A syndrome involving intrauterine growth retardation, microcephaly, cerebellar hypoplasia, B lymphocyte deficiency, and progressive pancytopenia

Pediatrics. 2000 Mar;105(3):E39. doi: 10.1542/peds.105.3.e39.

Abstract

We report a new complex syndrome involving profound failure to thrive with severe intrauterine growth retardation, cerebellar abnormalities, microcephaly, a complete lack of B lymphocyte development, and secondary, progressive marrow aplasia. B cell differentiation was found to be blocked at the pro-B cell stage. Although not strictly proven, a genetic origin is likely, according to similar cases reported in the literature. Three candidate genes, PAX5, encoding B cell-specific activator protein, a factor involved in B cell lineage commitment, stromal cell-derived factor 1, and CXCR4, encoding a chemokine and its receptor, respectively, were thought to be responsible for this disease, given the similarity between the phenotype of the corresponding knock-out mice and the clinical features of the patient. However, the genomic DNA sequences of these 3 genes were normal, and normal amounts of stromal cell-derived factor 1 and CXCR4 were present. These data strongly suggest that another molecule is involved in early B cell differentiation, hematopoiesis, and cerebellar development in humans.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Agammaglobulinemia / congenital
  • Agammaglobulinemia / genetics
  • B-Lymphocytes / cytology
  • Cell Differentiation
  • Cerebellum / abnormalities*
  • Chemokine CXCL12
  • Chemokines, CXC / blood
  • Chemokines, CXC / genetics
  • DNA-Binding Proteins*
  • Female
  • Fetal Growth Retardation* / genetics
  • Humans
  • Infant, Newborn
  • Microcephaly* / genetics
  • PAX5 Transcription Factor
  • Pancytopenia / congenital*
  • Pancytopenia / genetics
  • Proteins / genetics
  • Receptors, CXCR4 / genetics
  • Syndrome
  • Transcription Factors*

Substances

  • CXCL12 protein, human
  • Chemokine CXCL12
  • Chemokines, CXC
  • DNA-Binding Proteins
  • PAX5 Transcription Factor
  • PAX5 protein, human
  • Proteins
  • Receptors, CXCR4
  • Transcription Factors