Allergy Asthma and Clinical Immunology最新文献

{"title":"Sequential anaphylaxis and delayed rituximab-induced serum sickness with skin testing findings: a case report.","authors":"Shiori Mori, Utako Okata-Karigane, Masaki Arai, Chiaki Takahashi, Umi Tahara, Ayano Fukushima-Nomura, Eri Matsuki, Katsunori Masaki, Takeya Adachi","doi":"10.1186/s13223-026-01050-0","DOIUrl":"10.1186/s13223-026-01050-0","url":null,"abstract":"<p><strong>Background: </strong>Rituximab can cause immediate and delayed immune reactions, including rituximab-induced serum sickness (RISS). However, the temporal and mechanistic relationship between these reactions is unclear. We report a case in which RISS developed several days after rituximab-induced anaphylaxis.</p><p><strong>Case presentation: </strong>A 70-year-old woman with stage I mucosa-associated lymphoid tissue lymphoma was initially treated with weekly rituximab monotherapy. Four days after the third infusion, she developed fever, nonpruritic erythema, and arthralgia, which resolved spontaneously. When rituximab was re-administered, she immediately developed symptoms consistent with anaphylaxis, prompting treatment discontinuation. One year later, rituximab was re-started due to slight disease progression. The infusion induced nasal congestion and hoarseness but was completed under hydrocortisone. Eight days later, she presented with fever, widespread pruritic plaques, vomiting, diarrhea, and hypotension. Laboratory testing revealed elevated inflammatory markers without evidence of bacterial infection. Human anti-chimeric antibody levels were markedly elevated (> 5000 ng/mL). Intradermal testing with rituximab induced both an immediate wheal and a delayed erythematous flare lasting several days, indicating coexisting immediate-type hypersensitivity and RISS.</p><p><strong>Conclusions: </strong>This case demonstrates that RISS may occur several days after the resolution of rituximab-induced anaphylaxis. When immediate reactions occur after rituximab administration, patient education to ensure prompt reporting of delayed reactions is essential. Skin testing supported the identification of sequential immediate and delayed rituximab hypersensitivity in this case.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":" ","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-07-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13617842/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148392455","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"A qualitative study of the impact of the COVID-19 pandemic on healthcare access and service delivery for people living with chronic obstructive pulmonary disease and asthma.","authors":"Katelyn Brehon, Mirsol Choi, Pam Hung, Maxi Miciak, Rachel K Lim, Mohit Bhutani, Kadija Perreault, Paul E Ronksley, Micheal K Stickland, Jason Weatherald, Douglas P Gross, Grace Y Lam","doi":"10.1186/s13223-026-01052-y","DOIUrl":"10.1186/s13223-026-01052-y","url":null,"abstract":"<p><strong>Background: </strong>Incidence of chronic obstructive pulmonary disease and asthma diagnosis were lower during and after the Coronavirus disease 2019 pandemic in Alberta, Canada. However, it is unknown whether incidences were actually lower or if the pandemic created circumstances where patients did not seek care. As such, the objective of the current study was to explore the impact of COVID-19 on patient and clinician experiences of healthcare access and delivery.</p><p><strong>Methods: </strong>The study was conducted between October 2023 and July 2024. We used interpretive description, a qualitative approach with the end-goal of informing clinical decisions. Analysis was informed by Braun and Clarke's six phases of reflexive thematic analysis.</p><p><strong>Results: </strong>We completed thirteen interviews. Two key themes were generated: (1) The pandemic impacted care-seeking behaviours; and (2) A time and place for virtual and in-person care. Clinicians discussed how access to entry points to the health system were impacted by the pandemic and highlighted how strategies to manage health and stressors impacted symptoms and subsequent care-seeking behaviours. Participants highlighted the positives of virtual and in-person care with the consensus that both are valuable.</p><p><strong>Conclusion: </strong>Future use of virtual care modalities should include a visual element at minimum and prioritize the therapeutic relationship.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":" ","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-06-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13617704/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148340893","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Preventive effect of heat-killed Lactiplantibacillus plantarum FM8 in an ovalbumin-induced food allergy murine model.","authors":"Hideaki Takahashi, Tadashi Fujii, Chikako Yamada, Kotoyo Fujiki, Kento Kuramitsu, Shoei Okuda, Mamoru Tanaka, Takayuki Asahina, Kohei Funasaka, Eizaburo Ohno, Yoshiki Hirooka, Takumi Tochio","doi":"10.1186/s13223-026-01048-8","DOIUrl":"10.1186/s13223-026-01048-8","url":null,"abstract":"<p><strong>Background: </strong>Paraprobiotics, the non-viable microbial cells with health benefits, have gained interest as candidates for preventing food allergies owing to their safety and stability. Heat-killed Lactiplantibacillus plantarum FM8 (FM8) has been reported to induce interleukin (IL)-10 production in dendritic cells in vitro; however, its in vivo efficacy as a standalone intervention remains unexplored. We investigated the preventive effect of heat-killed FM8 in a murine model of ovalbumin (OVA)-induced food allergy.</p><p><strong>Methods: </strong>BALB/c mice were assigned to control, OVA-induced allergy, or three FM8 treatment groups receiving FM8 at low (2 × 10⁹ CFU/day), medium (1 × 10¹⁰ CFU/day), or high (5 × 10¹⁰ CFU/day) doses. Food allergy was induced by repeated OVA sensitization and challenge. Allergy symptoms, OVA-specific immunoglobulin E (IgE), and IL-10 levels were measured. Tight junction gene expression, mucin production, gut microbiota composition, and short-chain fatty acids (SCFAs) were also analyzed.</p><p><strong>Results: </strong>FM8 supplementation attenuated allergic responses in a dose-dependent manner. Allergy symptom scores were reduced in the High group, and the rectal temperature decline following OVA challenge was ameliorated in both the Med and High groups. The sensitization-induced increase in OVA-specific IgE was also attenuated in these groups. IL-4 expression in Peyer's patches was reduced, whereas colonic IL-10 expression and serum IL-10 levels were increased in the High group. FM8 supplementation was further associated with increased ileal expression of tight junction-related genes (Occludin, Claudin-1 and Zo-1) and Muc2, along with a trend toward increased fecal mucin levels; however, no comparable changes in tight junction-related gene expression were observed in the colon. Although taxa with reported SCFA-producing potential were enriched in the High group, cecal acetate and propionate levels remained unchanged, while butyrate levels were decreased.</p><p><strong>Conclusions: </strong>FM8 may prevent food allergy by promoting IL-10-associated immunoregulation and enhancing gut barrier-related responses, without a corresponding increase in cecal SCFA concentrations. The contribution of SCFA-related mechanisms remains to be clarified, and further studies are needed to determine clinically feasible dosing and efficacy in humans.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":" ","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-06-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13555864/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148334330","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Mepolizumab reduces healthcare resource utilization in patients with chronic rhinosinusitis with nasal polyps: a linked EMR and claims-based pre-post study.","authors":"Anna Swenson, Waseem Ahmed, Jared Silver, Shiyuan Zhang, Amy Edgecomb, Lingjiao Zhang, Rafael Alfonso-Cristancho, Richard Gliklich","doi":"10.1186/s13223-026-01038-w","DOIUrl":"10.1186/s13223-026-01038-w","url":null,"abstract":"<p><strong>Background: </strong>Real-world evidence on the effectiveness of mepolizumab at reducing healthcare resource utilization (HCRU) and clinical symptoms in patients with chronic rhinosinusitis with nasal polyps (CRSwNP) is limited.</p><p><strong>Methods: </strong>This real-world study used linked electronic medical record and claims data from the OM1 Real-World Data Cloud of clinician networks in the US, including an ear, nose and throat physician registry. CRSwNP-related HCRU, procedures, concomitant medications, and sign and symptom outcomes were assessed in adult patients with CRSwNP who initiated mepolizumab on/after July 29, 2021 (index date), with ≥1 mepolizumab record, data available for ≥12 months pre- and ≥6 months post-index (follow-up/post-mepolizumab period).</p><p><strong>Results: </strong>There was a significant difference in CRSwNP-related HCRU 6-months post- versus pre-mepolizumab initiation (N = 245), mean difference in outpatient visits (95% confidence interval): -0.82(-1.10, -0.53), p < 0.0001; otolaryngologist visits: -0.35(-0.54, -0.16), p = 0.0004; allergist visits: -0.28(-0.43, -0.14), p = 0.0001. Similar reductions were observed for all-cause HCRU. At 6-months post-mepolizumab, proportion of patients with oral corticosteroid and oral antibiotic prescriptions substantially reduced (36% pre- vs. 27% post-; and 38% pre- vs. 23% post-, respectively). There were clinically meaningful improvements in symptoms recorded 6-months post- versus pre-mepolizumab, with fewer patients experiencing anosmia (-53%), facial pain/pressure (-35%), nasal discharge (-32%) and nasal obstruction (-29%).</p><p><strong>Conclusions: </strong>In real-world practice, mepolizumab is associated with reduced HCRU and improved clinical symptoms, potentially alleviating medication burden in patients with CRSwNP. These results indicate, beyond improvements shown in randomized controlled trials, mepolizumab may reduce HCRU disease burden as early as 6-months post-initiation. This study offers real-world insight that may support clinical decision-making with mepolizumab.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":" ","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-06-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13602526/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148297075","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Triggers, clinical spectra and outcomes of pediatric anaphylaxis in a tertiary center: impact of comorbidities and cofactors on severity.","authors":"Necmiye Keser-Ozturk, Yasir Maghdeed, Selcen Bozkurt, Melek Yorgun-Altunbas, Salim Can, Razin Amirov, Ramin Mahmudov, Burkay Cagan Colak, Emel Eksi-Alp, Sevgi Bilgic-Eltan, Ahmet Ozen, Safa Baris, Elif Karakoc-Aydiner","doi":"10.1186/s13223-026-01033-1","DOIUrl":"10.1186/s13223-026-01033-1","url":null,"abstract":"<p><strong>Purpose: </strong>Anaphylaxis is a potentially life-threatening systemic hypersensitivity reaction. While triggers, clinical manifestations, and severity are influenced by age and sociocultural factors, most evidence regarding the impact of comorbidities and cofactors comes from adult studies. This study aimed to characterize the triggers, clinical features, and outcomes of pediatric anaphylaxis in a tertiary care center, with a particular emphasis on risk factors for severity.</p><p><strong>Methods: </strong>We retrospectively reviewed records from August 2023-August 2024 at the European Allergy Academy and Clinical Immunology Center of Excellence in Istanbul. Children aged 0-18 years (n = 100) with anaphylaxis as defined by the 2020 World Allergy Organization (WAO) criteria were included. Demographic, clinical, and follow-up data were collected. The updated 2024 (WAO) grading system was used to classify severity.</p><p><strong>Results: </strong>Of the 3,959 records, 100 children fulfilled the inclusion criteria. A female predominance was noted in those ≤ 4 years, with males predominating thereafter. Medications were the leading trigger, followed by food, venom, and idiopathic causes. Foods were more prevalent among outpatients, and drugs were more prevalent among inpatients. Comorbidities were more common in hospital-onset anaphylaxis. Atopy was more common in food-induced anaphylaxis, whereas infections and polypharmacy were more common in drug-induced cases. Food-related reactions with endogenous factors were typically milder, whereas drug-related reactions-especially in older patients with polypharmacy or hospital-onset-tended to be more severe. Biphasic reactions were associated with delayed presentation, elevated WBC/ANC, and older age. Intramuscular adrenaline was administered in 95% of the patients, but prehospital autoinjector use was rare.</p><p><strong>Conclusion: </strong>Our findings underscore the need for early recognition and management, systematic evaluation of contributing factors, risk-adapted monitoring, and individualized follow-up in pediatric anaphylaxis patients.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":" ","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13531739/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148284260","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Declining venom immunotherapy: patient characteristics and clinical outcomes.","authors":"Simon Ueberschaar, Axel Trautmann, Johanna Stoevesandt","doi":"10.1186/s13223-026-01046-w","DOIUrl":"10.1186/s13223-026-01046-w","url":null,"abstract":"<p><strong>Background: </strong>A largely unknown proportion of Hymenoptera venom-allergic patients do not undergo venom immunotherapy (VIT) despite positive allergy testing and counselling. We aimed to identify factors associated with the refusal of VIT, and evaluate the natural course of venom allergy in untreated individuals.</p><p><strong>Methods: </strong>Out of 1163 candidates for VIT, 271 (23.3%) declined or postponed treatment for at least 12 months. Complete data from 166 of these patients, who were interviewed and counselled during routine follow-up, were available for retrospective evaluation.</p><p><strong>Results: </strong>Patients declining VIT were significantly more likely to be female (P = 0.012) and had a lower grade of index sting-induced anaphylaxis (P < 0.001) compared to those who accepted treatment. Main reasons for deciding against VIT were that it was considered too time-consuming (39.8%) or perceived as unnecessary (24.1%). A minority of patients (6.6%) declined VIT due to fear of side effects; 7.2% perceived the VIT-associated risk as high to extremely high. Most (69.9%) reported routinely carrying an epinephrine autoinjector. The re-sting rate was 39.2% over a median follow-up of 5 years. Among 65 re-exposed patients, 13 (20%) reported an anaphylactic sting-reaction, with only two cases classified as severe. In no case did a severe relapse occur following a mild index sting reaction.</p><p><strong>Conclusion: </strong>In venom-allergic patients hesitant about VIT, treatment choices should be guided by a shared decision-making process considering risk factors for severe anaphylaxis and the degree of sting exposure, together with the patient's personal needs and preferences.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":"22 1","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-06-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13270586/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148260558","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Longitudinal FeNO monitoring in pediatric asthma: association of FeNO and its trajectory patterns with asthma control status.","authors":"Xiaolan Ying, Yiting Chen, Lu Liu, Yufen Wu, Jiajun Yuan, Li Hong, Yong Yin","doi":"10.1186/s13223-026-01044-y","DOIUrl":"10.1186/s13223-026-01044-y","url":null,"abstract":"<p><strong>Background: </strong>Fractional exhaled nitric oxide (FeNO) is a non-invasive biomarker of type 2 inflammation with potential for personalized asthma management. However, the longitudinal patterns of FeNO and their association with childhood asthma control remain insufficiently characterized.</p><p><strong>Methods: </strong>This cohort study enrolled children with newly diagnosed asthma from Shanghai Children's Medical Center. Participants underwent baseline assessments and serial FeNO measurements at diagnosis and at 3-month intervals over 12 months. Asthma control was assessed at 12 months using Global Initiative for Asthma (GINA) criteria. Longitudinal trajectories of FeNO were identified using group-based trajectory modeling (GBTM). Associations of time-specific FeNO levels and FeNO trajectory group with uncontrolled asthma at 12 months were analyzed using logistic regression.</p><p><strong>Results: </strong>Among 142 children (mean age 6.92 ± 2.08 years; 71.1% male), 125 (88.0%) achieved controlled asthma and 17 (12.0%) had uncontrolled asthma at 12 months. Elevated FeNO at 3 and 12 months was significantly associated with increased odds of uncontrolled asthma after adjusting for covariates. Longitudinal FeNO levels showed two distinct trajectories: low-stable (80.3% of participants) and high-rising (19.7%). Compared with the low-stable group, the high-rising group had significantly older age, later wheezing onset, higher serum total immunoglobulin E (IgE) and eosinophil percentages (all p < 0.05). Children in the high-rising trajectory group had higher odds of uncontrolled asthma (OR = 5.86; 95% CI: 1.28-29.57; p = 0.025).</p><p><strong>Conclusions: </strong>Elevated FeNO levels during follow-up and a high-rising FeNO trajectory are associated with uncontrolled asthma at 12 months in children with newly diagnosed asthma. Longitudinal FeNO monitoring may help identify children at increased risk of poor asthma control and support more individualized asthma management.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":" ","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-06-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13560344/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148240717","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Oral immunotherapy for the treatment of pumpkin seed allergy: a real-world case series.","authors":"Victor Paradis, Meghan Kanou, Louis Paradis, Roxane Labrosse, Kathryn Samaan, Philippe Bégin, Florence Gingras-Lessard, François Graham, Anne Des Roches","doi":"10.1186/s13223-026-01043-z","DOIUrl":"10.1186/s13223-026-01043-z","url":null,"abstract":"<p><strong>Background: </strong>Pumpkin seed, a member of the Cucurbitaceae family, is increasingly consumed because of its high protein content and perceived health benefits. Along with its growing use, cases of pumpkin seed allergy are being reported. However, data on pumpkin seed allergy and oral immunotherapy (OIT) remain scarce.</p><p><strong>Methods: </strong>We conducted a retrospective chart review at a tertiary pediatric center (Sainte-Justine University Hospital Center, Montreal, Canada) including all patients who initiated or completed pumpkin seed OIT since 2019. OIT protocols were individualized, with dose increases typically performed every four weeks. Target maintenance doses were at least 300 mg of pumpkin seed protein.</p><p><strong>Results: </strong>Eleven patients (median age at OIT initiation: 6.5 years; range 1-12) underwent pumpkin seed OIT. Ten patients (91%) reached maintenance dosing within a median of 9.5 months (range 6-22) while one patient discontinued OIT due to persistent abdominal pain. No anaphylactic reactions occurred at home during treatment. Two anaphylactic reactions requiring epinephrine occurred during in-clinic up-dosing visits in a single patient, despite this, the patient ultimately achieved maintenance. Gastrointestinal and oral symptoms were the most frequent adverse events and were generally managed with temporary premedication. Most patients (91%) underwent concomitant multi-food OIT. Patients with significant adverse reactions had high ratios of pumpkin-specific IgE to total IgE.</p><p><strong>Conclusion: </strong>Pumpkin seed OIT appears feasible in a highly atopic pediatric population, with a safety profile comparable to that reported for OIT to other food allergens. Given the increasing dietary exposure to pumpkin seeds, larger prospective studies are needed to better define risk factors and long-term outcomes.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":" ","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-06-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13450486/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"148151350","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Comprehensive care for hereditary angioedema: lessons learned from HAEmophilia Treatment Centers.","authors":"Kelsey Uminski, Dawn Goodyear","doi":"10.1186/s13223-026-01042-0","DOIUrl":"10.1186/s13223-026-01042-0","url":null,"abstract":"<p><p>Hereditary angioedema (HAE) is a rare, chronic disorder that requires coordinated, multidisciplinary care extending beyond acute attack management. Drawing on the successful comprehensive care framework established in Hemophilia Treatment Centers, this manuscript proposes a structured model for HAE care that emphasizes centralized care coordination, multidisciplinary teams, emergency preparedness, patient and caregiver education, psychosocial support, equitable access to emerging therapies, and integrated research and advocacy. By adapting lessons learned from hemophilia care, the proposed model aims to improve patient outcomes, empower individuals living with HAE, and provide a roadmap for implementing standardized, patient-centered care in rare disease management.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":"22 1","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-05-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13169828/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147935204","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}

{"title":"Unrecognized ciliary motility disorders in neutrophilic severe asthma exacerbations.","authors":"Sarita Thawanaphong, Lucia Gonzalez-Bravo, Katherine Radford, Melanie Kjarsgaard, Carmen Venegas Garrido, Sergey Fedosenko, Terence Ho, Lindsey Dyment, Nadia Suray Tan, Kayla Zhang, Adil Adatia, Manali Mukherjee, Myrna Dolovich, Parameswaran Nair","doi":"10.1186/s13223-026-01041-1","DOIUrl":"10.1186/s13223-026-01041-1","url":null,"abstract":"<p><strong>Background: </strong>Airway bacterial infections are frequent in severe asthma and are often under-appreciated as contributors to symptoms and exacerbations. We report our experience using integrative diagnostic methods to identify ciliary motility disorders as contributors to neutrophilic exacerbations in patients with severe asthma.</p><p><strong>Methods: </strong>Targeted exome sequencing for primary ciliary dyskinesia (PCD) was performed on 52 patients with severe asthma who met predefined criteria (≥ 3 respiratory infections or intense sputum neutrophilia, within a 2-year period, along with evidence of type 2 inflammation, including peripheral or sputum eosinophilia, elevated fractional exhaled nitric oxide (FeNO), or elevated serum immunoglobulin E), and without other obvious immunodeficiencies. A subset of patients with PCD-related gene variant(s) underwent ciliary beat frequency (CBF) analysis from nasal epithelial brushings (N = 20 with analyzable data), nasal nitric oxide (nNO) measurement (N = 15), and T1/2/17 sputum cytokine assays (N = 18).</p><p><strong>Results: </strong>Among 52 patients (mean age 54.2 ± 15.1 years; 59.6% female), 32(61.5%) had PCD-related gene variants. CBF was reduced in 19/20(95%) patients who underwent motility studies (mean CBF of 8.8 ± 2.7 Hz; normal 14.2 ± 1.0 Hz), and correlated significantly with FEV₁ (r_s=0.65, P = 0.0017) and inversely with peak sputum neutrophils (r_s=-0.62, P = 0.0097). Subnormal nNO levels (< 250 nL/min) were observed in 12/15(80%) patients; 3/15(20%) were < 77 nL/min (PCD threshold). 5 patients showed FeNO discordance (> 25 ppb). Cytokines associated with inflammasome activation were increased in sputum in majority of patients with PCD-related gene variants. Sputum-guided strategy and 7% hypertonic saline treatment enable significant inhaled corticosteroid doses reduction across the entire population and airway-eosinophilia subgroup (P = 0.0026 and P = 0.0273). Oral corticosteroid doses were reduced in 6/8(75%), and biologics were not initiated in 26/32(81.3%). FEV₁ improved by 130 ± 291mL, P = 0.0273.</p><p><strong>Conclusions: </strong>Ciliary dyskinesias are prevalent in patients with severe asthma. Identifying ciliary motility disorders can guide effective treatment and reduce unnecessary biologics use in severe asthma when symptoms are infection-driven. Even variants of uncertain significance in PCD-related genes may be associated with airway infections due to ciliary dysfunction, proven by ciliary beat frequency analysis. nNO could be a useful screening tool, but its optimal cutoff in this population needs further study.</p>","PeriodicalId":51302,"journal":{"name":"Allergy Asthma and Clinical Immunology","volume":" ","pages":""},"PeriodicalIF":2.4,"publicationDate":"2026-05-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13321632/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147857645","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}