Primary isolated amyloidosis in the extraocular muscle as a rare cause of ophthalmoplegia: A case report and literature review
Purpose: To report a case of external ophthalmoplegia due to an uncommon form of amyloidosis exclusively affecting the lateral rectus muscle, and to discuss the clinical manifestation, diagnostic challenges, and management pitfalls of isolated amyloidosis in the extraocular muscle. Observations: A 6...
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doaj-3b781262cb854b36aa7530521f4ac5f72021-05-22T04:37:58ZengElsevierAmerican Journal of Ophthalmology Case Reports2451-99362021-06-0122101052Primary isolated amyloidosis in the extraocular muscle as a rare cause of ophthalmoplegia: A case report and literature reviewNoriko Nishikawa0Yuriya Kawaguchi1Ami Konno2Yuya Kitani3Hidehiro Takei4Yasuo Yanagi5Department of Ophthalmology, Asahikawa Medical University, 2-1-1 Midorigaoka, Higashi, Asahikawa, Hokkaido, 078-8510, Japan; Corresponding author.Department of Ophthalmology, Asahikawa Medical University, 2-1-1 Midorigaoka, Higashi, Asahikawa, Hokkaido, 078-8510, JapanDepartment of Ophthalmology, Asahikawa Medical University, 2-1-1 Midorigaoka, Higashi, Asahikawa, Hokkaido, 078-8510, JapanDivision of Cardiology, Nephrology, Pulmonology and Neurology, Department of Internal Medicine, Asahikawa Medical University, 2-1-1 Midorigaoka, Higashi, Asahikawa, Hokkaido, 078-8510, JapanDepartment of Diagnostic Pathology, Asahikawa Medical University, 2-1-1 Midorigaoka, Higashi, Asahikawa, Hokkaido, 078-8510, JapanDepartment of Ophthalmology, Asahikawa Medical University, 2-1-1 Midorigaoka, Higashi, Asahikawa, Hokkaido, 078-8510, Japan; Singapore National Eye Centre, Singapore Eye Research Institute, 11 Third Hospital Ave, 168751, SingaporePurpose: To report a case of external ophthalmoplegia due to an uncommon form of amyloidosis exclusively affecting the lateral rectus muscle, and to discuss the clinical manifestation, diagnostic challenges, and management pitfalls of isolated amyloidosis in the extraocular muscle. Observations: A 64-year-old woman presented with diplopia in her left gaze lasting for six months. She had orthophoria in the primary position and abduction limitation in the left eye. Routine laboratory examinations were unremarkable. Orbital magnetic resonance imaging showed fusiform enlargement of the left lateral rectus muscle, without tendon involvement. Extraocular muscle biopsy was recommended to make a diagnosis, which revealed amyloid deposition in the lateral rectus muscle. A systemic work-up showed no evidence of systemic amyloidosis. Therefore, a diagnosis of primary isolated amyloidosis was made. Orthophoria in the primary position and diplopia in the lateral gaze persisted at the six-month follow-up. Conclusions and importance: Atypical extraocular muscle enlargement should alert clinicians to the need for tissue biopsy to identify uncommon etiologies, such as amyloidosis. There are no pathognomonic or radiological features to distinguish localized from systemic amyloidosis. Therefore, if amyloidosis of the extraocular muscles is diagnosed, a systemic work-up is needed to rule out systemic amyloidosis, which is potentially life-threatening.http://www.sciencedirect.com/science/article/pii/S2451993621000438External ophthalmoplegiaDiplopiaExtraocular muscle enlargementExtraocular muscle biopsyOrbital amyloidosis |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Noriko Nishikawa Yuriya Kawaguchi Ami Konno Yuya Kitani Hidehiro Takei Yasuo Yanagi |
spellingShingle |
Noriko Nishikawa Yuriya Kawaguchi Ami Konno Yuya Kitani Hidehiro Takei Yasuo Yanagi Primary isolated amyloidosis in the extraocular muscle as a rare cause of ophthalmoplegia: A case report and literature review American Journal of Ophthalmology Case Reports External ophthalmoplegia Diplopia Extraocular muscle enlargement Extraocular muscle biopsy Orbital amyloidosis |
author_facet |
Noriko Nishikawa Yuriya Kawaguchi Ami Konno Yuya Kitani Hidehiro Takei Yasuo Yanagi |
author_sort |
Noriko Nishikawa |
title |
Primary isolated amyloidosis in the extraocular muscle as a rare cause of ophthalmoplegia: A case report and literature review |
title_short |
Primary isolated amyloidosis in the extraocular muscle as a rare cause of ophthalmoplegia: A case report and literature review |
title_full |
Primary isolated amyloidosis in the extraocular muscle as a rare cause of ophthalmoplegia: A case report and literature review |
title_fullStr |
Primary isolated amyloidosis in the extraocular muscle as a rare cause of ophthalmoplegia: A case report and literature review |
title_full_unstemmed |
Primary isolated amyloidosis in the extraocular muscle as a rare cause of ophthalmoplegia: A case report and literature review |
title_sort |
primary isolated amyloidosis in the extraocular muscle as a rare cause of ophthalmoplegia: a case report and literature review |
publisher |
Elsevier |
series |
American Journal of Ophthalmology Case Reports |
issn |
2451-9936 |
publishDate |
2021-06-01 |
description |
Purpose: To report a case of external ophthalmoplegia due to an uncommon form of amyloidosis exclusively affecting the lateral rectus muscle, and to discuss the clinical manifestation, diagnostic challenges, and management pitfalls of isolated amyloidosis in the extraocular muscle. Observations: A 64-year-old woman presented with diplopia in her left gaze lasting for six months. She had orthophoria in the primary position and abduction limitation in the left eye. Routine laboratory examinations were unremarkable. Orbital magnetic resonance imaging showed fusiform enlargement of the left lateral rectus muscle, without tendon involvement. Extraocular muscle biopsy was recommended to make a diagnosis, which revealed amyloid deposition in the lateral rectus muscle. A systemic work-up showed no evidence of systemic amyloidosis. Therefore, a diagnosis of primary isolated amyloidosis was made. Orthophoria in the primary position and diplopia in the lateral gaze persisted at the six-month follow-up. Conclusions and importance: Atypical extraocular muscle enlargement should alert clinicians to the need for tissue biopsy to identify uncommon etiologies, such as amyloidosis. There are no pathognomonic or radiological features to distinguish localized from systemic amyloidosis. Therefore, if amyloidosis of the extraocular muscles is diagnosed, a systemic work-up is needed to rule out systemic amyloidosis, which is potentially life-threatening. |
topic |
External ophthalmoplegia Diplopia Extraocular muscle enlargement Extraocular muscle biopsy Orbital amyloidosis |
url |
http://www.sciencedirect.com/science/article/pii/S2451993621000438 |
work_keys_str_mv |
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