Allergy & RhinologyPub Date : 2020-06-22eCollection Date: 2020-01-01DOI: 10.1177/2152656720937694
Neha Sanan, Jason Schend, Marija Rowane, Robert Hostoffer
{"title":"Expedited Desensitization to Canakinumab.","authors":"Neha Sanan, Jason Schend, Marija Rowane, Robert Hostoffer","doi":"10.1177/2152656720937694","DOIUrl":"https://doi.org/10.1177/2152656720937694","url":null,"abstract":"<p><strong>Introduction: </strong>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.</p><p><strong>Case report: </strong>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.</p><p><strong>Discussion: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":" ","pages":"2152656720937694"},"PeriodicalIF":2.2,"publicationDate":"2020-06-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1177/2152656720937694","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"38108620","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Allergy & RhinologyPub Date : 2020-06-16eCollection Date: 2020-01-01DOI: 10.1177/2152656720934472
Ibrahim Sumaily, Ibrahim Alarifi, Abdussalam Alahmari, Mohammad Aloulah, Saad Alsaleh
{"title":"Sphenoid Sinus Involvement in Chronic Rhinosinusitis Without Polyps.","authors":"Ibrahim Sumaily, Ibrahim Alarifi, Abdussalam Alahmari, Mohammad Aloulah, Saad Alsaleh","doi":"10.1177/2152656720934472","DOIUrl":"https://doi.org/10.1177/2152656720934472","url":null,"abstract":"<p><strong>Background: </strong>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.</p><p><strong>Methods: </strong>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.</p><p><strong>Results: </strong>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 (<i>P</i> = .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.</p><p><strong>Conclusions: </strong>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.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":" ","pages":"2152656720934472"},"PeriodicalIF":2.2,"publicationDate":"2020-06-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1177/2152656720934472","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"38099596","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Allergy & RhinologyPub Date : 2020-06-05eCollection Date: 2020-01-01DOI: 10.1177/2152656720928245
Rosario B Jaime-Lara, Valentina Parma, Carol H Yan, Farhoud Faraji, Paule V Joseph
{"title":"Role of Olfaction in Human Health: A Focus on Coronaviruses.","authors":"Rosario B Jaime-Lara, Valentina Parma, Carol H Yan, Farhoud Faraji, Paule V Joseph","doi":"10.1177/2152656720928245","DOIUrl":"10.1177/2152656720928245","url":null,"abstract":"","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":"11 ","pages":"2152656720928245"},"PeriodicalIF":2.3,"publicationDate":"2020-06-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/01/90/10.1177_2152656720928245.PMC7278301.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10487380","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Allergy & RhinologyPub Date : 2020-06-02eCollection Date: 2020-01-01DOI: 10.1177/2152656720928062
Jack Jeskey, Akash Parida, Kelsey Graven, Robert Hostoffer
{"title":"Novel Gene Deletion in NLRC4 Expanding the Familial Cold Inflammatory Syndrome Phenotype.","authors":"Jack Jeskey, Akash Parida, Kelsey Graven, Robert Hostoffer","doi":"10.1177/2152656720928062","DOIUrl":"https://doi.org/10.1177/2152656720928062","url":null,"abstract":"<p><p>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.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":" ","pages":"2152656720928062"},"PeriodicalIF":2.2,"publicationDate":"2020-06-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1177/2152656720928062","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"38048255","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Allergy & RhinologyPub Date : 2020-06-02eCollection Date: 2020-01-01DOI: 10.1177/2152656720928065
Jack Jeskey, Akash Parida, Kelsey Graven, Robert Hostoffer
{"title":"Specific Antibody Immunodeficiency Presenting With <i>Streptococcal pneumonia</i>-Induced Spontaneous Bacterial Peritonitis.","authors":"Jack Jeskey, Akash Parida, Kelsey Graven, Robert Hostoffer","doi":"10.1177/2152656720928065","DOIUrl":"https://doi.org/10.1177/2152656720928065","url":null,"abstract":"<p><p>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. <i>Streptococcus pneumoniae</i> 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 <i>S. pneumoniae</i>-induced peritonitis as the presenting sign for SAD.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":" ","pages":"2152656720928065"},"PeriodicalIF":2.2,"publicationDate":"2020-06-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1177/2152656720928065","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"38048256","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Allergy & RhinologyPub Date : 2020-05-20eCollection Date: 2020-01-01DOI: 10.1177/2152656720927703
Marija Rowane, Reimus Valencia, Jason Schend, Devi Jhaveri, Robert Hostoffer
{"title":"Something to Sweat About: Two Cases of Dupilumab-Induced Hyperhidrosis and Bromhidrosis.","authors":"Marija Rowane, Reimus Valencia, Jason Schend, Devi Jhaveri, Robert Hostoffer","doi":"10.1177/2152656720927703","DOIUrl":"https://doi.org/10.1177/2152656720927703","url":null,"abstract":"<p><strong>Introduction: </strong>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.</p><p><strong>Objective: </strong>We report the first cases of hyperhidrosis and bromhidrosis as side effects from dupilumab (Dupixent®) for the treatment of AD.</p><p><strong>Case reports: </strong>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.</p><p><strong>Discussion: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":" ","pages":"2152656720927703"},"PeriodicalIF":2.2,"publicationDate":"2020-05-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1177/2152656720927703","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"38003264","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Allergy & RhinologyPub Date : 2020-04-21eCollection Date: 2020-01-01DOI: 10.1177/2152656720918874
Carly A Clark, Cameron P Worden, Brian D Thorp, Charles S Ebert, Adam M Zanation, Brent A Senior, Steven M Johnson, Wade G McClain, Adam J Kimple
{"title":"Extramedullary Hematopoiesis in the Sinonasal Cavity: A Case Report and Review of the Literature.","authors":"Carly A Clark, Cameron P Worden, Brian D Thorp, Charles S Ebert, Adam M Zanation, Brent A Senior, Steven M Johnson, Wade G McClain, Adam J Kimple","doi":"10.1177/2152656720918874","DOIUrl":"https://doi.org/10.1177/2152656720918874","url":null,"abstract":"<p><strong>Background: </strong>Extramedullary hematopoiesis (EMH) occurs in patients with hematologic disorders, but rarely within the paranasal sinuses. We report a case of EMH in a 17-year-old male with sickle cell disease (SCD) who presented with occipital pain and sinusitis. A computed tomography (CT) scan demonstrated heterogeneous opacification of the right maxillary sinus concerning for allergic fungal sinusitis or a fungal ball with bony erosion. He was taken to the operating room for endoscopic biopsy and a limited endoscopic sinus surgery. Grossly, his maxillary sinus was filled with spiculated osseous tissue. Final pathology demonstrated active hematopoietic bone marrow filling the sinus.</p><p><strong>Methods: </strong>We present a case report and literature review of sinonasal EMH.</p><p><strong>Results: </strong>We identified 14 articles with 15 patients. EMH was typically associated with SCD or beta thalassemia. The average age of presentation was 30. There was a male sex predilection with a ratio of 11:15. The most common presenting symptom was a headache and nasal obstruction (33% for both). The most common finding on CT was a soft tissue expansile mass (73%). The most commonly affected location was the maxillary sinus (60%).</p><p><strong>Conclusions: </strong>This case report serves as a reminder to consider EMH as an uncommon cause of sinus opacification, particularly in patients with SCD or beta thalassemia. The expansion of hematopoietic tissue may be identified as a sinus mass on CT. By recognizing the potential manifestations of chronic anemia, an accurate and timely diagnosis can be made.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":" ","pages":"2152656720918874"},"PeriodicalIF":2.2,"publicationDate":"2020-04-21","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1177/2152656720918874","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"37897163","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Epithelial-Myoepithelial Carcinoma of the Lacrimal Sac and Literature Review of the Lacrimal System.","authors":"Dhruv Sharma, Ashley Neiweem, Kyle Davis, Mark Prendes, Rao Chundury, Elisa Illing","doi":"10.1177/2152656720920600","DOIUrl":"https://doi.org/10.1177/2152656720920600","url":null,"abstract":"<p><strong>Background: </strong>Epithelial-myoepithelial carcinomas make up less than 0.1% of head and neck malignancies and are regarded as rare, low-grade malignant neoplasms of the salivary gland. They are thought to arise from intercalated ducts with histopathology showing a classic biphasic morphology of an outer layer of myoepithelial cells and inner layer of epithelial cells. These tumors most commonly occur in the parotid gland; however, rare cases have also been described in the nasal cavity, nasopharynx, subglottis, base of tongue, and the lacrimal gland.</p><p><strong>Objective: </strong>To describe the clinical presentation, surgical management, and histopathology of the first reported case of lacrimal sac epithelial-myoepithelial carcinoma. To conduct a literature review of this malignancy, which is present in the lacrimal system.</p><p><strong>Methods: </strong>Case report (n = 1) and literature review.</p><p><strong>Results: </strong>We report a case of a 72-year-old man presenting with epiphora and a lacrimal sac mass with intranasal extension on imaging and nasal endoscopy. A combined endoscopic endonasal and open approach provided successful definitive treatment for final pathologic diagnosis of epithelial-myoepithelial carcinoma of the lacrimal sac, with orbital reconstruction and lacrimal stenting providing good cosmetic and functional results.</p><p><strong>Conclusions: </strong>After PubMed database search for any case series or reports of lacrimal system epithelial-myoepithelial carcinomas, we believe this is the first documented case originating from the lacrimal sac. Although the histopathology of this tumor is distinct, unusual location and clinical presentation may pose significant diagnostic difficulties.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":" ","pages":"2152656720920600"},"PeriodicalIF":2.2,"publicationDate":"2020-04-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1177/2152656720920600","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"37878480","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Allergy & RhinologyPub Date : 2020-03-31eCollection Date: 2020-01-01DOI: 10.1177/2152656720915701
Lauren Fill, Marija Rowane, Kelsey Graven, Neha Sanan, Shan Shan Wu, Robert W Hostoffer
{"title":"Post Autologous Bone Marrow Transplant Associated With a Resultant Mixed Polyclonal/Monoclonal Hyper-IgG3.","authors":"Lauren Fill, Marija Rowane, Kelsey Graven, Neha Sanan, Shan Shan Wu, Robert W Hostoffer","doi":"10.1177/2152656720915701","DOIUrl":"https://doi.org/10.1177/2152656720915701","url":null,"abstract":"<p><p>There have been few studies illustrating the post immunological phenotype of patients receiving autologous bone marrow transplant (ABMT) for the treatment of diffuse large B-cell lymphoma. High-dose chemotherapy and autologous bone marrow transplantation have been shown to be the only potential curative treatment modalities for B-cell lymphoma. Autologous bone marrow transplantation, although widely utilized in patients with non-Hodgkin lymphoma recurrence, does have an association with immunologic side effects, although serologic changes where rarely reported unless accompanied by recurrent infections. We report the first case of a 62-year-old female patient who experienced recurrent infections, namely, sinusitis and pneumonia, after receiving an ABMT with subsequent hyper-IgG3 phenotype.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":" ","pages":"2152656720915701"},"PeriodicalIF":2.2,"publicationDate":"2020-03-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1177/2152656720915701","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"37831090","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Allergy & RhinologyPub Date : 2020-03-13eCollection Date: 2020-01-01DOI: 10.1177/2152656720911605
Sofia Dimitri-Pinheiro, Raquel Soares, Pedro Barata
{"title":"The Microbiome of the Nose-Friend or Foe?","authors":"Sofia Dimitri-Pinheiro, Raquel Soares, Pedro Barata","doi":"10.1177/2152656720911605","DOIUrl":"https://doi.org/10.1177/2152656720911605","url":null,"abstract":"<p><p>Recently, multiple studies regarding the human microbiota and its role on the development of disease have emerged. Current research suggests that the nasal cavity is a major reservoir for opportunistic pathogens, which can then spread to other sections of the respiratory tract and be involved in the development of conditions such as allergic rhinitis, chronic rhinosinusitis, asthma, pneumonia, and otitis media. However, our knowledge of how nasal microbiota changes originate nasopharyngeal and respiratory conditions is still incipient. Herein, we describe how the nasal microbiome in healthy individuals varies with age and explore the effect of nasal microbiota changes in a range of infectious and immunological conditions. We also describe the potential health benefits of human microbiota modulation through probiotic use, both in disease prevention and as adjuvant therapy. Current research suggests that patients with different chronic rhinosinusitis phenotypes possess distinct nasal microbiota profiles, which influence immune response and may be used in the future as biomarkers of disease progression. Probiotic intervention may also have a promising role in the prevention and adjunctive treatment of acute respiratory tract infections and allergic rhinitis, respectively. However, further studies are needed to define the role of probiotics in the chronic rhinosinusitis.</p>","PeriodicalId":45192,"journal":{"name":"Allergy & Rhinology","volume":" ","pages":"2152656720911605"},"PeriodicalIF":2.2,"publicationDate":"2020-03-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1177/2152656720911605","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"37765973","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}