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Minocycline-associated rimmed vacuolar myopathy in a patient with rheumatoid arthritis

BACKGROUND: The autophagic vacuolar myopathies (AVM) are a group of inherited myopathies defined by the presence of autophagic vacuoles in pathological muscle specimens. AVM can be categorized into three groups: acid maltase deficiency, myopathies characterized by autophagic vacuoles with unique sar...

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Autores principales: Bokuda, Kota, Sugaya, Keizo, Tamura, Shunichiro, Miyamoto, Kazuhito, Matsubara, Shiro, Komori, Takashi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2012
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3522006/
https://www.ncbi.nlm.nih.gov/pubmed/23171360
http://dx.doi.org/10.1186/1471-2377-12-140
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author Bokuda, Kota
Sugaya, Keizo
Tamura, Shunichiro
Miyamoto, Kazuhito
Matsubara, Shiro
Komori, Takashi
author_facet Bokuda, Kota
Sugaya, Keizo
Tamura, Shunichiro
Miyamoto, Kazuhito
Matsubara, Shiro
Komori, Takashi
author_sort Bokuda, Kota
collection PubMed
description BACKGROUND: The autophagic vacuolar myopathies (AVM) are a group of inherited myopathies defined by the presence of autophagic vacuoles in pathological muscle specimens. AVM can be categorized into three groups: acid maltase deficiency, myopathies characterized by autophagic vacuoles with unique sarcolemmal features, and rimmed vacuolar myopathies (RVM). While the pathogeneses of these conditions are still being elucidated, some drugs (e.g., chloroquine, its analog, hydroxychloroquine, and colchicine) can also cause AVM. Minocycline is a disease-modifying anti-rheumatic drug that may be used in the treatment of rheumatoid arthritis (RA). Here, we describe the first case of minocycline-associated AVM with rimmed vacuole formation. CASE PRESENTATION: A 75-year-old woman suffering from RA has been continuously treated with minocycline (200 mg/day) for the past 7 years. During this time, she developed a myopathy that predominantly affected her lower limbs. Histological studies of biopsied muscle revealed scattered atrophic myofibers with rimmed vacuoles that contained pigment granules. Histochemical staining revealed that the pigment comprised both iron and melanin, which is consistent with type II minocycline-induced cutaneous pigmentation. Under electron microscopy, autophagic vacuoles were consistently observed in association with numerous collections of pigment granules. CONCLUSIONS: This is the first report of minocycline-induced pigmentation in skeletal muscle. The strong association between autophagic vacuoles and the accumulation of minocycline-induced pigments suggest that long-term minocycline treatment induced pigment accumulation, leading to elevation of autophagic activity and RVM. It might also be possible that minocycline directly activated autophagy, as the observed pigments are known to form complexes containing minocycline and/or its metabolites. As long-term minocycline treatment is expected to be used more widely in the future, we must draw attention to this adverse effect.
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spelling pubmed-35220062012-12-14 Minocycline-associated rimmed vacuolar myopathy in a patient with rheumatoid arthritis Bokuda, Kota Sugaya, Keizo Tamura, Shunichiro Miyamoto, Kazuhito Matsubara, Shiro Komori, Takashi BMC Neurol Case Report BACKGROUND: The autophagic vacuolar myopathies (AVM) are a group of inherited myopathies defined by the presence of autophagic vacuoles in pathological muscle specimens. AVM can be categorized into three groups: acid maltase deficiency, myopathies characterized by autophagic vacuoles with unique sarcolemmal features, and rimmed vacuolar myopathies (RVM). While the pathogeneses of these conditions are still being elucidated, some drugs (e.g., chloroquine, its analog, hydroxychloroquine, and colchicine) can also cause AVM. Minocycline is a disease-modifying anti-rheumatic drug that may be used in the treatment of rheumatoid arthritis (RA). Here, we describe the first case of minocycline-associated AVM with rimmed vacuole formation. CASE PRESENTATION: A 75-year-old woman suffering from RA has been continuously treated with minocycline (200 mg/day) for the past 7 years. During this time, she developed a myopathy that predominantly affected her lower limbs. Histological studies of biopsied muscle revealed scattered atrophic myofibers with rimmed vacuoles that contained pigment granules. Histochemical staining revealed that the pigment comprised both iron and melanin, which is consistent with type II minocycline-induced cutaneous pigmentation. Under electron microscopy, autophagic vacuoles were consistently observed in association with numerous collections of pigment granules. CONCLUSIONS: This is the first report of minocycline-induced pigmentation in skeletal muscle. The strong association between autophagic vacuoles and the accumulation of minocycline-induced pigments suggest that long-term minocycline treatment induced pigment accumulation, leading to elevation of autophagic activity and RVM. It might also be possible that minocycline directly activated autophagy, as the observed pigments are known to form complexes containing minocycline and/or its metabolites. As long-term minocycline treatment is expected to be used more widely in the future, we must draw attention to this adverse effect. BioMed Central 2012-11-21 /pmc/articles/PMC3522006/ /pubmed/23171360 http://dx.doi.org/10.1186/1471-2377-12-140 Text en Copyright ©2012 Bokuda et al.; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Bokuda, Kota
Sugaya, Keizo
Tamura, Shunichiro
Miyamoto, Kazuhito
Matsubara, Shiro
Komori, Takashi
Minocycline-associated rimmed vacuolar myopathy in a patient with rheumatoid arthritis
title Minocycline-associated rimmed vacuolar myopathy in a patient with rheumatoid arthritis
title_full Minocycline-associated rimmed vacuolar myopathy in a patient with rheumatoid arthritis
title_fullStr Minocycline-associated rimmed vacuolar myopathy in a patient with rheumatoid arthritis
title_full_unstemmed Minocycline-associated rimmed vacuolar myopathy in a patient with rheumatoid arthritis
title_short Minocycline-associated rimmed vacuolar myopathy in a patient with rheumatoid arthritis
title_sort minocycline-associated rimmed vacuolar myopathy in a patient with rheumatoid arthritis
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3522006/
https://www.ncbi.nlm.nih.gov/pubmed/23171360
http://dx.doi.org/10.1186/1471-2377-12-140
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