Spectrum of Pulmonary Aspergillosis in Hyper-IgE Syndrome with Autosomal-Dominant STAT3 Deficiency

被引：23

作者：

Dureault, Amelie ^{[1
]}

Tcherakian, Colas ^{[2
,3
,4
]}

Poiree, Sylvain ^{[5
]}

Catherinot, Emilie ^{[2
,3
]}

Danion, Francois ^{[1
]}

Jouvion, Gregory ^{[6
,7
]}

Bougnoux, Marie Elisabeth ^{[8
]}

Mahlaoui, Nizar ^{[9
,10
,11
,12
]}

Givel, Claire ^{[2
,3
]}

Castelle, Martin ^{[13
]}

Picard, Capucine ^{[9
,10
,11
,12
]}

Chansdesris, Marie Olivia ^{[13
]}

Lortholary, Olivier ^{[1
,14
]}

Lanternier, Fanny ^{[1
,14
]}

Coignard, H. ^{[1
]}

Amazzough, K. ^{[1
]}

Suarez, F. ^{[13
]}

Blanche, S. ^{[11
]}

Sendid, B. ^{[15
,16
]}

Cornu, M. ^{[17
]}

Bervar, J. F. ^{[18
]}

Deschildre, A. ^{[19
]}

Wemeau, L. ^{[20
]}

Fieschi, C. ^{[21
]}

Alanio, A. ^{[22
,23
]}

Menetrey, C. ^{[24
]}

Senechal, A. ^{[25
]}

Ader, F. ^{[25
,26
]}

Tattevin, P. ^{[27
]}

Pison, C. ^{[28
,29
]}

Grandiere-Perez, L. ^{[30
]}

Garcia-Hermoso, D. ^{[31
]}

Botterel-Chartier, F. ^{[32
,33
]}

机构：

[1] Univ Paris 05, Hop Necker Enfants Malad, AP HP, Serv Malad Infect & Trop, Paris, France

[2] Hop Foch, Serv Pneumol, Suresnes, France

[3] Univ Versailles St Quentin En Yvelines, Fac Sci Sante Simone Veil, Versailles, France

[4] Natl Referral Ctr Hypereosinophil CEREO, Suresnes, France

[5] Hop Necker Enfants Malad, AP HP, Serv Radiol, Paris, France

[6] Inst Pasteur, Unite Neuropathol Expt, Paris, France

[7] Sorbonne Univ, Hop Trousseau, AP HP, Dept Genet Med, Paris, France

[8] Hop Necker Enfants Malad, AP HP, Microbiol Serv, Paris, France

[9] Hop Necker Enfants Malad, AP HP, CEDI, Paris, France

[10] Hop Necker Enfants Malad, AP HP, Ctr Reference Deficits Immunitaires Hereditaires, CEREDIH, Paris, France

[11] Hop Necker Enfants Malad, AP HP, Serv Immunol Hematol Pediat, Paris, France

[12] Univ Paris 05, Imagine Inst, INSERM, UMR1163, Paris, France

[13] Hop Necker Enfants Malad, AP HP, Serv Hematol, Paris, France

[14] Inst Pasteur, CNRS, Ctr Natl Reference Mycoses Invas & Antifong, Unite Mycol Mol, Paris, France

[15] Univ Lille, CHU Lille, Team Fungal Associate Inflammatory & Invas Dis 2, Inserm,U995,LIRIC, Lille, France

[16] Univ Lille, Lab Parasitol Mycol, Lille, France

[17] CHU Lille, Lab Parasitol Mycol, Lille, France

[18] CHRU Lille, Hop Calmette, Clin Malad Resp, Lille, France

[19] Univ Nord France, Hop Jeanne Flandre, CHU Lille, Pediat Pulmonol & Allergy Unit, Lille, France

[20] Hop Calmette, CHRU Lille, Clin Malad Resp, Lille, France

[21] Hop St Louis, AP HP, Dept Clin Immunol, Paris, France

[22] Hop St Louis, AP HP, Ctr Natl Reference Mycoses Invas & Antifong, Inst Pasteur,CNRS,Unite Mycol Mol, Paris, France

[23] Hop St Louis, AP HP, Lab Parasitol Mycol, Paris, France

[24] Hop Mere & Enfant, CHU Limoges, Serv Pediat, Limoges, France

[25] Hosp Civils Lyon, Hop Croix Rousse, Serv Malad Infect & Trop, Lyon, France

[26] Univ Claude Bernard Lyon 1, Ctr Rech Infectiol CIRI, INSERM, U1111, Lyon, France

[27] CHU Pontchaillou, Malad Infect & Reanimat Med, Rennes, France

[28] Ctr Hosp & Univ Grenoble, Serv Hosp Univ Pneumol Physiol, Pole Thorax & Vaisseaux, Grenoble, France

[29] Univ Grenoble Alpes, Lab Bioenerget Fondamentale & Appl, Inserm1055, Grenoble, France

[30] Ctr Hosp Le Mans, Serv Malad Infect & Trop, Le Mans, France

[31] Inst Pasteur, CNRS, Ctr Natl Reference Mycoses Invas & Antifong, Unite Mycol Mol, Paris, France

[32] CHU Henri Mondor, AP HP, Unite Parasitol Mycol, Creteil, France

[33] EA Dynamyc UPEC, ENVA, Creteil, France

来源：

JOURNAL OF ALLERGY AND CLINICAL IMMUNOLOGY-IN PRACTICE | 2019年 / 7卷 / 06期

关键词：

Aspergillosis; STAT3-deficient patient; Allergic bronchopulmonary aspergillosis; Cavitary chronic pulmonary aspergillosis; Aspergilloma; ALLERGIC BRONCHOPULMONARY ASPERGILLOSIS; INVASIVE FUNGAL DISEASE; BACTERIAL-INFECTION; CLASSIFICATION;

D O I：

10.1016/j.jaip.2019.02.041

中图分类号：

R392 [医学免疫学];

学科分类号：

100102 ;

摘要：

BACKGROUND: Autosomal-dominant signal transducer and activator of transcription 3 (STAT3) deficiency predisposes to recurrent bacterial pneumonia, complicated by bronchiectasis and cavitations. Aspergillosis is a major cause of morbidity in these patients. However, its diagnosis, classification, and treatment are challenging. OBJECTIVE: We aimed to assess the prevalence and describe the clinical, mycological, and radiological presentation and related therapy and outcome of Aspergillus infections of the respiratory tract in the STAT3-deficient patients of the National French cohort. METHODS: We performed a retrospective study of all pulmonary aspergillosis cases in STAT3-deficient patients (n = 74). Clinical and mycological data were collected up to October 2015 and imaging was centralized. RESULTS: Twenty-one episodes of pulmonary aspergillosis in 13 (17.5%) STAT3-deficient patients were identified. The median age at first episode was 13 years (interquartile range, 10-26 years). Ninety percent of patients had previous bronchiectasis or cavitations. Infections were classified as follows: 5 single aspergilloma, 9 chronic cavity pulmonary aspergillosis, 5 allergic bronchopulmonary aspergillosis-like disease, and 2 mixed forms of concomitant allergic bronchopulmonary aspergillosis-like disease and chronic cavity pulmonary aspergillosis. No invasive aspergillosis cases were identified. Aspergillus species were isolated in 71% of episodes and anti-Aspergillus antibodies in 93%. Eleven episodes were breakthrough infections. Antifungal treatment was prolonged, with a median of 13 months, and 6 patients (7 episodes) required surgery, with a high rate of postsurgical complications. One patient died and 6 had a relapse. CONCLUSIONS: Chronic and allergic forms of aspergillosis occurred in 17.5% of STAT3-deficient patients, mostly in lung cavities. Almost half had recurrences, despite prolonged antifungal treatment and/or surgery. (C) 2019 American Academy of Allergy, Asthma & Immunology

引用

页码：1986 / +

页数：13

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