| Literature DB >> 30120985 |
Mitsuru Arima1, Shoko Tsukamoto2, Rumi Akiyama2, Kei Nishiyama3, Ri-Ichiro Kohno2, Takashi Tachibana2, Akira Hayashida2, Miwa Murayama2, Toshio Hisatomi2, Kandai Nozu4, Kazumoto Iijima4, Shouichi Ohga3, Koh-Hei Sonoda2.
Abstract
Pierson syndrome, an autosomal recessive disorder caused by a mutation in laminin ß2 (LAMB2) gene, is characterized by congenital nephrotic syndrome and various ocular abnormalities. The ocular findings in Pierson syndrome are not well understood, because the incidence of this syndrome is very rare. We report ocular findings in a 5-month-old boy with Pierson syndrome with a novel mutation in LAMB2. We performed a pupilloplasty for his microcoria. Ophthalmic examinations after surgery revealed that he had cataract, severe retinal degeneration, and high myopia. Optical coherence tomography showed the collapse of retinal layer structures and a marked decrease of choroidal thickness. Immunohistochemistry and electron microscopy examinations revealed abnormal iris differentiation and thinning or defect of basal membranes. These results suggest that the development of the iris, lens, retina, and choroid are affected in this type of mutation.Entities:
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Year: 2018 PMID: 30120985 DOI: 10.1016/j.jaapos.2018.03.016
Source DB: PubMed Journal: J AAPOS ISSN: 1091-8531 Impact factor: 1.220