Literature DB >> 1447652

Fatal infantile liver failure associated with mitochondrial DNA depletion.

M R Mazziotta1, E Ricci, E Bertini, C Dionisi Vici, S Servidei, A B Burlina, G Sabetta, A Bartuli, G Manfredi, G Silvestri.   

Abstract

A 3-month-old girl was admitted to the hospital because of hypotonia and frequent vomiting. She had severe metabolic acidosis and her liver function was abnormal. Hepatomegaly and rapidly progressive liver failure developed, and she died at 4 months of age. Two half-siblings from a different mother had died in infancy of an undiagnosed myopathy. The liver was fatty and hepatocytes were filled with large and small lipid droplets. Other tissues were morphologically normal. The respiratory chain enzymes containing subunits encoded by mitochondrial DNA were markedly decreased in liver, partially decreased in muscle, but normal in other tissues. Southern blot analysis showed 90% depletion of mitochondrial DNA in liver, 53% depletion in muscle, and normal amounts in other tissues. This is the second case of fatal infantile liver failure associated with mitochondrial DNA depletion. This pathogenetic mechanism should be considered in infants with multiple respiratory chain defects and variable tissue expression.

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Year:  1992        PMID: 1447652     DOI: 10.1016/s0022-3476(05)80335-x

Source DB:  PubMed          Journal:  J Pediatr        ISSN: 0022-3476            Impact factor:   4.406


  17 in total

1.  Mitochondrial DNA depletion syndrome is expressed in amniotic fluid cell cultures.

Authors:  J C Blake; J W Taanman; A M Morris; R G Gray; J M Cooper; P J McKiernan; J V Leonard; A H Schapira
Journal:  Am J Pathol       Date:  1999-07       Impact factor: 4.307

2.  Fumarate hydratase deficiency: increased fumaric acid in amniotic fluid of two affected pregnancies.

Authors:  N J Manning; S E Olpin; R J Pollitt; M Downing; A F Heeley; I D Young
Journal:  J Inherit Metab Dis       Date:  2000-11       Impact factor: 4.982

3.  Fatal neonatal outcome in a case of muscular mitochondrial DNA depletion.

Authors:  G M Poggi; E Lamantea; F Ciani; M A Donati; F Carrara; L Bartalena; B Garavaglia; E Zammarchi
Journal:  J Inherit Metab Dis       Date:  2000-11       Impact factor: 4.982

4.  Potentially diagnostic electron paramagnetic resonance spectra elucidate the underlying mechanism of mitochondrial dysfunction in the deoxyguanosine kinase deficient rat model of a genetic mitochondrial DNA depletion syndrome.

Authors:  Brian Bennett; Daniel Helbling; Hui Meng; Jason Jarzembowski; Aron M Geurts; Marisa W Friederich; Johan L K Van Hove; Michael W Lawlor; David P Dimmock
Journal:  Free Radic Biol Med       Date:  2016-01-08       Impact factor: 7.376

5.  Fatal neonatal liver failure and depletion of mitochondrial DNA in three children of one family.

Authors:  H D Bakker; C Van den Bogert; H R Scholte; R Zwart; F A Wijburg; J N Spelbrink
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

Review 6.  Respiratory-chain and pyruvate metabolism defects: Italian collaborative survey on 72 patients.

Authors:  U Caruso; A Adami; E Bertini; A B Burlina; F Carnevale; R Cerone; C Dionisi-Vici; G Giordano; E Leuzzi; G Parenti; S Savasta; G Uziel; M Zeviani
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

Review 7.  Genetic counselling and prenatal diagnosis in disorders of the mitochondrial energy metabolism.

Authors:  W Ruitenbeek; U Wendel; B C Hamel; J M Trijbels
Journal:  J Inherit Metab Dis       Date:  1996       Impact factor: 4.982

8.  Fulminant hepatic failure in a child as a potential adverse effect of trimethoprim-sulphamethoxazole.

Authors:  B Simma; B Meister; J Deutsch; W Sperl; F Fend; D Ofner; R Margreiter; W Vogel
Journal:  Eur J Pediatr       Date:  1995-07       Impact factor: 3.183

9.  Early-onset fatal encephalomyopathy associated with severe mtDNA depletion.

Authors:  V Paquis-Flucklinger; J F Pellissier; J Camboulives; B Chabrol; A Saunières; M F Monfort; H Giudicelli; C Desnuelle
Journal:  Eur J Pediatr       Date:  1995-07       Impact factor: 3.183

10.  Nuclear complementation restores mtDNA levels in cultured cells from a patient with mtDNA depletion.

Authors:  A G Bodnar; J M Cooper; I J Holt; J V Leonard; A H Schapira
Journal:  Am J Hum Genet       Date:  1993-09       Impact factor: 11.025

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