Trends in Natural Decannulation in Patients with Robin Sequence: A Twenty-five Year Retrospective Review.

OBJECTIVE: Robin sequence (RS) consists of micrognathia and glossoptosis that result in upper airway obstruction (UAO). In RS patients who undergo tracheostomy, long-term goals include natural decannulation (ND) without further surgical airway intervention. The objective of this study was to identify long-term trends in the rate and length of time to ND. METHODS: Retrospective chart review on 144 patients with RS treated from 1995 to 2020 at a pediatric tertiary care center. Patients were grouped by year of tracheostomy. Demographic data, UAO management, postoperative care, complications, and time to decannulation were recorded. RESULTS: Thirty-six patients met the inclusion criteria. Tracheostomy was performed at a median age of 45.5 days. 19 (53%) patients experienced ND at a median time of 66.1 months. ND rate was higher in non-syndromic patients (93% non-syndromic vs 27% syndromic; P < .0001) and during the first study period (1995-2006: 78%, 2007-2020: 28%; P = .003). Cox proportional-hazard regression demonstrated that white race [aHR 0.15 (0.03-0.8); P = .023] and higher birthweight [aHR 0.9 (0.8-0.98); P = .018] were associated with a higher likelihood of ND while a syndromic diagnosis had a negative association with ND [aHR 12.5 (3.3-50.0); P < .001]. CONCLUSIONS: Our study documented that ND in patients with RS who underwent tracheostomy was significantly associated with ethnicity, birthweight, and syndromic status. The negative impact on successful ND was most observed in patients with syndromic associations. Additionally, ND rates are lower in the 2007 to 2020 subgroup. We suspect this is because alternative management techniques such as tongue lip adhesion and mandibular distraction osteogenesis became primary surgical treatment in severe RS upper airway obstruction at our institution starting in 2007.

Early Health Economic Modeling of Novel Therapeutics in Age-Related Hearing Loss.

Background: Health systems face challenges to accelerate access to innovations that add value and avoid those unlikely to do so. This is very timely to the field of age-related sensorineural hearing loss (ARHL), where a significant unmet market need has been identified and sizeable investments made to promote the development of novel hearing therapeutics (NT). This study aims to apply health economic modeling to inform the development of cost-effective NT. Methods: We developed a decision-analytic model to assess the potential costs and effects of using regenerative NT in patients ≥50 with ARHL. This was compared to the current standard of care including hearing aids and cochlear implants. Input data was collected from systematic literature searches and expert opinion. A UK NHS healthcare perspective was adopted. Three different but related analyses were performed using probabilistic modeling: (1) headroom analysis, (2) scenario analyses, and (3) threshold analyses. Results: The headroom analysis shows an incremental net monetary benefit (iNMB) of £20,017[£11,299-£28,737] compared to the standard of care due to quality-adjusted life-years (QALY) gains and cost savings. Higher therapeutic efficacy and access for patients with all degrees of hearing loss yields higher iNMBs. Threshold analyses shows that the ceiling price of the therapeutic increases with more severe degrees of hearing loss. Conclusion: NT for ARHL are potentially cost-effective under current willingness-to-pay (WTP) thresholds with considerable room for improvement in the current standard of care pathway. Our model can be used to help decision makers decide which therapeutics represent value for money and are worth commissioning, thereby paving the way for urgently needed NT.

The Potential Added Value of Novel Hearing Therapeutics: An Early Health Economic Model for Hearing Loss.

OBJECTIVE: To construct an early health economic model to assess the potential added value of novel hearing therapeutics, compared with the current standard of care. We use idiopathic sudden sensorineural hearing loss (ISSNHL) as a case example, because it is a lead indication for several emerging hearing therapeutics. METHODS: A decision analytic model was developed to assess the costs and effects of using novel hearing therapeutics for patients with ISSNHL. This was compared to the current standard of care. Input data were derived from literature searches and expert opinion. The study adopted a healthcare perspective of the UK National Health Service. Four analyses were conducted: 1) headroom, 2) scenario, 3) threshold, 4) sensitivity. RESULTS: The decision analytic model showed that novel therapeutics for ISSNHL have potential value both in terms of improved patient outcomes, as well as incremental net monetary benefit (iNMB). The base case analysis revealed an iNMB of £39,032 for novel therapeutics compared with the current standard of care. Results of the threshold and scenario analysis revealed that age of treatment and severity of ISSNHL are major determinants of iNMB for novel therapeutics. CONCLUSION: This article describes the first health economic model for novel therapeutics for hearing loss; and shows that novel hearing therapeutics can be cost-effective under NICE's cost-effectiveness threshold, with considerable room for improvement in the current standard of care. Our model can be used to inform the development of cost-effective hearing therapeutics; and help decision makers decide which therapeutics represent value for money.

Systematic Review and Network Meta-analysis of Cognitive and/or Behavioral Therapies (CBT) for Tinnitus.

OBJECTIVE: To evaluate the efficacy of cognitive and/or behavioral therapies in improving health-related quality of life (HRQOL), depression, and anxiety associated with tinnitus. DATA SOURCES: EMBASE, MEDLINE, PubMed, PsycINFO, and the Cochrane Registry were used to identify English studies from database inception until February 2018. STUDY SELECTION: Randomized controlled trials (RCTs) comparing cognitive and/or behavioral therapies to one another or to waitlist controls for the treatment of tinnitus were included. DATA EXTRACTION: Quality and risk were assessed using GRADE and Cochrane's Risk of Bias tool respectively. DATA SYNTHESIS: Pairwise meta-analysis (12 RCTs: 1,144 patients) compared psychological interventions to waitlist controls. Outcomes were measured using standardized mean differences (SMDs) and 95% confidence intervals (CI). I and subgroup analyses were used to assess heterogeneity. Network meta-analysis (NMA) (19 RCTS: 1,543 patients) compared psychological therapies head-to-head. Treatment effects were presented by network diagrams, interval plots, and ranking diagrams indicating SMDs with 95% CI. Direct and indirect results were further assessed by inconsistency plots. CONCLUSIONS: Results are consistent with previously published guidelines indicating that CBT is an effective therapy for tinnitus. While guided self-administered forms of CBT had larger effect sizes (SMD: 3.44; 95% CI: -0.022, 7.09; I: 99%) on tinnitus HRQOL, only face-to-face CBT was shown to make statistically significant improvements (SMD: 0.75; 95% CI: 0.53, 0.97; I: 0%). Guided self-administered CBT had the highest likelihood of being ranked first in improving tinnitus HRQOL (75%), depression (83%), and anxiety (87%), though statistically insignificant. This NMA is the first of its kind in this therapeutic area and provides new insights on the effects of different forms of cognitive and/or behavioral therapies for tinnitus.

Early phase trials of novel hearing therapeutics: Avenues and opportunities.

Novel hearing therapeutics are rapidly progressing along the innovation pathway and into the clinical trial domain. Because these trials are new to the hearing community, they come with challenges in terms of trial design, regulation and delivery. In this paper, we address the key scientific and operational issues and outline the opportunities for interdisciplinary and international collaboration these trials offer. Vital to the future successful implementation of these therapeutics is to evaluate their potential for adoption into healthcare systems, including consideration of their health economic value. This requires early engagement with all stakeholder groups along the hearing innovation pathway.

The use of video glasses improved learning of tonsillectomy and adenoidectomy surgery: A randomized controlled trial.

OBJECTIVE: One of the most common challenges in surgical education for trainees is gaining practical experience through observing procedures in the operating room. Due to the nature of some procedures, a narrow surgical view severely limits the learning experience. Video glasses are new devices that offer the potential to project the primary surgeon's exact view to learners in real-time, allowing for an enhanced operative learning experience. STUDY DESIGN: Single center randomized prospective trial. SETTING: Tertiary care pediatric hospital. PARTICIPANTS: Using block randomization, medical students and surgical residents observed either a tonsillectomy or adenoidectomy, either directly at table-side or by real-time video feed from the surgeon's video glasses projected to a screen in the operating room, in random order. Participants then completed a survey comparing aspects of their learning experience viewing the procedure through the video feed in comparison to direct observation. MAIN OUTCOME MEASURES: Evaluating the hypothesis that video glasses provided an improved overall learning experience and a realistic simulation of the open surgical procedures tested. RESULTS: 23 trainees participated in the study. Survey results demonstrated that the overall learning experience with the use of video glasses was significantly improved when compared to direct visualization (average Visual Analog Scale (VAS) score 82/100 vs. 64/100, p = 0.021). Video glasses were shown to be superior when comparing the view of the surgical field (83/100 vs. 54/100 on VAS, p < 0.001) and the ability to identify anatomical structures (79/100 vs. 56/100 on VAS, p = 0.001). The ease of following surgical steps with video glasses was also shown to be better than by direct visualization (81/100 vs. 69/100 on VAS, p = 0.039). All participants stated that video glasses closely simulated the learning environment of the real-life open procedure. CONCLUSION: This study showed that the use of video glasses was beneficial for surgical education and a realistic tool for learners at varying levels of training. Video glasses may significantly improve the learning experience for procedures with a narrow field of view.

An Uncommon Osseous Frontal Sinus Tumor: Monostotic Paget's Disease.

Paget's disease of the bone is a disorder characterized by abnormal breakdown and formation of bone tissue. The cause is believed to be either viral or genetic in origin. Most of the time, patients are asymptomatic. Diagnosis is supported by findings from multiple modalities including serum markers, imaging, bone scans, and histology. We present a rare case of Paget's disease of the bone involving the frontal sinus. We review the relevant clinical, diagnostic, and histological findings. We also suggest indications for the management of monostotic Paget's disease of the frontal sinus.