Literature DB >> 2751974

Ocular complications in homocystinuria--early and late treated.

J P Burke1, M O'Keefe, R Bowell, E R Naughten.   

Abstract

Homocystinuria due to cystathionine-beta-synthetase deficiency is an autosomal recessive disorder of methionine metabolism with an incidence in Ireland of 1 in 52,544 births. Ocular complications in untreated patients include ectopia lentis, secondary glaucoma, optic atrophy, and retinal detachment. There are no characteristic signs or symptoms in infancy, and early detection relies on screening of newborn babies. Nineteen patients with homocystinuria were studied; 14 received dietary treatment and vitamin supplementation starting in the newborn period. Of these, none developed ectopia lentis after a mean follow-up of 8.2 years, compared with a 70% dislocation rate in untreated patients with a similar follow-up period. Ectopia lentis developed and progressed in five patients diagnosed later in life, despite tight biochemical control. The risk of ocular complications in homocystinuria can be substantially reduced in patients started on treatment within six weeks of birth.

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Year:  1989        PMID: 2751974      PMCID: PMC1041762          DOI: 10.1136/bjo.73.6.427

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


  10 in total

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Journal:  Am J Ophthalmol       Date:  1973-03       Impact factor: 5.258

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10.  The natural history of homocystinuria due to cystathionine beta-synthase deficiency.

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  10 in total
  11 in total

1.  Visual outcome and incidence of glaucoma in patients with microspherophakia.

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Journal:  Eye (Lond)       Date:  2014-11-14       Impact factor: 3.775

Review 2.  Molecular pathogenesis and management strategies of ectopia lentis.

Authors:  A Chandra; D Charteris
Journal:  Eye (Lond)       Date:  2014-01-10       Impact factor: 3.775

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Authors:  H Neunhöffer; L Goldammer; V Gravenhorst; N Feltgen; H Hoerauf
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Review 5.  Optic atrophies in metabolic disorders.

Authors:  Marjan Huizing; Brian P Brooks; Yair Anikster
Journal:  Mol Genet Metab       Date:  2005-09-27       Impact factor: 4.797

6.  Ophthalmologic Findings in Patients with Neuro-metabolic Disorders.

Authors:  Narjes Jafari; Karl Golnik; Mansoor Shahriari; Parvaneh Karimzadeh; Sayena Jabbehdari
Journal:  J Ophthalmic Vis Res       Date:  2018 Jan-Mar

7.  A child with Myhre syndrome presenting with corectopia and tetralogy of Fallot.

Authors:  Marianna Alagia; Gerarda Cappuccio; Michele Pinelli; Annalaura Torella; Raffaella Brunetti-Pierri; Francesca Simonelli; Giuseppe Limongelli; Guido Oppido; Vincenzo Nigro; Nicola Brunetti-Pierri
Journal:  Am J Med Genet A       Date:  2017-12-12       Impact factor: 2.802

8.  High Myopia: A Pointer of an Inborn Error of Metabolism.

Authors:  Sanjay Kumar Sahu; Jyoti Ranjan Behera; Jishnu Kr; Moparthi Puramjai; Ramakrushna Gudu; Arun K Dash; Amit R Rup; Sibabratta Patnaik
Journal:  Cureus       Date:  2022-01-04

Review 9.  Homocystinuria and ocular complications - A review.

Authors:  Mehzabeen Rahman; Mohita Sharma; Pragati Aggarwal; Silkee Singla; Neha Jain
Journal:  Indian J Ophthalmol       Date:  2022-07       Impact factor: 2.969

10.  Unilateral and Spontaneous Complete Anterior Dislocation of the Crystalline Lens in a Patient With Homocystinuria.

Authors:  Arezoo Miraftabi; Amin Zand; Kaveh Abri Aghdam
Journal:  Cureus       Date:  2021-04-23
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