Literature DB >> 14736770

Leopard spot retinal pigmentation in infancy indicating a peroxisomal disorder.

C J Lyons1, G Castano, A Q McCormick, D Applegarth.   

Abstract

BACKGROUND: Neonatal adrenoleucodystrophy (NALD) is a rare disorder resulting from abnormal peroxisomal biogenesis. Affected patients present in infancy with developmental delay, hypotonia, and seizures. Blindness and nystagmus are prominent features. The authors suggest a characteristic leopard spot pigmentary pattern in the peripheral retina to be diagnostic.
METHODS: Three patients are reported with this presentation; the characteristic retinal appearance resulted in early diagnosis for one of these.
CONCLUSION: Leopard spot retinopathy in an infant with hypotonia, seizures, developmental delay, with or without dysmorphic features and hearing impairment, is a clue to the diagnosis of NALD.

Entities:  

Mesh:

Year:  2004        PMID: 14736770      PMCID: PMC1772012          DOI: 10.1136/bjo.2003.023010

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


  13 in total

1.  UVEAL EFFUSION. 1. CLINICAL PICTURE.

Authors:  C L SCHEPENS; R J BROCKHURST
Journal:  Arch Ophthalmol       Date:  1963-08

2.  Adrenoleukodystrophy. Preliminary report of a connatal case. Light- and electron microscopical, immunohistochemical and biochemical findings.

Authors:  J Ulrich; N Herschkowitz; P Heitz; T Sigrist; P Baerlocher
Journal:  Acta Neuropathol       Date:  1978-08-07       Impact factor: 17.088

3.  'Leopard spot' retinopathy in Warburg syndrome.

Authors:  R A Barth; R A Pagon; A H Bunt-Milam
Journal:  Ophthalmic Paediatr Genet       Date:  1986-08

4.  Ocular pathologic findings in neonatal adrenoleukodystrophy.

Authors:  B J Glasgow; H H Brown; J B Hannah; R Y Foos
Journal:  Ophthalmology       Date:  1987-08       Impact factor: 12.079

5.  Retinal pigment epithelial abnormalities in leukemic disease.

Authors:  H M Clayman; J T Flynn; K Koch; C Israel
Journal:  Am J Ophthalmol       Date:  1972-09       Impact factor: 5.258

6.  Ocular histopathologic and biochemical studies of the cerebrohepatorenal syndrome (Zellweger's syndrome) and its relationship to neonatal adrenoleukodystrophy.

Authors:  S M Cohen; F R Brown; L Martyn; H W Moser; W Chen; M Kistenmacher; H Punnett; Z C de la Cruz; N R Chan; W R Green
Journal:  Am J Ophthalmol       Date:  1983-10       Impact factor: 5.258

7.  Cerebro-hepato-renal (Zellweger) syndrome and neonatal adrenoleukodystrophy: similarities in phenotype and accumulation of very long chain fatty acids.

Authors:  F R Brown; A J McAdams; J W Cummins; R Konkol; I Singh; A B Moser; H W Moser
Journal:  Johns Hopkins Med J       Date:  1982-12

8.  Adrenoleukodystrophy with disease of the eye and optic nerve.

Authors:  S H Wray; D G Cogan; T Kuwabara; H H Schaumburg; J M Powers
Journal:  Am J Ophthalmol       Date:  1976-09       Impact factor: 5.258

9.  Ocular histopathologic studies of neonatal and childhood adrenoleukodystrophy.

Authors:  S M Cohen; W R Green; Z C de la Cruz; F R Brown; H W Moser; M W Luckenbach; D J Dove; I H Maumenee
Journal:  Am J Ophthalmol       Date:  1983-01       Impact factor: 5.258

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Authors:  H W Moser; A E Moser; I Singh; B P O'Neill
Journal:  Ann Neurol       Date:  1984-12       Impact factor: 10.422

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  2 in total

1.  Leopard-like retinopathy and severe early-onset portal hypertension expand the phenotype of KARS1-related syndrome: a case report.

Authors:  Francesca Peluso; Viviana Palazzo; Giuseppe Indolfi; Francesco Mari; Roberta Pasqualetti; Elena Procopio; Claudia Nesti; Renzo Guerrini; Filippo Santorelli; Sabrina Giglio
Journal:  BMC Med Genomics       Date:  2021-01-21       Impact factor: 3.063

2.  Peroxisome biogenesis disorders.

Authors:  Catherine Argyriou; Maria Daniela D'Agostino; Nancy Braverman
Journal:  Transl Sci Rare Dis       Date:  2016-11-07
  2 in total

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