Literature DB >> 17684749

[Phenotype of the visual system in oculocutaneous and ocular albinism].

B Käsmann-Kellner1, B Seitz.   

Abstract

In spite of albinism being one of the visual impairments which has been known for over a century, it has only been known for a few decades that albinism is correlated to severe cerebral morphological developmental alterations. The increasing knowledge about the role of melanin in the development and orientation of cerebral neurons not only renders more insight into albinism, but also a greater insight in the physiological neuronal and cerebral development in man. Concerning the morphological and visual phenotype there are new clinical findings which enlarge the known spectrum of albinism. In a representative group of 506 persons with oculocutaneous and ocular albinism who are in care at the Department of Ophthalmology at the University of Saarland (UKS), we present a staging of morphological findings of the iris, retinal pigment epithelium and macula, and of the optic nerve head which has been in use for 10 years. Albinism may present with a remarkably mild ocular phenotype and a near to normal functional phenotype. We present correlations between molecular genetic types of albinism, ocular phenotype and visual function. Of great importance concerning later visual acuity is the dysplasia of the optic nerve head (ONH), which is a frequent finding in albinism. The appearance of the ONH should always be included in any clinical description of an albinism patient. It is highly possible that due to a moderate phenotype there are still many patients who have not been diagnosed yet. Visual acuity of 30/20 to 20/20 and no nystagmus do not rule out albinism. In addition, when performing albino VEPs in phenotypically normal children with infantile strabismus, small ONHs, but normal visual acuity and no nystagmus, the classical atypical chiasmal crossing is sometimes found. Therefore, the number of persons having undiagnosed albinism is probably quite high, perhaps there even is a very broad transition zone from normal to albinotic.

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Year:  2007        PMID: 17684749     DOI: 10.1007/s00347-007-1571-4

Source DB:  PubMed          Journal:  Ophthalmologe        ISSN: 0941-293X            Impact factor:   1.059


  28 in total

1.  The molecular basis of oculocutaneous albinism type 1 (OCA1): sorting failure and degradation of mutant tyrosinases results in a lack of pigmentation.

Authors:  K Toyofuku; I Wada; R A Spritz; V J Hearing
Journal:  Biochem J       Date:  2001-04-15       Impact factor: 3.857

2.  Positive angle kappa in albinism.

Authors:  C Gail Summers; Richard A King; Kimberly S Merrill; Jane D Lavoie
Journal:  Am J Ophthalmol       Date:  2004-12       Impact factor: 5.258

3.  P gene mutations associated with oculocutaneous albinism type II (OCA2).

Authors:  William S Oetting; Sarah Savage Garrett; Marcia Brott; Richard A King
Journal:  Hum Mutat       Date:  2005-03       Impact factor: 4.878

4.  Spectrum of foveal development in albinism detected with optical coherence tomography.

Authors:  Patti S Harvey; Richard A King; C Gail Summers
Journal:  J AAPOS       Date:  2006-06       Impact factor: 1.220

5.  Might the refractive state in oculocutaneous albino patients be a clue for distinguishing between tyrosinase-positive and tyrosinase-negative forms of oculocutaneous albinism?

Authors:  B Käsmann; K W Ruprecht
Journal:  Ger J Ophthalmol       Date:  1996-11

6.  Heterologous expression of tyrosinase recapitulates the misprocessing and mistrafficking in oculocutaneous albinism type 2: effects of altering intracellular pH and pink-eyed dilution gene expression.

Authors:  Li Ni-Komatsu; Seth J Orlow
Journal:  Exp Eye Res       Date:  2005-09-30       Impact factor: 3.467

7.  Correlation between rod photoreceptor numbers and levels of ocular pigmentation.

Authors:  Philippe Donatien; Glen Jeffery
Journal:  Invest Ophthalmol Vis Sci       Date:  2002-04       Impact factor: 4.799

Review 8.  Oculocutaneous albinism type 1: the last 100 years.

Authors:  William S Oetting; James P Fryer; Sabitha Shriram; Richard A King
Journal:  Pigment Cell Res       Date:  2003-06

9.  Diagnostic DNA testing for X-linked ocular albinism (OA1) with a hierarchical mutation screening protocol.

Authors:  M Hegde; R A Lewis; C S Richards
Journal:  Genet Test       Date:  2002

10.  Organization of the visual cortex in human albinism.

Authors:  Michael B Hoffmann; David J Tolhurst; Anthony T Moore; Antony B Morland
Journal:  J Neurosci       Date:  2003-10-01       Impact factor: 6.167

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  7 in total

1.  [Hermansky-Pudlak syndrome].

Authors:  A Atili; J Lübke; M Shoukier; M P Schittkowski
Journal:  Ophthalmologe       Date:  2013-02       Impact factor: 1.059

2.  Clinical Insights Into Foveal Morphology in Albinism.

Authors:  Brandon K McCafferty; Melissa A Wilk; John T McAllister; Kimberly E Stepien; Adam M Dubis; Murray H Brilliant; Jennifer L Anderson; Joseph Carroll; C Gail Summers
Journal:  J Pediatr Ophthalmol Strabismus       Date:  2015 May-Jun       Impact factor: 1.402

3.  [Selected aspects of pediatric ophthalmology for non-pediatric ophthalmologists. Part 2: The odd looking optic nerve head in infants].

Authors:  B Käsmann-Kellner; B Seitz
Journal:  Ophthalmologe       Date:  2012-06       Impact factor: 1.059

4.  [Selected aspects of pediatric ophthalmology for the non-pediatric ophthalmologist. Part 1: Basic knowledge and essential diagnostics to identify the need for specialized action].

Authors:  B Käsmann-Kellner; B Seitz
Journal:  Ophthalmologe       Date:  2012-02       Impact factor: 1.059

Review 5.  [Oculocutaneous and ocular albinism].

Authors:  A S Kubasch; M Meurer
Journal:  Hautarzt       Date:  2017-11       Impact factor: 0.751

6.  Albinism: particular attention to the ocular motor system.

Authors:  Richard W Hertle
Journal:  Middle East Afr J Ophthalmol       Date:  2013 Jul-Sep

Review 7.  Ocular findings and strabismus surgery outcomes in Chinese children with Angelman syndrome: Three case reports.

Authors:  Haiyun Ye; Xiaoping Lan; Qingyu Liu; Yidan Zhang; Siying Wang; Ce Zheng; Yue Di; Tong Qiao
Journal:  Medicine (Baltimore)       Date:  2019-12       Impact factor: 1.889

  7 in total

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