Literature DB >> 11078225

Mitochondrial encephalomyopathy with lactic acidosis and stroke like episodes (MELAS) with prominent degeneration of the intestinal wall and cactus-like cerebellar pathology.

O Mori1, M Yamazaki, Y Ohaki, Y Arai, T Oguro, H Shimizu, G Asano.   

Abstract

A 67-year-old woman had frequent subacute ileus, hearing difficulty, muscle atrophy and stroke-like episodes. Computed tomography revealed multiple low-density areas, which did not correlate with the vascular supply, in the cerebral cortex. She had metabolic disturbance comprising lactic acidosis and elevated pyruvate level. Her skeletal muscle biopsy specimen showed ragged-red fibers, and mitochondrial DNA analysis revealed a point mutation at position 3243, findings consistent with MELAS. Examination of her small intestine revealed a necrotic zone and numerous abnormal large mitochondria in the smooth muscle cells, vascular media and endothelium, and intestinal ganglion cells. The cerebral cortex showed multiple microcystic necrotic foci in cerebral cortex. Cactus-like pathology resembling the changes associated with Menkes' kinky hair disease and torpedoes were observed in the cerebellar Purkinje cells. The intestinal dysmotility due to MELAS and cerebellar changes were presumed to be associated with a disturbance of copper metabolism.

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Year:  2000        PMID: 11078225     DOI: 10.1007/s004010000209

Source DB:  PubMed          Journal:  Acta Neuropathol        ISSN: 0001-6322            Impact factor:   17.088


  8 in total

1.  p62/sequestosome-1 knockout delays neurodegeneration induced by Drp1 loss.

Authors:  Tatsuya Yamada; Yoshihiro Adachi; Toru Yanagawa; Miho Iijima; Hiromi Sesaki
Journal:  Neurochem Int       Date:  2017-05-18       Impact factor: 3.921

2.  Biallelic TBCD Mutations Cause Early-Onset Neurodegenerative Encephalopathy.

Authors:  Noriko Miyake; Ryoko Fukai; Chihiro Ohba; Takahiro Chihara; Masayuki Miura; Hiroshi Shimizu; Akiyoshi Kakita; Eri Imagawa; Masaaki Shiina; Kazuhiro Ogata; Jiu Okuno-Yuguchi; Noboru Fueki; Yoshifumi Ogiso; Hiroshi Suzumura; Yoshiyuki Watabe; George Imataka; Huey Yin Leong; Aviva Fattal-Valevski; Uri Kramer; Satoko Miyatake; Mitsuhiro Kato; Nobuhiko Okamoto; Yoshinori Sato; Satomi Mitsuhashi; Ichizo Nishino; Naofumi Kaneko; Akira Nishiyama; Tomohiko Tamura; Takeshi Mizuguchi; Mitsuko Nakashima; Fumiaki Tanaka; Hirotomo Saitsu; Naomichi Matsumoto
Journal:  Am J Hum Genet       Date:  2016-09-22       Impact factor: 11.025

Review 3.  Neuropathological aspects of mitochondrial DNA disease.

Authors:  Joanne Betts; Robert N Lightowlers; Douglass M Turnbull
Journal:  Neurochem Res       Date:  2004-03       Impact factor: 3.996

4.  Familial Pernicious Chronic Intestinal Pseudo-obstruction with a Mitochondrial DNA A3243G Mutation.

Authors:  Junichiro Suzuki; Mai Iwata; Hideyuki Moriyoshi; Suguru Nishida; Takeshi Yasuda; Yasuhiro Ito
Journal:  Intern Med       Date:  2017-05-01       Impact factor: 1.271

5.  Cerebellar ataxia in patients with mitochondrial DNA disease: a molecular clinicopathological study.

Authors:  Nichola Zoe Lax; Philippa Denis Hepplewhite; Amy Katherine Reeve; Victoria Nesbitt; Robert McFarland; Evelyn Jaros; Robert William Taylor; Douglass Matthew Turnbull
Journal:  J Neuropathol Exp Neurol       Date:  2012-02       Impact factor: 3.685

6.  Development of passive CLARITY and immunofluorescent labelling of multiple proteins in human cerebellum: understanding mechanisms of neurodegeneration in mitochondrial disease.

Authors:  Jonathan Phillips; Alex Laude; Robert Lightowlers; Chris M Morris; Doug M Turnbull; Nichola Z Lax
Journal:  Sci Rep       Date:  2016-05-16       Impact factor: 4.379

Review 7.  The genetics and pathology of mitochondrial disease.

Authors:  Charlotte L Alston; Mariana C Rocha; Nichola Z Lax; Doug M Turnbull; Robert W Taylor
Journal:  J Pathol       Date:  2016-11-02       Impact factor: 7.996

Review 8.  Review: Central nervous system involvement in mitochondrial disease.

Authors:  N Z Lax; G S Gorman; D M Turnbull
Journal:  Neuropathol Appl Neurobiol       Date:  2016-07-07       Impact factor: 8.090

  8 in total

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