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Idiopathic Bilateral Extraocular Myositis in a Subject With Poorly Controlled Type 2 Diabetes Mellitus: Case Report

Background: Extraocular myositis is characterized by acute onset of orbital pain, extraocular muscle swelling, absence of thyroid disease, and effectiveness of steroid therapy. While oculomotor nerve paralysis is often observed in subjects with diabetes mellitus, extraocular muscle paralysis is very...

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Autores principales: Tatsumi, Fuminori, Fushimi, Yoshiro, Sanada, Junpei, Shimoda, Masashi, Kohara, Kenji, Kimura, Tomohiko, Obata, Atsushi, Nakanishi, Shuhei, Mune, Tomoatsu, Kaku, Kohei, Kaneto, Hideaki
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8514759/
https://www.ncbi.nlm.nih.gov/pubmed/34660622
http://dx.doi.org/10.3389/fmed.2021.700307
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author Tatsumi, Fuminori
Fushimi, Yoshiro
Sanada, Junpei
Shimoda, Masashi
Kohara, Kenji
Kimura, Tomohiko
Obata, Atsushi
Nakanishi, Shuhei
Mune, Tomoatsu
Kaku, Kohei
Kaneto, Hideaki
author_facet Tatsumi, Fuminori
Fushimi, Yoshiro
Sanada, Junpei
Shimoda, Masashi
Kohara, Kenji
Kimura, Tomohiko
Obata, Atsushi
Nakanishi, Shuhei
Mune, Tomoatsu
Kaku, Kohei
Kaneto, Hideaki
author_sort Tatsumi, Fuminori
collection PubMed
description Background: Extraocular myositis is characterized by acute onset of orbital pain, extraocular muscle swelling, absence of thyroid disease, and effectiveness of steroid therapy. While oculomotor nerve paralysis is often observed in subjects with diabetes mellitus, extraocular muscle paralysis is very rare among various diabetic mononeuropathies. In addition, while most diabetic mononeuropathies are observed as sporadic and/or unilateral neuropathy, bilateral mononeuropathy is also very rare. Case presentation: A 58-year-old male visited our institution due to diplopia. He was diagnosed as type 2 diabetes mellitus about 10 years before and treated with oral diabetes agents. To examine the cause of his symptom, he was hospitalized in our institution. Slight ptosis was observed, and failure of adduction was observed in the right eye. Glycemic control was poor; HbA1c was 9.3%. Liver, renal, and thyroid function were within normal range. Immunoglobulin (Ig) G was slightly high, but IgA, IgM, and IgG4 were within normal range. Various antibodies were all negative. Angiotensin-converting enzyme level was within normal range. There were no abnormalities in brain magnetic resonance imaging (MRI). After admission, to alleviate glucose toxicity, we started insulin therapy. On day 17, adduction failure of the left eye was observed in addition to the right eye. Vertical movement was also impaired in both eyes. Slight ptosis was observed in both eyes, and the right eye was completely close. In orbital MRI, some high signal was detected in both extraocular muscles. We performed steroid pulse therapy twice. About 4 months later, ptosis and vertical and horizontal movements in both eyes were almost completely recovered. Finally, we diagnosed him as idiopathic bilateral extraocular myositis. Conclusions: We should bear in mind the possibility of idiopathic bilateral extraocular myositis especially in subjects with poor glycemic control, although its incident rate is extremely rare.
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spelling pubmed-85147592021-10-15 Idiopathic Bilateral Extraocular Myositis in a Subject With Poorly Controlled Type 2 Diabetes Mellitus: Case Report Tatsumi, Fuminori Fushimi, Yoshiro Sanada, Junpei Shimoda, Masashi Kohara, Kenji Kimura, Tomohiko Obata, Atsushi Nakanishi, Shuhei Mune, Tomoatsu Kaku, Kohei Kaneto, Hideaki Front Med (Lausanne) Medicine Background: Extraocular myositis is characterized by acute onset of orbital pain, extraocular muscle swelling, absence of thyroid disease, and effectiveness of steroid therapy. While oculomotor nerve paralysis is often observed in subjects with diabetes mellitus, extraocular muscle paralysis is very rare among various diabetic mononeuropathies. In addition, while most diabetic mononeuropathies are observed as sporadic and/or unilateral neuropathy, bilateral mononeuropathy is also very rare. Case presentation: A 58-year-old male visited our institution due to diplopia. He was diagnosed as type 2 diabetes mellitus about 10 years before and treated with oral diabetes agents. To examine the cause of his symptom, he was hospitalized in our institution. Slight ptosis was observed, and failure of adduction was observed in the right eye. Glycemic control was poor; HbA1c was 9.3%. Liver, renal, and thyroid function were within normal range. Immunoglobulin (Ig) G was slightly high, but IgA, IgM, and IgG4 were within normal range. Various antibodies were all negative. Angiotensin-converting enzyme level was within normal range. There were no abnormalities in brain magnetic resonance imaging (MRI). After admission, to alleviate glucose toxicity, we started insulin therapy. On day 17, adduction failure of the left eye was observed in addition to the right eye. Vertical movement was also impaired in both eyes. Slight ptosis was observed in both eyes, and the right eye was completely close. In orbital MRI, some high signal was detected in both extraocular muscles. We performed steroid pulse therapy twice. About 4 months later, ptosis and vertical and horizontal movements in both eyes were almost completely recovered. Finally, we diagnosed him as idiopathic bilateral extraocular myositis. Conclusions: We should bear in mind the possibility of idiopathic bilateral extraocular myositis especially in subjects with poor glycemic control, although its incident rate is extremely rare. Frontiers Media S.A. 2021-09-30 /pmc/articles/PMC8514759/ /pubmed/34660622 http://dx.doi.org/10.3389/fmed.2021.700307 Text en Copyright © 2021 Tatsumi, Fushimi, Sanada, Shimoda, Kohara, Kimura, Obata, Nakanishi, Mune, Kaku and Kaneto. 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 Medicine
Tatsumi, Fuminori
Fushimi, Yoshiro
Sanada, Junpei
Shimoda, Masashi
Kohara, Kenji
Kimura, Tomohiko
Obata, Atsushi
Nakanishi, Shuhei
Mune, Tomoatsu
Kaku, Kohei
Kaneto, Hideaki
Idiopathic Bilateral Extraocular Myositis in a Subject With Poorly Controlled Type 2 Diabetes Mellitus: Case Report
title Idiopathic Bilateral Extraocular Myositis in a Subject With Poorly Controlled Type 2 Diabetes Mellitus: Case Report
title_full Idiopathic Bilateral Extraocular Myositis in a Subject With Poorly Controlled Type 2 Diabetes Mellitus: Case Report
title_fullStr Idiopathic Bilateral Extraocular Myositis in a Subject With Poorly Controlled Type 2 Diabetes Mellitus: Case Report
title_full_unstemmed Idiopathic Bilateral Extraocular Myositis in a Subject With Poorly Controlled Type 2 Diabetes Mellitus: Case Report
title_short Idiopathic Bilateral Extraocular Myositis in a Subject With Poorly Controlled Type 2 Diabetes Mellitus: Case Report
title_sort idiopathic bilateral extraocular myositis in a subject with poorly controlled type 2 diabetes mellitus: case report
topic Medicine
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8514759/
https://www.ncbi.nlm.nih.gov/pubmed/34660622
http://dx.doi.org/10.3389/fmed.2021.700307
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