The journal of allergy and clinical immunology. Global最新文献_第7页

Depression in children with asthma: Testing a cholinergically mediated “endotype” 哮喘儿童抑郁：胆碱能介导的“内窥镜”检测

The journal of allergy and clinical immunology. Global

Pub Date : 2025-10-10 DOI: 10.1016/j.jacig.2025.100581

Heather K. Lehman MD, FAAAAI , Beatrice L. Wood PhD, ABPP , Quratulain Humayun MBBS, MS, MD , Weyman Lam MD , Bruce D. Miller MD

Background

Asthma is not a single uniform disease; instead, it comprises various disease sub entities, or “endotypes,” with different etiologies and pathophysiologies. As asthma endotypes are discerned, individuals with asthma can be matched with optimal therapies for their specific asthma subtype. Depression is common in persons with asthma and associated with increased asthma-related morbidity and mortality. Depression in child asthma is associated with a predominance of parasympathetic/cholinergic activation over sympathetic activation. Cholinergic activity mediates airway smooth muscle constriction and has effects on airway inflammatory cells and mucus-secreting goblet cells.

Objective

The goal of the current study was to examine whether children with asthma who have more depressive symptoms have cholinergically mediated disease that is differentially responsive to short-acting anticholinergic therapy.

Methods

A total of 39 children with asthma (aged 7-17 years) and baseline obstruction on spirometry were evaluated for depressive symptoms by using the Children’s Depression Inventory. Spirometry was used to evaluate the children for improvement in airway function following treatment with a short-acting anticholinergic medication. Inflammation was assessed by determining fractional exhaled nitric oxide levels and peripheral blood eosinophil counts, and atopic status was assessed by skin prick testing and measurement of total serum IgE level.

Results

There was a statistically significant positive correlation between depressive symptoms and increase in FEV1 percent predicted value in response to ipratropium administration (rs = 0.344; P = .032).

Conclusion

Higher depressive symptom scores are associated with increased airway responsiveness to inhaled ipratropium in children with asthma and active airway obstruction. These findings suggest the potential for definition of an endotype of asthma with comorbid depression that may benefit from anticholinergic asthma therapies.

背景：哮喘不是一种单一的疾病；相反，它由具有不同病因和病理生理学的各种疾病子实体或“内型”组成。随着哮喘内型的识别，哮喘患者可以根据其特定的哮喘亚型与最佳治疗相匹配。抑郁症在哮喘患者中很常见，并与哮喘相关的发病率和死亡率增加有关。儿童哮喘抑郁症与副交感神经/胆碱能激活优于交感神经激活有关。胆碱能活性介导气道平滑肌收缩，并对气道炎症细胞和粘液分泌杯状细胞有影响。目的：本研究的目的是研究有更多抑郁症状的哮喘儿童是否患有对短效抗胆碱能治疗有差异反应的胆碱能介导性疾病。方法采用儿童抑郁量表对39例哮喘患儿（7 ~ 17岁）进行抑郁症状评估。使用肺活量测定法评估儿童在短效抗胆碱能药物治疗后气道功能的改善情况。通过测定呼出一氧化氮分数水平和外周血嗜酸性粒细胞计数来评估炎症，通过皮肤点刺试验和血清总IgE水平测定来评估特应性状态。结果异丙托品治疗组抑郁症状与FEV1 %预测值升高有显著正相关（rs = 0.344; P = 0.032）。结论哮喘合并活动性气道梗阻患儿的抑郁症状评分越高，气道对吸入异丙托品的反应性越高。这些发现提示有可能定义一种伴有共病性抑郁的哮喘内型，这种哮喘可能受益于抗胆碱能哮喘治疗。

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引用次数: 0

Maternal beliefs and asthma medication adherence during pregnancy 怀孕期间母亲的信念与哮喘药物依从性

The journal of allergy and clinical immunology. Global

Pub Date : 2025-10-10 DOI: 10.1016/j.jacig.2025.100579

Kelly Colas DO, PhD , Jennifer Namazy MD , Diana Johnson MS , Alec L. Todd MA , Christina D. Chambers PhD, MPH

Background

Maintaining optimal control of asthma during pregnancy is critical, as poorly controlled asthma is associated with negative health implications for both mother and baby. However, asthma medications are frequently discontinued by patients during pregnancy.

Objective

This study aimed to examine maternal beliefs, maternal characteristics, and infant birth outcomes associated with asthma medication nonadherence during pregnancy.

Methods

Between 2014 and 2022, pregnant asthmatic women were invited to respond to a 29-question asthma adherence survey. Using the t test, chi-square test, or Fisher exact test, we compared maternal characteristics and beliefs about asthma medications between those survey respondents with more reported adherence and those survey respondents with less reported adherence. Additionally, we used logistic regression with adjustment for select covariates to compare birth outcomes between those survey participants with more and those participants with less adherence.

Results

Of the 341 participants who responded to the asthma medication adherence question, 40 (11.7%) reported less adherence. Less adherence was associated with agreement with the statement that taking asthma medications in pregnancy is unsafe (P = .001), concern that a preventive/controller medication would harm the baby (P = .003), and disagreement with the belief that asthma medication is effective in controlling symptoms in pregnancy (P = .021). Less adherence was not associated with preterm delivery (odds ratio [OR] = 0.54 [95% CI = 0.12-2.38]), small for gestational age from the standpoint of birth weight (adjusted OR = 0.31 [95% CI = 0.06-1.76]), or small for gestational age from the standpoint of birth length (adjusted OR = 0.45 [95% CI = 0.05-4.43]).

Conclusion

Although we did not find an association between any maternal characteristics and asthma adherence, concerns about the safety and efficacy of asthma medication were significantly associated with asthma medication adherence.

背景：在怀孕期间保持对哮喘的最佳控制是至关重要的，因为控制不良的哮喘对母亲和婴儿的健康都有负面影响。然而，患者在怀孕期间经常停药。目的本研究旨在探讨妊娠期间哮喘药物依从性与母亲信念、母亲特征和婴儿出生结局的关系。方法在2014年至2022年期间，邀请怀孕的哮喘妇女回答29个问题的哮喘依从性调查。使用t检验、卡方检验或Fisher精确检验，我们比较了报告依从性较高的调查对象和报告依从性较低的调查对象之间的母亲特征和对哮喘药物的信念。此外，我们对选定的协变量进行调整，使用逻辑回归来比较依从性较高和依从性较低的调查参与者的出生结果。结果在回答哮喘药物依从性问题的341名参与者中，40名（11.7%）报告依从性较低。依从性较低与以下因素相关：同意妊娠期服用哮喘药物不安全（P = 0.001），担心预防性/控制性药物会伤害婴儿（P = 0.003），以及不同意哮喘药物对妊娠期控制症状有效（P = 0.021）。依从性较低与早产无关（优势比[OR] = 0.54 [95% CI = 0.12-2.38]），与出生体重的比值较小（调整后的OR = 0.31 [95% CI = 0.06-1.76]），与出生长度的比值较小（调整后的OR = 0.45 [95% CI = 0.05-4.43]）。结论虽然我们没有发现任何母亲特征与哮喘依从性之间的关联，但对哮喘药物安全性和有效性的担忧与哮喘药物依从性显著相关。

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引用次数: 0

Biomarker-guided IL-5 blockade achieves steroid-free MPO-ANCA seroconversion in EGPA 生物标志物引导的IL-5阻断在EGPA中实现无类固醇MPO-ANCA血清转化

The journal of allergy and clinical immunology. Global

Pub Date : 2025-10-10 DOI: 10.1016/j.jacig.2025.100580

Osamu Matsuno MD, PhD, Tansri Wibowo MD, Yutaka Ishida MD, Atsuhsi Ogata MD, PhD

This report describes a case of biopsy-confirmed EGPA in which early initiation of mepolizumab, guided by biomarker surveillance, resulted in steroid-free MPO-ANCA seroconversion. These findings suggest a potential steroid-sparing strategy for ANCA-positive EGPA.

本报告描述了一例活检证实的EGPA，在生物标志物监测的指导下，早期开始使用mepolizumab，导致无类固醇MPO-ANCA血清转化。这些发现为anca阳性EGPA提供了一种潜在的类固醇节约策略。

引用次数: 0

Evidence for dysbiosis in the gut microbiome of patients with systemic mastocytosis 系统性肥大细胞增多症患者肠道菌群失调的证据

The journal of allergy and clinical immunology. Global

Pub Date : 2025-10-09 DOI: 10.1016/j.jacig.2025.100578

Lauren E. Krausfeldt PhD , Vivian Cao MS , Richard Rodrigues PhD , Wendy A. Henderson PhD , Robin Eisch RN , Linda M. Scott LNP , Dean D. Metcalfe MD, MS , Hirsh D. Komarow MD

Background

Limited research studies have investigated the role of the gut microbiome in systemic mastocytosis (SM), which is characterized by an aberrant expansion of clonal mast cells in specific tissues including the skin, marrow, liver, and the gastrointestinal tract.

Objectives

We sought to investigate the relationship between the intestinal microbiome and clinical manifestations of SM.

Methods

The V4 region of the 16S rRNA gene was sequenced from stool samples of 22 patients with SM and 9 healthy controls. Microbial community composition, diversity, and functional genes inferred from 16S rRNA gene sequences were analyzed. ClinicalTrials.gov Identifier NCT00044122.

Results

Changes in microbial community composition were associated with SM, KIT D816V, and tryptase (PERMANOVA, P = .004, P = .05, P = .005, respectively). The differences with SM were driven by the composition of Firmicutes (P = .04) and an increase in Bacteroidetes abundance (P = .04). Predicted functions of the gut microbiome suggested that there were differences in metabolite profiles, including short-chain fatty acids, increased virulence factors, and decreased bacterial defense mechanisms in patients with SM. Dietary components were associated with symptoms, quality of life, and markers of mast cell activation and inflammation, as well as changes in microbial composition and predicted function in patients with SM.

Conclusions

Dysbiosis of the gut microbiome is evident in patients with SM and is seemingly associated with mast cell activation. In addition, diet may further alter microbial composition and metabolism in the gut of patients with SM.

背景有限的研究调查了肠道微生物组在系统性肥大细胞增多症（SM）中的作用，该疾病的特征是克隆肥大细胞在特定组织（包括皮肤、骨髓、肝脏和胃肠道）中异常扩增。目的探讨肠道菌群与SM临床表现的关系。方法对22例SM患者和9例健康对照者粪便中16S rRNA基因V4区进行测序。分析了16S rRNA基因序列推断的微生物群落组成、多样性和功能基因。ClinicalTrials.gov标识符NCT00044122。结果微生物群落组成的变化与SM、KIT D816V和胰蛋白酶相关（PERMANOVA, P = 0.004, P = 0.05, P = 0.005）。与SM的差异是由厚壁菌门的组成（P = .04）和拟杆菌门丰度的增加（P = .04）引起的。肠道微生物组的预测功能表明，SM患者的代谢物谱存在差异，包括短链脂肪酸、毒力因子增加和细菌防御机制降低。饮食成分与SM患者的症状、生活质量、肥大细胞活化和炎症标志物以及微生物组成的变化和预测功能相关。结论SM患者肠道菌群失调明显，可能与肥大细胞活化有关。此外，饮食可能会进一步改变SM患者肠道中的微生物组成和代谢。

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引用次数: 0

Trends over a decade in the prevalence and eliciting dose of peanut and tree nut allergies in Japan 十年来日本花生和树坚果过敏的流行和诱发剂量的趋势

The journal of allergy and clinical immunology. Global

Pub Date : 2025-10-08 DOI: 10.1016/j.jacig.2025.100582

Hitomi Kubota MD , Tatsuki Fukuie MD, PhD , Sayaka Hamaguchi MD , Seiko Hirai MD , Kenji Toyokuni MD, PhD , Kiwako Yamamoto-Hanada MD, PhD , Akira Ishiguro MD, PhD , Yukihiro Ohya MD, PhD

Background

The prevalence of tree nuts allergy is rapidly increasing in Japan, yet the eliciting dose (ED) for tree nuts, particularly in Asian populations, has not been sufficiently investigated.

Objective

This study aimed to quantify EDs for peanut, cashew nut, and walnut in Japanese patients with food allergies and to investigate longitudinal changes in these values.

Methods

We retrospectively analyzed 1,275 oral food challenge results for patients with diagnosed peanut, cashew nut, or walnut allergy, conducted between November 2013 and December 2023 at a single allergy center. EDs were calculated using the Weibull, log-normal, and log-logistic models, and bivariate survival analysis was used to examine the relationship between allergen-specific IgE levels and ED causing reaction in 5% of the allergic population (ED₀₅).

Results

The ED₀₅ values were determined as 4.88 mg for peanut, 0.53 mg for cashew nut, and 4.37 mg for walnut. When the component-specific IgE was set at 50.0 kU_A/L, ED₀₅ decreased to 3.20 mg, 0.55 mg, and 1.92 mg, respectively. Notably, a marked decline in walnut ED was observed over time.

Conclusions

EDs in Japanese tree nut–allergic populations are decreasingly aligned with those reported in Western countries, reflecting shifts in dietary habits and allergen exposure. The findings of the dynamic nature of threshold dose distributions over time, even within the same region, emphasize the necessity for periodic reassessment of allergen risk thresholds to ensure optimal patient safety.

背景：在日本，树坚果过敏的患病率正在迅速上升，但树坚果的诱发剂量（ED），特别是在亚洲人群中，尚未得到充分的调查。目的本研究旨在量化日本食物过敏患者对花生、腰果和核桃的ed，并调查这些值的纵向变化。方法回顾性分析2013年11月至2023年12月在单一过敏中心进行的1275例诊断为花生、腰果或核桃过敏的患者口服食物挑战结果。使用Weibull、log-normal和log-logistic模型计算ED，并使用双变量生存分析来检查5%过敏人群中过敏原特异性IgE水平与ED引起反应之间的关系（ED05）。结果花生、腰果和核桃的ED05值分别为4.88 mg、0.53 mg和4.37 mg。当组分特异性IgE为50.0 kUA/L时，ED05分别降至3.20 mg、0.55 mg和1.92 mg。值得注意的是，随着时间的推移，核桃ED明显下降。结论：日本树坚果过敏人群的seds与西方国家报告的seds一致，反映了饮食习惯和过敏原暴露的变化。阈值剂量分布随时间的动态特性的研究结果，甚至在同一地区，强调了定期重新评估过敏原风险阈值以确保最佳患者安全的必要性。

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引用次数: 0

Drug-induced enterocolitis syndrome with amoxicillin-containing products: A review of cases reported to the FDA adverse event reporting system and published literature 含阿莫西林产品引起的药物性小肠结肠炎综合征：对FDA不良事件报告系统和已发表文献报告的病例回顾

The journal of allergy and clinical immunology. Global

Pub Date : 2025-10-03 DOI: 10.1016/j.jacig.2025.100576

Maya Beganovic PharmD, MPH, Rachna Kapoor PharmD, MBA, S. Christopher Jones PharmD, MPH, MS, Jenni Yoon Lee MD

Background

Drug-induced enterocolitis syndrome (DIES) is a rare, non–IgE-mediated allergic reaction occurring 1 to 4 hours after drug ingestion.

Objective

The purpose of this evaluation was to describe postmarketing reports of DIES in patients receiving amoxicillin-containing products.

Methods

We queried the US Food and Drug Administration Adverse Event Reporting System database and the medical literature (Embase, PubMed) for cases of DIES in patients receiving amoxicillin-containing products through September 26, 2024. Cases were included if DIES was diagnosed via a positive result of a graded challenge with vomiting occurring 1 to 4 hours after ingestion, absence of classic IgE-mediated symptoms, and at least 3 minor criteria having been met. Causality was assessed using the World Health Organization Uppsala Monitoring Centre system for standardized case causality assessment.

Results

After the application of inclusion and causality criteria, 12 cases were identified (median case patient age 6 years). Most of the case patients required medical intervention, including intravenous fluids (10 of 12), emergency department visits or hospitalization (9 of 12), and supportive treatments. Of the 12 case patients, 6 were reported as having leukocytosis with neutrophilia. All cases resolved within 24 hours after drug administration and supportive care.

Conclusion

This case series summarizes reports of DIES associated with amoxicillin-containing product use and highlights the management, pathogenesis, and recent changes to the US prescribing information.

药物性小肠结肠炎综合征（DIES）是一种罕见的非ige介导的过敏反应，发生在药物摄入后1 ~ 4小时。目的本评价的目的是描述服用含阿莫西林产品的患者死亡的上市后报告。方法查询美国食品药品监督管理局不良事件报告系统数据库和医学文献（Embase, PubMed），查询截至2024年9月26日服用含阿莫西林产品的患者死亡病例。如果在摄入后1至4小时出现呕吐，没有典型的ige介导的症状，并且至少满足3个次要标准，通过分级挑战阳性结果诊断为死亡，则纳入病例。使用世界卫生组织乌普萨拉监测中心标准化病例因果关系评估系统评估因果关系。结果应用纳入标准和因果关系标准后，确定了12例（中位病例患者年龄6岁）。大多数病例患者需要医疗干预，包括静脉输液（12例中的10例）、急诊或住院（12例中的9例）以及支持性治疗。在12例患者中，6例报告有白细胞增多伴中性粒细胞增多。所有病例均在给予药物治疗和支持性护理后24小时内解决。结论：本病例系列总结了与阿莫西林相关的死亡报告，并强调了管理、发病机制和美国处方信息的最新变化。

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引用次数: 0

Specialist care improves diagnosis and treatment for patients with chronic spontaneous urticaria: A real-world survey 专科护理改善慢性自发性荨麻疹患者的诊断和治疗：一项真实世界的调查

The journal of allergy and clinical immunology. Global

Pub Date : 2025-09-30 DOI: 10.1016/j.jacig.2025.100573

Giselle Mosnaim MD , James Hetherington BSc , Aaron Keal MSc , Michael Holden MD , Arpamas Seetasith PhD

Background

Although effective treatments and guidelines for chronic spontaneous urticaria (CSU) are available, patients often experience diagnostic delays and undertreatment.

Objective

Our aim was to assess diagnostic differences and receipt of biologic treatment in patients with CSU across physician specialties.

Methods

Data from the Adelphi Real World CSU Disease-Specific Programme survey of physicians, including specialists (allergists/immunologists [n = 49] and dermatologists [n = 51]) and primary care physicians (PCPs) (n = 20), and their consulting patients with CSU (n = 1082) (United States, December 2020-October 2021) were analyzed. Logistic and Cox proportional hazard regressions were used to assess associations between outcomes and physician specialty.

Results

CSU was most often diagnosed by a specialist, even though the physician whom most patients consulted first was a PCP. The median time to diagnosis was shorter for specialists than for PCPs: patients who first consulted a PCP were more likely to experience a delay to CSU diagnosis than were patients who first consulted a specialist (odds ratio for an allergist/immunologist = 4.89 [P < .001]). Further, patients who first consulted a PCP versus a specialist were more likely to receive an initial non-CSU diagnosis (odds ratio for an allergist/immunologist vs a PCP = 2.70, [P = .002]). Patients received biologic treatment faster if diagnosed by an allergist/immunologist (hazard ratio = 0.35 vs PCPs [P = .006]), and they were more likely to receive omalizumab at the recommended dose (300 mg every 4 weeks).

Conclusion

For patients with CSU, consultation with an allergist/immunologist or dermatologist versus with a PCP was associated with faster time to CSU diagnosis and guideline-recommended treatment with omalizumab. This is the first study providing clear evidence that patients with CSU would benefit from specialist consultation.

背景：虽然慢性自发性荨麻疹（CSU）有有效的治疗方法和指南，但患者经常经历诊断延迟和治疗不足。目的：我们的目的是评估不同医师专业的CSU患者的诊断差异和生物治疗的接受情况。方法分析来自Adelphi Real World CSU疾病特异性计划的医生调查数据，包括专科医生（过敏症专家/免疫学家[n = 49]和皮肤科医生[n = 51]）和初级保健医生（pcp）（n = 20），以及他们咨询的CSU患者（n = 1082）（美国，2020年12月- 2021年10月）。使用Logistic和Cox比例风险回归来评估结果与医生专业之间的关联。结果scsu多数由专科医生诊断，尽管大多数患者首先咨询的医生是PCP。专科医生的中位诊断时间比专科医生短：首次咨询专科医生的患者比首次咨询专科医生的患者更有可能延迟CSU诊断（过敏症专科医生/免疫学家的优势比= 4.89 [P < .001]）。此外，与专科医生相比，首次咨询PCP的患者更有可能获得最初的非csu诊断（过敏症专科医生/免疫学家与PCP的比值比= 2.70,[P = .002]）。如果由过敏症专家/免疫学家诊断，患者接受生物治疗的速度更快（风险比= 0.35 vs pcp [P = 0.006]），并且他们更有可能接受推荐剂量（每4周300 mg）的omalizumab。结论：对于CSU患者，与PCP相比，咨询过敏症专科医生/免疫学家或皮肤科医生与CSU诊断和指南推荐的omalizumab治疗的时间更快相关。这是第一个提供明确证据的研究，表明CSU患者可以从专家咨询中获益。

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引用次数: 0

Sublingual immunotherapy in children with asthma: A population-based register study 儿童哮喘的舌下免疫治疗：一项基于人群的登记研究

The journal of allergy and clinical immunology. Global

Pub Date : 2025-09-30 DOI: 10.1016/j.jacig.2025.100574

Jon R. Konradsen MD, PhD , Cecilia Lundholm PhD , Anna M. Hedman PhD , Caroline Stridsman PhD , Hanna Karim MD , Bronwyn K. Brew PhD , Emma Caffrey Osvald MD, PhD , Samuel Rhedin MD, PhD , Maria Ingemansson MD, PhD , Catarina Almqvist MD, PhD

Background

Daily sublingual immunotherapy (SLIT) for 3 years reduces symptoms of allergic disease and induces tolerance. Real-life data from children with asthma receiving SLIT are scarce.

Objective

We used population-based data to describe characteristics, SLIT duration, and changes in morbidity in children with asthma prescribed SLIT.

Methods

The study included children (5-17 years) with asthma who were prescribed SLIT, and who were registered in the Swedish National Airway Register (SNAR) before SLIT initiation (N = 1,514), of whom 782 had post-SLIT recordings in the SNAR. Age, sex, Asthma Control Test score (≤19 denotes uncontrolled asthma), spirometry data, number of SLIT tablets dispensed, and socioeconomic background were extracted from the SNAR and other national registers. SLIT duration was classified as <4, ≥4, >12, or >24 months.

Results

SLIT was more common in boys (69%) and adolescents (71%). Most children had parents with higher education (70%), and 25% had uncontrolled asthma. SLIT duration of ≥4, >12, and >24 months was identified in 86%, 50%, and 30%, respectively. Parents with higher education were associated with SLIT duration of ≥4 months (odds ratio = 3.69; 95% confidence interval 2.75-4.96). SLIT duration of >12 months was associated with lower risk of post-SLIT uncontrolled asthma (adjusted odds ratio = 0.49; 95% confidence interval, 0.25-0.99). No associations were found between SLIT duration of ≥4, >12, or >24 months and changes in spirometry.

Conclusion

Longer SLIT duration was associated with higher education in parents and lower risk of uncontrolled asthma. Measures to improve persistence in SLIT in children with asthma may have important clinical implications.

每日舌下免疫治疗（SLIT） 3年可减轻过敏性疾病的症状并诱导耐受性。接受SLIT治疗的哮喘儿童的真实数据很少。目的：我们使用基于人群的数据来描述使用SLIT治疗的哮喘儿童的特征、SLIT持续时间和发病率变化。方法本研究纳入了5-17岁的哮喘患儿，这些患儿在SLIT开始前已在瑞典国家气道登记（SNAR）中登记（N = 1514），其中782人在SLIT后的SNAR中有记录。年龄、性别、哮喘控制测试分数（≤19表示哮喘未控制）、肺量测定数据、SLIT片剂配发数量和社会经济背景从SNAR和其他国家登记册中提取。SLIT持续时间分为4个月、≥4个月、12个月和24个月。结果slit在男孩（69%）和青少年（71%）中较为常见。大多数儿童的父母受过高等教育（70%），25%患有未控制的哮喘。裂隙持续时间≥4个月、≥12个月和≥24个月的分别为86%、50%和30%。父母受过高等教育与SLIT持续时间≥4个月相关（优势比= 3.69；95%可信区间2.75-4.96）。SLIT持续时间为12个月与SLIT后未控制哮喘的风险较低相关（校正优势比= 0.49；95%可信区间为0.25-0.99）。SLIT持续时间≥4个月、12个月或24个月与肺活量测定变化无关联。结论SLIT持续时间越长，父母受教育程度越高，哮喘控制风险越低。改善哮喘患儿SLIT持续性的措施可能具有重要的临床意义。

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引用次数: 0

Berotralstat effectiveness and safety in patients with hereditary angioedema with normal C1 inhibitor 贝洛特司他在C1抑制剂正常的遗传性血管性水肿患者中的有效性和安全性

The journal of allergy and clinical immunology. Global

Pub Date : 2025-09-30 DOI: 10.1016/j.jacig.2025.100575

Matthew S. Buckland PhD, FRCP , Isabelle Boccon-Gibod MD , Claire De Moreuil MD, PhD , Sébastien Sanges MD, PhD

Background

Hereditary angioedema (HAE) with normal C1 inhibitor (nC1-INH) is the least common endotype of HAE, a rare disorder with localized, intermittent attacks of soft tissue swelling. Prophylactic treatments are available across HAE endotypes, including berotralstat, a once-daily oral inhibitor of plasma kallikrein.

Objective

This European multicenter case series aimed to report treatment-related outcomes with berotralstat in patients with HAE-nC1-INH.

Methods

A retrospective case series analysis including observational data from patients in United Kingdom and French centers was performed. Patients were included if they had a genetic assay demonstrating a known HAE-nC1-INH–associated variant; or had a family history of angioedema and normal C1-INH level and function, no response to antihistamine treatment, met predefined diagnostic criteria for HAE-nC1-INH, and had been prescribed berotralstat 150 mg daily for at least 6 months. Data were collected from the patients’ records using a standardized form.

Results

Four female and 2 male patients with HAE-nC1-INH were included from 4 centers in France and the United Kingdom. Duration of berotralstat treatment at analysis ranged from 6 to 23 months. Five patients showed a response to berotralstat, observed as a 29-100% reduction in attack rates. Three patients experienced a reduction in attack severity from moderate to minor or mild. One patient reported adverse events during berotralstat initiation: nausea and diarrhea.

Conclusion

Long-term prophylactic berotralstat was effective in reducing HAE attacks for 5 of 6 patients with HAE-nC1-INH. No significant safety signals were noted.

背景：遗传性血管性水肿（HAE）伴正常C1抑制剂（nC1-INH）是HAE最不常见的内型，是一种罕见的疾病，伴有局部、间歇性软组织肿胀发作。预防性治疗可用于HAE内型，包括贝曲司他，一种每日一次的口服血浆钾likrein抑制剂。目的：该欧洲多中心病例系列旨在报告HAE-nC1-INH患者使用贝曲司他的治疗相关结果。方法采用回顾性病例系列分析，包括来自英国和法国中心的患者观察资料。如果患者有基因检测显示已知的hae - nc1 - inh相关变异，则纳入患者；或有血管性水肿家族史，C1-INH水平和功能正常，抗组胺治疗无反应，符合HAE-nC1-INH的预先诊断标准，并且服用贝格曲司他150mg /天至少6个月。使用标准化表格从患者记录中收集数据。结果来自法国和英国4个中心的HAE-nC1-INH患者，女性4例，男性2例。贝曲司他治疗的持续时间从6个月到23个月不等。5例患者对贝曲司他有反应，发作率降低29-100%。三名患者的发作严重程度从中度降至轻微或轻度。一名患者报告了贝曲司他起始治疗期间的不良事件：恶心和腹泻。结论6例HAE- nc1 - inh患者中有5例长期预防性贝曲司他可有效减少HAE发作。没有注意到明显的安全信号。

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引用次数: 0

Accelerated Hymenoptera testing in pediatric patients 加速膜翅目昆虫在儿科患者中的检测

The journal of allergy and clinical immunology. Global

Pub Date : 2025-09-26 DOI: 10.1016/j.jacig.2025.100570

Elizabeth S. Brunner MD, Jeffrey M. Chambliss MD, Timothy G. Chow MD

Background

Skin testing is an important component of evaluating Hymenoptera sensitization in patients presenting with a suspected Hymenoptera systemic reaction. Accelerated skin testing protocols have been reported in adults, but they have not been evaluated in children.

Objective

Our aim was to assess the safety of an accelerated Hymenoptera testing protocol in the evaluation of Hymenoptera venom hypersensitivity systemic reactions in pediatric patients.

Methods

A retrospective chart review of pediatric patients evaluated for Hymenoptera venom allergy at an academic allergy clinic from January 2017 through December 2024 was conducted. The children included in the study had a history consistent with a systemic reaction, as determined by a board-certified allergist to a sting and completed Hymenoptera allergy testing. The variables collected included demographic factors, age at initial reaction, age at skin testing, comorbid atopy, Mueller severity score reaction grade, reagents and concentrations used for skin testing, and skin testing results.

Results

A total of 61 patients were included. Of those patients, 80% presented with systemic reactions having a Mueller grade of 2 to 4. The median age at initial sting reaction was 5 years (interquartile range 2.75-8 years), and the median age at skin testing was 6 years (interquartile range 3-8 years). In all, 36 patients underwent accelerated intradermal testing (12 for flying Hymenoptera allergy, 24 for fire ant allergy); there were no adverse events or systemic reactions to testing.

Conclusion

Our findings provide initial evidence suggesting that an accelerated Hymenoptera skin testing protocol is safe in the pediatric population and encouraging subsequent larger, multicenter studies evaluating the safety of this diagnostic approach.

背景：皮肤试验是评估膜翅目全身反应疑似患者的膜翅目致敏性的重要组成部分。在成人中已有加速皮肤试验方案的报道，但尚未对儿童进行评估。目的：我们的目的是评估加速膜翅目检测方案在评估儿科患者膜翅目毒液过敏全身反应中的安全性。方法回顾性分析2017年1月至2024年12月在某学术变态反应诊所接受膜翅目毒液过敏评估的儿科患者。研究中包括的儿童有与全身性反应相一致的病史，由委员会认证的过敏专科医生对刺痛和完成的膜翅目过敏测试确定。收集的变量包括人口统计学因素、初始反应年龄、皮肤试验年龄、合并症特应性、穆勒严重程度评分反应等级、皮肤试验所用试剂和浓度以及皮肤试验结果。结果共纳入61例患者。在这些患者中，80%出现全身反应，穆勒评分为2至4级。首次刺痛反应时的中位年龄为5岁（四分位数范围2.75 ~ 8岁），皮试时的中位年龄为6岁（四分位数范围3 ~ 8岁）。总共有36例患者接受了皮内加速试验（12例为膜翅目昆虫过敏，24例为火蚁过敏）；没有不良事件或系统反应的测试。结论：我们的研究结果提供了初步证据，表明加速膜翅目皮肤检测方案在儿科人群中是安全的，并鼓励后续更大规模的多中心研究来评估这种诊断方法的安全性。

{"title":"Accelerated Hymenoptera testing in pediatric patients","authors":"Elizabeth S. Brunner MD, Jeffrey M. Chambliss MD, Timothy G. Chow MD","doi":"10.1016/j.jacig.2025.100570","DOIUrl":"10.1016/j.jacig.2025.100570","url":null,"abstract":"<div><h3>Background</h3><div>Skin testing is an important component of evaluating Hymenoptera sensitization in patients presenting with a suspected Hymenoptera systemic reaction. Accelerated skin testing protocols have been reported in adults, but they have not been evaluated in children.</div></div><div><h3>Objective</h3><div>Our aim was to assess the safety of an accelerated Hymenoptera testing protocol in the evaluation of Hymenoptera venom hypersensitivity systemic reactions in pediatric patients.</div></div><div><h3>Methods</h3><div>A retrospective chart review of pediatric patients evaluated for Hymenoptera venom allergy at an academic allergy clinic from January 2017 through December 2024 was conducted. The children included in the study had a history consistent with a systemic reaction, as determined by a board-certified allergist to a sting and completed Hymenoptera allergy testing. The variables collected included demographic factors, age at initial reaction, age at skin testing, comorbid atopy, Mueller severity score reaction grade, reagents and concentrations used for skin testing, and skin testing results.</div></div><div><h3>Results</h3><div>A total of 61 patients were included. Of those patients, 80% presented with systemic reactions having a Mueller grade of 2 to 4. The median age at initial sting reaction was 5 years (interquartile range 2.75-8 years), and the median age at skin testing was 6 years (interquartile range 3-8 years). In all, 36 patients underwent accelerated intradermal testing (12 for flying Hymenoptera allergy, 24 for fire ant allergy); there were no adverse events or systemic reactions to testing.</div></div><div><h3>Conclusion</h3><div>Our findings provide initial evidence suggesting that an accelerated Hymenoptera skin testing protocol is safe in the pediatric population and encouraging subsequent larger, multicenter studies evaluating the safety of this diagnostic approach.</div></div>","PeriodicalId":75041,"journal":{"name":"The journal of allergy and clinical immunology. Global","volume":"4 4","pages":"Article 100570"},"PeriodicalIF":0.0,"publicationDate":"2025-09-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145264782","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

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