Radiologic changes in infancy in McKusick cartilage hair hypoplasia

Am J Med Genet. 1999 Oct 8;86(4):312-5. doi: 10.1002/(sici)1096-8628(19991008)86:4<312::aid-ajmg2>3.0.co;2-8.

Abstract

Cartilage hair hypoplasia (CHH), or metaphyseal dysplasia McKusick type, classically comprises short stature and scant fine hair. In this skeletal dysplasia there is a high incidence of immune deficiency and Hirschsprung disease, as well as a higher rate of malignancy. Clinical findings may be subtle in young children, and radiographic changes may be elusive. We present four children below age 2 in whom the clinical diagnosis of CHH was confirmed radiographically. We emphasize radiologically and clinically discernable anterior angulation of the entire sternum, a sign not previously described in this dysplasia.

Publication types

  • Case Reports

MeSH terms

  • Cartilage / abnormalities*
  • Cartilage / diagnostic imaging
  • Consanguinity
  • Female
  • Genes, Recessive
  • Hair / abnormalities*
  • Humans
  • Immunologic Deficiency Syndromes / genetics
  • Infant
  • Infant, Newborn
  • Male
  • Osteochondrodysplasias / diagnostic imaging*
  • Osteochondrodysplasias / genetics
  • Radiography
  • Syndrome