A high prevalence of neutrophil-specific antibodies in ELANE-mutated severe congenital neutropenia

Pediatr Blood Cancer. 2023 Apr;70(4):e30247. doi: 10.1002/pbc.30247. Epub 2023 Feb 3.

Abstract

An assay for neutrophil-specific antibodies is frequently used in the workup of chronic severe neutropenia and is suggestive of autoimmune, or sporadically alloimmune neutropenia, rather than severe congenital neutropenia (SCN). We analyzed a neutropenia consortium database for the outcomes of antibody testing initiated before receiving genetic diagnosis in Polish SCN cohort. Test results, performed in a single reference laboratory, were available for 14 patients with ELANE-mutated SCN or cyclic neutropenia, and were frequently positive (36%). We note that the trigger for genetic studies in severe neutropenia should not be affected by antibody-positivity and should be clinically driven.

Keywords: ELANE; autoimmune neutropenia; neutropenia; neutrophil-specific antibodies; severe congenital neutropenia.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Autoantibodies
  • Congenital Bone Marrow Failure Syndromes
  • Humans
  • Leukocyte Elastase / genetics
  • Mutation
  • Neutropenia* / congenital
  • Neutropenia* / genetics
  • Neutrophils*
  • Prevalence

Substances

  • Leukocyte Elastase
  • Autoantibodies

Supplementary concepts

  • Neutropenia, Severe Congenital, Autosomal Recessive 3