Infection

, Volume 28, Issue 3, pp 171–174

Visceral Leishmaniasis and Other Severe Infections in an Adult Patient with p47-phox-Deficient Chronic Granulomatous Disease

  • V. Asensi
  • L. Tricas
  • A. Meana
  • D. Roos
  • J.A. Carton
  • J.A. Maradona
  • M.F. Fresno
  • E. Valle
  • J. Fierer
  • J.M. Arribas
Case Report

DOI: 10.1007/s150100050074

Cite this article as:
Asensi, V., Tricas, L., Meana, A. et al. Infection (2000) 28: 171. doi:10.1007/s150100050074

Summary

We report a rare case of a male patient without known immunodeficiency consecutively diagnosed with visceral leishmaniasis, brain abscess and cavitating pneumonia in the 3rd decade of life. Chronic granulomatous disease (CGD) was diagnosed by a nitroblue tetrazolium test. A p47-phox mutation of the NADPH oxidase of the leukocytes was suspected by immunoblotting and confirmed by DNA analysis. The patient was homozygous for this mutation while his mother and sister were heterozygous asymptomatic carriers. After the CGD diagnosis the patient started a chronic prophylactic regimen with subcutaneous interferon-γ (0.05 mg/m2 of body surface/three times a week), and oral trimethoprim-sulfamethoxazole and itraconazole (both at 5 mg/kg/day) with no subsequent infections after 12 months of follow-up.

Key Words Adult chronic granulomatous diseasep47-phox mutationLeishmaniaBrain abscessPneumonia

Copyright information

© Urban & Vogel Medien und Medizin Verlagsgesellschaft 2000

Authors and Affiliations

  • V. Asensi
    • 1
  • L. Tricas
    • 2
  • A. Meana
    • 3
  • D. Roos
    • 4
  • J.A. Carton
    • 1
  • J.A. Maradona
    • 1
  • M.F. Fresno
    • 5
  • E. Valle
    • 6
  • J. Fierer
    • 7
  • J.M. Arribas
    • 1
  1. 1.Infectious Diseases Unit, Hospital Central de Asturias, Oviedo University Medical School, c/ Celestino Villamil s/n, E-33006 Oviedo, Spain; e-mail: vesensia@medynet.comES
  2. 2.Immunodeficiencies Unit, Hospital Central de Asturias, Oviedo University Medical School, Oviedo, SpainES
  3. 3.Principado de Asturias Transfusion Center, Hospital Central de Asturias, Oviedo University Medical School, Oviedo, SpainES
  4. 4.Netherlands Red Cross Transfusion Service, Amsterdam, The NetherlandsNL
  5. 5.Pathology Service, Hospital Central de Asturias, Oviedo University Medical School, Oviedo, SpainES
  6. 6.Department of Biochemistry, Oviedo University Medical School, Oviedo, SpainES
  7. 7.Departments of Medicine and Pathology, Veterans Administration Medical Center, University of California, San Diego, USAUS