Literature DB >> 24012100

Systemic diagnostic testing in patients with apparently isolated uveal coloboma.

Nancy Huynh1, Delphine Blain, Tanya Glaser, E Lauren Doss, Wadih M Zein, David M Lang, Eva H Baker, Suvimol Hill, Carmen C Brewer, Jeffrey B Kopp, Tanya M Bardakjian, Irene H Maumenee, Bronwyn J Bateman, Brian P Brooks.   

Abstract

PURPOSE: To investigate the frequency and types of systemic findings in patients with apparently isolated uveal coloboma.
DESIGN: Cross-sectional observational study.
METHODS: setting: Single-center ophthalmic genetics clinic. study population: Ninety-nine patients with uveal coloboma seen at the National Eye Institute. observational procedure: Results of audiology testing, echocardiogram, brain magnetic resonance imaging, renal ultrasound, and total spine radiographs. main outcome measure: Prevalence of abnormal findings on systemic testing.
RESULTS: Uveal coloboma affected only the anterior segment in 8 patients, only the posterior segment in 23 patients, and both anterior and posterior segments in 68 patients. Best-corrected visual acuity (BCVA) of eyes with coloboma was ≥20/40 in 45% of eyes; 23% of eyes had BCVA of ≤20/400. The majority of patients (74%) had good vision (>20/60) in at least 1 eye. Ten of the 19 patients (53%) who underwent echocardiography had abnormalities, with ventral septal defects being the most prevalent. Abnormal findings were observed in 5 of 72 patients (7%) who had a renal ultrasound and in 5 of 29 patients (17%) who underwent a brain MRI. Audiology testing revealed abnormalities in 13 of 75 patients (17%), and spine radiographs showed anomalies in 10 of 77 patients (13%). Most findings required no acute intervention.
CONCLUSIONS: Although some patients with coloboma had evidence of extraocular abnormalities, the majority of findings on routine clinical examination did not require acute intervention, but some warranted follow-up. Results from the systemic evaluation of patients with coloboma should be interpreted with caution and in view of their clinical context. Published by Elsevier Inc.

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Year:  2013        PMID: 24012100      PMCID: PMC4167417          DOI: 10.1016/j.ajo.2013.06.037

Source DB:  PubMed          Journal:  Am J Ophthalmol        ISSN: 0002-9394            Impact factor:   5.258


  21 in total

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Review 2.  Uveal coloboma: clinical and basic science update.

Authors:  Lan Chang; Delphine Blain; Stefano Bertuzzi; Brian P Brooks
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3.  Visual acuity in children with coloboma: clinical features and a new phenotypic classification system.

Authors:  S J Hornby; S Adolph; C E Gilbert; L Dandona; A Foster
Journal:  Ophthalmology       Date:  2000-03       Impact factor: 12.079

4.  Chorioretinal colobomas in a pediatric population.

Authors:  D R Daufenbach; M S Ruttum; J S Pulido; R V Keech
Journal:  Ophthalmology       Date:  1998-08       Impact factor: 12.079

Review 5.  Unilateral renal agenesis: a systematic review on associated anomalies and renal injury.

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6.  Congenital heart disease in CHARGE association.

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8.  Epidemiology of congenital eye malformations in 131,760 consecutive births.

Authors:  C Stoll; Y Alembik; B Dott; M P Roth
Journal:  Ophthalmic Paediatr Genet       Date:  1992-09

9.  Genitourinary anomalies in the CHARGE association.

Authors:  D C Ragan; A J Casale; R C Rink; M P Cain; D D Weaver
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10.  Predicting visual acuity in children with colobomas involving the optic nerve.

Authors:  T W Olsen; C G Summers; W H Knobloch
Journal:  J Pediatr Ophthalmol Strabismus       Date:  1996 Jan-Feb       Impact factor: 1.402

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  6 in total

1.  Ocular and Systemic Findings in Adults with Uveal Coloboma.

Authors:  Malena Daich Varela; Laryssa A Huryn; Robert B Hufnagel; Wadih M Zein; Delphine Blain; Brian P Brooks
Journal:  Ophthalmology       Date:  2020-05-17       Impact factor: 12.079

2.  EFTUD2 deficiency in vertebrates: Identification of a novel human mutation and generation of a zebrafish model.

Authors:  Brett Deml; Linda M Reis; Sanaa Muheisen; David Bick; Elena V Semina
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3.  A 3-Day-Old Girl Referred From Her Pediatrician for Oral Ulcerations.

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4.  Uveal coloboma: about 3 cases at the University Teaching Hospital, Yaounde, Cameroon.

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Journal:  Pan Afr Med J       Date:  2016-07-08

5.  High-throughput custom capture sequencing identifies novel mutations in coloboma-associated genes: Mutation in DNA-binding domain of retinoic acid receptor beta affects nuclear localization causing ocular coloboma.

Authors:  Vijay K Kalaskar; Ramakrishna P Alur; LeeAnn K Li; James W Thomas; Yuri V Sergeev; Delphine Blain; Robert B Hufnagel; Tiziana Cogliati; Brian P Brooks
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Review 6.  Ocular coloboma-a comprehensive review for the clinician.

Authors:  Gopal Lingam; Alok C Sen; Vijaya Lingam; Muna Bhende; Tapas Ranjan Padhi; Su Xinyi
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  6 in total

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