, Deiva K, Hemingway C, Nytrova P, Woodhall M, et al. Myelin oligodendrocyte glycoprotein antibodies are associated with a non-MS course in children. Neurol Neuroimmunol Neuroinflamm. 2015;2(2):e81. doi: 10.1212/NXI.0000000000000081.
  • 6. Solomon AJ, Arrambide G, Brownlee WJ, Brownlee WJ, Flanagan EP, Amato MP, et al. Differential diagnosis of suspected multiple sclerosis: an updated consensus approach. Lancet Neurol. 2023;22(8):750-68. doi: 10.1016/S1474-4422(23)00148-5.
  • 7. Waters PJ, McKeon A, Leite MI, Rajasekharan S, Lennon VA, Villalobos A, et al. Serologic diagnosis of NMO: a multicenter comparison of aquaporin-4-IgG assays. Neurology. 2012;78(9):665-71; discussion 669. doi: 10.1212/WNL.0b013e318248dec1.
  • 8. López-Chiriboga AS, Majed M, Fryer J, Dubey D, McKeon A, Flanagan EP, et al. Association of MOG-IgG Serostatus With Relapse After Acute Disseminated Encephalomyelitis and Proposed Diagnostic Criteria for MOG-IgG-Associated Disorders. JAMA Neurol. 2018;75(11):1355-63. doi: 10.1001/jamaneurol.2018.1814.
  • 9. Jurynczyk M, Messina S, Woodhall MR, Raza N, Everett R, Roca-Fernandez A, et al. Clinical presentation and prognosis in MOG-antibody disease: a UK study. Brain. 2017;140(12):3128-38. doi: 10.1093/brain/awx276. Erratum in: Brain. 2018 Apr 1;141(4):e31.
  • 10. Banwell B, Bennett JL, Marignier R, Kim HJ, Brilot F, Flanagan EP, et al. Diagnosis of myelin oligodendrocyte glycoprotein antibody-associated disease: International MOGAD Panel proposed criteria. Lancet Neurol. 2023;22(3):268-82. doi: 10.1016/S1474-4422(22)00431-8. Epub 2023 Jan 24. PMID: 36706773.
  • 11. Shahriari M, Sotirchos ES, Newsome SD, Yousem DM. MOGAD: How It Differs From and Resembles Other Neuroinflammatory Disorders. AJR Am J Roentgenol. 2021;216(4):1031-9. doi: 10.2214/AJR.20.24061. 12. Sechi E, Cacciaguerra L, Chen JJ, Mariotto S, Fadda G, Dinoto A, et al. Myelin Oligodendrocyte Glycoprotein Antibody-Associated Disease (MOGAD): A Review of Clinical and MRI Features, Diagnosis, and Management. Front Neurol. 2022;13:885218. doi: 10.3389/fneur.2022.885218.
  • 13. Hegen H, Reindl M. Recent developments in MOG-IgG associated neurological disorders. Ther Adv Neurol Disord. 2020;13:1756286420945135. doi: 10.1177/1756286420945135.
  • 14. Healy S, ElHadd KT, Gibbons E, Whittam D, Griffiths MJ, Jacob A, et al. Treatment of myelin oligodendrocyte glycoprotein immunoglobulin G–associated disease. Clin Exp Neuroimmunol. 2021;12:22–41.
  • 15. Waters PJ, Komorowski L, Woodhall M, Lederer S, Majed M, Fryer J, et al. A multicenter comparison of MOG-IgG cell-based assays. Neurology. 2019;92(11):e1250-5. doi: 10.1212/WNL.0000000000007096.
  • 16. Whittam DH, Karthikeayan V, Gibbons E, Kneen R, Chandratre S, Ciccarelli O, et al. Treatment of MOG antibody associated disorders: results of an international survey. J Neurol. 2020;267(12):3565-77. doi: 10.1007/s00415-020-10026-y.
  • 17. Chen JJ, Flanagan EP, Bhatti MT, Jitprapaikulsan J, Dubey D, Lopez Chiriboga ASS, et al. Steroid-sparing maintenance immunotherapy for MOG-IgG associated disorder. Neurology. 2020;95(2):e111-20. doi: 10.1212/WNL.0000000000009758.
  • 18. Lana-Peixoto MA, Talim N. Neuromyelitis optica spectrum disorder and anti-MOG syndromes. Biomedicines. 2019;7(2):42.
  • 19. Ambrosius W, Michalak S, Kozubski W, Kalinowska A. Myelin oligodendrocyte glycoprotein antibody-associated disease: current insights into the disease pathophysiology, diagnosis, and management. Int J Mol Sci. 2020;22(1):100.
Toplam 18 adet kaynakça vardır.

Ayrıntılar

Birincil Dil İngilizce
Konular Beyin ve Sinir Cerrahisi (Nöroşirurji)
Bölüm Araştırma Makalesi
Yazarlar

Alihan Abdullah Akbaş Bu kişi benim 0009-0005-6098-5096

Ömer Elçi Bu kişi benim 0009-0006-0657-0057

Beyzanur Bozkurt Bu kişi benim 0000-0001-6896-3821

Vasfiye Sezer 0000-0002-2982-8210

Abdulkadir Tunç 0000-0002-9747-5285

Erken Görünüm Tarihi 7 Haziran 2024
Yayımlanma Tarihi 30 Haziran 2024
Gönderilme Tarihi 23 Şubat 2024
Kabul Tarihi 8 Mayıs 2024
Yayımlandığı Sayı Yıl 2024

Kaynak Göster

AMA Akbaş AA, Elçi Ö, Bozkurt B, Sezer V, Tunç A. Clinical Characteristics and Treatment Outcomes of MOGAD: A Detailed Analysis from a Single-Center Four-Year Retrospective Cohort. Sakarya Tıp Dergisi. Haziran 2024;14(2):164-171. doi:10.31832/smj.1438799

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