Journal of Allergy and Clinical Immunology-In Practice最新文献

{"title":"Chronic norovirus infection in immunodeficiency: A UK national case series","authors":"Alexandros Grammatikos FRCPath , Anisha Mangtani MBBS , Mark Ponsford FRCPath , Stephen Jolles FRCPath , Elizabeth McDermott FRCPath , Sarah Johnston FRCPath , Marina Frleta-Gilchrist MRCP , Patrick Yong FRCPath , Fiona Moghaddas FRACP , Moira Thomas FRCPath , Smita Patel FRCPath , Caitlin Blundell MBiochem , Phil Bright FRCPath , Elizabeth Drewe FRCPath , Suzanne Elcombe FRCPath , Archana Herwadkar FRCPath , Sai Murng FRCPath , Da-In Kim MRCP , Mark Gompels FRCPath , David Lowe FRCPath","doi":"10.1016/j.jaip.2025.02.026","DOIUrl":"10.1016/j.jaip.2025.02.026","url":null,"abstract":"","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Pages 1489-1492"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143538072","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Five Patients With Two Novel Homozygous Variants in CD3 Subunits and Comprehensive Review of the Literature","authors":"Gamze Sonmez MD ,&nbsp;Alp Kazancıoglu MD ,&nbsp;Yigit Yazarkan ,&nbsp;Ates Kutay Tenekeci MD ,&nbsp;Ismail Yaz PhD ,&nbsp;Saliha Esenboga MD ,&nbsp;Diclehan Orhan MD, PhD ,&nbsp;Mirjam van der Burg PhD ,&nbsp;Ozden Sanal MD ,&nbsp;Ilhan Tezcan MD, PhD ,&nbsp;Deniz Cagdas MD, PhD","doi":"10.1016/j.jaip.2025.03.022","DOIUrl":"10.1016/j.jaip.2025.03.022","url":null,"abstract":"<div><h3>Background</h3><div>CD3 subunit deficiency (CD3SD) causes combined immunodeficiency (CID) and severe CID (SCID).</div></div><div><h3>Objective</h3><div>To elucidate the clinical, laboratory, and genetic features of patients with different CD3SD subtypes.</div></div><div><h3>Methods</h3><div>We evaluated the data of five patients with CD3ε (two), CD3γ (two), and CD3δ (one) deficiencies from our institution. In addition, we reviewed the medical literature for cases of CD3SD.</div></div><div><h3>Results</h3><div>We identified two novel homozygous CD3 variants. In addition, we identified 44 CD3SD cases in the literature. In total, we analyzed the results of 49 patients. Our review of the medical literature revealed 11, 12, 18, and three patients with CD3ε, CD3γ, CD3δ, and CD3ζ deficiency, respectively. Of the 49 patients, 40 had an SCID profile, whereas nine with CD3γ variants had a CID profile. The patients with SCID presented with typical symptoms, including recurrent infections (18 of 40; 45%), diarrhea (13 of 40; 33%), and candidiasis (12 of 40, 30%). Recurrent sinopulmonary infections (four of nine; 45%), thyroiditis (four of nine; 45%), and bronchiectasis (four of nine; 45%) were common in patients with CID. Almost 70% of patients with SCID (27 of 40) underwent hematopoietic stem cell transplantation.</div></div><div><h3>Conclusions</h3><div>Our results show that patients with CD3δ, CD3ε, and CD3ζ deficiencies typically present with a classic SCID phenotype. In contrast, patients with CD3γ deficiency may either show an SCID phenotype or a milder, less severe CID phenotype. Importantly, autoimmunity may be the sole manifestation of CD3γ deficiency.</div></div>","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Pages 1430-1439.e7"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143732940","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Hereditary α-tryptasemia is Associated With Anaphylaxis to Antibiotics and Monoclonal Antibodies","authors":"Peter Korošec PhD ,&nbsp;Jonathan J. Lyons MD ,&nbsp;Manca Svetina MSc ,&nbsp;Monika Koudová MD ,&nbsp;Martina Bittóová MSc ,&nbsp;Mihaela Zidarn MD, PhD ,&nbsp;Lenka Sedláčková MD ,&nbsp;Matija Rijavec PhD ,&nbsp;Peter Kopač MD, PhD","doi":"10.1016/j.jaip.2025.04.013","DOIUrl":"10.1016/j.jaip.2025.04.013","url":null,"abstract":"<div><h3>Background</h3><div>Hereditary α-tryptasemia, a genetic trait caused by increased α-tryptase copy number, is associated with idiopathic and venom anaphylaxis.</div></div><div><h3>Objective</h3><div>We aimed to determine the impact of tryptase genotypes on drug-induced anaphylaxis.</div></div><div><h3>Methods</h3><div>A prospective discovery cohort of 99 patients from a referral center in Slovenia with acute anaphylaxis to drugs underwent tryptase genotyping by droplet digital PCR. For validation, we included a cohort of 26 patients from the Czech Republic. Associated inciting agents and the severity of the reactions were subsequently examined.</div></div><div><h3>Results</h3><div>Hereditary α-tryptasemia was associated with drug-induced anaphylaxis with a prevalence of 13% (n = 13 of 99) in the discovery cohort and 15% in the validation cohort (n = 4 of 26). Hereditary α-tryptasemia was identified in every individual with elevated basal serum tryptase levels (11.6-21.9 ng/mL; n = 14) within both cohorts of patients. Hereditary α-tryptasemia was more prevalent in individuals with antibiotic- or mAb-induced anaphylaxis in both the discovery and validation cohorts (n = 13 of 51; 26%) compared to those with anaphylaxis resulting from neuromuscular blocking agents, nonsteroidal anti-inflammatory drugs, contrast, chlorhexidine, or other drugs (n = 5 of 74; 7%; <em>P</em> = .02; odds ratio = 4.1; 95% CI, 1.3-11.1). Overall, we found fewer individuals with no ⍺-tryptase than in the general population, and there was a trend for subjects with more ⍺-tryptase copies to have more severe reactions. Thus, among subjects with three ⍺-tryptase copies, the prevalence of severe anaphylaxis was 73%, compared with 59% with one to two ⍺-tryptase copies and 58% for subjects without ⍺-tryptase.</div></div><div><h3>Conclusions</h3><div>Risk for anaphylaxis to antibiotics and biologics is associated with inherited differences in α-tryptase–encoding copies at <em>Tryptase α/β1</em>.</div></div>","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Pages 1449-1456.e4"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144028061","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Reply to “The pathogenesis of primary acquired cold urticaria”","authors":"Alan Wanderer MD","doi":"10.1016/j.jaip.2025.03.041","DOIUrl":"10.1016/j.jaip.2025.03.041","url":null,"abstract":"","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Pages 1497-1498"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144212573","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Indoor Allergen Interventions in Homes and Schools for Managing Asthma","authors":"","doi":"10.1016/j.jaip.2025.05.006","DOIUrl":"10.1016/j.jaip.2025.05.006","url":null,"abstract":"","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Page 1266"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144213239","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Long-Term Efficacy and Safety of Benralizumab Treatment for PDGFRA-Negative Hypereosinophilic Syndrome","authors":"Fei Li Kuang MD, PhD ,&nbsp;Michelle A. Makiya MS ,&nbsp;JeanAnne M. Ware MSN, MPH ,&nbsp;Lauren Wetzler MHC, PA-C ,&nbsp;Celeste Nelson MS, CRNP ,&nbsp;Thomas Brown BSN, CCRP ,&nbsp;Paneez Khoury MD, MHS ,&nbsp;Amy D. Klion MD","doi":"10.1016/j.jaip.2025.03.016","DOIUrl":"10.1016/j.jaip.2025.03.016","url":null,"abstract":"<div><h3>Background</h3><div>Therapies for hypereosinophilic syndromes (HES) have variable efficacy and significant toxicity. Benralizumab (BENRA), an afucosylated monoclonal antibody that binds interleukin 5 receptor α expressed on eosinophils, reduced blood and tissue eosinophils in a 48-week, phase 2 study in <em>PDGFRA</em>-negative HES.</div></div><div><h3>Objective</h3><div>To assess safety and efficacy of long-term BENRA treatment in <em>PDGFRA</em>-negative HES.</div></div><div><h3>Methods</h3><div>Twenty <em>PDGFRA</em>-negative HES patients with severe and diverse clinical manifestations were treated in a double-blind, placebo-controlled clinical trial of BENRA (NCT02130882). Fourteen were followed on an open-label extension. Efficacy and safety were reviewed at each visit.</div></div><div><h3>Results</h3><div>Among 14 long-term participants, 3 withdrew owing to loss of efficacy, and 1 owing to death unrelated to drug after 2 to 6 years. The remaining 10 participants received BENRA for at least 8 years (maximum exposure 10.1 y). Peripheral eosinophil counts remained suppressed in all participants. Eight participants attempted to taper from monthly to every-2-month dosing, of which 5 resumed monthly dosing owing to increased symptoms and/or eosinophil count. There were 11 serious adverse events: 10 hospitalizations (3 for infections, 5 for surgeries, and 2 for drug desensitization or observation) and 1 death from antibiotic-associated toxic epidermal necrolysis. None were attributed to BENRA. Eight participants reported injection-site reactions (23 total), all mild in severity and self-limited. Three malignancies and no parasitic infections were reported. Despite sustained eosinophil depletion, immunoglobulin levels and peripheral T-, B-, and natural killer–cell counts were stable for the duration of the study.</div></div><div><h3>Conclusions</h3><div>Long-term BENRA therapy is effective and well-tolerated in HES patients without significant adverse effects.</div></div>","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Pages 1421-1429.e2"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143694460","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Phenotypic Characteristics of Occupational Asthma Caused by Persulfate Salts in Hairdressers: A Multicenter Cohort Study","authors":"Virginie Doyen MD, PhD ,&nbsp;Robin Thirionet MD ,&nbsp;Nicolas Migueres MD, PhD ,&nbsp;Olivier Vandenplas MD, PhD ,&nbsp;Joaquin Sastre MD ,&nbsp;Marcela Valverde-Monge MD ,&nbsp;Thomas Blix Grydeland MD, PhD ,&nbsp;Xavier Munoz MD, PhD ,&nbsp;Christian Romero-Mesones MD, PhD ,&nbsp;Hille Suojalehto MD, PhD ,&nbsp;Katri Suuronen PhD ,&nbsp;Vera van Kampen PhD ,&nbsp;Christian Eisenhawer MD ,&nbsp;Ilenia Folletti MD ,&nbsp;Iben Brock Jacobsen MD ,&nbsp;Jolanta Walusiak-Skorupa MD ,&nbsp;Paola Mason MD, PhD ,&nbsp;Alexandra M. Preiser MD ,&nbsp;Santiago Quirce MD, PhD ,&nbsp;Gareth Walters MB, ChB, MD ,&nbsp;Cecilie Svanes MD, PhD","doi":"10.1016/j.jaip.2025.03.015","DOIUrl":"10.1016/j.jaip.2025.03.015","url":null,"abstract":"<div><h3>Background</h3><div>The clinical and inflammatory characteristics of occupational asthma (OA) caused by persulfate salts (PS) in hair bleaches have not yet been comprehensively characterized.</div></div><div><h3>Objective</h3><div>This study aimed to compare the phenotypic characteristics of PS-induced OA with those of OA due to other low-molecular-weight (LMW) agents.</div></div><div><h3>Methods</h3><div>This study was conducted in a retrospective multicenter cohort of subjects with OA ascertained by a positive specific inhalation challenge (SIC). The clinical and inflammatory characteristics of hairdressers with PS-induced OA (n = 107) were compared with those of subjects who showed a positive SIC to isocyanates (n = 128) or various other LMW agents (n = 164).</div></div><div><h3>Results</h3><div>Subjects with PS-induced OA had a longer duration of exposure to the offending agent before the onset of asthma than those with OA caused the other LMW agents. They reported more frequently work-related rhinitis (76%) and showed a lower post-SIC level of fractional exhaled nitric oxide (median, 18 ppb [25th-75th percentile, 13–26]) compared with OA caused by both isocyanates (36%, <em>P</em> &lt; .001 and 35 ppb [21–80], <em>P</em> &lt; .001, respectively) and the other LMW agents (53%, <em>P</em> &lt; .001 and 27 ppb [14–52], <em>P</em> &lt; .001). Subjects with PS-induced OA showed the highest rate of isolated late asthmatic reactions (49%), but the difference reached statistical significance only when compared with LMW agents other than isocyanates (31%, <em>P</em> &lt; .002).</div></div><div><h3>Conclusions</h3><div>The PS-induced OA is associated with a higher prevalence of work-related rhinitis and lower levels of fractional exhaled nitric oxide compared with OA caused by other LMW agents. These findings further indicate substantial phenotypic heterogeneity among this broad category of agents.</div></div>","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Pages 1397-1404.e5"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143694465","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Reply to “Interrupting a vicious cycle in order to produce cessation of refractory chronic cough”","authors":"Woo-Jung Song MD, PhD ,&nbsp;Deepti Vellaichamy Manian MD ,&nbsp;Yeonhee Kim MD ,&nbsp;Mengru Zhang MD ,&nbsp;Alyn H. Morice MD","doi":"10.1016/j.jaip.2025.03.045","DOIUrl":"10.1016/j.jaip.2025.03.045","url":null,"abstract":"","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Pages 1502-1503"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144212570","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Early-Life Allergen Exposure and Its Influence on Risk of Atopic Disease","authors":"Elizabeth Huiwen Tham MRCPCH ,&nbsp;Soo-Jong Hong MD, PhD ,&nbsp;Eun Lee MD, PhD ,&nbsp;James E. Gern MD","doi":"10.1016/j.jaip.2025.02.043","DOIUrl":"10.1016/j.jaip.2025.02.043","url":null,"abstract":"<div><div>Childhood allergic diseases and asthma have their origins in early life, and allergen exposures during this period could be a critical determinant of the progression to tolerance versus disease. Mechanisms for sensitization may be different but overlapping for food and aeroallergen sensitization in children. This suggests differences in how exposure to food and aeroallergens influence allergic sensitization. For food allergy, introducing foods such as peanut and egg proteins into the diet at an early age reduces the risk of peanut and egg allergy, respectively, across a broad demographic, whereas evidence is less established for other foods. The relationship between allergen exposure and sensitization to aeroallergens is more complex but critical, given the close relationship between specific immunoglobulin E and respiratory disease. Several factors could mediate the progression from allergen exposure and allergic sensitization versus tolerance, including epithelial barrier function and altered immune development at the skin and mucosal surfaces, exposure to irritants and pollutants, and genetic susceptibility. Collectively, the current evidence base provides a compelling rationale for the primary prevention of food allergy by introducing common allergens such as peanut and egg early. In contrast, primary prevention of aeroallergen sensitization is more complex and perhaps more challenging to achieve by manipulating allergen exposures. Even so, recent advances in understanding how the microbiome and environmental toxins and irritants modulate the mucosal immune response have identified potential new strategies for primary prevention of food and aeroallergen sensitization.</div></div>","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Pages 1243-1253"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143694458","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Diagnostic Utility of Biomarkers in Anaphylaxis: A Systematic Review and Meta-Analysis","authors":"Roy Khalaf ,&nbsp;Connor Prosty MD ,&nbsp;William Davalan ,&nbsp;Elissa Abrams MD ,&nbsp;Mohammed Kaouache PhD ,&nbsp;Moshe Ben-Shoshan MD","doi":"10.1016/j.jaip.2025.04.008","DOIUrl":"10.1016/j.jaip.2025.04.008","url":null,"abstract":"<div><h3>Background</h3><div>Anaphylaxis is a life-threatening allergic reaction commonly triggered by food, venom, or drugs. Clinical criteria are central to diagnosing anaphylaxis. However, laboratory biomarkers could provide valuable confirmation when clinical diagnosis is challenging.</div></div><div><h3>Objective</h3><div>We aimed to evaluate key biomarkers including tryptase, histamine, platelet-activating factor (PAF), PAF-acetylhydrolase (PAF-AH), and urinary prostaglandin D2 (PGD2) for their diagnostic utility in anaphylaxis.</div></div><div><h3>Methods</h3><div>A systematic review was conducted following PRISMA-DTA (Preferred Reporting Items for a Systematic Review and Meta-analysis of Diagnostic Test Accuracy studies) guidelines. Studies published between 2004 and 2024 from Embase and MEDLINE were included if they evaluated the diagnostic test accuracy of tryptase, histamine, PAF, PAF-AH, or urinary PGD2 in confirmed anaphylaxis cases. Pooled sensitivity and specificity estimates were calculated using the diagmeta package in R.</div></div><div><h3>Results</h3><div>Twenty-eight studies with 18,749 patients were included, of whom 3329 had anaphylaxis. Tryptase was the most frequently studied biomarker (24 studies), with a pooled sensitivity and specificity of 0.49 and 0.82, respectively. Histamine had a pooled sensitivity of 0.76 and a specificity of 0.69. Limited data were available for PAF, PAF-AH, and urinary PGD2.</div></div><div><h3>Conclusions</h3><div>Studies suggest that tryptase remains the most widely used and accessible biomarker for diagnosing anaphylaxis mainly using the “Rule of Twos” diagnosis strategy. Histamine and urinary PGD2 show potential, though their application is limited by practical challenges. Further research is needed to establish the diagnostic roles of PAF and PAF-AH, particularly in non–IgE-mediated anaphylaxis pathways.</div></div>","PeriodicalId":51323,"journal":{"name":"Journal of Allergy and Clinical Immunology-In Practice","volume":"13 6","pages":"Pages 1342-1349.e12"},"PeriodicalIF":8.2,"publicationDate":"2025-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144057293","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":1,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}