The Many Faces of a Monogenic Autoinflammatory Disease: Adenosine Deaminase 2 Deficiency

Curr Rheumatol Rep. 2020 Aug 26;22(10):64. doi: 10.1007/s11926-020-00944-1.

Abstract

Purpose of review: We aim to describe the pathophysiology, clinical findings, diagnosis, and treatment of deficiency of adenosine deaminase 2 (DADA2).

Recent findings: DADA2 is a multi-organ disease of children and less often adults, which can present with wide-ranging manifestations including strokes, medium vessel vasculitis, hematologic disease, and immunodeficiency. Diagnosis is through detection of reduced activity level of the adenosine deaminase 2 (ADA2) enzyme and/or identification of bi-allelic mutations in the ADA2 gene. Outside of high-dose glucocorticoids, conventional immunosuppression has been largely ineffective in treating this relapsing and remitting disease. Vasculitic-predominant manifestations respond extremely well to tumor necrosis factor-α inhibition. Hematopoietic stem cell transplantation can lead to normalization of enzyme activity, as well as resolution of vasculitic, hematologic, and immunologic manifestations, although treatment-related adverse effects are not uncommon. Early detection of this disease across multiple disciplines could prevent devastating clinical outcomes, especially in genetically pre-disposed populations.

Keywords: Adenosine; Adenosine deaminase 2 deficiency; Monogenic disease; Polyarteritis nodosa; Vasculitis.

Publication types

  • Review

MeSH terms

  • Adenosine Deaminase / deficiency
  • Adenosine Deaminase / genetics
  • Hereditary Autoinflammatory Diseases* / diagnosis
  • Hereditary Autoinflammatory Diseases* / genetics
  • Humans
  • Intercellular Signaling Peptides and Proteins / deficiency
  • Mutation
  • Polyarteritis Nodosa*
  • Severe Combined Immunodeficiency* / diagnosis
  • Severe Combined Immunodeficiency* / genetics
  • Severe Combined Immunodeficiency* / therapy

Substances

  • Intercellular Signaling Peptides and Proteins
  • ADA2 protein, human
  • Adenosine Deaminase