Objectives/Hypothesis
Hypoglossal nerve (HGN) stimulation is a novel therapy for obstructive sleep apnea (OSA) in adults. Its efficacy and safety in children with Down syndrome (DS) was previously reported in a preliminary case series of six adolescents.
Study Design
Case series.
Methods
Twenty nonobese children and adolescents (aged 10–21 years) with DS and severe OSA (apnea‐hypopnea index [AHI] >10 and <50 events/hr) despite prior adenotonsillectomy were enrolled. Participants had failed a trial of continuous positive airway pressure therapy and underwent sleep endoscopy confirming surgical candidacy. The primary outcome was to assess safety and monitor for adverse events. Secondary outcomes included efficacy in reducing AHI (% reduction in AHI), adherence to therapy, and change in a validated quality‐of‐life instrument, the OSA‐18 survey.
Results
All 20 children (median age = 16.0 years [interquartile range = 13–17 years], 13 male) were implanted with no long‐term complications. We report two interval adverse events, both of which were corrected with revision surgery. Twenty participants completed the 2‐month polysomnogram, with median percent reduction in titration AHI of 85% (interquartile range = 75%–92%). The median nightly usage for these children was 9.21 hours/night. There was a median change in the OSA‐18 score of 1.15, indicating a moderate, yet significant, clinical change.
Conclusions
HGN stimulation was safe and effective in the study population. Two minor surgical complications were corrected surgically. Overall, these data suggest that pediatric HGN stimulation appears to be a safe and effective therapy for children with DS and refractory severe OSA.
Level of Evidence
4 Laryngoscope, 130:E263–E267, 2020
Inpatient surgical release of lingual frenulums rose 10-fold between 1997 and 2012 despite insufficient evidence that frenotomy for ankyloglossia is associated with improvements in breastfeeding. Clear indications for surgical release remain murky, and best practice guidelines have yet to be developed.OBJECTIVE To determine whether infants referred for frenotomy to treat breastfeeding difficulties should undergo procedures after comprehensive feeding examination, during which the primary cause of feeding issues was identified, and targeted intervention was provided.
DESIGN, SETTING, AND PARTICIPANTSThis observational quality improvement study followed mother-infant dyads between March and December of 2018 who were referred to our tertiary care center for difficulty with breastfeeding. All infants underwent a comprehensive feeding evaluation by speech and language pathologists who examined the infants' ability to breastfeed prior to a surgical consultation for initial frenotomy. Data analysis was performed between January 2019 and May 2019.INTERVENTIONS A multidisciplinary feeding evaluation that examined infants' oral structure and function and their ability to breastfeed and that offered techniques for mothers to address any feeding difficulties prior to surgical intervention was developed. Infants either found success in feeding and weight gain through this program or underwent procedures.
MAIN OUTCOMES AND MEASURESThe primary outcome was the percentage of frenotomy procedures following implementation of a multidisciplinary feeding team evaluation. The secondary outcome was the percentage of infants referred for lingual frenotomy who later had either combined lingual and labial frenotomy or labial frenotomy alone.RESULTS Included in the study were 115 patients (median age, 34 days [interquartile range, 19-56 days], 68 (59%) were male) referred for surgical division of the lingual frenum. Following the development of a program with feeding examination with a pediatric speech and language pathologist, 72 (62.6%) patients subsequently did not undergo surgical procedures. Although all of the referrals were for lingual frenotomy, 10 (8.7%) underwent labial frenotomy alone and 32 (27.8%) underwent both labial and lingual frenotomy.
CONCLUSIONS AND RELEVANCEThe majority of patients referred for ankyloglossia may benefit from alternative intervention strategies following comprehensive feeding evaluation. Close collaboration and formation of multidisciplinary teams are imperative for treating these children.
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