Allergy & Rhinology最新文献

Background: Nasopharyngeal oncocytic lesions are a spectrum of benign lesions that represent a reactive or hyperplastic response to chronic inflammation. Though oncocytic lesions are typically asymptomatic, unilateral, and benign, this article discusses a rare case of large, bilateral oncocytic cysts and downstream otologic sequelae with a focus on identifying and discussing similar disease processes.

Methods: Case report and literature review.Case Presentation: A 67-year-old patient with 57 pack year smoking history presented for one year of left sided hearing loss and aural fullness. Clinic endoscopic exam demonstrated severe inflammatory and cystic changes lining the bilateral tori. Imaging and tissue sampling confirmed the cause was minor salivary gland cysts with papillary projections lined by oncocytic cells within bilateral tori tubarius. He was successfully treated with myringotomy with pressure equalizing tube, counseling on tobacco cessation, and surveillance with serial nasopharyngoscopy.

Conclusions: Chronic eustachian tube dysfunction is a possible rare presentation and sequelae of large oncocytic cysts of the nasopharynx. Oncocytic cysts should be considered on the differential diagnosis for nasopharyngeal masses causing such dysfunction.

Introduction: Eosinophilic esophagitis (EoE) is a clinico-pathological diagnosis characterized by esophageal dysfunction and eosinophilic infiltration of the esophagus. Demonstration of esophageal eosinophilia (more than 15 eosinophils/hpf) in biopsy specimen obtained by esophagogastroduodenoscopy (EGD) continues to be the gold standard for diagnosis and monitoring of response to therapy. There is a growing necessity for non-invasive biomarkers that can accurately diagnose this condition and assess response to therapy. While microRNAs (miRNA) are being investigated in allergic diseases, including EoE, not many studies have explored the role of salivary miRNAs in EoE. MiR-4668-5p is a particularly interesting candidate, as it is predicted to regulate TGF-beta signaling and has not previously been identified as a target in any allergy disease. We sought to further investigate the role of miR-4668 as a biomarker to characterize and monitor response to treatment with swallowed topical glucocorticoids.

Methods: After IRB approval, twenty-two adult patients with EoE were randomly enrolled to provide a saliva sample before and after 2 months of swallowed fluticasone therapy. Differences of miRNA expression before and after treatment were analyzed by paired T-test. A significance cutoff of <0.05 was used for all analyses.

Results: Expression of miR-4668 was higher in EoE vs. non-EoE subjects. The level of miR-4668 decreased in all subjects except one, with a mean fold change 0.49 ± 0.25. There was an association between miRNA expression and number of positive aeroallergens. The miR-4668 high group had a higher number of positive aeroallergen tests, while the miR-4668 low group had a greater number of subjects with drug allergies.

Conclusions: In this study, we identified that salivary miRNAs may serve as biomarkers to characterize EoE and response to topical corticosteroids. We specifically identified miR-4668 as a novel potential biomarker, which was not previously discovered as a target in EoE or any other allergic disease.

This is a series of 4 cases (3 therapeutic failure and 1 early relapse) in adult patients treated with allergen immunotherapy (AIT) for allergic rhinitis (AR) in our immunotherapy clinic, which treats 110 new patients per year. AIT includes both subcutaneous and sublingual routes. The current national/international AIT recommendations and the literature have been searched to identify guidance for the optimal management of therapeutic failure of AIT in AR. There is scant information available to support clinicians when treatment failure and/or intolerable side effects occur. The importance is highlighted for developing the guidance and evidence base for the benefit of this patient subgroup. The potential strategies that clinicians have proposed are discussed in this article, though it is acknowledged that these are mostly not evidence-based.

Background: Cryoablation of the posterior nasal nerve at the middle meatus has been shown to successfully treat nasal obstruction and symptoms of chronic rhinitis. Cryoablation of both the middle and inferior meatus has not yet been studied.

Objectives: To evaluate the safety and feasibility of cryoablation of the posterior nasal nerve at both the middle and inferior meatus locations to treat chronic rhinitis.

Methods: Participants underwent bilateral cryoablation of the posterior nasal nerve at both the middle meatus and inferior meatus and were assessed through 3 months post treatment. The primary endpoint is the change from baseline to 3-month follow-up in the reflective Total Nasal Symptom Score (rTNSS). Other assessments include additional patient-reported outcomes, physician assessment, and independent review and scoring of imaging.

Results: Thirty participants were enrolled at 3 US centers. There was a significant improvement from baseline in the median rTNSS (-4.0, P < .001) at 3 months. Statistically significant improvements from baseline (P < .001) were also observed with the Nasal Obstruction Symptom Score (NOSE), nasal symptom visual analog scale (VAS), Sino-Nasal Outcomes Score (SNOT-22), and mini Rhinoconjunctivitis Quality of Life Questionnaire (mini RQLQ). Clinical Global Impression - Improvement (CGI-I) indicated that 89.7% (26/29) of participants experienced improvement at 3 months. No serious adverse events were reported.

Conclusion: Cryoablation at both the middle meatus and inferior meatus appears to be a safe and feasible option for treatment of chronic rhinitis. In this feasibility study, there is significant improvement in symptoms post treatment. Adverse events are minor and transient.

Background: Although the close relationship between the upper and lower airways has been highlighted previously, little is known about the association between lung function and the recurrence of chronic rhinosinusitis with nasal polyps (CRSwNP). This study aimed to evaluate the factors associated with pulmonary function that affect CRSwNP recurrence after surgery.

Methods: We performed a series of routine pulmonary function tests for general anesthesia prior to CRSwNP surgery. The values for each parameter were compared in the presence or absence of recurrence.

Results: Sixty-nine patients with CRSwNP were included. The percent predicted forced expiratory volume in one second (%FEV₁) in the recurrent group was significantly lower than that in the non-recurrent group (P = .005). A multivariable logistic regression model revealed that %FEV₁ was a positive predictor of recurrence (odds ratio: 0.96, 95% CI: 0.92-0.99, P = .023). There were no significant differences in the other pulmonary functions between the two groups.

Conclusions: We found that %FEV₁ may be a predictor of CRSwNP recurrence after surgery. As %FEV₁ is a pulmonary function test that is routinely performed before surgery, this parameter is readily applicable. Moreover, as %FEV₁ appears to have the potential to reveal concealed asthma, %FEV₁ might be a particularly useful tool for the prediction of CRSwNP recurrence after surgery.

Introduction: Interleukin-1 (IL-1) antagonists have been successful in the management of monogenic auto-inflammatory diseases, notably classic hereditary fever syndromes, such as Familial Mediterranean Fever (FMF). Anakinra (Kineret®), a human recombinant IL-1 receptor antagonist (IL-1Ra), has been clinically effective in the management of persistent auto-inflammation, such as FMF. Few studies report anaphylaxis in response to anakinra, which were resolved with an anakinra desensitization or the anti-IL-1β monoclonal antibody canakinumab (ILARIS®). We describe the first reported desensitization protocol to canakinumab.

Case report: A 51-year-old man with a prior history of FMF presented with history of failed colchicine, nonsteroidal anti-inflammatory drug, and anakinra trials. Anakinra desensitization and canakinumab intradermal testing (IDT) resulted in anaphylactic and allergic symptoms, respectively. Expedited desensitization to canakinumab was successfully performed with 15-minute intervals between 13 doses of incremental increase to 150 mg.

Discussion: Biological agents are immune modulators that may evoke unanticipated hypersensitivity reactions, including anaphylaxis. These anaphylactic reactions to biologics have been infrequently reported, but the expanding market may increase the risk of IgE-mediated hypersensitivities and subsequent need for desensitization protocols. The current, expedited desensitization evaluated several published protocols involving anakinra desensitization to determine appropriate dosing for canakinumab.

Conclusion: We report the gastrointestinal intolerance and continued FMF flares on colchicine, followed by anaphylactic responses to anakinra and allergic reaction to IDT of canakinumab, in the present case of FMF. Our novel, expedited canakinumab desensitization protocol serves as an effective and alternative therapy in cases when other appropriate biologic agents are not tolerated.

Background: Chronic rhinosinusitis (CRS) is a common chronic disease. It has 2 main clinical subtypes: CRS with nasal polyposis (CRSwNP) and without nasal polyps (CRSsNP). The sphenoid sinus appears to be less frequently involved in CRSsNP cases. Thus, we aimed to compare the incidence of sphenoid sinus involvement between CRSsNP and CRSwNP cases.

Methods: A retrospective chart review of CRS cases was performed. The clinical and imaging findings, including age, sex, adenoid, and inferior turbinate hypertrophy (ITH), deviation of the nasal septum (DNS), presence of polyps, Lund-McKay scores, and the final diagnosis, were assessed. The incidence of sphenoid sinus involvement in each CRS subtype and its correlation with the aforementioned variables were studied.

Results: Of the 289 cases, 151 met the inclusion criteria including 82 CRSwNP and 69 CRSsNP cases. The mean patient age was 35.48 ± 11.88 years. The incidence of men and women were 66.9% and 33.1%, respectively. The sphenoid sinus involvement was 89% and 65.2% in the CRSwNP and CRSsNP cases (P = .0001), respectively. The involvement of other paranasal sinuses showed no statistically significant differences between the 2 phenotypes. No other evaluated variables, including age, gender, DNS, ITH, or adenoid hypertrophy, significantly correlated with the incidence of sphenoid sinus involvement.

Conclusions: This is the first study to demonstrate that the sphenoid sinus is less frequently involved in CRSsNP cases. Further studies should investigate the underlying factors causing the lower incidence of sphenoid sinus involvement in CRSsNP.

Familial cold inflammatory syndrome (FCAS) is a rare, inherited inflammatory disease characterized by episodes of fever, rash, and arthralgias after exposure to cold stimuli. Previous literature has established FCAS linked to autosomal dominant mutations in the NLRP3 (CIAS1) and NLRP12 genes. Moreover, there has been recent evidence of NLRC4-inflammasomopathies. Although there have been cases of FCAS secondary to missense mutations in NLRC4, we report the first symptomatic case associated with a 93-base-pair in-frame deletion within Exon 5 of the leucine rich repeat domain.

Specific antibody immunodeficiency (SAD) is a primary immunodeficiency disorder characterized by normal levels of serum immunoglobulins (IgG, IgA, and IgM) associated with a dysfunctional immune response. SAD is associated with recurrent infections in the setting of an insufficient response to polysaccharide vaccinations. Streptococcus pneumoniae is a well-established cause of respiratory infections in SAD. However, there has been a paucity of evidence of pneumococcal peritonitis in SAD patients, being reported as spontaneous in acquired immunodeficiency such as AIDS. We report the first case of S. pneumoniae-induced peritonitis as the presenting sign for SAD.

Introduction: Atopic dermatitis (AD, eczema) is familial chronic inflammatory skin disease of complex etiology and increasing prevalence. Dupilumab is an IL-4 receptor subunit alpha (IL-4Rα) antagonist that is the first Food and Drug Administration-approved biological therapy for moderate-to-severe adult AD inadequately controlled with topical therapies. Adverse effects reported in the literature include injection site reactions, conjunctivitis, headache, and nasopharyngitis.

Objective: We report the first cases of hyperhidrosis and bromhidrosis as side effects from dupilumab (Dupixent®) for the treatment of AD.

Case reports: Case 1 is a 20-year-old woman with controlled allergic rhinitis and severe AD reported axillary hyperhidrosis with bromhidrosis, comparable to sweat from high-intensity exercise, with no relief from several different over-the-counter antiperspirants. Case 2 is a 61-year-old woman with history of chronic asthma, allergic contact dermatitis, allergic rhinitis, and AD noticed markedly increased sweating with bromhidrosis that was reminiscent of her menopausal symptomology, about 3 months after initiating dupilimab.

Discussion: Traditional immunosuppressive agents and corticosteroids have limited efficacy, numerous side effects, and increased risk of infection. The safety profile and efficacy of the newly approved IL-4Rα antagonist dupilumab may be favorable to oral immunosuppressants, but its use remains limited to severe recalcitrant cases, due to financial implications and lack of long-term safety data and comparative head-to-head trials.

Conclusion: We report improved outcomes with dupilumab, in addition to unpublished cases of bromhidrosis and hyperhidrosis in 2 patients with AD. This report of additional complications may inspire further clinical research and assist clinicians in considering the option of dupilumab for uncontrolled AD, despite aggressive traditional treatment.