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Titolo:
Multifocal osteomyelitis caused by nontuberculous mycobacteria in patientswith a genetic defect of the interferon-gamma receptor
Autore:
Arend, SM; Janssen, R; Gosen, JJ; Waanders, H; de Boer, T; Ottenhoff, THM; van Dissel, JT;
Indirizzi:
Leiden Univ, Med Ctr, Dept Infect Dis, NL-2300 RC Leiden, Netherlands Leiden Univ Leiden Netherlands NL-2300 RC NL-2300 RC Leiden, Netherlands Rijnland Hosp, Dept Pediat, Leiderdorp, Netherlands Rijnland Hosp Leiderdorp Netherlands pt Pediat, Leiderdorp, Netherlands T Lange Land Hosp, Dept Internal Med, Zoetermeer, Netherlands T Lange LandHosp Zoetermeer Netherlands l Med, Zoetermeer, Netherlands Leiden Univ, Med Ctr, Dept Immunohematol & Blood Transfus, Leiden, Netherlands Leiden Univ Leiden Netherlands ol & Blood Transfus, Leiden, Netherlands
Titolo Testata:
NETHERLANDS JOURNAL OF MEDICINE
fascicolo: 3, volume: 59, anno: 2001,
pagine: 140 - 151
SICI:
0300-2977(200109)59:3<140:MOCBNM>2.0.ZU;2-9
Fonte:
ISI
Lingua:
ENG
Soggetto:
CALMETTE-GUERIN INFECTION; DISSEMINATED BCG INFECTION; AVIUM COMPLEX INFECTION; INTRACELLULAR BACTERIA; TYPE-1 CYTOKINES; SUSCEPTIBILITY; DEFICIENCY; TUBERCULOSIS; IMMUNITY; CHILD;
Keywords:
Mycobacterium avium; nontuberculous mycobacteria; BCG; infection; osteomyelitis; genetic; interferon-gamma receptor;
Tipo documento:
Article
Natura:
Periodico
Settore Disciplinare:
Clinical Medicine
Citazioni:
50
Recensione:
Indirizzi per estratti:
Indirizzo: van Dissel, JT Leiden Univ, Med Ctr, Dept Infect Dis, C5P,POB 9600, NL-2300 RC Leiden, Netherlands Leiden Univ C5P,POB 9600 Leiden Netherlands NL-2300 RC nds
Citazione:
S.M. Arend et al., "Multifocal osteomyelitis caused by nontuberculous mycobacteria in patientswith a genetic defect of the interferon-gamma receptor", NETH J MED, 59(3), 2001, pp. 140-151

Abstract

We describe three patients with multifocal osteomyelitis caused by Mycobacterium avium and a family history of one or more first degree family members diagnosed with various clinical presentations of infections with nontuberculous mycobacteria. There was a significant delay in the diagnosis and they had a protracted course of their illness, which responded only slowly to prolonged multi-drug treatment. In one patient, additional treatment with interferon-gamma (IFN-gamma) was necessary. Macrophages of these patients had decreased in vitro responsiveness to IFN-gamma. Genomic sequencing revealed that these patients and their affected family members were heterozygous for a previously described dominant negative mutation in the gene encoding the IFN-gamma binding receptor-1 chain. The clinical presentations of the infections with nontuberculous mycobacteria in these families, with spread limited to skin, bone and lymph nodes. is discussed in the light of the immune mechanisms that are responsible for the clearance of otherwise poorly pathogenic environmental mycobacteria. (C) 2001 Elsevier Science B.V. All rights reserved.

ASDD Area Sistemi Dipartimentali e Documentali, Università di Bologna, Catalogo delle riviste ed altri periodici
Documento generato il 03/07/20 alle ore 16:06:19