Skip to main content
SpringerLink
Log in

Intermittent ketoconazole therapy of chronic mucocutaneous candidiasis in childhood

  • Original Investigations
  • Published:
European Journal of Pediatrics Aims and scope Submit manuscript

Abstract

We report the clinical and laboratory findings in two children with chronic mucocutaneous condidiasis (CMC) treated successfully with intermittent long-term ketoconazole therapy. Both had chronic infection of the nails, skin and mucous membranes with positive cultures for candida. Both were resistant to multiple local and systemic antifungal agents. After institution of ketoconazole therapy there was a dramatic improvement with clearing of the oral (one week), skin (two months) and nail lesions (5 months). After 8 months the drug was stopped and clinical remission persisted for 10 and 7 months respectively. Relapse of oral candidiasis was treated with a short course of ketoconazole (4–16 weeks) leading to complete healing of the lesions. Clinical improvement was not related to an amelioration in lymphocyte transformation. There was no change in the progressive deterioration of the lymphocyte responses to candida antigen which was probably due to persisting candida cell wall components (e.g. mannan).

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

References

  1. Axelsen HM, Kirkpatrick CH, Buckley RH (1974) Precipitins to candida albicans in chronic mucocutaneous candidiasis studied by immunoelectrophoresis with intermediate gel. Correlation with clinical and immunological findings. Clin Exp Immunol 17: 385–394

    PubMed  Google Scholar 

  2. Bennet JE (1977) Fluocytosine. Ann Intern Med 86:319–322

    PubMed  Google Scholar 

  3. Blizzard RM, Gibbs, JH (1968) Candidiasis: studies pertaining to its association with endocrinopathies and pernicious anemia. Pediatrics 42: 231–237

    PubMed  Google Scholar 

  4. Buckley RH, Lucas ZJ, Hattler BG (1968) Defective cellular immunity associated with chronic mucocutaneous moniliasis and staphylococcal botryomycosis: immunologic reconsitution by allogeneic bone marrow. Clin Exp Immun 3:153–169

    PubMed  Google Scholar 

  5. De Brabander M, Aerts F, Van Cutsem J, Van Den Bossche H, Borgers M (1980) The activity of ketoconazole in mixed cultures of leukocytes and candida albicans. Sabouraudia 18:197–210

    PubMed  Google Scholar 

  6. Dixon D, Shadomy S, Shadomy HJ, Espinel-Ingroff A, Kerkering TM (1978) Comparison of the in vitro antifungal activities of miconazole and a new imidazole R 41,400. J Infect Dis 138:245–248

    PubMed  Google Scholar 

  7. Drouhet E, Dupont B (1980) Chronic mucocutaneous candidiasis and other superficial and systemic mycosis successfully treated with ketoconazole. Rev Infect Dis 2:606–619

    PubMed  Google Scholar 

  8. Fischer A, Ballet JJ, Griscelli C (1978) Specific inhibition of in vitro candida-induced lymphocyte proliferation by polysaccharidic antigens present in the serum of patients with chronic mucocutaneous candidiasis. J Clin Invest 62:1005–1013

    PubMed  Google Scholar 

  9. Fischer A, Pichat L, Audinot M, Griscelli C (1982) Defective handling of mannan by monocytes in patients with chronic mucocutaneous candidiasis resulting in a specific cellular unresponsiveness. Clin Exp Immunol 47:653–660

    PubMed  Google Scholar 

  10. Fischer TJ, Klein RB, Kershnar HE, Borut TC, Stiehm ER (1977) Miconazole in the treatment of chronic mucocutaneous candidiasis: A preliminary report. J Pediatr 91:815–819

    PubMed  Google Scholar 

  11. Galgiani JN, Stevens DA (1978) Turbidimetric studies of growth inhibition of yeast with three drugs: inquiry into inoculum-dependent susceptibility testing, time of onset of drug effect and implications for current and newer methods. Antimicr Agents & Chemother 13:249–254

    Google Scholar 

  12. Graybill JR, Herndon JH, Kniker WT, Levine HB (1980) Ketoconazole treatment of chronic mucocutaneous candidiasis. Arch Dermatol 116:1137–1141

    PubMed  Google Scholar 

  13. Hay RJ, Wells RS, Clayton YM, Winfield H (1980) Treatment of chronic mucocutaneous cendidiasis with oral ketoconazole — a study of 12 cases. Rev Infect Dis 2:600–605

    PubMed  Google Scholar 

  14. Janssen Pharmaceutica (1980) Ketoconazole investigators brochure. New Brunswick, New Jersey

  15. Kennedy CTC, Valdimarsson H, Hay RJ (1981) Chronic mucocutaneous candidiasis with a serum-dependent neutrophil defect: response to ketoconazole. J Royal Soc Med 74:158–163

    Google Scholar 

  16. Kirkpatrick CH, Rich RR, Graw RG, Smith TK, Rogentine GN (1971) Treatment of chronic mucocutaneous moniliasis by immunologic reconstitution. Clin Exp Immun 9:733–748

    PubMed  Google Scholar 

  17. Kirkpatrick CH, Alling DW (1978) Treatment of chronic oral candidiasis with clotrimazole troches: A controlled clinical trial. N Engl J Med 299:1201–1203

    PubMed  Google Scholar 

  18. Kirkpatrick CH, Greenberg LE (1979) Treatment of chronic mucocutaneous candidiasis with transfer factor. In: Kahn A, Kirkpatrick CH, Hill NO (eds) Immune modulators in transfer factor. Academic Press, New York, pp 547–562

    Google Scholar 

  19. Legendre R, Steltz MA (1980) A multicenter double-blind comparison of ketoconazole and griseofulvin in the treatment of dermatophyte infections. Rev Infect Dis 2 (4:586–591

    PubMed  Google Scholar 

  20. Lehner T, Wilton JMA, Ivanyi L (1972) Immunodeficiencies in chronic mucocutaneous candidiasis. Immunology 22:775–787

    PubMed  Google Scholar 

  21. Levy RL, Huang SW, Bach ML, Bach FH, Hony R, Ammann AJ, Bortin M, Kay HEM (1971) Thymic transplantation in a case of chronic mucocutaneous candidiasis. Lancet 2:898–900

    PubMed  Google Scholar 

  22. Medoff G, Kobayashi GS (1980) Strategies in the treatment of systemic fungal infections. N Engl J Med 302:145–155

    PubMed  Google Scholar 

  23. Montes LF, Cooper MD, Bradford LG (1971) Prolonged oral treatment of chronic mucocutaneous candidiasis with amphothericin B. Arch Dermatol 104:45–56

    PubMed  Google Scholar 

  24. Petersen EA, Alling DW, Kirkpatrick CH (1980) Treatment of chronic mucocutaneous candidiasis with ketoconazole. Ann Intern Med 93:791–795

    PubMed  Google Scholar 

  25. Robertson M, Parker F, Hanfin JM (1980) Oral ketoconazole for griseofulvin-resistant dermatophytosis. Clin Res 28:252A, Abstract

    Google Scholar 

  26. Rosenblatt HM, Byrne W, Ament ME (1980) Successful treatment of chronic mucocutaneous candidiasis with ketoconazole. J Pediatr 97:657–660

    PubMed  Google Scholar 

  27. Rosenblatt HM, Byrne W, Ament ME Stiehm ER (1980) Successful treatment of severe chronic mucocutaneous candidiasis with a new oral agent, ketoconazole. In: Nelson JD, Grassi C (eds) Current chemotherapy and infectious diseases. American Society for Microbiology, Washington DC, pp 963–965

    Google Scholar 

  28. Seger RA, Issler Ch, Joller PW, Hitzig WH (1981) Chronische mucocutane Candidiasis: Therapeutische Erfahrungen mit Ketoconazol. Helv Paediat Acta [Suppl] 46:31, Abstract

    Google Scholar 

  29. Symoens J, Moens M, Dom J, Scheijgrond H, Dony J, Schuermans V, Legendre R, Finestine N (1980) An evaluation of two years' clinical experience with ketoconazole. Rev Infect Dis 2:674–687

    PubMed  Google Scholar 

  30. Valdimarsson H, Higgs JM, Wells RS, Yamamura M, Hobbs JR, Holt PJL (1973) Immune abnormalities associated with chronic mucocutaneous candidiasis. Cell Immunol 6:348–361

    PubMed  Google Scholar 

  31. Wells RS, Higgs JM, Macdonald A, Valdimarsson H, Holt PJL (1972) Familial chronic mucocutaneous candidiasis. J Med Genet 9:302–310

    PubMed  Google Scholar 

Download references

Author information

Author notes

  1. S. Fanconi

    Present address: Abteilung für Intensivpflege, Universitätskinderklinik Bern, CH-3010, Bern, Schweiz

Authors and Affiliations

  1. Department of Pediatrics, University of Zürich, Steinwiesstr 75, Ch-8032, Zürich, Switzerland

    S. Fanconi, R. Seger, P. Joller & C. Issler

  2. Department of Microbiology, University of Zürich, Gloriastr. 32, CH-8032, Zürich, Switzerland

    G. Schär

Authors
  1. S. Fanconi
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. R. Seger
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. P. Joller
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. C. Issler
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. G. Schär
    View author publications

    You can also search for this author in PubMed Google Scholar

Additional information

Dedicated to Prof. W. Hitzig on the occasion of his 60th birthday

Rights and permissions

Reprints and permissions

About this article

Cite this article

Fanconi, S., Seger, R., Joller, P. et al. Intermittent ketoconazole therapy of chronic mucocutaneous candidiasis in childhood. Eur J Pediatr 139, 176–180 (1982). https://doi.org/10.1007/BF01377351

Download citation

  • Received:

  • Accepted:

  • Issue Date:

  • DOI: https://doi.org/10.1007/BF01377351

Key words

Search

Navigation