Allergic manifestations of inborn errors of immunity and their impact on the diagnosis: A worldwide study

World Allergy Organization Journal

Dalia H. El-Ghoneimy, MD, PhD¹  Kathleen Sullivan, MD, PhD²  Surjit Singh, MD, DCH (Lon.), FRCP (Lon.), FRCPCH (Lon.), FAMS³  Maryam A. Al-Nesf, MD, ABHS⁴  Waleed Al-Herz, MD⁵  Nadia Kechout, MD, PhD⁶  Sara Espinosa-Padilla, PhD⁶  Nesrine Radwan, MD, PhD⁷  Sangeetha Siniah, MBBS, MRCPCH⁸  Kohsuke Imai, MD, PhD⁸  Brian Eley, MD⁹  Theresa Cole, MD, PhD^1,10  Adam Klocperk, MD, PhD^1,11  Monia Ouederni, MD^1,12  Michael Levin, MD, PhD^1,12  Ebtihal Kamal, MD^1,13  Claudio Pignata, MD, PhD^1,14  Sonia Qureshi, MBBS, FCPS, MSc^1,15  Nima Rezaei, MD, PhD^1,16  Intisar Abdel-Hakam Taha, MD, SMSB^1,17  Nita Radhakrishnan, MD, PhD^1,18  Isabella Quinti, MD, PhD^1,18  Ramsay Fuleihan, MD^1,19  Roberto Paganelli, MD^2,20  Antonio Condino-Neto, MD, PhD^2,21  Elizabeth Garabedian, RN, MSLS^2,22  Carla Irani, MD, MSCE^2,23  Alla Volokha, MD, PhD, DSc^2,24  Tomas Milota, MD, PhD^2,25  Ben Van Dort, BN^2,26  Luciana K. Tanno, MD, Ph.D^2,27  Nelson A. Rosario Filho, MD, PhD^2,28  Emilia Faria, MD^2,29  Mary Hintermeyer, BN^3,30  Nahla H.H. Erwa, DipRCPATH^3,31  José A. Ortega-Martell, MD^3,32  Juan C. Aldave, MD^3,32  Zeinab A. El-Sayed, MD, PhD^3,32  Nermeen Galal, MD, PhD^3,33  John Routes, MD, PhD^3,34  Farah N. Qamar, MBBS, FCPS, MSc, DHPE, FRCP^3,35 

[1] Corresponding author. Madinaty, V 202, G 12, Cairo, 19519, Egypt.;Desbrest Institute of Epidemiology and Public Health, UMR UA11, INSERM, University of Montpellier, Montpellier, France;Network of Immunity in Infection, Malignancy and Autoimmunity (NIIMA), Universal Scientific Education and Research Network (USERN), Tehran, Iran;Allergy and Clinical Immunology Unit, Pediatric Department, AlSabah Hospital, Faculty of Medicine, Kuwait University, Kuwait;Allergy and Clinical Immunology, Hospital Nacional Edgardo Rebagliati Martins, Peru;Allergy and Immunology Unit- Soba University Hospital and Faculty of Medicine, University of Khartoum, Sudan;Clínica de Alergia Pediátrica- Mexico;Department of Allergy and Clinical Immunology- Children's Hospital of Wisconsin, Medical College of Wisconsin, USA;Department of Allergy and Immunology- Royal Children's Hospital, Australia;Department of Immunology, Institute of Biomedical Sciences, University of Sao Paulo, Sao Paulo, Brazil;Department of Immunology, Pasteur Institute of Algeria, Algeria;Department of Immunology, University Hospital Motol and the 2nd Faculty of Medicine, Charles University, Czech Republic;Department of Internal Medicine and Clinical Immunology, Hotel Dieu de France Hospital, Beirut, Lebanon;Department of Medicine and Sciences of Aging, University “G. D’Annunzio”, Chieti-Pescara – and YDA, Institute of Clinical Immunotherapy and Advanced Biological Treatments, Pescara, Italy;Department of Molecular Medicine, PID Reference Centre, Sapienza University of Rome, Italy;Department of Pediatric Hematology Oncology, Super Specialty Pediatric Hospital and Post Graduate Teaching Institute Noida, Delhi NCR 201303, India;Department of Pediatrics and Chief, Allergy Immunology Unit, Advanced Pediatrics Centre, Post Graduate Institute of Medical Education and Research (PGIMER), Chandigarh, India;Department of Pediatrics and Child Health- Aga Khan University Hospital, Karachi, Pakistan;Department of Pediatrics, Federal University of Parana, Brazil;Department of Pediatrics, Immuno-hematology and Stem Cell Transplantation- Bone Marrow Transplantation Center Tunisia;Department of Pediatrics, Pulmonary Division- Allergy and Immunology Section, Qatar;Department of Pediatrics- Division of Immunology, Cairo University, Egypt;Department of Pediatrics-Tokyo Medical and Dental University, Japan;Department of Pulmonology, Division of Allergy, University Hospital of Montpellier, Montpellier, France;Division of Pediatric Allergy, University of Cape Town, South Africa;Gaafer Ibn Ouf Specialist Paediatric Hospital, Sudan;Hospital Tunku Azizah, Women and Children Hospital Kuala Lumpur, Malaysia;Immunoallergy Department, Coimbra Hospital and University Centre (CHUC), Portugal;Instituto Nacional de Pediatría, Immunodeficiencies Laboratory, Mexico;National Human Genome Research Institute, National Institute of Health, USA;Paediatric Infectious Diseases Unit, Red Cross War Memorial Children's Hospital and the Department of Paediatrics and Child Health, University of Cape Town, South Africa;Pediatric Allergy, Immunology and Rheumatology Unit, Children's Hospital, Ain Shams University, Cairo, Egypt;Pediatric Allergy, Immunology and Rheumatology, Columbia University Medical Center, New York, USA;Research Center for Immunodeficiencies, Children's Medical Center, Tehran University of Medical Sciences, Tehran, Iran;Translational Medical Sciences-Pediatric Immunology, Italy;

关键词: Primary immunodeficiency;    Asthma;    Atopic dermatitis;    IVIG;    Omalizumab;    Anaphylaxis;   

DOI  :  

来源: DOAJ

【 摘 要 】

Background: Allergies have long been observed in Inborn Errors of Immunity (IEI) and might even be the first presentation resulting in delayed diagnosis or misdiagnosis in some cases. However, data on the prevalence of allergic diseases among IEI patients are limited and contradictory. Objective: To provide a worldwide view of allergic diseases, across a broad spectrum of IEI, and their impact on the timely diagnosis of IEI. Methods: This is a worldwide study, conceived by the World Allergy Organization (WAO) Inborn Errors of Immunity Committee. A questionnaire was developed and pilot-tested and was sent via email to collect data from 61 immunology centers known to treat pediatric and/or adult IEI patients in 41 countries. In addition, a query was submitted to The United States Immunodeficiency Network (USIDNET) at its website. Results: Thirty centers in 23 countries caring for a total of 8450 IEI patients responded. The USIDNET dataset included 2332 patients. Data from responders showed that a median (IQR) of 16.3% (10–28.8%) of patients experienced allergic diseases during the course of their IEI as follows: 3.6% (1.3–11.3%) had bronchial asthma, 3.6% (1.9–9.1%) atopic dermatitis, 3.0% (1.0–7.8%) allergic rhinitis, and 1.3% (0.5–3.3%) food allergy. As per the USIDNET data, the frequency of allergy among IEI patients was 68.8% (bronchial asthma in 46.9%). The percentage of IEI patients who presented initially with allergic disorders was 8% (5–25%) and diagnosis delay was reported in 7.5% (0.9–20.6%). Predominantly antibody deficiencies had the highest frequency of allergic disease followed by combined immunodeficiency with a frequency of 40.3% (19.2–62.5%) and 20.0% (10–32%) respectively. As per the data of centers, anaphylaxis occurred in 25/8450 patients (0.3%) whereas per USIDNET dataset, it occurred in 249/2332 (10.6%); drugs and food allergy were the main causes in both datasets. Conclusions: This multinational study brings to focus the relation between allergic diseases and IEI. Major allergies do occur in IEI patients but were less frequent than the general population. Initial presentation with allergy could adversely affect the timely diagnosis of IEI. There is a need for policies to raise awareness and educate primary care and other referring specialties on the association of allergic diseases with IEI. This study provides a network among centers for future prospective studies in the field.

【 授权许可】

Unknown