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Investigating the association between neoplasms and MOG antibody-associated disease

INTRODUCTION: The association of myelin oligodendrocyte glycoprotein (MOG) antibody associated disease (MOGAD) and tumors has seldom been reported. We aim to investigate the occurrence of tumors in a cohort of patients with MOGAD and to describe their clinical features, in addition to previously rep...

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Autores principales: Trentinaglia, Milena, Dinoto, Alessandro, Carta, Sara, Chiodega, Vanessa, Ferrari, Sergio, Andreone, Vincenzo, Maniscalco, Giorgia Teresa, Mariotto, Sara
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10289293/
https://www.ncbi.nlm.nih.gov/pubmed/37360349
http://dx.doi.org/10.3389/fneur.2023.1193211
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author Trentinaglia, Milena
Dinoto, Alessandro
Carta, Sara
Chiodega, Vanessa
Ferrari, Sergio
Andreone, Vincenzo
Maniscalco, Giorgia Teresa
Mariotto, Sara
author_facet Trentinaglia, Milena
Dinoto, Alessandro
Carta, Sara
Chiodega, Vanessa
Ferrari, Sergio
Andreone, Vincenzo
Maniscalco, Giorgia Teresa
Mariotto, Sara
author_sort Trentinaglia, Milena
collection PubMed
description INTRODUCTION: The association of myelin oligodendrocyte glycoprotein (MOG) antibody associated disease (MOGAD) and tumors has seldom been reported. We aim to investigate the occurrence of tumors in a cohort of patients with MOGAD and to describe their clinical features, in addition to previously reported cases. METHODS: We retrospectively identified patients with MOGAD (i.e., compatible clinical phenotype and positive MOG antibodies analysed with a live cell-based assay) from 1/1/2015 to 1/1/2023 who had a neoplasm diagnosed within 2  years from MOGAD onset. Furthermore, we performed systematic review of literature to identify previously reported cases. Clinical, paraclinical and oncological findings were collected and reported as median (range) or number (percentage). RESULTS: Two of 150 MOGAD patients (1%) had a concomitant neoplasm in our cohort. Fifteen additional cases were retrieved from literature. Median age was 39 (16–73) years-old, 12 patients were female. ADEM (n = 4;23.5%), encephalomyelitis (n = 3;17.6%), and monolateral optic neuritis (n = 2;11.8%) were the most frequent phenotypes. Median number of treatments was 1 (range 1–4), improvement was reported in 14/17 cases (82.4%). Oncological accompaniments were teratoma (n = 4), CNS (n = 3), melanoma (n = 2), lung (n = 2), hematological (n = 2), ovary (n = 1), breast (n = 1), gastrointestinal (n = 1), and thymic (n = 1) neoplasms. Median time from tumor diagnosis to MOGAD onset was 0 (range − 60 to 20) months. MOG expression in neoplastic tissue was reported in 2/4 patients. Median PNS-CARE score was 3 (range 0–7): 11 patients were classified as “non-PNS,” 5 as “possible PNS,” and 1 as “probable PNS.” DISCUSSION: Our study confirms that MOG is a low-risk antibody for paraneoplastic neurological syndromes and that the clinical presentation and oncological accompaniments are extremely variable. Most of these patients were classified as non-PNS, whereas only a minority was diagnosed with possible/probable PNS, frequently in association with ovarian teratoma. These findings support the notion that MOGAD is not a paraneoplastic disease.
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spelling pubmed-102892932023-06-24 Investigating the association between neoplasms and MOG antibody-associated disease Trentinaglia, Milena Dinoto, Alessandro Carta, Sara Chiodega, Vanessa Ferrari, Sergio Andreone, Vincenzo Maniscalco, Giorgia Teresa Mariotto, Sara Front Neurol Neurology INTRODUCTION: The association of myelin oligodendrocyte glycoprotein (MOG) antibody associated disease (MOGAD) and tumors has seldom been reported. We aim to investigate the occurrence of tumors in a cohort of patients with MOGAD and to describe their clinical features, in addition to previously reported cases. METHODS: We retrospectively identified patients with MOGAD (i.e., compatible clinical phenotype and positive MOG antibodies analysed with a live cell-based assay) from 1/1/2015 to 1/1/2023 who had a neoplasm diagnosed within 2  years from MOGAD onset. Furthermore, we performed systematic review of literature to identify previously reported cases. Clinical, paraclinical and oncological findings were collected and reported as median (range) or number (percentage). RESULTS: Two of 150 MOGAD patients (1%) had a concomitant neoplasm in our cohort. Fifteen additional cases were retrieved from literature. Median age was 39 (16–73) years-old, 12 patients were female. ADEM (n = 4;23.5%), encephalomyelitis (n = 3;17.6%), and monolateral optic neuritis (n = 2;11.8%) were the most frequent phenotypes. Median number of treatments was 1 (range 1–4), improvement was reported in 14/17 cases (82.4%). Oncological accompaniments were teratoma (n = 4), CNS (n = 3), melanoma (n = 2), lung (n = 2), hematological (n = 2), ovary (n = 1), breast (n = 1), gastrointestinal (n = 1), and thymic (n = 1) neoplasms. Median time from tumor diagnosis to MOGAD onset was 0 (range − 60 to 20) months. MOG expression in neoplastic tissue was reported in 2/4 patients. Median PNS-CARE score was 3 (range 0–7): 11 patients were classified as “non-PNS,” 5 as “possible PNS,” and 1 as “probable PNS.” DISCUSSION: Our study confirms that MOG is a low-risk antibody for paraneoplastic neurological syndromes and that the clinical presentation and oncological accompaniments are extremely variable. Most of these patients were classified as non-PNS, whereas only a minority was diagnosed with possible/probable PNS, frequently in association with ovarian teratoma. These findings support the notion that MOGAD is not a paraneoplastic disease. Frontiers Media S.A. 2023-06-09 /pmc/articles/PMC10289293/ /pubmed/37360349 http://dx.doi.org/10.3389/fneur.2023.1193211 Text en Copyright © 2023 Trentinaglia, Dinoto, Carta, Chiodega, Ferrari, Andreone, Maniscalco and Mariotto. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Neurology
Trentinaglia, Milena
Dinoto, Alessandro
Carta, Sara
Chiodega, Vanessa
Ferrari, Sergio
Andreone, Vincenzo
Maniscalco, Giorgia Teresa
Mariotto, Sara
Investigating the association between neoplasms and MOG antibody-associated disease
title Investigating the association between neoplasms and MOG antibody-associated disease
title_full Investigating the association between neoplasms and MOG antibody-associated disease
title_fullStr Investigating the association between neoplasms and MOG antibody-associated disease
title_full_unstemmed Investigating the association between neoplasms and MOG antibody-associated disease
title_short Investigating the association between neoplasms and MOG antibody-associated disease
title_sort investigating the association between neoplasms and mog antibody-associated disease
topic Neurology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10289293/
https://www.ncbi.nlm.nih.gov/pubmed/37360349
http://dx.doi.org/10.3389/fneur.2023.1193211
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