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Underlying chronic granulomatous disease in a patient with bronchocentric granulomatosis
  1. Y Moltyaner1,
  2. W H Geerts1,
  3. D W Chamberlain2,
  4. P G Heyworth4,
  5. D Noack5,
  6. J Rae4,
  7. J J Doyle3,
  8. G P Downey1
  1. 1Division of Respirology, University of Toronto, Toronto, Ontario, Canada
  2. 2Department of Pathology, University of Toronto, Toronto, Ontario, Canada
  3. 3Division of Hematology, Hospital for Sick Children, Toronto, Ontario, Canada
  4. 4Department of Molecular and Experimental Medicine, The Scripps Research Institute, La Jolla, California, USA
  5. 5Department of Immunology, Genentech Inc, South San Francisco, California, USA
  1. Correspondence to:
    Dr W H Geerts
    Sunnybrook & Women’s College Health Sciences Center, 2075 Bayview Avenue, Toronto, Ontario, M4N 3M5 Canada; william.geertsswchsc.on.ca

Abstract

We present a case of bronchocentric granulomatosis in a woman with no history of asthma who was colonised with Aspergillusfumigatus. A family history of chronic granulomatous disease prompted further testing that demonstrated severely depressed neutrophil oxidant production and gp91phox deficiency compatible with the X linked carrier state of chronic granulomatous disease. Only one report of the association of these two rare diseases has previously appeared in the literature. We postulate that an ineffective immune response led to the prolonged colonisation of Afumigatus resulting in a hypersensitivity reaction that was manifest clinically as bronchocentric granulomatosis.

  • chronic granulomatous disease
  • bronchocentric granulomatosis
  • Aspergillus

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Footnotes

  • This work was supported in part by a CIHR grant MT–10994 to G Downey and NIH grants CA68276 and AI24838 to P G Heyworth and J Rae.