Recurrent pericarditis in Myhre syndrome

Am J Med Genet A. 2013 May;161A(5):1164-6. doi: 10.1002/ajmg.a.35892.

Abstract

Myhre syndrome is a rare disorder characterized by pre- and postnatal short stature, brachydactyly, facial dysmorphism (short palpebral fissures, maxillary hypoplasia, prognathism and short philtrum), thick skin, muscular-appearing body build, decreased joint mobility, mixed hearing loss, and cleft lip and palate. Other clinical features include skeletal dysplasia, developmental delay with intellectual disability and/or behavioral disturbance, cardiac defects, cryptorchidism, and bone anomalies. The disease is caused by recently identified SMAD4 mutations. Here we describe a 7-year-old boy with a molecularly proven Myhre syndrome who presented life-threatening recurrent pericarditis and systemic inflammatory symptoms that required treatment with steroid and recombinant interleukin-1 receptor antagonist.

Publication types

  • Case Reports

MeSH terms

  • Child
  • Cryptorchidism / complications*
  • Cryptorchidism / drug therapy
  • Cryptorchidism / genetics
  • Facies
  • Growth Disorders / complications*
  • Growth Disorders / drug therapy
  • Growth Disorders / genetics
  • Hand Deformities, Congenital / complications*
  • Hand Deformities, Congenital / drug therapy
  • Hand Deformities, Congenital / genetics
  • Humans
  • Hypertrophy / complications*
  • Hypertrophy / drug therapy
  • Hypertrophy / genetics
  • Intellectual Disability / complications*
  • Intellectual Disability / drug therapy
  • Intellectual Disability / genetics
  • Interleukin 1 Receptor Antagonist Protein / therapeutic use*
  • Joint Diseases / complications*
  • Joint Diseases / drug therapy
  • Joint Diseases / genetics
  • Male
  • Pericarditis / complications*
  • Pericarditis / drug therapy
  • Pericarditis / genetics
  • Prednisone / therapeutic use*
  • Recurrence
  • Smad4 Protein / genetics*
  • Treatment Outcome

Substances

  • Interleukin 1 Receptor Antagonist Protein
  • Smad4 Protein
  • Prednisone

Supplementary concepts

  • Growth mental deficiency syndrome of Myhre