Blepharophimosis, corneal vascularization, deafness, and acroosteolysis: a "new" syndrome?

Am J Med Genet A. 2006 Dec 15;140(24):2709-13. doi: 10.1002/ajmg.a.31543.

Abstract

We report on a patient with blepharophimosis who after unsuccessful surgery developed progressive corneal vascularization. The patient had conductive hearing loss, acroosteolysis of the phalanges, arthropathy, loss of subcutaneous fat of the hands, feet and face, and oligospermia. He had had spontaneous pneumothorax four times. We have found no similar case reports in the literature and suggest that this is a new syndrome, which must be differentiated from hereditary mucoepithelial dysplasia, mandibuloacral dysplasia, keratitis-ichthyosis-deafness syndrome, Hajdu-Cheney syndrome, Penttinen syndrome, and mucopolysaccharidoses.

Publication types

  • Case Reports

MeSH terms

  • Acro-Osteolysis / complications*
  • Acro-Osteolysis / genetics
  • Adult
  • Blepharophimosis / complications*
  • Blepharophimosis / genetics
  • Connective Tissue / pathology
  • Corneal Neovascularization / complications*
  • Corneal Neovascularization / genetics
  • Craniofacial Abnormalities / complications
  • Craniofacial Abnormalities / genetics
  • Diagnosis, Differential
  • Ectodermal Dysplasia / complications
  • Ectodermal Dysplasia / genetics
  • Hearing Loss, Conductive / complications*
  • Hearing Loss, Conductive / genetics
  • Humans
  • Male
  • Oligospermia / complications
  • Oligospermia / genetics
  • Syndactyly / complications
  • Syndactyly / genetics
  • Syndrome