A transient myelodysplastic/myeloproliferative neoplasm in a patient with cardio-facio-cutaneous syndrome and a germline BRAF mutation

Am J Med Genet A. 2013 Oct;161A(10):2600-3. doi: 10.1002/ajmg.a.36107. Epub 2013 Aug 15.

Abstract

A male infant, born at 32 weeks gestation by cesarean because of hydrops fetalis, presented with multiple anomalies, such as sparse and curly scalp hair, absent eyebrows, frontal bossing, an atrial septal defect, pulmonary artery stenosis, and whole myocardial thickening. He was clinically diagnosed with cardio-facio-cutaneous (CFC) syndrome, and was confirmed to have a germline V-raf murine sarcoma viral oncogene homologue B1 (BRAF) c.721 A>C mutation. At 1 month of age, he presented with a transient myelodysplastic/myeloproliferative neoplasm (MDS/MPN), which improved within a month without the administration of antineoplastic agents. This is the first report of CFC syndrome with MDS/MPN. The coexistence of MDS/MPN may be related to this BRAF c.721 A>C mutation.

Keywords: BRAF; RAS/MAPK syndromes; cardio-facio-cutaneous syndrome; juvenile myelomonocytic leukemia; myelodysplastic/myeloproliferative neoplasm.

Publication types

  • Case Reports

MeSH terms

  • Amino Acid Substitution
  • Codon
  • Down Syndrome / complications*
  • Down Syndrome / diagnosis
  • Ectodermal Dysplasia / complications*
  • Ectodermal Dysplasia / diagnosis
  • Ectodermal Dysplasia / genetics*
  • Facies
  • Failure to Thrive / complications*
  • Failure to Thrive / diagnosis
  • Failure to Thrive / genetics*
  • Genotype
  • Germ-Line Mutation*
  • Heart Defects, Congenital / complications*
  • Heart Defects, Congenital / diagnosis
  • Heart Defects, Congenital / genetics*
  • Humans
  • Infant, Newborn
  • Leukemoid Reaction / complications*
  • Leukemoid Reaction / diagnosis
  • Male
  • Phenotype
  • Proto-Oncogene Proteins B-raf / genetics*

Substances

  • Codon
  • Proto-Oncogene Proteins B-raf

Supplementary concepts

  • Cardiofaciocutaneous syndrome
  • Myeloproliferative Syndrome, Transient