| Occurrence and pattern of ocular disease in children with cholestatic disorders. | |
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MedLine Citation:
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PMID: 20384607 Owner: NLM Status: In-Data-Review |
Abstract/OtherAbstract:
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Purpose: To describe visual function and ocular manifestations in patients with onset of cholestasis during the neonatal period. Methods: Patients with neonatal cholestasis, either transitory or chronic, who came for assessment to our tertiary referral centre were included in a cross-sectional study and underwent ophthalmological examinations including fundus photography. A total of 57 patients (24 girls and 33 boys), aged 0.4-18.0 years, were included. Of these, 28 patients had biliary atresia, 11 had Alagille's syndrome, five had progressive familiar intrahepatic cholestasis and nine had different disorders such as pituitary insufficiency, alpha-1-antitrypsin deficiency, mitochondriopathy, congenital infections or cholestasis caused by unknown reasons. Results: Visual dysfunction and/or one or several ocular manifestations occurred in 39 out of 57 patients. Major ocular malformations occurred in five patients. Out of three patients with biliary atresia, one patient had severe visual impairment caused by microphthalmia and chorioretinal coloboma, one patient with Cat Eye syndrome had bilateral uveochorioretinal coloboma and one patient had Rieger's anomaly. Two patients, both with pituitary deficiency and transient cholestasis, had severe unilateral visual impairment caused by optic nerve hypoplasia. Conclusion: The majority (68%) of the patients with cholestasis had ocular manifestations. Although the severity of ocular complications varied with diagnosis, and was most apparent among patients with biliary atresia or pituitary deficiency, no conclusion can be drawn regarding the connections between these conditions from the present study. Nevertheless, ocular assessment is important for diagnostic purposes and for early intervention in patients with cholestasis. |
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Authors:
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Kristina Teär Fahnehjelm; Björn Fischler; Lene Martin; Antal Nemeth |
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Publication Detail:
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Type: Journal Article |
Journal Detail:
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Title: Acta ophthalmologica Volume: 89 ISSN: 1755-3768 ISO Abbreviation: Acta Ophthalmol Publication Date: 2011 Mar |
Date Detail:
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Created Date: 2011-02-25 Completed Date: - Revised Date: - |
Medline Journal Info:
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Nlm Unique ID: 101468102 Medline TA: Acta Ophthalmol Country: England |
Other Details:
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Languages: eng Pagination: 143-50 Citation Subset: IM |
Copyright Information:
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© 2010 The Authors. Journal compilation © 2010 Acta Ophthalmol. |
Affiliation:
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Department of Clinical Neuroscience, Karolinska Institutet, Stockholm, Sweden Department of Paediatric Ophthalmology and Strabismus, St Erik Eye Hospital, Karolinska University Hospital, Huddinge, Stockholm, Sweden Department of Clinical Science, Intervention and Technology Clintec, Karolinska Institutet, Stockholm, Sweden Department of Paediatrics, Karolinska University Hospital, Huddinge, Stockholm, Sweden. |
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From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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