Academic Journal

Clinical features of candidiasis in patients with inherited interleukin 12 receptor ß1 deficiency

التفاصيل البيبلوغرافية
العنوان: Clinical features of candidiasis in patients with inherited interleukin 12 receptor ß1 deficiency
المؤلفون: Barbouche R., Broides A., Goudouris E., Franco J.L., Parvaneh N., Reisli I., Strickler A., Shcherbina A., Somer A., Segal A., Angel-Moreno A., Lezana-Fernandez J.L., Bejaoui M., Kachboura S., Bobadilla-Del Valle M., Sentongo T., Ben-Mustapha I., Bustamante J., Picard C., Puel A., Boisson-Dupuis S., Abel L., Casanova J.-L., Rodríguez-Gallego C., Sanal O., Ouederni M., Ikinciogullari A., Tezcan I., Dogu F., Sologuren I., Pedraza-Sánchez S., Keser M., Tanir G., Nieuwhof C., Colino E., Kumararatne D., Levy J., Kutukculer N., Aytekin C., Herrera-Ramos E., Bhatti M., Karaca N.
سنة النشر: 2014
المجموعة: Ege University Institutional Repository
مصطلحات موضوعية: Interleukin-12 receptor ß1 chain, Candida, Mycobacterium, Primary immunodeficiency, Salmonella
الوصف: PubMed ID: 24186907 ; Background. Interleukin 12Rß1 (IL-12Rß1)-deficient patients are prone to clinical disease caused by mycobacteria, Salmonella, and other intramacrophagic pathogens, probably because of impaired interleukin 12-dependent interferon 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-12Rß1 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-Guérin.Conclusions. Patients who are deficient in IL-12Rß1 may have candidiasis, usually mucocutaneous, which is frequently recurrent or persistent. Candidiasis may be the first clinical manifestation in these patients. © The Author 2013. ; 1Pediatric Hematology-Immunology Unit, National Bone Marrow Transplantation Center, Tunis, 2Faculty of Medicine, University of Tunis El Manar, ...
نوع الوثيقة: article in journal/newspaper
اللغة: English
تدمد: 1058-4838
Relation: Makale - Uluslararası Hakemli Dergi - Kurum Öğretim Elemanı; Clinical Infectious Diseases; https://hdl.handle.net/11454/26148; https://doi.org/10.1093/cid/cit722; 58; 204; 213
DOI: 10.1093/cid/cit722
الاتاحة: https://hdl.handle.net/11454/26148
https://doi.org/10.1093/cid/cit722
Rights: open
رقم الانضمام: edsbas.D3779A24
قاعدة البيانات: BASE
الوصف
تدمد:10584838
DOI:10.1093/cid/cit722