Department of Otolaryngology- Head & Neck Surgery Faculty Papers

Permanent URI for this collectionhttps://digital.lib.washington.edu/handle/1773/15669

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    Descriptive Summary and Experience Introducing the Orthodontic Airway Plate to Treat Upper Airway Obstruction for Infants with Robin Sequence: Lessons Learned
    (2025-04-08) Case, Hannah F.; Layuno Matos, Marielena; Boven, Lindsay; Brodie, Kara; Barbour, Michael; Benedict, Lindsey; Billimoria, Zeenia C.; Bly, Randall; Brundage, Tracy; Chen, Maida; Dahl, Jake; Friedman, Seth D.; Moreman, Lucy; Sheller, Barbara; Evans, Kelly; Tang, Kayen
    Objectives  Share our experience, summarize our protocol and describe initial outcomes implementing an orthodontic airway plate (OAP) care pathway for infants with Robin sequence (RS) and upper airway obstruction (UAO).      Design   Narrative experience of a care pathway for neonates with RS and UAO with retrospective review of initial series of patients with RS treated with this pathway.     Setting  The neonatal intensive care and acute care units of a tertiary Children's Hospital, with transition to outpatient multidisciplinary craniofacial clinic care.     Patients and Participants   Neonates with RS and severe base of tongue UAO not stabilized with positioning, and without mechanical ventilation, profound dysphagia, or microstomia.       Interventions  Creation of an orthodontic airway plate clinical pathway     Main Outcome Measure(s)  Lessons learned, clinical course, respiratory and sleep metrics      Results   In August 2023 our institution launched an OAP treatment pathway. Critical lessons learned highlight the importance of multidisciplinary communication, patient selection, and patience. Nine of eleven patients completed OAP therapy. oAHI values before OAP were median 71 (range 16-189, StDev 55.1).  After OAP treatment, oAHI improved to median oAHI 7.9 (range 1.1-12.7, StDev 3.8). Pre-treatment, seven patients were on high-flow nasal cannula, one on low-flow nasal cannula, and one on CPAP. All OAP graduates advanced to room air.   Conclusions  The OAP can be a safe and effective nonsurgical intervention for UAO in RS. Interdisciplinary collaboration is key to OAP program success. Infants undergoing OAP treatment can safely transition from hospital to home and experience sustained respiratory improvements.  Keywords: Robin Sequence, Upper airway obstruction, Orthodontic airway plate
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    Automated atlas-based segmentation for skull base surgical planning
    (2020-09-09) Konuthula, Neeraja; Perez, Francisco A; Maga, A Murat; Abuzeid, Waleed M.; Moe, Kris; Hannaford, Blake; Bly, Randall A.
    Purpose: Computational surgical planning tools could help develop novel skull base surgical approaches that improve safety and patient outcomes. This defines a need for automated skull base segmentation to improve the usability of surgical planning software. The objective of this work was to design and validate an algorithm for atlas-based automated segmentation of skull base structures in individual scans in skull base surgical planning. Methods: Advanced Normalization Tools software was used to construct a synthetic CT template from 6 subject scans, and skull base structures were manually segmented to create a reference atlas. Structures were also segmented on scans from 30 additional subjects to serve as ground truth scans for accuracy testing. Landmark registration followed by Elastix deformable registration were applied to the template to register it to each of the 30 patient scans. Dice coefficient, average Hausdorff distance, and clinical usability scoring were used to compare the atlas segmentations to those of the ground truth scans. Results: The upper limit of the 95% confidence intervals for the average Hausdorff distance for all structures was less than 2 mm. For structures greater than 2.5mL in volume, the average Dice coefficient was 0.73 (range 0.59-0.82), and for structures less than 2.5 mL in volume the Dice coefficient was less than 0.7. The usability scoring survey was completed by three experts, and all structures met the criteria for acceptable effort except for the foramen spinosum, rotundum, and carotid artery, which required more than minor corrections. Conclusion: Currently available open-source algorithms, such as the Elastix deformable algorithm, can be used for automated atlas-based segmentation of skull base structures with acceptable clinical accuracy and minimal corrections with the use of the proposed atlas. The first publicly available CT template and anterior skull base segmentation atlas being released with this paper will allow for general use of automated atlas-based segmentation of the skull base. Screen reader support enabled.
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    A novel WFS1 mutation in a family with dominant low frequency sensorineural hearing loss with normal VEMP and EcochG findings
    (2008) Bramhall, Naomi F.; Kallman, Jeremy C.; Verrall, Aimee M.; Street, Valerie A.
    Background: Low frequency sensorineural hearing loss (LFSNHL) is an uncommon clinical finding. Mutations within three different identified genes (DIAPH1, MYO7A, and WFS1) are known to cause LFSNHL. The majority of hereditary LFSNHL is associated with heterozygous mutations in the WFS1 gene (wolframin protein). The goal of this study was to use genetic analysis to determine if a small American family's hereditary LFSNHL is linked to a mutation in the WFS1 gene and to use VEMP and EcochG testing to further characterize the family's audiovestibular phenotype. Methods: The clinical phenotype of the American family was characterized by audiologic testing, vestibular evoked myogenic potentials (VEMP), and electrocochleography (EcochG) evaluation. Genetic characterization was performed by microsatellite analysis and direct sequencing of WFS1 for mutation detection. Results: Sequence analysis of the WFS1 gene revealed a novel heterozygous mutation at c.2054G>C predicting a p.R685P amino acid substitution in wolframin. The c.2054G>C mutation segregates faithfully with hearing loss in the family and is absent in 230 control chromosomes. The p.R685 residue is located within the hydrophilic C-terminus of wolframin and is conserved across species. The VEMP and EcochG findings were normal in individuals segregating the WFS1 c.2054G>C mutation. Conclusion: We discovered a novel heterozygous missense mutation in exon 8 of WFS1 predicting a p.R685P amino acid substitution that is likely to underlie the LFSNHL phenotype in the American family. For the first time, we describe VEMP and EcochG findings for individuals segregating a heterozygous WFS1 mutation.
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    A prospective study of hearing changes after beginning zidovudine or didanosine in HIV-1 treatment-naive people
    (2006) Schouten, Jeffrey T.; Lockhart, David W.; Rees, Thomas S.; Collier, Ann C.; Marra, Christina M.
    Background: While hearing loss in HIV-infected people after beginning nucleoside reverse transcriptase inhibitors (NRTIs) has been reported, there have been no prospective studies that measured hearing changes longitudinally in treatment-naive HIV-infected subjects following initiation of regimens containing NRTIs. The goal of this study was to conduct a prospective assessment of the contribution of zidovudine (ZDV) and didanosine (ddI) to hearing loss Methods/design: A prospective observational pilot study to determine whether ZDV or ddI, alone or in combination, are associated with sensorineural hearing loss in HIV-infected persons. Changes in hearing levels at all frequencies and in low and high frequency pure tone averages were measured at baseline, 16, and 32 weeks after initiating antiretroviral therapy. Discussion: Treatment with ZDV and ddI did not result in loss of hearing, even after taking into account noise exposure, immune status and age. The results of this prospective pilot study do not support the notion that treatment with nucleoside antiretrovirals damages hearing.