Optimizing Inspire Hypoglossal Nerve Stimulator Settings in Pediatric Obstructive Sleep Apnea.

INTRODUCTION: Hypoglossal nerve stimulation (HGNS) is an effective therapy for carefully selected pediatric patients with Down syndrome (DS) and obstructive sleep apnea (OSA), but optimization of device settings has been focused on stimulation settings. The potential benefits of sensing lead setting optimization have not been investigated. We describe a series of three cases of HGNS in children with DS where optimization of both the stimulation lead and sensing lead settings, as well as utilization of drug-induced sleep endoscopy (DISE), when indicated, was more effective than stimulation lead optimization alone. METHODS: Using our novel titration algorithm, among the three identified pediatric patients with DS in whom HGNS initially failed to reduce the apnea-hypopnea index (AHI), two patients in whom sleep studies were characterized as hypopnea-dominant were assigned to advanced titration (optimization of stimulation settings and sensing lead settings). The third patient, with an obstructive apnea-dominant sleep study, was referred for interrogated DISE. Advanced titration included adjustment of both stimulation settings and sensing lead settings. RESULTS: Application of the advanced titration algorithm, tailored to obstructive apnea- versus hypopnea-dominant sleep patterns, resulted in approximately 50% or greater reduction in AHI in all three patients. "Normal to mild OSA" AHI (AHI <5) was achieved in two patients. CONCLUSIONS: A novel diagnostic and therapeutic titration algorithm for the Inspire HGNS system significantly reduced AHI in three pediatric patients with DS and OSA who had failed to respond to the standard device titration that focused solely on stimulation settings. LEVEL OF EVIDENCE: 4 Laryngoscope, 2024.

Epstein-Barr virus infection status among first year undergraduate university students.

OBJECTIVE: Epstein-Barr virus (EBV) is the cause of infectious mononucleosis, which disproportionately affects university students. This population has the potential to benefit from a prophylactic EBV vaccine trial. Our objectives were to determine EBV infection status and associated demographic/lifestyle factors among first year undergraduate university students at the beginning and end of first year. METHODS: EBV infection status was assessed by testing for circulating IgG class antibodies against EBV viral capsid antigen. RESULTS: Of 198 starting students; 56.1% were positive for EBV antibodies with a higher rate in women (64.8%) than male (41.1%); p = 0.002. A history of deep kissing was associated with a higher rate of EBV antibody positivity. On follow-up 8 months later at the end of freshman year, 22.4% had acquired EBV antibodies for a primary infection incidence of 33.6/100 person years. CONCLUSION: These findings indicate that our first year undergraduate population contains sufficient EBV-naïve subjects for a prophylactic vaccine trial.

Can Red Blood Cell Distribution Width Predict Laryngectomy Complications or Survival Outcomes?

OBJECTIVE: Red blood cell distribution width (RDW), a reported biomarker for morbidity and mortality in chronic disease and following certain surgeries, has not been well-studied in head and neck cancer patients. The aim of the study was to examine the association of RDW with postoperative complications and survival among patients who underwent primary or salvage laryngectomy. METHODS: We analyzed a retrospective case series study of patients diagnosed with squamous cell carcinoma of the larynx treated with total laryngectomy. Survival outcomes were examined using Kaplan-Meier analysis. RESULTS: One hundred seventy-seven patients were included in the final analysis. The most common tumor subsite was the supraglottis (60%). On bivariate analysis, patients with RDW ≥14.5 had higher prevalence of non-surgical, systemic complications, including deep venous thrombosis, pneumonia, cardiovascular events, and difficulty weaning from mechanical ventilation. However, there was no significant difference in laryngectomy-specific post-operative complications, including pharyngocutaneous fistula, wound infection, stoma complications, and chyle leak. RDW was not found to be associated with survival outcomes following laryngectomy. CONCLUSIONS: Among laryngectomy patients, RDW ≥14.5 is associated with higher prevalence of systemic morbidity, but not with specific local surgical complications or decreased survival.

Pembrolizumab Induced Acute Persistent Airway Disease in a Patient with Recurrent Respiratory Papillomatosis (RRP).

OBJECTIVE: To present an uncommon but serious, recently identified complication of checkpoint inhibitor therapy in a patient treated with pembrolizumab infusion for disseminated recurrent respiratory papillomatosis (RRP). METHODS: Case report. RESULTS: A 43-year-old woman with underlying asthma developed acute hypoxic respiratory failure within 24 hours of her third infusion of pembrolizumab for treatment of intractable, disseminated recurrent respiratory papillomatosis. Pulmonary function testing revealed a severe intra-thoracic obstructive ventilatory defect. Discontinuation of pembrolizumab, ventilatory support, and treatment with systemic and inhaled corticosteroids resulted in resolution of respiratory failure; however, her underlying asthma remains poorly controlled. CONCLUSION: To our knowledge, this case is the first report of pembrolizumab-induced obstructive respiratory failure in a patient being treated for RRP.

Ultrasound and Sialogram Correlates to Parotid Immunoglobulin G4-Related Disease.

Immunoglobulin G4 (IgG4)-related disease is an immune-mediated disorder that commonly manifests in the salivary glands. As a recently described disorder, the description and classification of IgG4-related disease is an ongoing process. Diagnosis of IgG4-related disease requires integration of clinical history, histopathology, and radiographic findings, including ultrasonography and sialography. In this case report, we correlate parotid ultrasonographic and sialographic findings in a patient with proven IgG4-related disorder confirmed from analysis of previous submandibular gland resections. We aim to highlight the utility of multimodality imaging in the diagnosis of IgG4-related disease.

Not All Küttner Tumors Are IgG4-Related Disease (IgG4-RD).

Küttner tumor is an uncommon cause of salivary gland enlargement that most frequently affects the submandibular gland. More recently it has been considered a manifestation of IgG4-related disease (IgG4-RD) and synonymous with chronic sclerosing sialadenitis (CSS). We present a series of cases to emphasize this clinical entity as a disease pattern and presentation that is separate from IgG4-RD. Retrospective case series of 3 patients with unilateral salivary gland enlargement, or "Küttner tumor," histologically classified as "chronic sclerosing sialadenitis," The clinical history, radiology reports, laboratory studies, and pathology slides were reviewed for each case. Radiology showed discrete unilateral mass-like lesions in all three cases. Immunohistochemistry showed reduced tissue IgG4-positive plasma cells in two cases and increased numbers in one case, but insufficient to diagnose IgG4-RD. Storiform fibrosis was not seen in all cases and did not coincide with increased IgG4-positive plasma cells. A systemic workup, including serum IgG4 levels in two cases, was normal. A brief review of the literature on the spectrum of salivary gland involvement by IgG4-RD is presented. Küttner tumor is not necessarily the same as chronic sclerosing sialadenitis and is not always associated with IgG4-related disease. This report includes the second documented case of Küttner tumor of the sublingual gland.

Sialographic Analysis of Radioiodine-Associated Chronic Sialadenitis.

OBJECTIVES/HYPOTHESIS: To apply a novel sialography classification system to identify parotid and submandibular ductal findings following I-131 therapy and to assess correlates to dose and duration of symptoms. STUDY DESIGN: Retrospective single-center case series. METHODS: Patients who underwent sialography between February 2008 and February 2019 after previously receiving I-131 treatment were identified via a retrospective chart review. Their sialograms were systematically evaluated and scored by applying the Iowa parotid sialogram scale to also include submandibular gland analysis. RESULTS: From 337 sialograms, 30 (five submandibular, 25 parotid) underwent analysis. Ductal stenosis was identified in all sialograms and was graded as moderate (>50%-75%) in 7/30 cases and severe (>75%) in 15/30 cases. The distal (main) duct was narrowed in 23/30 cases. No association was identified between degree of ductal stenosis and I-131 dose (P = .39), age (P = .81), or time from I-131 therapy to sialogram (P = .97). CONCLUSIONS: The Iowa parotid sialogram scale was successfully applied to report abnormalities of the parotid and submandibular ductal system. The most common manifestation of I-131-associated sialadenitis was a severe stenosis within the distal salivary duct. No statistically significant association was found between degree of ductal stenosis and dose of I-131, age, or duration of symptoms. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:E1450-E1456, 2021.