Literature DB >> 22602837

Neonatal liver failure due to deoxyguanosine kinase deficiency.

Susana Nobre1, Manuela Grazina, Francisco Silva, Carla Pinto, Isabel Gonçalves, Luísa Diogo.   

Abstract

Deoxyguanosine kinase (dGK) deficiency, a rare severe cause of mitochondrial DNA (mtDNA) depletion, has two forms of presentation: hepatocerebral syndrome and isolated hepatic disease. The authors report three cases with neonatal liver failure due to dGK deficiency. Consanguinity was present in all patients. One patient had a brother who died with a probable diagnosis of neonatal haemochromatosis. All patients had progressive cholestatic liver failure, hypoglycaemia, hyperlactacidaemia, elevated ferritin levels and nystagmus, since first day of life. Liver tissue study revealed: cholestasis, iron deposits, microvesicular steatosis and fibrosis/cirrhosis. Only one patient was submitted to liver transplantation. The other two died, at 2 and 5 months of age. mtDNA quantification and DGUOK gene study should be considered in infants/neonates with acute liver failure and systematically performed in patients with hepatocerebral presentation. Differential diagnosis with neonatal haemochromatosis is needed. Liver transplantation might be a therapeutic option. Early diagnosis is important for genetic counselling.

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Year:  2012        PMID: 22602837      PMCID: PMC3339167          DOI: 10.1136/bcr.12.2011.5317

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  10 in total

1.  Clinical, biochemical and morphological features of hepatocerebral syndrome with mitochondrial DNA depletion due to deoxyguanosine kinase deficiency.

Authors:  François Labarthe; Dries Dobbelaere; Louise Devisme; Anne De Muret; Claude Jardel; Jan-Willem Taanman; Frédéric Gottrand; Anne Lombès
Journal:  J Hepatol       Date:  2005-08       Impact factor: 25.083

2.  Orthotopic liver transplantation for mitochondrial respiratory chain disorders: a study of 5 children.

Authors:  B Dubern; P Broue; C Dubuisson; V Cormier-Daire; D Habes; C Chardot; D Devictor; A Munnich; O Bernard
Journal:  Transplantation       Date:  2001-03-15       Impact factor: 4.939

3.  Hepatocerebral mitochondrial DNA depletion syndrome caused by deoxyguanosine kinase (DGUOK) mutations.

Authors:  Peter Freisinger; Nancy Fütterer; Erwin Lankes; Klaus Gempel; Thomas M Berger; Johannes Spalinger; Alexandra Hoerbe; Claudia Schwantes; Martin Lindner; René Santer; Martin Burdelski; Hansjörg Schaefer; Bernhard Setzer; Ulrich A Walker; Rita Horváth
Journal:  Arch Neurol       Date:  2006-08

4.  Deoxyguanosine kinase mutations and combined deficiencies of the mitochondrial respiratory chain in patients with hepatic involvement.

Authors:  A Slama; I Giurgea; D Debrey; D Bridoux; P de Lonlay; P Levy; D Chretien; M Brivet; A Legrand; P Rustin; A Munnich; A Rötig
Journal:  Mol Genet Metab       Date:  2005-11-02       Impact factor: 4.797

5.  Liver transplantation in mitochondrial respiratory chain disorders.

Authors:  E M Sokal; R Sokol; V Cormier; F Lacaille; P McKiernan; F J Van Spronsen; O Bernard; J M Saudubray
Journal:  Eur J Pediatr       Date:  1999-12       Impact factor: 3.183

6.  Deoxyguanosine kinase deficiency presenting as neonatal hemochromatosis.

Authors:  Neil A Hanchard; Oleg A Shchelochkov; Angshumoy Roy; Joanna Wiszniewska; Jing Wang; Edwina J Popek; Saul Karpen; Lee-Jun C Wong; Fernando Scaglia
Journal:  Mol Genet Metab       Date:  2011-03-11       Impact factor: 4.797

7.  Clinical and molecular features of mitochondrial DNA depletion due to mutations in deoxyguanosine kinase.

Authors:  D P Dimmock; Q Zhang; C Dionisi-Vici; R Carrozzo; J Shieh; L-Y Tang; C Truong; E Schmitt; M Sifry-Platt; S Lucioli; F M Santorelli; C H Ficicioglu; M Rodriguez; K Wierenga; G M Enns; N Longo; M H Lipson; H Vallance; W J Craigen; F Scaglia; L-J Wong
Journal:  Hum Mutat       Date:  2008-02       Impact factor: 4.878

8.  Abnormal neurological features predict poor survival and should preclude liver transplantation in patients with deoxyguanosine kinase deficiency.

Authors:  David P Dimmock; J Kay Dunn; Annette Feigenbaum; Anthony Rupar; Rita Horvath; Peter Freisinger; Bénédicte Mousson de Camaret; Lee-Jun Wong; Fernando Scaglia
Journal:  Liver Transpl       Date:  2008-10       Impact factor: 5.799

9.  Mitochondrial respiratory chain defect: a new etiology for neonatal cholestasis and early liver insufficiency.

Authors:  I Goncalves; D Hermans; D Chretien; P Rustin; A Munnich; J M Saudubray; F Van Hoof; R Reding; J de Ville de Goyet; J B Otte
Journal:  J Hepatol       Date:  1995-09       Impact factor: 25.083

10.  Clinical and molecular features of mitochondrial DNA depletion syndromes.

Authors:  A Spinazzola; F Invernizzi; F Carrara; E Lamantea; A Donati; M Dirocco; I Giordano; M Meznaric-Petrusa; E Baruffini; I Ferrero; M Zeviani
Journal:  J Inherit Metab Dis       Date:  2008-12-27       Impact factor: 4.982

  10 in total
  6 in total

Review 1.  Neonatal hemochromatosis.

Authors:  Amy G Feldman; Peter F Whitington
Journal:  J Clin Exp Hepatol       Date:  2013-11-27

Review 2.  Gastrointestinal and hepatic manifestations of mitochondrial disorders.

Authors:  Shamima Rahman
Journal:  J Inherit Metab Dis       Date:  2013-05-15       Impact factor: 4.982

3.  Mitochondrial hepatopathies: advances in genetics, therapeutic approaches, and outcomes.

Authors:  Way Seah Lee; Ronald J Sokol
Journal:  J Pediatr       Date:  2013-06-28       Impact factor: 4.406

4.  Antenatal manifestations of mitochondrial disorders.

Authors:  Mariana Vide Tavares; Maria João Santos; Ana Patrícia Domingues; João Pratas; Cândida Mendes; Marta Simões; Paulo Moura; Luísa Diogo; Manuela Grazina
Journal:  J Inherit Metab Dis       Date:  2013-01-30       Impact factor: 4.982

5.  A fatal case of mitochondrial DNA depletion syndrome with novel compound heterozygous variants in the deoxyguanosine kinase gene.

Authors:  Weiyuan Fang; Peng Song; Xinbao Xie; Jianshe Wang; Yi Lu; Gang Li; Kuerbanjiang Abuduxikuer
Journal:  Oncotarget       Date:  2017-09-15

6.  Clinical and molecular characterization of three patients with Hepatocerebral form of mitochondrial DNA depletion syndrome: a case series.

Authors:  Ghazale Mahjoub; Parham Habibzadeh; Hassan Dastsooz; Malihe Mirzaei; Arghavan Kavosi; Laila Jamali; Haniyeh Javanmardi; Pegah Katibeh; Mohammad Ali Faghihi; Seyed Alireza Dastgheib
Journal:  BMC Med Genet       Date:  2019-10-29       Impact factor: 2.103

  6 in total

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