Milder ocular findings in Hermansky-Pudlak syndrome type 3 compared with Hermansky-Pudlak syndrome type 1

Ophthalmology. 2004 Aug;111(8):1599-603. doi: 10.1016/j.ophtha.2003.12.058.

Abstract

Purpose: To compare clinically 2 different subtypes of Hermansky-Pudlak syndrome (HPS), type 1 (HPS-1) and type 3 (HPS-3).

Design: Cross-sectional study of a series of patients.

Participants: Sixteen patients with HPS-1 and 14 patients with HPS-3 were studied.

Methods: Complete eye examination, including best-corrected visual acuity and photographs and photographic grading of iris transillumination and macular transparency using a previously established grading system.

Results: Snellen visual acuity was 20/160-2 in the HPS-1 group and 20/125+2 in the HPS-3 group (P = 0.017). Iris grading was statistically significant for less translucence in the HPS-3 patients. The HPS-3 patients also tended to have less transparent maculas, but the difference was not statistically significant.

Conclusions: Patients with HPS-3 have less severe ophthalmic manifestations than patients with HPS-1. Ophthalmologists treating patients with albinism should consider HPS in their differential diagnosis even in the case of mild iris and macular hypopigmentation.

Publication types

  • Comparative Study

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Carrier Proteins / genetics*
  • Child
  • Child, Preschool
  • Cross-Sectional Studies
  • Hermanski-Pudlak Syndrome / classification*
  • Hermanski-Pudlak Syndrome / genetics
  • Humans
  • Intracellular Signaling Peptides and Proteins
  • Iris / pathology*
  • Membrane Proteins / genetics*
  • Middle Aged
  • Phenotype
  • Photography
  • Retina / pathology*
  • Visual Acuity

Substances

  • Carrier Proteins
  • HPS1 protein, human
  • HPS3 protein, human
  • Intracellular Signaling Peptides and Proteins
  • Membrane Proteins