Literature DB >> 7726239

Myopathy, lactic acidosis, and sideroblastic anemia: a new syndrome.

A Inbal1, N Avissar, M Shaklai, A Kuritzky, A Schejter, E Ben-David, S Shanske, B Z Garty.   

Abstract

We describe 2 sibs (brother and sister) with myopathy, sideroblastic anemia, lactic acidosis, mental retardation, microcephaly, high palate, high philtrum, distichiasis, and micrognathia. Very low levels of cytochromes a, b, and c were detected in the patients' muscle mitochondria. Deposition of iron within the mitochondria of bone marrow erythroblasts was observed on electron microscopy. Irregular and enlarged mitochondria with paracrystalline inclusions were also seen on electron microscopy of the patients' muscle specimen. Examination of DNA from the affected sibs showed no deletions in the mitochondrial DNA nor the mutations identified in the syndromes of mitochondrial myopathy, encephalopathy, lactic acidosis, and strokelike episodes (MELAS) or myoclonus, and epilepsy associated with rugged-red fibers (MERRF). Since the parents were first cousins and 2 of 6 sibs (male and female) were affected, we suggest that the syndrome expressed by our patients represents a previously unknown autosomal recessive disorder that includes mitochondrial myopathy, lactic acidosis, and sideroblastic anemia.

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Year:  1995        PMID: 7726239     DOI: 10.1002/ajmg.1320550325

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  15 in total

1.  Multiple presentation of mitochondrial disorders.

Authors:  A Nissenkorn; A Zeharia; D Lev; A Fatal-Valevski; V Barash; A Gutman; S Harel; T Lerman-Sagie
Journal:  Arch Dis Child       Date:  1999-09       Impact factor: 3.791

2.  A genetic-phenotypic classification for syndromic micrognathia.

Authors:  Qiming Chen; Yan Zhao; Yifeng Qian; Chenpei Lu; Guofang Shen; Jiewen Dai
Journal:  J Hum Genet       Date:  2019-07-04       Impact factor: 3.172

3.  Nonsense mutation in pseudouridylate synthase 1 (PUS1) in two brothers affected by myopathy, lactic acidosis and sideroblastic anaemia (MLASA).

Authors:  Erika Fernandez-Vizarra; Angela Berardinelli; Lucia Valente; Valeria Tiranti; Massimo Zeviani
Journal:  BMJ Case Rep       Date:  2009-06-09

Review 4.  Hereditary sideroblastic anemia: pathophysiology and gene mutations.

Authors:  Hideo Harigae; Kazumichi Furuyama
Journal:  Int J Hematol       Date:  2010-09-17       Impact factor: 2.490

5.  Clinical Features, Molecular Heterogeneity, and Prognostic Implications in YARS2-Related Mitochondrial Myopathy.

Authors:  Ewen W Sommerville; Yi Shiau Ng; Charlotte L Alston; Cristina Dallabona; Micol Gilberti; Langping He; Charlotte Knowles; Sophie L Chin; Andrew M Schaefer; Gavin Falkous; David Murdoch; Cheryl Longman; Marianne de Visser; Laurence A Bindoff; John M Rawles; John C S Dean; Richard K Petty; Maria E Farrugia; Tobias B Haack; Holger Prokisch; Robert McFarland; Douglass M Turnbull; Claudia Donnini; Robert W Taylor; Gráinne S Gorman
Journal:  JAMA Neurol       Date:  2017-06-01       Impact factor: 18.302

6.  A novel homozygous YARS2 mutation causes severe myopathy, lactic acidosis, and sideroblastic anemia 2.

Authors:  Junya Nakajima; Tuba F Eminoglu; Goksel Vatansever; Mitsuko Nakashima; Yoshinori Tsurusaki; Hirotomo Saitsu; Hisashi Kawashima; Naomichi Matsumoto; Noriko Miyake
Journal:  J Hum Genet       Date:  2014-01-16       Impact factor: 3.172

7.  Missense mutation in pseudouridine synthase 1 (PUS1) causes mitochondrial myopathy and sideroblastic anemia (MLASA).

Authors:  Yelena Bykhovskaya; Kari Casas; Emebet Mengesha; Aida Inbal; Nathan Fischel-Ghodsian
Journal:  Am J Hum Genet       Date:  2004-04-22       Impact factor: 11.025

8.  Mitochondrial myopathy, lactic acidosis, and sideroblastic anemia (MLASA) plus associated with a novel de novo mutation (m.8969G>A) in the mitochondrial encoded ATP6 gene.

Authors:  Lindsay C Burrage; Sha Tang; Jing Wang; Taraka R Donti; Magdalena Walkiewicz; J Michael Luchak; Li-Chieh Chen; Eric S Schmitt; Zhiyv Niu; Rodrigo Erana; Jill V Hunter; Brett H Graham; Lee-Jun Wong; Fernando Scaglia
Journal:  Mol Genet Metab       Date:  2014-06-30       Impact factor: 4.797

9.  Haematological abnormalities in mitochondrial disorders.

Authors:  Josef Finsterer; Marlies Frank
Journal:  Singapore Med J       Date:  2015-07       Impact factor: 1.858

10.  Clinical and molecular study in a long-surviving patient with MLASA syndrome due to novel PUS1 mutations.

Authors:  Michelangelo Cao; Marta Donà; M Lucia Valentino; Lucia Valentino; Claudio Semplicini; Alessandra Maresca; Matteo Cassina; Alessandra Torraco; Eva Galletta; Valeria Manfioli; Gianni Sorarù; Valerio Carelli; Roberto Stramare; Enrico Bertini; Rosalba Carrozzo; Leonardo Salviati; Elena Pegoraro
Journal:  Neurogenetics       Date:  2015-11-10       Impact factor: 2.660

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