Successful granulocyte colony-stimulating factor treatment of relapsing Candida Albicans meningoencephalitis caused by CARD9 deficiency

Pediatric Infectious Disease Journal

Volumn 35, Issue 4, 2016, Pages 428-431

  Celmeli, Fatih ^a   Oztoprak, Nefise ^a   Turkkahraman, Doga ^b   Seyman, Derya ^a   Mutlu, Esvet ^c   Frede, Natalie ^d,e   Köksoy, Sadi ^c   Grimbacher, Bodo ^d,e,f  

^a Antalya Education and Research Hospital   (Turkey)

^b Akdeniz University Medical School Hospital ^*  (Turkey)

^c AKDENIZ UNIVERSITY   (Turkey)

^d UNIVERSITY OF FREIBURG   (Germany)

^e DZIF Standort Freiburg   (Germany)

^f UNIVERSITY COLLEGE LONDON   (United Kingdom)

Author keywords

CARD9 deficiency; Chronic mucocutaneous candidiasis; G CSF treatment

Indexed keywords

AMOXICILLIN PLUS CLAVULANIC ACID; AMPHOTERICIN B; CASPASE; CASPASE ASSOCIATED RECRUITMENT DOMAIN 9; CASPOFUNGIN; FILGRASTIM; FLUCONAZOLE; GAMMA INTERFERON; GLUCOSE; INTERLEUKIN 17; UNCLASSIFIED DRUG; VORICONAZOLE; ANTIFUNGAL AGENT; CARD9 PROTEIN, HUMAN; CASPASE RECRUITMENT DOMAIN SIGNALING PROTEIN; FUNGUS ANTIBODY; GRANULOCYTE COLONY STIMULATING FACTOR;

ADD ON THERAPY; ADULT; ANTIBIOTIC THERAPY; ANTIMICROBIAL THERAPY; ARTICLE; AUTOSOMAL RECESSIVE DISORDER; BACTERIUM CULTURE; CANDIDA ALBICANS; CANDIDA MENINGITIS; CASE REPORT; CASPASE ASSOCIATED RECRUITMENT DOMAIN 9 DEFICIENCY; CEREBROSPINAL FLUID CULTURE; CEREBROSPINAL FLUID LEVEL; CONTROLLED STUDY; CYTOKINE PRODUCTION; CYTOKINE RELEASE; DRUG WITHDRAWAL; ENZYME DEFICIENCY; ENZYME LINKED IMMUNOSPOT ASSAY; GENE MUTATION; HEADACHE; HOMOZYGOSITY; HUMAN; IMMUNE DEFICIENCY; LUMBAR PUNCTURE; LYMPHOCYTE COUNT; MALE; MEDICAL HISTORY; MENINGOENCEPHALITIS; MONOTHERAPY; MUCOCUTANEOUS CANDIDIASIS; NITROBLUE TETRAZOLIUM TEST; OROPHARYNGEAL MONILIASIS; OROPHARYNX CANDIDIASIS; PHYSICAL EXAMINATION; PRIORITY JOURNAL; PROTEIN SYNTHESIS; RECURRENT INFECTION; REMISSION; RESPIRATORY BURST; STIFF NECK; THRUSH; TINEA VERSICOLOR; TONSILLITIS; TREATMENT DURATION; VOMITING; BLOOD; CANDIDIASIS, INVASIVE; DEFICIENCY; HOMOZYGOTE; IMMUNOLOGY; LYMPHOCYTE SUBPOPULATION; METABOLISM; MUTATION; RECURRENT DISEASE; TREATMENT OUTCOME;

ADULT; ANTIBODIES, FUNGAL; ANTIFUNGAL AGENTS; CANDIDA ALBICANS; CANDIDIASIS, INVASIVE; CARD SIGNALING ADAPTOR PROTEINS; GRANULOCYTE COLONY-STIMULATING FACTOR; HOMOZYGOTE; HUMANS; INTERLEUKIN-17; LYMPHOCYTE SUBSETS; MALE; MENINGOENCEPHALITIS; MUTATION; RECURRENCE; TREATMENT OUTCOME;

EID: 84949571244     PISSN: 08913668     EISSN: 15320987     Source Type: Journal    
DOI: 10.1097/INF.0000000000001028     Document Type: Article

References (10)

1. Hsu YM, Zhang Y, You Y, et al. The adaptor protein CARD9 is required for innate immune responses to intracellular pathogens. Nat Immunol. 2007;8:198-205.
2. Glocker EO, Hennigs A, Nabavi M, et al. A homozygous CARD9 mutation in a family with susceptibility to fungal infections. N Engl J Med. 2009;361:1727-1735.
3. Drewniak A, Gazendam RP, Tool AT, et al. Invasive fungal infection and impaired neutrophil killing in human CARD9 deficiency. Blood. 2013;121:2385-2392.
4. Lanternier F, Barbati E, Meinzer U, et al. Inherited CARD9 deficiency in 2 unrelated patients with invasive Exophiala infection. J Infect Dis. 2015;211:1241-1250.
5. Gavino C, Cotter A, Lichtenstein D, et al. CARD9 deficiency and spontaneous central nervous system candidiasis: complete clinical remission with GM-CSF therapy. Clin Infect Dis. 2014;59:81-84.
6. Boisson B, Wang C, Pedergnana V, et al. An ACT1 mutation selectively abolishes interleukin-17 responses in humans with chronic mucocutaneous candidiasis. Immunity. 2013;39:676-686.
7. Puel A, Cypowyj S, Bustamante J, et al. Chronic mucocutaneous candidiasis in humans with inborn errors of interleukin-17 immunity. Science. 2011;332:65-68.
8. Gaviria JM, van Burik JA, Dale DC, et al. Comparison of interferon-gamma, granulocyte colony-stimulating factor, and granulocyte-macrophage colony-stimulating factor for priming leukocyte-mediated hyphal damage of opportunistic fungal pathogens. J Infect Dis. 1999;179:1038-1041.
9. Wildbaum G, Shahar E, Katz R, et al. Continuous G-CSF therapy for isolated chronic mucocutaneous candidiasis: complete clinical remission with restoration of IL-17 secretion. J Allergy Clin Immunol. 2013;132:761-764.
10. Pappas PG, Kauffman CA, Andes D, et al; Infectious Diseases Society of America. Clinical practice guidelines for the management of candidiasis: 2009 update by the Infectious Diseases Society of America. Clin Infect Dis. 2009;48:503-535.