Goltz syndrome (focal dermal hypoplasia)

  • PORCN gene mutation: expressed in skin, craniofacial, long bones, tooth buds and eyes, protein involved in Wnt signaling, fibroblast proliferation, inhibits adipogenesis and induces osteogenesis, early limb patterning, tooth formation
  • XL-D
  • At birth: vermiculate dermal atrophy/telangiectasias
  • Later: hypo/hyperpigmentation + fat “herniations”
  • “Raspberry-like” papillomas + verrucous surface and fibrovascular core, esp. in anogenital area, lips, larynx, and acral sites
  • Ectrodactyly, asymmetric syndactyly, oligodactyly, long bone reduction defects
  • Eye involvement
  • Ectodermal (nails, hair, teeth, dysmorphic facies)
  • Aplasia cutis congenita, cleft lip/ palate
  • Spine, urinary tract and uterus anomalies
  • Radiographs of lower extremities midportion, osteopathia striata
  • H&E: ↓dermal thickness + fewer adnexal structures
  • Genetic diagnosis/counselling
  • Multidisciplinary care
  • Supportive treatment
  • Telangiectasia: pulsed dye laser 
  • Exophytic papillomas: curettage or photodynamic therapy