- Myofibrillar Myopathy: A Case Report.
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Jee Young Kim, Eun Hae Jeong, Kee Duk Park, Heasoo Koo
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Korean J Pathol. 2010;44(4):426-430.
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DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.4.426
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Abstract
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- Myofibrillar myopathies (MFMs) are a genetically or clinically heterogeneous group of diseases that are characterized by focal myofibrillar dissolution associated with accumulation of myofibrillar degradation products and ectopic expression of multiple proteins. Since MFMs show morphologically distinct features but consist of genetically and clinically heterogeneous diseases, muscle biopsy is important for the diagnosis. A 20-year-old man complained of progressive weakness and atrophy of both legs for two years.
He had a dysmorphic face and short stature. The light microscopic examination of his muscle biopsy showed mixed myopathic and neurogenic changes. Many myofibers with multiple clusters of blue red rod-like structures and cytoplasmic inclusions were noted. Immunohistochemistry showed a focal positive reaction in sarcoplasm to desmin and myotilin antibodies. An electron microscope study revealed variable abnormalities of myofibrillar structures. To the best of our knowledge, this is the first reported case of MFM with pathology findings in Korea.
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- A case of myofibrillary myopathy due to Bcl2-Associated Athanogene 3 (BAG3) mutation complicated by peripheral neuropathy
Risa Nagatomo, Yujiro Higuchi, Jun Takei, Tomonori Nakamura, Hiroaki Hashiguchi, Hiroshi Takashima Rinsho Shinkeigaku.2023; 63(12): 836. CrossRef
- Myopathy due to Chronic Clevudine Therapy: A Case Report.
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Jee Young Kim, Young Shin Yoon, Kee Duk Park, Heasoo Koo
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Korean J Pathol. 2009;43(6):575-579.
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DOI: https://doi.org/10.4132/KoreanJPathol.2009.43.6.575
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3,676
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Abstract
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- A 40-year-old man with chronic hepatitis B complained of progressive weakness of the proximal muscles and edema of both legs. He had been receiving long-term clevudine (nucleoside analogue reverse transcriptase inhibitor, NRTI) therapy for his hepatitis. The serum creatine kinase level was increased on the laboratory tests. His electromyography showed a generalized myopathic process. The muscle biopsy showed numerous ragged-red fibers, degenerating myofibers with variable sized cytoplasmic bodies, the prominence of type 1 fibers with type 2 fiber atrophy and an endomysial mononuclear cell infiltration. The electron microscopic examination revealed necrotic myofibers, including extremely dysmorphic mitochondria with extensive loss, blunting and focal clumping of the cristae and concentric cristae.
Although clevudine is known to be a less cytotoxic agent among the various NRTIs, careful clinical attention should be paid to the patients who are receiving long-term clevudine therapy for the occurrence of myopathy.
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Citations
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- A randomized phase 1b trial of the active site polymerase inhibitor nucleotide ATI‐2173 in patients with chronic hepatitis B virus infection
Katherine E. Squires, Lauren Ogilvie, Alina Jucov, Igor Anastasiy, Nelli Ghicavii, Jade Huguet, Rebeca Melara, Martin Constantineau, Abel De La Rosa, Douglas L. Mayers Journal of Viral Hepatitis.2023; 30(1): 19. CrossRef - Active site polymerase inhibitor nucleotides (ASPINs): Potential agents for chronic HBV cure regimens
Robert G Gish, Tarik Asselah, Katherine Squires, Douglas Mayers Antiviral Chemistry and Chemotherapy.2022; 30: 204020662211387. CrossRef - Clevudine Induced Mitochondrial Myopathy
Soo-Hyun Park, Kyung-Seok Park, Nam-Hee Kim, Joong-Yang Cho, Moon Soo Koh, Jin Ho Lee Journal of Korean Medical Science.2017; 32(11): 1857. CrossRef - In silico elucidation of the inclusion phenomenon and permeation behavior of a zidovudine–cyclodextrin complex via static lattice atomistic simulation
Pradeep Kumar, Yahya E. Choonara, Lisa C. Toit, Viness Pillay Journal of Inclusion Phenomena and Macrocyclic Chemistry.2014; 78(1-4): 445. CrossRef
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