Literature DB >> 8151637

Juvenile Kearns-Sayre syndrome initially misdiagnosed as a psychosomatic disorder.

S Nørby1, P Lestienne, I Nelson, I M Nielsen, H Schmalbruch, O Sjö, M Warburg.   

Abstract

We have investigated a 15 year old girl with progressive external ophthalmoplegia, including bilateral ptosis and retinal rod and cone cell dysfunction with atypical retinal pigmentation, complicated by cerebellar ataxia, partial cardiac conduction block, and diabetes mellitus. In infancy she had a severe crisis of bone marrow depression, and as a child she suffered from hypersensitivity to light, increasing fatigue, and vertigo, signs that were initially though to be psychosomatic. Histological examination showed mitochondrial myopathy, and subsequent mitochondrial DNA (mtDNA) analysis showed a deletion of approximately 5500 base pairs in 35 to 40% of her muscle mtDNA. We therefore conclude that this patient has developed the Kearns-Sayre syndrome after a Pearson syndrome-like crisis in her first year of life.

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Year:  1994        PMID: 8151637      PMCID: PMC1049598          DOI: 10.1136/jmg.31.1.45

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  36 in total

1.  Widespread tissue distribution of mitochondrial DNA deletions in Kearns-Sayre syndrome.

Authors:  S Shanske; C T Moraes; A Lombes; A F Miranda; E Bonilla; P Lewis; M A Whelan; C A Ellsworth; S DiMauro
Journal:  Neurology       Date:  1990-01       Impact factor: 9.910

2.  Segregation of mitochondrial genomes in a heteroplasmic lineage with Leber hereditary optic neuroretinopathy.

Authors:  J Vilkki; M L Savontaus; E K Nikoskelainen
Journal:  Am J Hum Genet       Date:  1990-07       Impact factor: 11.025

Review 3.  Mitochondrial DNA mutations in human diseases: a review.

Authors:  P Lestienne
Journal:  Biochimie       Date:  1992-02       Impact factor: 4.079

4.  Deletion of blood mitochondrial DNA in pancytopenia.

Authors:  A Rötig; M Colonna; S Blanche; A Fischer; F Le Deist; J Frezal; J M Saudubray; A Munnich
Journal:  Lancet       Date:  1988-09-03       Impact factor: 79.321

5.  Transcription and translation of deleted mitochondrial genomes in Kearns-Sayre syndrome: implications for pathogenesis.

Authors:  H Nakase; C T Moraes; R Rizzuto; A Lombes; S DiMauro; E A Schon
Journal:  Am J Hum Genet       Date:  1990-03       Impact factor: 11.025

6.  Sequence and organization of the human mitochondrial genome.

Authors:  S Anderson; A T Bankier; B G Barrell; M H de Bruijn; A R Coulson; J Drouin; I C Eperon; D P Nierlich; B A Roe; F Sanger; P H Schreier; A J Smith; R Staden; I G Young
Journal:  Nature       Date:  1981-04-09       Impact factor: 49.962

7.  Spontaneous Kearns-Sayre/chronic external ophthalmoplegia plus syndrome associated with a mitochondrial DNA deletion: a slip-replication model and metabolic therapy.

Authors:  J M Shoffner; M T Lott; A S Voljavec; S A Soueidan; D A Costigan; D C Wallace
Journal:  Proc Natl Acad Sci U S A       Date:  1989-10       Impact factor: 11.205

8.  Evidence for intramitochondrial complementation between deleted and normal mitochondrial DNA in some patients with mitochondrial myopathy.

Authors:  S R Hammans; M G Sweeney; I J Holt; J M Cooper; A Toscano; J B Clark; J A Morgan-Hughes; A E Harding
Journal:  J Neurol Sci       Date:  1992-01       Impact factor: 3.181

9.  A new syndrome of refractory sideroblastic anemia with vacuolization of marrow precursors and exocrine pancreatic dysfunction.

Authors:  H A Pearson; J S Lobel; S A Kocoshis; J L Naiman; J Windmiller; A T Lammi; R Hoffman; J C Marsh
Journal:  J Pediatr       Date:  1979-12       Impact factor: 4.406

10.  The retinal manifestations of mitochondrial myopathy. A study of 22 cases.

Authors:  M A Mullie; A E Harding; R K Petty; H Ikeda; J A Morgan-Hughes; M D Sanders
Journal:  Arch Ophthalmol       Date:  1985-12
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  1 in total

Review 1.  Roles of Mitochondrial DNA Mutations in Stem Cell Ageing.

Authors:  Tianhong Su; Doug M Turnbull; Laura C Greaves
Journal:  Genes (Basel)       Date:  2018-03-27       Impact factor: 4.096

  1 in total

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