skip to main content
OSTI.GOV title logo U.S. Department of Energy
Office of Scientific and Technical Information

Title: Two novel mutations in the glycine-rich region of human PAX6 gene: Implications for an association of cataracts and anosmia with aniridia

Journal Article · · American Journal of Human Genetics
OSTI ID:134257
; ; ;  [1]
  1. Univ. of Texas M.D. Anderson Cancer Center, Houston, TX (United States)

Aniridia (iris hyplasia) is a autosomal dominant congenital disorder of the eye. Mutations in the human aniridia (PAX6) gene have now been identified in many patients from various ethnic groups. In the present study we describe new mutations in this gene. Out of four mutations found, three were novel mutations; the fourth one is identical to the previously reported mutations (C{yields}T transition at nt 240). The three novel mutations analyzed were in the glycine-rich region (two) and in the proline/serine/threonine-rich (PST) region (one). Previously no mutations were reported for the glycine-rich region in humans. One of the mutations found in this region is associated with cataracts in an aniridia family. The other splice mutation found in the PST domain is associated with anosmia (lack of sensation to smell) in a sporadic aniridia case. Two of the mutations presented here, one in the glycine-rich region and the other in the PST domain, were not detected by SSCR. These mutations could be detected by using MDE gel and heteroduplex information. All mutations found in the present study are similar in that 32 of 33 PAX6 mutations result in protein truncation and haploinsufficiency.

OSTI ID:
134257
Report Number(s):
CONF-941009-; ISSN 0002-9297; TRN: 95:005313-0993
Journal Information:
American Journal of Human Genetics, Vol. 55, Issue Suppl.3; Conference: 44. annual meeting of the American Society of Human Genetics, Montreal (Canada), 18-22 Oct 1994; Other Information: PBD: Sep 1994
Country of Publication:
United States
Language:
English

Similar Records

Paired box mutations in familial and sporadic aniridia predicts truncated aniridia proteins
Journal Article · Sun May 01 00:00:00 EDT 1994 · American Journal of Human Genetics; (United States) · OSTI ID:134257

Mutation of the PAX6 gene in a sporadic patient with atypical aniridia
Journal Article · Thu Sep 01 00:00:00 EDT 1994 · American Journal of Human Genetics · OSTI ID:134257

Mutation of the PAX6 gene in patients with autosomal dominant keratitis
Journal Article · Fri Sep 01 00:00:00 EDT 1995 · American Journal of Human Genetics · OSTI ID:134257