Cargando…

Case report: Coexistence of triple-seronegative myasthenia gravis and pathology-proven cryptogenic organizing pneumonia

OBJECTIVE: Emerging evidence shows that patients with myasthenia gravis (MG) were at a higher risk for the co-occurrence of other autoimmune diseases, which reflects phenotypic heterogeneity in MG. The coexistence of MG and cryptogenic organizing pneumonia (COP) has rarely been reported. The present...

Descripción completa

Detalles Bibliográficos
Autores principales: Huang, Shi-Qi, Wang, Bin, Gao, Lin, Wang, Meng, Zhao, Hong-Dong, Shi, Jian-Quan
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10690590/
https://www.ncbi.nlm.nih.gov/pubmed/38046596
http://dx.doi.org/10.3389/fneur.2023.1295374
_version_ 1785152553349021696
author Huang, Shi-Qi
Wang, Bin
Gao, Lin
Wang, Meng
Zhao, Hong-Dong
Shi, Jian-Quan
author_facet Huang, Shi-Qi
Wang, Bin
Gao, Lin
Wang, Meng
Zhao, Hong-Dong
Shi, Jian-Quan
author_sort Huang, Shi-Qi
collection PubMed
description OBJECTIVE: Emerging evidence shows that patients with myasthenia gravis (MG) were at a higher risk for the co-occurrence of other autoimmune diseases, which reflects phenotypic heterogeneity in MG. The coexistence of MG and cryptogenic organizing pneumonia (COP) has rarely been reported. The present case is to report the coexistence of triple-seronegative MG and pathology-proven COP in a patient. METHODS: The clinical data of the patient were derived from medical records of Nanjing First Hospital, Nanjing Medical University, China. Written informed consent was obtained from the patient. RESULTS: We presented a 56-year-old man with acute respiratory syndrome, who was diagnosed with COP based on the intra-alveolar fibroinflammatory buds (Masson's bodies) in the pathology of bronchoscopy biopsy. Oral prednisone induced dramatic symptomatic improvement and complete resolution of previous lung lesions. After a stable course of no respiratory symptom for 2 months, he was referred to the neurology department with complaints of fluctuating generalized muscle weakness. He was diagnosed with triple-seronegative MG based on fluctuating weakness, neostigmine test-positivity and RNS-positivity. After three-month treatment with pyridostigmine in combination with tacrolimus, the symptoms gradually improved and he achieved minimal symptom expression. CONCLUSIONS: This case highlights the rare coexistence of triple-seronegative MG and pathology-proven COP. However, a causal association between COP and MG cannot be explicitly ascertained. In future, more data are needed to clarify the relationship, taking into account the limited number of cases reported with this coexistence of the diseases.
format Online
Article
Text
id pubmed-10690590
institution National Center for Biotechnology Information
language English
publishDate 2023
publisher Frontiers Media S.A.
record_format MEDLINE/PubMed
spelling pubmed-106905902023-12-02 Case report: Coexistence of triple-seronegative myasthenia gravis and pathology-proven cryptogenic organizing pneumonia Huang, Shi-Qi Wang, Bin Gao, Lin Wang, Meng Zhao, Hong-Dong Shi, Jian-Quan Front Neurol Neurology OBJECTIVE: Emerging evidence shows that patients with myasthenia gravis (MG) were at a higher risk for the co-occurrence of other autoimmune diseases, which reflects phenotypic heterogeneity in MG. The coexistence of MG and cryptogenic organizing pneumonia (COP) has rarely been reported. The present case is to report the coexistence of triple-seronegative MG and pathology-proven COP in a patient. METHODS: The clinical data of the patient were derived from medical records of Nanjing First Hospital, Nanjing Medical University, China. Written informed consent was obtained from the patient. RESULTS: We presented a 56-year-old man with acute respiratory syndrome, who was diagnosed with COP based on the intra-alveolar fibroinflammatory buds (Masson's bodies) in the pathology of bronchoscopy biopsy. Oral prednisone induced dramatic symptomatic improvement and complete resolution of previous lung lesions. After a stable course of no respiratory symptom for 2 months, he was referred to the neurology department with complaints of fluctuating generalized muscle weakness. He was diagnosed with triple-seronegative MG based on fluctuating weakness, neostigmine test-positivity and RNS-positivity. After three-month treatment with pyridostigmine in combination with tacrolimus, the symptoms gradually improved and he achieved minimal symptom expression. CONCLUSIONS: This case highlights the rare coexistence of triple-seronegative MG and pathology-proven COP. However, a causal association between COP and MG cannot be explicitly ascertained. In future, more data are needed to clarify the relationship, taking into account the limited number of cases reported with this coexistence of the diseases. Frontiers Media S.A. 2023-11-16 /pmc/articles/PMC10690590/ /pubmed/38046596 http://dx.doi.org/10.3389/fneur.2023.1295374 Text en Copyright © 2023 Huang, Wang, Gao, Wang, Zhao and Shi. 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
Huang, Shi-Qi
Wang, Bin
Gao, Lin
Wang, Meng
Zhao, Hong-Dong
Shi, Jian-Quan
Case report: Coexistence of triple-seronegative myasthenia gravis and pathology-proven cryptogenic organizing pneumonia
title Case report: Coexistence of triple-seronegative myasthenia gravis and pathology-proven cryptogenic organizing pneumonia
title_full Case report: Coexistence of triple-seronegative myasthenia gravis and pathology-proven cryptogenic organizing pneumonia
title_fullStr Case report: Coexistence of triple-seronegative myasthenia gravis and pathology-proven cryptogenic organizing pneumonia
title_full_unstemmed Case report: Coexistence of triple-seronegative myasthenia gravis and pathology-proven cryptogenic organizing pneumonia
title_short Case report: Coexistence of triple-seronegative myasthenia gravis and pathology-proven cryptogenic organizing pneumonia
title_sort case report: coexistence of triple-seronegative myasthenia gravis and pathology-proven cryptogenic organizing pneumonia
topic Neurology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10690590/
https://www.ncbi.nlm.nih.gov/pubmed/38046596
http://dx.doi.org/10.3389/fneur.2023.1295374
work_keys_str_mv AT huangshiqi casereportcoexistenceoftripleseronegativemyastheniagravisandpathologyprovencryptogenicorganizingpneumonia
AT wangbin casereportcoexistenceoftripleseronegativemyastheniagravisandpathologyprovencryptogenicorganizingpneumonia
AT gaolin casereportcoexistenceoftripleseronegativemyastheniagravisandpathologyprovencryptogenicorganizingpneumonia
AT wangmeng casereportcoexistenceoftripleseronegativemyastheniagravisandpathologyprovencryptogenicorganizingpneumonia
AT zhaohongdong casereportcoexistenceoftripleseronegativemyastheniagravisandpathologyprovencryptogenicorganizingpneumonia
AT shijianquan casereportcoexistenceoftripleseronegativemyastheniagravisandpathologyprovencryptogenicorganizingpneumonia