Literature DB >> 30120985

Ocular findings in a case of Pierson syndrome with a novel mutation in laminin ß2 gene.

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.
Copyright © 2018 American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved.

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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


  2 in total

1.  Pierson Syndrome Associated with Hypothyroidism and Septic Shock.

Authors:  Areeba Ejaz; Meher B Ali; Fatima Siddiqui; Mashal B Ali; Ammarah Jamal
Journal:  Sultan Qaboos Univ Med J       Date:  2020-12-21

2.  OCULAR MANIFESTATIONS OF PORETTI-BOLTSHAUSER SYNDROME: FINDINGS FROM MULTIMODAL IMAGING AND ELECTROPHYSIOLOGY.

Authors:  Cindy X Cai; Michelle Go; Michael P Kelly; Sandra Holgado; Cynthia A Toth
Journal:  Retin Cases Brief Rep       Date:  2020-03-17
  2 in total

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