Literature DB >> 10340975

Ocular ochronosis in alkaptonuria patients carrying mutations in the homogentisate 1,2-dioxygenase gene.

U Felbor1, Y Mutsch, F Grehn, C R Müller, W Kress.   

Abstract

AIMS: To assess the involvement of the recently identified human homogentisate 1,2-dioxygenase gene (HGO) in alkaptonuria (AKU) in two unrelated patients with ochronosis of the conjunctiva, sclera, and cornea.
METHODS: A mutation screen of the entire coding region of the HGO gene was performed using single stranded conformational analysis after polymerase chain reaction with oligonucleotide primers flanking all 14 exons of the HGO gene. Fragments showing aberrant mobility were directly sequenced.
RESULTS: Two homozygous missense mutations, L25P and M368V, were identified, each of which leads to the replacement of a highly conserved amino acid in the HGO protein.
CONCLUSIONS: The authors describe a novel mutation, L25P, in the German population and bring to 18 the total number of known HGO mutations.

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Year:  1999        PMID: 10340975      PMCID: PMC1723057          DOI: 10.1136/bjo.83.6.680

Source DB:  PubMed          Journal:  Br J Ophthalmol        ISSN: 0007-1161            Impact factor:   4.638


  19 in total

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Journal:  Proc Natl Acad Sci U S A       Date:  1989-04       Impact factor: 11.205

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Review 6.  New concepts in the biology and biochemistry of ascorbic acid.

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Journal:  N Engl J Med       Date:  1986-04-03       Impact factor: 91.245

7.  The molecular basis of alkaptonuria.

Authors:  J M Fernández-Cañón; B Granadino; D Beltrán-Valero de Bernabé; M Renedo; E Fernández-Ruiz; M A Peñalva; S Rodríguez de Córdoba
Journal:  Nat Genet       Date:  1996-09       Impact factor: 38.330

8.  Molecular defects in alkaptonuria.

Authors:  A Gehrig; S R Schmidt; C R Müller; S Srsen; K Srsnova; W Kress
Journal:  Cytogenet Cell Genet       Date:  1997

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Journal:  Am J Med Genet       Date:  1978

10.  Fungal metabolic model for human type I hereditary tyrosinaemia.

Authors:  J M Fernández-Cañón; M A Peñalva
Journal:  Proc Natl Acad Sci U S A       Date:  1995-09-26       Impact factor: 11.205

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  8 in total

1.  [A 69-year-old patient with brown-black pigmentation of the sclera].

Authors:  D Schmidt
Journal:  Ophthalmologe       Date:  2011-12       Impact factor: 1.059

2.  An update on molecular genetics of Alkaptonuria (AKU).

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Journal:  J Inherit Metab Dis       Date:  2011-07-01       Impact factor: 4.982

3.  High frequency of alkaptonuria in Slovakia: evidence for the appearance of multiple mutations in HGO involving different mutational hot spots.

Authors:  A Zatková; D B de Bernabé; H Poláková; M Zvarík; E Feráková; V Bosák; V Ferák; L Kádasi; S R de Córdoba
Journal:  Am J Hum Genet       Date:  2000-10-02       Impact factor: 11.025

4.  Mutation spectrum of homogentisic acid oxidase (HGD) in alkaptonuria.

Authors:  Thierry Vilboux; Michael Kayser; Wendy Introne; Pim Suwannarat; Isa Bernardini; Roxanne Fischer; Kevin O'Brien; Robert Kleta; Marjan Huizing; William A Gahl
Journal:  Hum Mutat       Date:  2009-12       Impact factor: 4.878

5.  A late and difficult diagnosis of ochronosis.

Authors:  Laura Groseanu; Rodica Marinescu; Dan Laptoiun; Iozefina Botezatu; Florica Staniceanu; Sabina Zurac; Ruxandra lonescu
Journal:  J Med Life       Date:  2010 Oct-Dec

Review 6.  On the ocular findings in ochronosis: a systematic review of literature.

Authors:  Moritz Lindner; Thomas Bertelmann
Journal:  BMC Ophthalmol       Date:  2014-01-30       Impact factor: 2.209

7.  In Vivo Confocal Microscopy and Anterior Segment Optic Coherence Tomography Findings in Ocular Ochronosis.

Authors:  Elif Demirkilinc Biler; Suzan Guven Yilmaz; Melis Palamar; Pedram Hamrah; Afsun Sahin
Journal:  Case Rep Ophthalmol Med       Date:  2015-12-15

8.  Alkaptonuria: A case report.

Authors:  Nirupama Damarla; Prathima Linga; Mallika Goyal; Sanjay Reddy Tadisina; G Satyanarayana Reddy; Hymavathi Bommisetti
Journal:  Indian J Ophthalmol       Date:  2017-06       Impact factor: 1.848

  8 in total

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