Clinical Features of Candidiasis in Patients With Inherited Interleukin 12 Receptor ?1 Deficiency

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dc.contributor.authorOuederni, Monia
dc.contributor.authorSanal, Ozden
dc.contributor.authorIkinciogullari, Aydan
dc.contributor.authorTezcan, Ilhan
dc.contributor.authorDogu, Figen
dc.contributor.authorSologuren, Ithaisa
dc.contributor.authorPedraza-Sanchez, Sigifredo
dc.date.accessioned2024-02-23T14:21:00Z
dc.date.available2024-02-23T14:21:00Z
dc.date.issued2014
dc.departmentNEÜen_US
dc.description.abstractBackground. Interleukin 12R beta 1 (IL-12R beta 1)-deficient patients are prone to clinical disease caused by mycobacteria, Salmonella, and other intramacrophagic pathogens, probably because of impaired interleukin 12-dependent interferon gamma production. About 25% of patients also display mucocutaneous candidiasis, probably owing to impaired interleukin 23-dependent interleukin 17 immunity. The clinical features and outcome of candidiasis in these patients have not been described before, to our knowledge. We report here the clinical signs of candidiasis in 35 patients with IL-12R beta 1 deficiency. Results. Most (n = 71) of the 76 episodes of candidiasis were mucocutaneous. Isolated oropharyngeal candidiasis (OPC) was the most common presentation (59 episodes, 34 patients) and was recurrent or persistent in 26 patients. Esophageal candidiasis (n = 7) was associated with proven OPC in 2 episodes, and cutaneous candidiasis (n = 2) with OPC in 1 patient, whereas isolated vulvovaginal candidiasis (VVC; n = 3) was not. Five episodes of proven invasive candidiasis were documented in 4 patients; 1 of these episodes was community acquired in the absence of any other comorbid condition. The first episode of candidiasis occurred earlier in life (median age +/- standard deviation, 1.5 +/- 7.87 years) than infections with environmental mycobacteria (4.29 +/- 11.9 years), Mycobacterium tuberculosis (4 +/- 3.12 years), or Salmonella species (4.58 +/- 4.17 years) or other rare infections (3 +/- 11.67 years). Candidiasis was the first documented infection in 19 of the 35 patients, despite the vaccination of 10 of these 19 patients with live bacille Calmette-Guerin. Conclusions. Patients who are deficient in IL-12R beta 1 may have candidiasis, usually mucocutaneous, which is frequently recurrent or persistent. Candidiasis may be the first clinical manifestation in these patients.en_US
dc.description.sponsorshipFondo de Investigaciones Sanitarias, Ministerio de Economia y Competitividad [PI06/1031, PI10/01718]; European Regional Development Fund-European Social Fund (FEDER-FSE); Fundacion Canaria de Investigacion y Salud (Canarian government) [INREDCAN 05/06]; Foundation Caja Rural de Canarias-Chil y Naranjo; Universidad de Las Palmas de Gran Canaria; INSERM; University Paris Descartes; Rockefeller University; National Center for Research Resources; National Center for Advancing Sciences of the National Institutes of Health [8UL1TR000043]; Laboratoire d'Excellence Integrative Biology of Emerging Infectious Diseases [ANR-10-LABX-62-IBEID]; European Research Council, Agence Nationale de la Recherche [GENCMCD 11-BSV3-005-01]; St Giles Foundation; Candidoser Association; Consejo Nacional de Ciencia y Tecnologia de Mexico [69992, 182817]en_US
dc.description.sponsorshipThis work was supported by Fondo de Investigaciones Sanitarias, Ministerio de Economia y Competitividad (grants PI06/1031 and PI10/01718), the European Regional Development Fund-European Social Fund (FEDER-FSE), Fundacion Canaria de Investigacion y Salud (Canarian government; INREDCAN 05/06), Foundation Caja Rural de Canarias-Chil y Naranjo (research prize 2004), Universidad de Las Palmas de Gran Canaria (fellowship to E. H. R), INSERM, University Paris Descartes, Rockefeller University, National Center for Research Resources and the National Center for Advancing Sciences of the National Institutes of Health (grant 8UL1TR000043), Laboratoire d'Excellence Integrative Biology of Emerging Infectious Diseases (grant ANR-10-LABX-62-IBEID), the European Research Council, Agence Nationale de la Recherche (grant GENCMCD 11-BSV3-005-01), the St Giles Foundation, the Candidoser Association, and Consejo Nacional de Ciencia y Tecnologia de Mexico (grants 69992 and 182817).en_US
dc.identifier.doi10.1093/cid/cit722
dc.identifier.endpage213en_US
dc.identifier.issn1058-4838
dc.identifier.issn1537-6591
dc.identifier.issue2en_US
dc.identifier.pmid24186907en_US
dc.identifier.scopus2-s2.0-84891754308en_US
dc.identifier.scopusqualityQ1en_US
dc.identifier.startpage204en_US
dc.identifier.urihttps://doi.org/10.1093/cid/cit722
dc.identifier.urihttps://hdl.handle.net/20.500.12452/13402
dc.identifier.volume58en_US
dc.identifier.wosWOS:000329131300012en_US
dc.identifier.wosqualityQ1en_US
dc.indekslendigikaynakWeb of Scienceen_US
dc.indekslendigikaynakScopusen_US
dc.indekslendigikaynakPubMeden_US
dc.language.isoenen_US
dc.publisherOxford Univ Press Incen_US
dc.relation.ispartofClinical Infectious Diseasesen_US
dc.relation.publicationcategoryMakale - Uluslararası Hakemli Dergi - Kurum Öğretim Elemanıen_US
dc.rightsinfo:eu-repo/semantics/openAccessen_US
dc.subjectInterleukin-12 Receptor Beta 1 Chainen_US
dc.subjectPrimary Immunodeficiencyen_US
dc.subjectCandidaen_US
dc.subjectMycobacteriumen_US
dc.subjectSalmonellaen_US
dc.titleClinical Features of Candidiasis in Patients With Inherited Interleukin 12 Receptor ?1 Deficiencyen_US
dc.typeArticleen_US

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