Document Detail


Severe Peters Plus syndrome-like phenotype with anterior eye staphyloma and hypoplastic left heart syndrome: proposal of a new syndrome.
MedLine Citation:
PMID:  20584037     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Peters Plus syndrome is a very rare autosomal recessive condition characterized by ocular defects (typically Peters anomaly) and other systemic major/minor anomalies. Mutations in the B3GALTL gene encoding beta 1,3-glucosyltransferase have been found in virtually all patients with typical Peters Plus syndrome. We report on a female patient with unusually severe manifestations of Peters Plus syndrome, including anterior eye staphyloma, cleft lip and palate, and hypoplastic left heart syndrome (HLHS). Analysis of the B3GALTL gene revealed no mutation in the patient. To our knowledge, HLHS has not previously been reported in Peters Plus syndrome so far, and anterior staphyloma, a most severe defect of the anterior eye chamber, is also apparently rare in the syndrome. Our patient might represent a new syndrome of severe Peters Plus syndrome-like phenotype with anterior eye staphyloma and HLHS.
Authors:
Reiko Shimizu; Ryota Saito; Kenji Hoshino; Kiyoshi Ogawa; Takashi Negishi; Jiro Nishimura; Norimasa Mitsui; Makiko Osawa; Hirofumi Ohashi
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, Non-U.S. Gov't     Date:  2010-06-24
Journal Detail:
Title:  Congenital anomalies     Volume:  50     ISSN:  1741-4520     ISO Abbreviation:  Congenit Anom (Kyoto)     Publication Date:  2010 Sep 
Date Detail:
Created Date:  2010-08-24     Completed Date:  2010-12-20     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9306292     Medline TA:  Congenit Anom (Kyoto)     Country:  Japan    
Other Details:
Languages:  eng     Pagination:  197-9     Citation Subset:  IM    
Affiliation:
Department of Pediatrics, School of Medicine, Tokyo Women's Medical University, 8-1 Kawada-cho, Shinjuku-ku, Tokyo, Japan. rmuto@ped.twmu.ac.jp
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MeSH Terms
Descriptor/Qualifier:
Abnormalities, Multiple / diagnosis*
Cornea / abnormalities
Fatal Outcome
Female
Galactosyltransferases / genetics
Glucosyltransferases / genetics
Growth Disorders / diagnosis
Humans
Hypoplastic Left Heart Syndrome / diagnosis*
Infant, Newborn
Limb Deformities, Congenital / diagnosis
Chemical
Reg. No./Substance:
EC 2.4.1.-/B3GALTL protein, human; EC 2.4.1.-/Galactosyltransferases; EC 2.4.1.-/Glucosyltransferases; EC 2.4.1.-/beta3Glc-T glucosyltransferase, human

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