Haematological characteristics of MYH9 disorders due to MYH9 R702 mutations

Eur J Haematol. 2007 Mar;78(3):220-6. doi: 10.1111/j.1600-0609.2006.00806.x. Epub 2007 Jan 16.

Abstract

Objective: MYH9 disorders are characterised by giant platelets, thrombocytopenia, and Döhle body-like cytoplasmic granulocyte inclusion bodies that result from mutations in MYH9, the gene for non-muscle myosin heavy chain-IIA (NMMHC-IIA). MYH9 R702 mutations are highly associated with Alport manifestations and result in Epstein syndrome. The aim of our study was to determine the haematological characteristics of MYH9 disorders as a result of R702 mutations to aid in making a proper diagnosis.

Patients and methods: Platelet size of patients with MYH9 disorders was determined as platelet diameter by microscopic observation of 200 platelets on stained peripheral blood smears. Double in situ hybridisation using a biotinylated oligo(dT) probe and immunofluorescence analysis of neutrophil NMMHC-IIA was performed on peripheral blood smears.

Results: Patients carrying R702 mutations had significantly larger platelets than those with other MYH9 mutations. Although granulocyte inclusion bodies were mostly invisible on stained blood smears, immunofluorescence analysis for NMMHC-IIA showed an abnormal type II localisation in all neutrophils. We first showed that poly(A)+ RNA coincided with accumulated NMMHC-IIA at inclusion bodies in patients with MYH9 disorders. However, no condensation of poly(A)+ RNA at inclusion bodies was observed in patients with R702 mutations.

Conclusion: Our study shows that R702 mutations result in especially large platelets and inclusion bodies being faint and mostly invisible on conventionally stained blood smears. We further demonstrated that poly(A)+ RNA content but not NMMHC-IIA accumulation is responsible for the morphological appearance/stainability of inclusion bodies on stained blood smears and the amount of poly(A)+ RNA is decreased in those with R702 mutations.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Arginine / genetics*
  • Arginine / metabolism
  • Blood Platelet Disorders / genetics
  • Blood Platelet Disorders / pathology
  • Blood Platelets / pathology*
  • Cell Size
  • Child
  • Child, Preschool
  • Female
  • Humans
  • Inclusion Bodies / metabolism
  • Infant
  • Male
  • Molecular Motor Proteins / genetics*
  • Molecular Motor Proteins / metabolism*
  • Mutation
  • Myosin Heavy Chains / genetics*
  • Myosin Heavy Chains / metabolism*
  • Neutrophils / metabolism
  • Neutrophils / pathology
  • Poly A / genetics
  • Thrombocytosis / genetics*
  • Thrombocytosis / pathology*

Substances

  • MYH9 protein, human
  • Molecular Motor Proteins
  • Poly A
  • Arginine
  • Myosin Heavy Chains