Ankyloglossia and Tethered Oral Tissue: An Evidence-Based Review.

Tethered oral tissue may represent anatomic variation or true pathology with functional limitations. The most prevalent functional limitation is impaired feeding due to ankyloglossia. Treatment options include conservative management with feeding support versus intervention such as frenotomy or frenuloplasty. The benefits of intervention have yet to be fully elucidated. Many can agree that intervention may improve maternal pain during breastfeeding, but the impact of these procedures on feeding efficiency and intake is debated. Alternate ties such as posterior ankyloglossia and lip tie have been proposed as pathologic oral ties, but their true impact on feeding and speech articulation remains unclear.

Sclerotherapy for Benign Cystic Lesions of the Head and Neck: Systematic Review of 474 Cases.

OBJECTIVE: The role of sclerotherapy for vascular lesions of the head and neck is well established. However, the efficacy of sclerotherapy for benign cystic lesions of the head and neck is less clear. The objective of this review is to determine the efficacy and safety of sclerotherapy for benign cystic lesions of the head and neck. DATA SOURCES: PubMed/MEDLINE, Cochrane Library, and Embase. REVIEW METHODS: The PRISMA guidelines (Preferred Reporting Systems for Systematic Reviews and Meta-analyses) were followed for this systematic review. Studies of patients with benign head and neck cystic masses treated primarily with sclerotherapy were included. Thirty-two studies met criteria for inclusion. RESULTS: A total of 474 cases of sclerotherapy were reviewed. Agents comprised OK-432, ethanol, doxycycline, tetracycline, and bleomycin. Lesions in the analysis were ranula, thyroglossal duct cyst, branchial cleft cyst, benign lymphoepithelial cyst, parotid cyst, thoracic duct cyst, and unspecified lateral neck cyst. A total of 287 patients (60.5%) had a complete response; 132 (27.9%) had a partial response; and 55 (11.6%) had no response. OK-432 was the most widely utilized agent, with a higher rate of complete response than that of ethanol (62.0% vs 39.4%, P = .015). Fifty-three cases (11.2%) required further surgical management. One case of laryngeal edema was reported and managed nonoperatively. CONCLUSION: Sclerotherapy appears to be a safe and efficacious option for benign cystic lesions if malignancy is reliably excluded. Efficacy rates are comparable to those of sclerotherapy for vascular malformations. The rate of serious complications is low, with 1 incident of airway edema reported in the literature.

Vocal Fold Motion Impairment Following Chemotherapy Administration: Case Reports and Review of the Literature.

OBJECTIVE: Chemotherapy-induced vocal fold motion impairment (CIVFMI) is a rare complication of cancer therapy with potential for airway compromise. The objective of this review is to present 2 new cases of CIVFMI to add to the literature as well as characterize the demographics, symptoms, exam findings, airway complication rates and prognosis of CIVFMI. METHODS: A search of Pubmed/MEDLINE (1970 to May 1, 2020), Embase (1970 to May 1, 2020), and Cochrane Library using medical study heading (MeSH) terms related to chemotherapy (drug therapy, chemotherapy, vincristine, vinblastine, paclitaxel) and vocal cord motion impairment (vocal cord, cords, vocal folds, immobility, hypomobility) was performed. Exploratory pooling of data without formal meta-analysis was performed. RESULTS: A preliminary search yielded 148 abstracts, review articles and studies. A total of 23 studies met inclusion criteria. There were 35 total cases presented in the literature, with a mean age of 29.5 (0.4-78). The most common cancer diagnosis was acute lymphoblastic leukemia (n = 15, 42.9%), and the most common agent was vincristine (n = 30, 85.7%). Dysphagia, bilateral CIVFMI, and vocal fold immobility rather than hypomobility were more common in pediatric patients. There were 8 cases of surgical airway intervention, including tracheostomy and posterior cordotomy. The duration of symptoms was 7 to 420 days, and spontaneous resolution was reported in 32 cases. CONCLUSIONS: CIVFMI has potential for airway complications requiring surgical intervention. Spontaneous resolution after cessation of the offending agent is the most likely outcome. Bilateral CIVFMI, dysphagia and vocal fold immobility are more common in the pediatric population.

International Pediatric Otolaryngology Group (IPOG) survey: Efforts to avoid complications in home tracheostomy care.

OBJECTIVE: To provide guidance for home care tracheostomy management in the pediatric population. The mission of the IPOG is to develop expertise-based recommendations for the management of pediatric otolaryngologic disorders with the goal of improving patient care. METHODS: Survey of expert opinion by the members of the International Pediatric Otolaryngology Group (IPOG). RESULTS: Survey results provide guidance for caregiver teaching, the reuse of tracheostomies and suction catheters while inpatient and following discharge, acceptable sterilization practices for tracheostomies, tracheitis workup and management, and outpatient follow-up practices. CONCLUSION: This presentation of common home tracheostomy care practices are aimed at improving patient-centered care in the pediatric population.

Assessment of Pain and Analgesic Use in Children Following Otologic Surgery.

OBJECTIVE: To compare postoperative pain and analgesic use in children following transcanal endoscopic ear surgery (TEES) vs non-transcanal endoscopic ear surgery (non-TEES). STUDY DESIGN: Prospective case series. SETTING: Tertiary care center. METHODS: Surveys using the Wong-Baker FACES Pain Rating Scale and recording the frequency and dosage of consumed analgesics were administered prospectively to caregivers of children undergoing otologic surgery between May 2018 to February 2020. Pain intensity and medication use were recorded twice daily for 6 days, starting on postoperative day 0. Mean pain scores and mean number of consumed analgesic doses were compared between groups. RESULTS: Survey response rate was 57.9%. Among 53 patients who completed the survey, 35 (66.0%) underwent TEES and 18 (34.0%) underwent non-TEES. Mean pain ratings on postoperative days 0 and 1 were significantly lower among children undergoing TEES (2.2 and 2.1) vs non-TEES (4.0 and 4.1), P = .045 and P = .008, respectively (Mann-Whitney U test). The mean pain ratings across the 6 days were similar in TEES (1.7) and non-TEES (2.6) (P = .140, Mann-Whitney U test). The mean number of analgesic doses consumed per half-day over the 6 days was significantly lower among children undergoing TEES (0.3) vs non-TEES (0.6; P = .049, Mann-Whitney U test). CONCLUSION: Postoperative pain following TEES and non-TEES in children was overall low. Children undergoing TEES had a small but statistically significant decrease in pain on postoperative days 0 and 1 and decreased use of pain medications compared to non-TEES.

Factors associated with frenotomy after a multidisciplinary assessment of infants with breastfeeding difficulties.

OBJECTIVE: Frenotomy for ankyloglossia has increased nearly 10-fold over the past few decades despite insufficient evidence that the procedure improves breastfeeding outcomes. There is no universally accepted method for identifying patients who may benefit from the procedure. The objective of this study is to determine if comprehensive feeding evaluations and targeted interventions can identify children who should undergo procedures, and to identify factors associated with lip or tongue frenotomy to treat breastfeeding difficulties. METHODS: This observational quality improvement study followed infant-mother dyads between March 2018 and December 2019 referred to our tertiary care center for breastfeeding difficulties. Speech and language pathologists performed comprehensive feeding evaluations on infants prior to surgical consultation for frenotomy. Infants' oral anatomy and function and their ability to breast and bottle feed were assessed, and techniques for mothers to address feeding difficulties without a procedure were offered prior to surgical consultation. Infants either found success over a short observation period or underwent procedures (lip and/or tongue frenotomy). RESULTS: 153 patients (mean age 47.0 days (stdev 39.0 days, 56.2% male) were referred for surgical division of the lingual frenulum. Following development of a program utilizing pediatric speech language pathologists to perform feeding evaluations prior to surgical consultation, 69.9% of patients subsequently did not undergo surgical procedures. 11 (23.9%) underwent labial frenotomy alone and 30 (65.2%) underwent both labial and lingual frenotomies. Frenotomy was associated with significantly increased worry subscale of the Feeding Swallow Impact Survey (FSIS) and decreased mean Breastfeeding Self Efficacy Scale score (p = 0.0001, p = 0.006, respectively). Tongue appearance was significantly associated with having a procedure, while lip appearance was not. The Bristol Breastfeeding Assessment Tool (BBAT) was lower in children undergoing tongue and/or lip frenotomy (p = 0.0006), while the Hazelbaker Assessment Tool for Lingual Frenulum Function (HATLFF) appearance and function scores were lower in children undergoing lingual frenotomy with or without lip frenotomy (p = 0.0008, p = 0.0009, respectively). CONCLUSIONS: The majority of patients referred for ankyloglossia may benefit from nonsurgical intervention strategies based on findings from comprehensive feeding evaluation. Frenotomy is associated with higher maternal feeding-related worry and reduced breastfeeding self-efficacy scores. While tongue appearance is associated with frenotomy, functional assessment is critical for identifying patients who may also benefit from lip frenotomy.

Quantifying the benefits from a care coordination program for tracheostomy placement in neonates.

OBJECTIVE: Value-based care models are becoming instrumental in structuring clinical care delivery in our healthcare climate. Our objective was to determine the value associated with implementation of a Family-Centered Care Coordination (FCCC) program for neonates undergoing tracheostomy. METHODS: A multi-disciplinary FCCC program was implemented at the Massachusetts Eye and Ear Infirmary and MassGeneral Hospital for Children in January 2013. This program is designed to ensure a safe transition out of the hospital for children undergoing tracheostomy, reduce re-admission rates, and increase caregiver quality of life (QOL). Study participants included neonates undergoing tracheostomy in 2012 and 2015. This retrospective cohort study examined length of stay (LOS), utilized time-driven activity-based costing to estimate the cost of care, assessed caregiver QOL with 1-month Pediatric Tracheostomy Health Status Instrument (PTHSI) scores, and assessed complications with 6-month Medical Complications Associated with Pediatric Tracheostomy (MCAT) scores. RESULTS: Following implementation of the FCCC program, average LOS decreased from 30.5 days (range 17-39) to 16.6 days (range 9-23). The largest process improvement (cost reduction of 61%) occurred in the discharge-planning phase. The overall cost per care cycle was reduced by 36%. A large clinically meaningful benefit was demonstrated for PTHSI (effect size 0.80) as well as MCAT scores (effect size 9.35). CONCLUSIONS: We demonstrated the higher outcomes, including reductions in caregiver burden and complication rates, and the lower costs associated with implementation of the FCCC program for neonates undergoing tracheostomy.

Pediatric Endoscopic Ossiculoplasty Following Surgery for Chronic Ear Disease.

OBJECTIVE: Rigid endoscopes can improve visualization of the tympanic space compared to traditional microscopic techniques. This study investigates whether use of transcanal endoscopic ossiculoplasty influences audiologic outcomes compared to microscopic ossiculoplasty following chronic ear surgery in children. STUDY DESIGN: Comparative cohort study at two tertiary care centers. METHODS: Retrospective review of pediatric chronic ear cases where ossiculoplasty was performed from February 2009 to March 2018. RESULTS: We identified 100 ears that underwent endoscopic ossiculoplasty and 100 ears that underwent microscopic ossiculoplasty. The mean age was 11 years (range, 4-18 years) with 63% males. There were no significant differences in these parameters between the two groups. Subjects underwent either primary ossiculoplasty or ossiculoplasty during second-look procedures. There was no significant difference in air conduction pure tone average (PTA) after microscopic cases compared to endoscopic cases (-12.5 dB vs. -10.5 dB, P = .40). These results were independent of prosthesis type. Microscopic ossiculoplasty was significantly more likely to use a post-auricular approach (P = .0001). There was no difference in complication rate between the two groups. The malleus was more likely to be absent or removed prior to endoscopic ossiculoplasty (P = .0004) with no significant difference in the change in PTA between groups. CONCLUSIONS: Transcanal endoscopic ossiculoplasty was found to have equivalent audiometric outcomes with significantly fewer post-auricular approaches and no increase in complications compared to microscopic ossiculoplasty. While the malleus was more likely to be absent in endoscopic cases, this did not appear to influence the change in PTA. LEVEL OF EVIDENCE: 4 Laryngoscope, 2020.

Vagal stimulation and laryngeal electromyography for recurrent laryngeal reinnervation in children.

Ansa-to-recurrent laryngeal nerve (ANSA-RLN) reinnervation procedures are now often first-line treatments for some children with unilateral vocal fold immobility. Although many describe that children with prolonged denervation and true vocal fold atrophy should not undergo this procedure, there has been no gold-standard means of identifying true denervation. Here, we describe a novel technique using evoked vagal electromyography to predict degree of chronic nerve injury prior to recurrent laryngeal nerve reinnervation in children. This is a simple, readily available technique that may play an important role in predicting likelihood of success with pediatric ANSA-RLN reinnervation. Laryngoscope, 130:747-751, 2020.

Update on hypoglossal nerve stimulation in children with down syndrome and obstructive sleep apnea.

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.

Long-term swallowing outcomes following type 1 laryngeal cleft injection.

INTRODUCTION: Interarytenoid injection augmentation (IIA) during initial diagnostic endoscopy for aspiration and dysphagia has been described as both a diagnostic and therapeutic technique in the evaluation of type 1 laryngeal cleft (LC-1). IIA is additionally hypothesized to be a temporizing measure that facilitates improvement of swallowing function and potentially obviates the need for future formal endoscopic suture repair of LC-1. However, long-term (>6 month) outcomes of IIA for LC-1 remain largely unknown. The objective of this study was to evaluate the effect of IIA on long-term swallowing outcomes and need for formal endoscopic suture repair in patients with LC-1. METHODS: This is a retrospective cohort study of patients age ≤24 months with pharyngeal phase dysphagia on preoperative videofluoroscopic swallow study (VFSS) who underwent IIA for LC-1 during diagnostic laryngoscopy and bronchoscopy at a single tertiary care academic subspecialty hospital from June 2017 to May 2018. Included patients underwent VFSS within 30 days of IIA and had documented SLP follow up at 6 months or more post-procedure. Exclusion criteria included prior cleft repair, gastrostomy tube dependence, additional procedures at the time of IIA, or lack of documented follow up. A total of 34 patients underwent LC-1 during study period with 24 included in final analyses. The primary outcome measure was improvement in safely swallowed consistency at 6 months or greater following injection. Secondary outcomes included need for formal suture LC-1 repair following IIA and comparison of 30-day and long-term swallowing function. RESULTS: Median [range] age at injection was 15.3 [10.3-19.1] months and 50% were female (n = 12). Improvement was noted in 12 (50%) patients within 30 days of IIA, with 11 of 12 demonstrating sustained improvement at long-term follow up (10.3 [9.3-14.0] months). Among all patients, 15 of 24 (63%) demonstrated improvement compared to preoperative baseline. Six of 24 (25%) required formal suture repair of LC-1. CONCLUSIONS: IIA is a safe procedure that may result in both immediate and long-term improvement in dysphagia in select patients with LC-1. Additional studies are required to determine impact of IIA on pulmonary complications and hospital utilization and as well as patient- and caregiver-related outcome measures.

Lessons learned to aid the successful outcome of pediatric recurrent laryngeal nerve reinnervation.

While Ansa to recurrent laryngeal nerve reinnervation is gaining popularity in the treatment of unilateral vocal fold immobility, little has been reported on commonly encountered surgical challenges with this procedure. Here, we present a cohort of 21 pediatric patients who underwent this procedure with a full description of techniques used to overcome common challenges with this procedure. We report vocal and swallowing outcomes for these patients, with an overall success rate of 19/21 (90.5%) patients, with success defined as improvement of a half-consistency or resolution of vocal issues.

Association of Feeding Evaluation With Frenotomy Rates in Infants With Breastfeeding Difficulties.

IMPORTANCE: 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 PARTICIPANTS: This 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 MEASURES: The 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 RELEVANCE: The 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.

Pharyngeal flap using carotid artery mobilization in 22q11.2 deletion syndrome with velopharyngeal insufficiency.

INTRODUCTION: 22q11.2 deletion syndrome is the most common microdeletion syndrome in children. Many patients with this disease develop craniofacial defects including cleft palate, bifid uvula, and velopharyngeal insufficiency. Our study adds to the current body of literature by describing a novel technique of carotid mobilization performed in conjunction with pharyngeal flap surgery in patients with extensive medialization of the carotid artery. METHODS: Carotid artery mobilization followed by insertion of a superiorly based pharyngeal flap was performed on two patients, a 10-year-old girl and a 5-year-old boy, with 22q11.2 deletion syndrome concurrent with velopharyngeal insufficiency. RESULTS: Neither patient experienced significant post-operative issues. Following the procedure, parents of both patients noted significant speech and voice improvement. Both patients had improvements in VPI Effects On Life Outcome (VELO) scores, nasometry, and production of paragraph passages following surgery. CONCLUSIONS: Our study describes a novel surgical treatment for children with 22q11.2 deletion syndrome with significant velopharyngeal insufficiency (VPI). The procedure wherein is characterized by an extensive mobilization of the carotid artery followed by implantation of a pharyngeal flap. This technique resulted in no significant intra-operative bleeding, and was measured to be successful as noted by nasometry scores and changes in pre- and post-op VELO scores.