Literature DB >> 8363469

Ocular clinicopathologic study of the mitochondrial encephalomyopathy overlap syndromes.

T S Chang1, D R Johns, D Walker, Z de la Cruz, I H Maumence, W R Green.   

Abstract

Recent advances in molecular genetics have led to a better understanding of mitochondrially inherited diseases. Mitochondrial encephalomyopathy overlap syndrome is one such group of diseases in which ocular abnormalities are frequently manifest. The authors describe the clinical, molecular genetic, and pathologic findings of two patients with the mitochondrial encephalomyopathy overlap syndrome. The patients shared a similar clinical course with features overlapping the three traditionally distinct clinical phenotypes (the Kearns-Sayre syndrome; the syndrome of mitochondrial encephalopathy, lactic acidosis, and stroke [MELAS], and the syndrome of myoclonus, epilepsy, and ragged red fibers [MERRF]). The patients had identical mitochondrial DNA mutations (at nucleotide position 3243) and had similar ultrastructural abnormalities, including abundant enlarged mitochondria with "whorled" and "tubular" cristae. These abnormal mitochondria appeared to be preferentially distributed in cells with high metabolic activity (retinal pigment epithelium, corneal endothelium, and extraocular muscles).

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Year:  1993        PMID: 8363469     DOI: 10.1001/archopht.1993.01090090106028

Source DB:  PubMed          Journal:  Arch Ophthalmol        ISSN: 0003-9950


  7 in total

Review 1.  The neuro-ophthalmology of mitochondrial disease.

Authors:  J Alexander Fraser; Valérie Biousse; Nancy J Newman
Journal:  Surv Ophthalmol       Date:  2010-05-14       Impact factor: 6.048

2.  Cone and rod dysfunction in the NARP syndrome.

Authors:  I Chowers; T Lerman-Sagie; O N Elpeleg; A Shaag; S Merin
Journal:  Br J Ophthalmol       Date:  1999-02       Impact factor: 4.638

3.  Central nervous system involvement in gyrate atrophy of the choroid and retina with hyperornithinaemia.

Authors:  M Valtonen; K Näntö-Salonen; S Jääskeläinen; K Heinänen; A Alanen; O J Heinonen; N Lundbom; M Erkintalo; O Simell
Journal:  J Inherit Metab Dis       Date:  1999-12       Impact factor: 4.982

4.  Human iPSC disease modelling reveals functional and structural defects in retinal pigment epithelial cells harbouring the m.3243A > G mitochondrial DNA mutation.

Authors:  Valeria Chichagova; Dean Hallam; Joseph Collin; Adriana Buskin; Gabriele Saretzki; Lyle Armstrong; Patrick Yu-Wai-Man; Majlinda Lako; David H Steel
Journal:  Sci Rep       Date:  2017-09-26       Impact factor: 4.379

5.  Mitochondrial A3243G mutation results in corneal endothelial polymegathism.

Authors:  Mathieu F Bakhoum; Wei-Pu Wu; Eugenia C White; Jesse D Sengillo; Christian Sanfilippo; Marcelle M Morcos; K Bailey Freund; Henry D Perry; David Sarraf; Stephen H Tsang
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  2018-01-29       Impact factor: 3.117

6.  Retinal and renal complications in patients with a mutation of mitochondrial DNA at position 3,243 (maternally inherited diabetes and deafness). A case-control study.

Authors:  P Massin; D Dubois-Laforgue; T Meas; M Laloi-Michelin; H Gin; B Bauduceau; C Bellanné-Chantelot; E Bertin; J-F Blickle; B Bouhanick; J Cahen-Varsaux; S Casanova; G Charpentier; P Chedin; O Dupuy; A Grimaldi; B Guerci; E Kaloustian; A Lecleire-Collet; F Lorenzini; A Murat; H Narbonne; F Olivier; V Paquis-Flucklinger; M Virally; M Vincenot; B Vialettes; J Timsit; P J Guillausseau
Journal:  Diabetologia       Date:  2008-06-26       Impact factor: 10.122

7.  [Central pigment epithelial defect in a 33 year-old female patient. A 33 year-old female patient with neurological symptoms, central pigment epithelial defect and limited upward gaze].

Authors:  A H Wolf; C Gass; A Kampik; G Rudolph
Journal:  Ophthalmologe       Date:  2004-08       Impact factor: 1.059

  7 in total

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