Literature DB >> 7849740

Molecular genetics of oculocutaneous albinism.

R A Spritz1.   

Abstract

Albinism is a group of genetic disorders characterized by deficient synthesis of melanin pigment. In oculocutaneous albinism (OCA) the pigment deficiency involves the skin, hair, and eyes, whereas in ocular albinism (OA) the defect involves principally the visual system. Type I (tyrosinase-deficient) OCA results from deficient catalytic activity of tyrosinase, which catalyzes at least three steps in the melanin biosynthetic pathway. Type II (tyrosinase-positive) OCA results from abnormalities of the 'P' polypeptide, which may be a melanosomal tyrosine transporter. At least some forms of OA appear to represent mild presentations of types I and II OCA. The causes of several other forms of albinism have not yet been identified. Recent application of molecular genetic techniques to the study of these disorders has led to greatly improved knowledge of their molecular pathogenesis and relationships, and paves the way to improved diagnosis, carrier detection and prenatal diagnosis, and even to eventual treatment.

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Year:  1994        PMID: 7849740     DOI: 10.1093/hmg/3.suppl_1.1469

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  23 in total

1.  Genetic variants in pigmentation genes, pigmentary phenotypes, and risk of skin cancer in Caucasians.

Authors:  Hongmei Nan; Peter Kraft; David J Hunter; Jiali Han
Journal:  Int J Cancer       Date:  2009-08-15       Impact factor: 7.396

Review 2.  Albinism: modern molecular diagnosis.

Authors:  S M Carden; R E Boissy; P J Schoettker; W V Good
Journal:  Br J Ophthalmol       Date:  1998-02       Impact factor: 4.638

3.  A microsatellite map of the pink-eyed dilution (p) deletion complex in mouse chromosome 7.

Authors:  M S Dhar; D K Johnson
Journal:  Mamm Genome       Date:  1997-02       Impact factor: 2.957

4.  Molecular analysis of 36 mutations at the mouse pink-eyed dilution (p) locus.

Authors:  D K Johnson; L J Stubbs; C T Culiat; C S Montgomery; L B Russell; E M Rinchik
Journal:  Genetics       Date:  1995-12       Impact factor: 4.562

5.  Melanoma: Stem cells, sun exposure and hallmarks for carcinogenesis, molecular concepts and future clinical implications.

Authors:  Athanassios Kyrgidis; Thrasivoulos-George Tzellos; Stefanos Triaridis
Journal:  J Carcinog       Date:  2010-04-01

6.  Frequent intragenic deletion of the P gene in Tanzanian patients with type II oculocutaneous albinism (OCA2).

Authors:  R A Spritz; K Fukai; S A Holmes; J Luande
Journal:  Am J Hum Genet       Date:  1995-06       Impact factor: 11.025

Review 7.  What are melanocytes really doing all day long...?

Authors:  P M Plonka; T Passeron; M Brenner; D J Tobin; S Shibahara; A Thomas; A Slominski; A L Kadekaro; D Hershkovitz; E Peters; J J Nordlund; Z Abdel-Malek; K Takeda; R Paus; J P Ortonne; V J Hearing; K U Schallreuter
Journal:  Exp Dermatol       Date:  2009-07-30       Impact factor: 3.960

8.  Genome-wide association study of tanning phenotype in a population of European ancestry.

Authors:  Hongmei Nan; Peter Kraft; Abrar A Qureshi; Qun Guo; Constance Chen; Susan E Hankinson; Frank B Hu; Gilles Thomas; Robert N Hoover; Stephen Chanock; David J Hunter; Jiali Han
Journal:  J Invest Dermatol       Date:  2009-04-02       Impact factor: 8.551

9.  Oculocutaneous albinism type 4 is one of the most common types of albinism in Japan.

Authors:  Katsuhiko Inagaki; Tamio Suzuki; Hiroshi Shimizu; Norihisa Ishii; Yoshinori Umezawa; Joji Tada; Noriaki Kikuchi; Minoru Takata; Kenji Takamori; Mari Kishibe; Michi Tanaka; Yoshinori Miyamura; Shiro Ito; Yasushi Tomita
Journal:  Am J Hum Genet       Date:  2004-02-11       Impact factor: 11.025

10.  Albinism and developmental delay: the need to test for 15q11-q13 deletion.

Authors:  Reem Saadeh; Emily C Lisi; Denise A S Batista; Iain McIntosh; Julie E Hoover-Fong
Journal:  Pediatr Neurol       Date:  2007-10       Impact factor: 3.372

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