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Title: Clinical features of candidiasis in patients with inherited interleukin 12 receptor β1 deficiency
Authors: Ouederni, Monia
Sanal, Ozden
Ikincioǧullari, Aydan
Tezcan, Ilhan
Dogu, Figen
Sologuren, Ithaisa
Pedraza-Sánchez, Sigifredo
Keser, Melike
Tanir, Gonul
Nieuwhof, Chris
Colino, Elena
Kumararatne, Dinakantha
Levy, Jacov
Kutukculer, Necil
Aytekin, Caner
Herrera-Ramos, Estefanía
Bhatti, Micah
Karaca, Neslihan
Barbouche, Ridha
Broides, Arnon
Goudouris, Ekaterini
Franco, José Luis
Parvaneh, Nima
Reisli, Ismail
Strickler, Alexis
Shcherbina, Anna
Somer, Ayper
Segal, Anthony
Angel-Moreno, Alfonso
Lezana-Fernandez, José Luis
Bejaoui, Mohamed
Bobadilla-Del Valle, Miriam
Kachboura, Salem
Sentongo, Timothy
Ben-Mustapha, Imen
Bustamante, Jacinta
Picard, Capucine
Puel, Anne
Boisson-Dupuis, Stéphanie
Abel, Laurent
Casanova, Jean Laurent
Rodríguez-Gallego, Carlos 
UNESCO Clasification: 32 Ciencias médicas
320505 Enfermedades infecciosas
Keywords: Chronic Mucocutaneous Candidiasis
Hyper-Ige Syndrome
Il-12R-Beta-1 Deficiency
Mycobacterial Disease, et al
Issue Date: 2014
Journal: Clinical Infectious Diseases 
Abstract: Background. 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.
ISSN: 1058-4838
DOI: 10.1093/cid/cit722
Source: Clinical Infectious Diseases[ISSN 1058-4838],v. 58, p. 204-213
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