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Severe congenital corneal staphyloma in a case with polydactyly Tomoko Haruki 1 , Fumie Ohtani 1 , Chizu Touge 1 , Yusuke Saeki 1 , Ryoko Ishikura 1 , Yoshitsugu Inoue 1 , Hiroko Katano 2 1Dept of Ophthalmol and Vis Sci,Tottori Univ Fac of Med 2Dept of Ophthalmol,Matsue City Hosp pp.929-932
Published Date 2010/6/15
DOI https://doi.org/10.11477/mf.1410103222
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Abstract. Background:Polydactyly is a congenital malformation that starts at about 6 weeks of gestation. Purpose:To report a case of progressive staphyloma associated with polydactyly. Case:A 4-day-old male infant presented with proptosis and cloudy cornea in his left eye. He was born of uneventful pregnancy with birthweight of 2,864 g. Findings:The left eye showed proptosis and clouded cornea with neovascularization. Polydactyly was present in the right hand. Magnetic resonance imaging(MRI)showed no tumorous lesion in the eye when performed at the age of 4 months. Proptosis progressed further and the left eye was eviscerated at 5 months. The iris was adherent to the posterior surface of the cornea. Corneal epithelium was keratinized and contained melanin granules. The cornea showed no Bowman membrane,Descemet membrane or endothelial cells. Conclusion:Presence of polydactyly suggests that corneal staphyloma in this patient is an instance of severe mesenchymal dysgenesis of anterior segment that develops by disorder of neural crest migration at 5 to 7th week of gestation.


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電子版ISSN 1882-1308 印刷版ISSN 0370-5579 医学書院

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