Inheritance pattern and clinical aspects of 93 Iranian patients with chronic granulomatous disease

J Clin Immunol. 2011 Oct;31(5):792-801. doi: 10.1007/s10875-011-9567-x. Epub 2011 Jul 26.

Abstract

Background: Chronic granulomatous disease (CGD) is a rare immunodeficiency due to a genetic defect in one of the NADPH-oxidase components. We studied CGD inheritance forms (autosomal recessive (AR) or X-linked (XL)) and AR-CGD subtypes in Iran.

Methods: Clinical and functional investigations were conducted in 93 Iranian CGD patients from 75 families.

Results: Most of the patients were AR-CGD (87.1%). This was related to consanguineous marriages (p = 0.001). The age of onset of symptoms and diagnosis were lower in XL-CGD compared with AR-CGD (p < 0.0001 for both). Among AR-CGD patients, p47phox defect was the predominant subtype (55.5%). The most common clinical features in patients were lymphadenopathy (65.6%) and pulmonary involvement (57%). XL-CGD patients were affected more frequently with severe infectious manifestations.

Conclusions: Although XL-CGD is the most common type of the disease worldwide, only 12 patients (12.9%) were XL-CGD in our study. The relatively high frequency of AR-CGD is probable due to widely common consanguineous marriages in Iran.

MeSH terms

  • Adolescent
  • Adult
  • Age of Onset
  • Child
  • Child, Preschool
  • Consanguinity
  • DNA Mutational Analysis
  • Female
  • Genes, Recessive / genetics
  • Genes, X-Linked / genetics
  • Granulomatous Disease, Chronic / epidemiology*
  • Granulomatous Disease, Chronic / genetics*
  • Granulomatous Disease, Chronic / physiopathology
  • Humans
  • Infant
  • Iran
  • Lymphatic Diseases
  • Male
  • Middle Aged
  • NADPH Oxidases / genetics*
  • Respiratory Tract Infections
  • Risk Factors

Substances

  • NADPH Oxidases
  • neutrophil cytosolic factor 1