Literature DB >> 12408187

A secondary respiratory chain defect in a patient with Fanconi-Bickel syndrome.

M H Odièvre1, A Lombès, P Dessemme, R Santer, M Brivet, B Chevallier, B Lagardère, M Odièvre.   

Abstract

A North African boy, the son of consanguineous parents, presented at 8 years of age with hypophosphataemic rickets due to De Toni-Debré-Fanconi syndrome. Hepatomegaly and abnormalities of carbohydrate metabolism were suggestive of Fanconi-Bickel syndrome. This was confirmed by the detection of a mutation within GLUT2, the gene encoding the liver-type facilitative glucose transporter. The study of the respiratory chain revealed a deficiency of complexes I, III and IV in muscle. Mechanisms responsible for an impairment ofmitochondrial function, which we interpret as a secondary phenomenon, are discussed.

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Year:  2002        PMID: 12408187     DOI: 10.1023/a:1020147716990

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  19 in total

1.  Mitochondrial activity in Pompe's disease.

Authors:  M A Selak; J P de Chadarevian; J J Melvin; W D Grover; L Salganicoff; E M Kaye
Journal:  Pediatr Neurol       Date:  2000-07       Impact factor: 3.372

2.  [Chronic aminoaciduria (amino acid diabetes or nephrotic-glucosuric dwarfism) in glycogen storage and cystine disease].

Authors:  G FANCONI; H BICKEL
Journal:  Helv Paediatr Acta       Date:  1949-11

3.  Hereditary mitochondrial myopathy with lactic acidemia, a De Toni-Fanconi-Debré syndrome, and a defective respiratory chain in voluntary striated muscles.

Authors:  J P Van Biervliet; L Bruinvis; D Ketting; P K De Bree; C Van der Heiden; S K Wadman
Journal:  Pediatr Res       Date:  1977-10       Impact factor: 3.756

4.  Glycogen storage disease, Fanconi nephropathy, abnormal galactose metabolism and mitochondrial myopathy.

Authors:  H Hurvitz; O N Elpeleg; V Barash; E Kerem; R M Reifen; W Ruitenbeek; C Mor; D Branski
Journal:  Eur J Pediatr       Date:  1989-10       Impact factor: 3.183

5.  Mitochondrial cytopathy with lactic acidosis, carnitine deficiency and DeToni-Fanconi-Debré syndrome.

Authors:  A Kitano; S Nishiyama; T Miike; S Hattori; Y Ohtani; I Matsuda
Journal:  Brain Dev       Date:  1986       Impact factor: 1.961

6.  The mutation spectrum of the facilitative glucose transporter gene SLC2A2 (GLUT2) in patients with Fanconi-Bickel syndrome.

Authors:  René Santer; Sebastian Groth; Martina Kinner; Anja Dombrowski; Gerard T Berry; Johannes Brodehl; James V Leonard; Shimon Moses; Svante Norgren; Flemming Skovby; Reinhard Schneppenheim; Beat Steinmann; Jürgen Schaub
Journal:  Hum Genet       Date:  2001-11-17       Impact factor: 4.132

7.  Mutations in GLUT2, the gene for the liver-type glucose transporter, in patients with Fanconi-Bickel syndrome.

Authors:  R Santer; R Schneppenheim; A Dombrowski; H Götze; B Steinmann; J Schaub
Journal:  Nat Genet       Date:  1997-11       Impact factor: 38.330

Review 8.  Glucose transporters: structure, function and consequences of deficiency.

Authors:  G K Brown
Journal:  J Inherit Metab Dis       Date:  2000-05       Impact factor: 4.982

Review 9.  Fanconi-Bickel syndrome--the original patient and his natural history, historical steps leading to the primary defect, and a review of the literature.

Authors:  R Santer; R Schneppenheim; D Suter; J Schaub; B Steinmann
Journal:  Eur J Pediatr       Date:  1998-10       Impact factor: 3.183

10.  de Toni-Fanconi-Debré syndrome with Leigh syndrome revealing severe muscle cytochrome c oxidase deficiency.

Authors:  H Ogier; A Lombes; H R Scholte; B T Poll-The; M Fardeau; J Alcardi; B Vignes; P Niaudet; J M Saudubray
Journal:  J Pediatr       Date:  1988-05       Impact factor: 4.406

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  4 in total

1.  Phenotypic variability in patients with fanconi-bickel syndrome with identical mutations.

Authors:  Elena Fridman; Avraham Zeharia; Tal Markus-Eidlitz; Yishai Haimi Cohen
Journal:  JIMD Rep       Date:  2014-04-10

2.  Functional and structural analysis of rare SLC2A2 variants associated with Fanconi-Bickel syndrome and metabolic traits.

Authors:  Osatohanmwen J Enogieru; Peter M U Ung; Sook Wah Yee; Avner Schlessinger; Kathleen M Giacomini
Journal:  Hum Mutat       Date:  2019-04-25       Impact factor: 4.878

3.  Fanconi-Bickel syndrome in two Palestinian children: marked phenotypic variability with identical mutation.

Authors:  Imad Mohammad Dweikat; Issa Shaher Alawneh; Sami Fares Bahar; Mutaz Idrees Sultan
Journal:  BMC Res Notes       Date:  2016-08-04

4.  Pyruvate carboxylase deficiency: An underestimated cause of lactic acidosis.

Authors:  F Habarou; A Brassier; M Rio; D Chrétien; S Monnot; V Barbier; R Barouki; J P Bonnefont; N Boddaert; B Chadefaux-Vekemans; L Le Moyec; J Bastin; C Ottolenghi; P de Lonlay
Journal:  Mol Genet Metab Rep       Date:  2014-11-28
  4 in total

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