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A case of Hermansky-Pudlak with dyspnea.

Ali Hossein Samadi TakaldaniNima JavanshirMaryam SalimiMohammad Negaresh
Published in: Oxford medical case reports (2023)
Hermansky-Pudlak syndrome (HPS) is a rare multisystem disorder inherited in an autosomal recessive manner. Its prevalence is 1 in 500 000 to 1 000 000 people worldwide. The cause of this disorder is genetic mutations that lead to defective organelles of lysosomes. In this report, a 49-year-old man is introduced who was referred to the medical center with ocular albinism and recently exacerbated shortness of breath. Imaging showed peripheral reticular opacities, ground-glass opacities of the lungs with subpleural sparing in some regions, and thickening of bronchovascular bundles, which were all in favor of non-specific interstitial pneumonia. This imaging pattern is an unusual finding in a patient with HPS.
Keyphrases
  • high resolution
  • case report
  • risk factors
  • genome wide
  • gene expression
  • intellectual disability
  • copy number
  • fluorescence imaging