Literature DB >> 2243133

Pearson's marrow-pancreas syndrome. A multisystem mitochondrial disorder in infancy.

A Rötig1, V Cormier, S Blanche, J P Bonnefont, F Ledeist, N Romero, J Schmitz, P Rustin, A Fischer, J M Saudubray.   

Abstract

Pearson's marrow-pancreas syndrome (McKusick No. 26056) is a fatal disorder of hitherto unknown etiology involving the hematopoietic system, exocrine pancreas, liver, and kidneys. The observation of high lactate/pyruvate molar ratios in plasma and abnormal oxidative phosphorylation in lymphocytes led us to postulate that Pearson's syndrome belongs to the group of mitochondrial cytopathies. Since rearrangements of the mitochondrial genome between direct DNA repeats were consistently found in all tissues tested, our results show that this disease is in fact a multisystem mitochondrial disorder, as suggested by the clinical course of the patients. Based on these observations, we would suggest giving consideration to the hypothesis of a defect of oxidative phosphorylation in elucidating the origin of other syndromes, especially those associated with an abnormal oxidoreduction status in plasma.

Entities:  

Mesh:

Substances:

Year:  1990        PMID: 2243133      PMCID: PMC296909          DOI: 10.1172/JCI114881

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  20 in total

1.  THE SYNDROME OF PANCREATIC INSUFFICIENCY AND BONE MARROW DYSFUNCTION.

Authors:  H SHWACHMAN; L K DIAMOND; F A OSKI; K T KHAW
Journal:  J Pediatr       Date:  1964-11       Impact factor: 4.406

2.  Duplications of mitochondrial DNA in mitochondrial myopathy.

Authors:  J Poulton; M E Deadman; R M Gardiner
Journal:  Lancet       Date:  1989-02-04       Impact factor: 79.321

3.  Mitochondrial DNA deletion in Pearson's marrow/pancreas syndrome.

Authors:  A Rotig; M Colonna; J P Bonnefont; S Blanche; A Fischer; J M Saudubray; A Munnich
Journal:  Lancet       Date:  1989-04-22       Impact factor: 79.321

4.  An autosomal dominant disorder with multiple deletions of mitochondrial DNA starting at the D-loop region.

Authors:  M Zeviani; S Servidei; C Gellera; E Bertini; S DiMauro; S DiDonato
Journal:  Nature       Date:  1989-05-25       Impact factor: 49.962

5.  Deletions of muscle mitochondrial DNA in mitochondrial myopathies: sequence analysis and possible mechanisms.

Authors:  I J Holt; A E Harding; J A Morgan-Hughes
Journal:  Nucleic Acids Res       Date:  1989-06-26       Impact factor: 16.971

6.  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

7.  The role of lymphocyte function-associated antigen 1 (LFA-1) in the adherence of T lymphocytes to B lymphocytes.

Authors:  F Mazerolles; C Lumbroso; O Lecomte; F Le Deist; A Fischer
Journal:  Eur J Immunol       Date:  1988-08       Impact factor: 5.532

8.  Deletions of mitochondrial DNA in Kearns-Sayre syndrome.

Authors:  M Zeviani; C T Moraes; S DiMauro; H Nakase; E Bonilla; E A Schon; L P Rowland
Journal:  Neurology       Date:  1988-09       Impact factor: 9.910

Review 9.  Mitochondrial DNA mutations and neuromuscular disease.

Authors:  D C Wallace
Journal:  Trends Genet       Date:  1989-01       Impact factor: 11.639

10.  An electron-transport system associated with the outer membrane of liver mitochondria. A biochemical and morphological study.

Authors:  G L Sottocasa; B Kuylenstierna; L Ernster; A Bergstrand
Journal:  J Cell Biol       Date:  1967-02       Impact factor: 10.539

View more
  106 in total

1.  Muscle carnitine acetyltransferase and carnitine deficiency in a case of mitochondrial encephalomyopathy.

Authors:  B Melegh; L Seress; T Bedekovics; G Kispál; B Sümegi; K Trombitás; K Méhes
Journal:  J Inherit Metab Dis       Date:  1999-10       Impact factor: 4.982

Review 2.  Clinical mitochondrial genetics.

Authors:  P F Chinnery; N Howell; R M Andrews; D M Turnbull
Journal:  J Med Genet       Date:  1999-06       Impact factor: 6.318

3.  Mitochondrial DNA deletion in an 8-year-old boy with Pearson syndrome.

Authors:  K E Baerlocher; A Feldges; M Weissert; H J Simonsz; A Rötig
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

4.  Detection of extremely low levels of wild-type mitochondrial DNA in the liver of a patient with Pearson syndrome by a sensitive PCR assay.

Authors:  D D de Vries; W Ruitenbeek; B A van Oost
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

Review 5.  Deletions of the mitochondrial genome.

Authors:  A E Harding; S R Hammans
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

6.  Duplications of mitochondrial DNA: implications for pathogenesis.

Authors:  J Poulton
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

7.  Pearson syndrome and the role of deletion dimers and duplications in the mtDNA.

Authors:  L J A M Jacobs; R J E Jongbloed; F A Wijburg; J B C de Klerk; J P M Geraedts; J G Nijland; H R Scholte; I F M de Coo; H J M Smeets
Journal:  J Inherit Metab Dis       Date:  2004       Impact factor: 4.982

8.  Autosomal dominant deletions of the mitochondrial genome in a case of progressive encephalomyopathy.

Authors:  V Cormier; A Rotig; M Tardieu; M Colonna; J M Saudubray; A Munnich
Journal:  Am J Hum Genet       Date:  1991-04       Impact factor: 11.025

9.  Genetic and clinical features of Chinese patients with mitochondrial ataxia identified by targeted next-generation sequencing.

Authors:  Hai-Lin Dong; Yin Ma; Quan-Fu Li; Yi-Chu Du; Lu Yang; Sheng Chen; Zhi-Ying Wu
Journal:  CNS Neurosci Ther       Date:  2018-05-13       Impact factor: 5.243

10.  Pearson bone marrow-pancreas syndrome with insulin-dependent diabetes, progressive renal tubulopathy, organic aciduria and elevated fetal haemoglobin caused by deletion and duplication of mitochondrial DNA.

Authors:  A Superti-Furga; E Schoenle; P Tuchschmid; R Caduff; V Sabato; D DeMattia; R Gitzelmann; B Steinmann
Journal:  Eur J Pediatr       Date:  1993-01       Impact factor: 3.183

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.