What is a tongue tie? Defining the anatomy of the in-situ lingual frenulum.

Surgical release of the lingual frenulum (frenotomy) has become an increasingly common procedure, performed from birth through to adulthood. Surprisingly, detailed anatomy of the in-situ lingual frenulum has never been described, and no anatomical basis has been proposed for the individual variability in frenulum morphology. The lingual frenulum is frequently referred to as a "cord" or "submucosal band" of connective tissue, yet there is no evidence to support this anatomical construct. This paper aims to describe the anatomy of the in-situ lingual frenulum and its relationship to floor of mouth structures. Fresh tissue microdissection of the lingual frenulum and floor of mouth was performed on nine adult cadavers with photo-documentation and description of findings. The lingual frenulum is a dynamic structure, formed by a midline fold in a layer of fascia that inserts around the inner arc of the mandible, forming a diaphragm-like structure across the floor of mouth. This fascia is located immediately beneath the oral mucosa, fusing centrally with the connective tissue on the tongue's ventral surface. The sublingual glands and submandibular ducts are enveloped by the fascial layer and anterior genioglossus fibers are suspended beneath it. Lingual nerve branches are located superficially on the ventral surface of the tongue, immediately deep to the fascia. The lingual frenulum is not a discrete midline structure. It is formed by dynamic elevation of a midline fold in the floor of mouth fascia. With this study, the clinical concept of ankyloglossia and its surgical management warrant revision. Clin. Anat. 32:749-761, 2019. © 2019 The Authors. Clinical Anatomy published by Wiley Periodicals, Inc. on behalf of American Association of Clinical Anatomists.

Defining the anatomy of the neonatal lingual frenulum.

The lingual frenulum is recognized as having the potential to limit tongue mobility, which may lead to difficulties with breastfeeding in some infants. There is extensive variation between individuals in the appearance of the lingual frenulum but an ambiguous relationship between frenulum appearance and functional limitation. An increasing number of infants are being diagnosed with ankyloglossia, with growing uncertainty regarding what can be considered "normal" lingual frenulum anatomy. In this study, microdissection of four fresh tissue premature infant cadavers shows that the lingual frenulum is a dynamic, layered structure formed by oral mucosa and the underlying floor of mouth fascia, which is mobilized into a midline fold with tongue elevation and/or retraction. Genioglossus is suspended from the floor of mouth fascia, and in some individuals can be drawn up into the fold of the frenulum. Branches of the lingual nerve are located superficially on the ventral surface of the tongue, immediately beneath the fascia, making them vulnerable to injury during frenotomy procedures. This research challenges the longstanding belief that the lingual frenulum is a midline structure formed by a submucosal "band" or "string" and confirms that the neonatal lingual frenulum structure replicates that recently described in the adult. This article provides an anatomical construct for understanding and describing variability in lingual frenulum morphology and lays the foundation for future research to assess the impact of specific anatomic variants of lingual frenulum morphology on tongue mobility. Clin. Anat. 32:824-835, 2019. © 2019 The Authors. Clinical Anatomy published by Wiley Periodicals, Inc. on behalf of American Association of Clinical Anatomists.

Interim Results of a Phase 1/2 Open-Label Study of INO-3107 for HPV-6 and/or HPV-11-Associated Recurrent Respiratory Papillomatosis.

OBJECTIVE: To evaluate the safety, immunogenicity, and efficacy of INO-3107, a DNA immunotherapy designed to elicit targeted T-cell responses against human papillomavirus (HPV) types 6 and 11, in adult patients with recurrent respiratory papillomatosis (RRP; NCT04398433). METHODS: Eligible patients required ≥2 surgical interventions for RRP in the year preceding dosing. INO-3107 was administered by intramuscular (IM) injection followed by electroporation (EP) on weeks 0, 3, 6, and 9. Patients underwent surgical debulking within 14 days prior to first dose, with office laryngoscopy and staging at screening and weeks 6, 11, 26, and 52. Primary endpoint was safety and tolerability, as assessed by treatment-emergent adverse events (TEAEs). Secondary endpoints included frequency of surgical interventions post-INO-3107 and cellular immune responses. RESULTS: An initial cohort of 21 patients was enrolled between October 2020 and August 2021. Fifteen (71.4%) patients had ≥1 TEAE; 11 (52.4%) were Grade 1, and 3 (14.3%) were Grade 3 (none treatment related). The most frequently reported TEAE was injection site or procedural pain (n = 8; 38.1%). Sixteen (76.2%) patients had fewer surgical interventions in the year following INO-3107 administration, with a median decrease of 3 interventions versus the preceding year. The RRP severity score, modified by Pransky, showed improvement from baseline to week 52. INO-3107 induced durable cellular responses against HPV-6 and HPV-11, with an increase in activated CD4 and CD8 T cells and CD8 cells with lytic potential. CONCLUSION: The data suggest that INO-3107 administered by IM/EP is tolerable and immunogenic and provides clinical benefit to adults with RRP. LEVEL OF EVIDENCE: 3 Laryngoscope, 133:3087-3093, 2023.

International Pediatric Otolaryngology Group (IPOG) management recommendations: Pediatric tracheostomy decannulation.

OBJECTIVES: To provide recommendations to otolaryngologists, pulmonologists, and allied clinicians for tracheostomy decannulation in pediatric patients. METHODS: An iterative questionnaire was used to establish expert recommendations by the members of the International Pediatric Otolaryngology Group. RESULTS: Twenty-six members completed the survey. Recommendations address patient criteria for decannulation readiness, airway evaluation prior to decannulation, decannulation protocol, and follow-up after both successful and failed decannulation. CONCLUSION: Tracheostomy decannulation recommendations are aimed at improving patient-centered care, quality and safety in children with tracheostomies.

Competency-Based Assessment Tool for Pediatric Esophagoscopy: International Modified Delphi Consensus.

OBJECTIVES/HYPOTHESIS: Create a competency-based assessment tool for pediatric esophagoscopy with foreign body removal. STUDY DESIGN: Blinded modified Delphi consensus process. SETTING: Tertiary care center. METHODS: A list of 25 potential items was sent via the Research Electronic Data Capture database to 66 expert surgeons who perform pediatric esophagoscopy. In the first round, items were rated as "keep" or "remove" and comments were incorporated. In the second round, experts rated the importance of each item on a seven-point Likert scale. Consensus was determined with a goal of 7 to 25 final items. RESULTS: The response rate was 38/64 (59.4%) in the first round and returned questionnaires were 100% complete. Experts wanted to "keep" all items and 172 comments were incorporated. Twenty-four task-specific and 7 previously-validated global rating items were distributed in the second round, and the response rate was 53/64 (82.8%) with questionnaires returned 97.5% complete. Of the task-specific items, 9 reached consensus, 7 were near consensus, and 8 did not achieve consensus. For global rating items that were previously validated, 6 reached consensus and 1 was near consensus. CONCLUSIONS: It is possible to reach consensus about the important steps involved in rigid esophagoscopy with foreign body removal using a modified Delphi consensus technique. These items can now be considered when evaluating trainees during this procedure. This tool may allow trainees to focus on important steps of the procedure and help training programs standardize how trainees are evaluated. LEVEL OF EVIDENCE: 5. Laryngoscope, 131:1168-1174, 2021.

Competency-Based Assessment Tool for Pediatric Tracheotomy: International Modified Delphi Consensus.

OBJECTIVES/HYPOTHESIS: Create a competency-based assessment tool for pediatric tracheotomy. STUDY DESIGN: Blinded, modified, Delphi consensus process. METHODS: Using the REDCap database, a list of 31 potential items was circulated to 65 expert surgeons who perform pediatric tracheotomy. In the first round, items were rated as "keep" or "remove," and comments were incorporated. In the second round, experts were asked to rate the importance of each item on a seven-point Likert scale. Consensus criteria were determined a priori with a goal of 7 to 25 final items. RESULTS: The first round achieved a response rate of 39/65 (60.0%), and returned questionnaires were 99.5% complete. All items were rated as "keep," and 137 comments were incorporated. In the second round, 30 task-specific and seven previously validated global rating items were distributed, and the response rate was 44/65 (67.7%), with returned questionnaires being 99.3% complete. Of the Task-Specific Items, 13 reached consensus, 10 were near consensus, and 7 did not achieve consensus. For the 7 previously validated global rating items, 5 reached consensus and two were near consensus. CONCLUSIONS: It is feasible to reach consensus on the important steps involved in pediatric tracheotomy using a modified Delphi consensus process. These items can now be considered to create a competency-based assessment tool for pediatric tracheotomy. Such a tool will hopefully allow trainees to focus on the important aspects of this procedure and help teaching programs standardize how they evaluate trainees during this procedure. LEVEL OF EVIDENCE: 5 Laryngoscope, 130:2700-2707, 2020.

Fatal and non-fatal food injuries among children (aged 0-14 years).

OBJECTIVE: To identify and characterize food items with high risk of airway obstruction in children younger than 15 years. METHODS: This retrospective study collected injury data from 1989 to 1998 for 26 pediatric hospitals in the United States and Canada. Aspiration, choking, ingestion, and insertion injuries due to food items were analyzed. The data included 1429 infants and children. Results were compared with fatality data published by the American Association of Pediatrics in 1984. RESULTS: The 10 food objects with the highest frequency for both injuries and fatalities were identified. Peanuts caused the highest frequency of injury, and hot dogs were most often associated with fatal outcomes. The severity of respiratory distress prior to hospital evaluation varied for different foods. Age younger than 3 years was the highest-risk factor. Key characteristics such as bite size, shape, and texture were analyzed and found to demonstrate relationships with severity of clinical outcomes. CONCLUSIONS: Children younger than 3 years remain at greatest risk of food injury and death. We found that hard, round foods with high elasticity or lubricity properties, or both, pose a significant level of risk. Consideration of the key characteristics of the most hazardous foods may greatly decrease airway obstruction injuries. Food safety education can help pediatricians and parents select, process, and supervise appropriate foods for children younger than 3 years to make them safer for this highest-risk population.

Breastfeeding difficulties and oral cavity anomalies: The influence of posterior ankyloglossia and upper-lip ties.

OBJECTIVE: Oral cavity anomalies may contribute to breastfeeding problems. The objective of this study was to describe our experience in a high-volume breastfeeding difficulty clinic with a focus on posterior ankyloglossia and upper-lip ties. METHODS: A retrospective review of patients from a dedicated breastfeeding difficulty clinic from January 2014 to December 2014 was performed. Those identified to have ankyloglossia and/or upper-lip ties underwent release procedures. Subjective breastfeeding changes were documented afterwards. RESULTS: Of the 618 total patients, 290 (47%) had anterior ankyloglossia, 120 (19%) had posterior ankyloglossia, and 14 (2%) had upper-lip tie. Some patients had both anterior ankyloglossia and upper lip-tie (6%), or posterior ankyloglossia and upper-lip tie (5%). For those with anterior ankyloglossia, 78% reported some degree of improvement in breastfeeding after frenotomy. For those with posterior ankyloglossia, 91% reported some degree of improvement in breastfeeding after frenotomy. Upper lip-tie release also led to improved breastfeeding (100%). CONCLUSIONS: Anterior and posterior ankyloglossia and upper-lip tie, or combinations thereof, were commonly recognized in our study population. Many newborns, however, also had no oral cavity anomalies. Although causation cannot be implied, these oral cavity anomalies may contribute to breastfeeding difficulties in some cases.

Long-term follow-up of pediatric recurrent respiratory papillomatosis managed with intralesional cidofovir.

OBJECTIVE: Cidofovir is an acyclic nucleotide phosphonate antiviral medication that has been used intralesionally for the treatment of severe respiratory papillomatosis (RRP) in pediatric patients. The long-term efficacy of this medication was assessed in 11 children with severe RRP who previously required operative debulking every 2 to 6 weeks to maintain airway patency. STUDY DESIGN: Clinical case series. RESULTS: Ten of these children have completed therapy. Five are disease free over a mean follow-up period of 51.6 months. Five other patients with active RRP decreased their mean severity scores from 17.8 (range 11-26) to 4.0 (range 2-6) and no longer require cidofovir. One patient continues to receive cidofovir for recurrent disease after an initial favorable response. Throughout the 6-year observational period, no patients demonstrated any adverse effects, laboratory abnormalities, or evidence of carcinogenesis. CONCLUSION: Intralesional cidofovir is a useful adjunct for managing children with tenuous airways caused by previously uncontrolled papilloma.

Nontuberculous mycobacterial infections of the head and neck.

Nontuberculous mycobacteria are ubiquitous in the environment. Immunocompetent children are commonly infected by these resilient organisms. Cervical lymphadenitis, the most frequent head and neck manifestation of NTM infection, often presents as chronic, unilateral lymphadenopathy with characteristic violaceous overlying skin changes. Diagnosis is ultimately dependent on culture or histopathologic examination of specimen obtained through excisional lymph node biopsy or FNA. The principal treatment of NTM infection remains the surgical excision of diseased tissue. Antibiotics augment surgical therapy and their potential role as a single-modality therapy continues to be investigated.

Management of subglottic hemangioma.

PURPOSE OF REVIEW: Subglottic hemangioma is a challenging congenital anomaly involving the larynx in children. Management is not uniform, and no single treatment modality has been accepted as ideal. During the last several years only a few articles have dealt with this topic. The purpose of this review is to examine the published literature and discuss the alternative treatments of SGH. The authors also describe their approach and the use of microdebrider as a new surgical tool. RECENT FINDINGS: Observation is an option for the rare case of SGH with minimal airway obstruction. However, most cases will require some additional intervention. Systemic steroids intralesional steroid injection, laser ablation with both the CO2 and KTP lasers, interferon (IFN) and open surgical excision have all been utilized. SUMMARY: The authors found few technical innovations in this field in the last 5 years. All techniques utilized have some degree of success. However, all approaches have downsides and are associated with complications, some of which can be very serious. Ideally, more structured research comparing techniques would be helpful to best determine operative and postoperative management.

Unilateral choanal atresia in identical twins: case report and literature review.

Choanal atresia is postulated to be secondary to an abnormality of the rupture of the buccopharyngeal membrane during the embryological period. This condition usually occurs sporadically, but has been described in siblings and successive generations. The genetics remain unclear. We present monozygotic twins with identical findings of unilateral choanal atresia and no other associated anomalies. To our knowledge, this is the first report of such an occurrence.

Current use of intralesional cidofovir for recurrent respiratory papillomatosis.

OBJECTIVES/HYPOTHESIS: The authors sought to define the indications, administration, and adverse events associated with intralesional cidofovir use for recurrent respiratory papillomatosis (RRP). STUDY DESIGN: Cross-sectional study. METHODS: A 21-question online survey was distributed to 115 selected adult and pediatric laryngeal surgeons internationally. Results were used to draft statements of best practice, which were approved by the full membership of the RRP Task Force. RESULTS: Eighty-two surgeons, who altogether presently manage 3,043 patients with RRP, responded to the survey. Seventy-four surgeons previously used cidofovir, reporting 1,248 patients in the last decade (estimated 801 adults and 447 children). Single indications for adjuvant cidofovir included six or more surgeries per year, increasing frequency of surgery, and extralaryngeal spread (in children). Most adult surgeons use 20 to 40 mg in <4 mL; pediatric surgeons use <20 mg in <2 mL. Scheduled administration following an initiation trial of five injections is common; cidofovir is discontinued following a complete response. Most surgeons biopsy routinely, use special informed consent, and are willing to participate in multi-institutional clinical trials on cidofovir uses, efficacy, and safety. CONCLUSIONS: Eighteen statements were approved by the RRP Task Force after discussion of the survey results. Intralesional cidofovir may be initiated if surgical debulking is required every 2 to 3 months. The concept of an adjuvant regimen with regular biopsy is favored. Administration should remain below established safe limits of dosing (3 mg/kg) and volume. Informed consent, including discussion of off-label use and acute kidney injury in children, is important. A special consent form sample is included. There remains a need for high-quality data.

Unilateral lymphangiomatous polyp of the palatine tonsil in a very young child: a clinicopathologic case report.

Childhood lymphangiomatous polyp of the palatine tonsil is a very unusual lesion found in the head and neck. Tonsillectomy has been reported to be the curative procedure of choice for this lesion. We report a case of a very young child with unilateral lymphangiomatous polyp of the palatine tonsil.

Defining ankyloglossia: a case series of anterior and posterior tongue ties.

INTRODUCTION: Ankyloglossia is a congenital condition in which tongue mobility is limited due to an abnormality of the lingual frenulum. The impact of ankyloglossia on breastfeeding is poorly understood but there is a recent trend toward more recognition of this condition and early intervention when needed. Currently, there lacks clear definition of ankyloglossia and different subtypes have been proposed with no clinical correlation. OBJECTIVE: To determine the prevalence of anterior versus posterior ankyloglossia in a large series of consecutive patients and to assess clinical outcomes after frenotomy. METHODS: Retrospective chart review of patients from July 2007 to July 2009 who were diagnosed with ankyloglossia and underwent office frenotomy. Baseline characteristics, specific feeding issues, type of ankyloglossia, and clinical outcomes after frenotomy were reviewed. RESULTS: Of the 341 total patients, 322 (94%) had anterior ankyloglossia and 19 (6%) had posterior ankyloglossia. Median age at presentation was 2.7 weeks (range 1 day of life to 24 weeks); 227 were males and 114 were females. Revision frenotomy rates were significantly higher for the posterior ankyloglossia group (3.7% anterior and 21.1% posterior, p=0.008). CONCLUSION: Anterior ankyloglossia is much more common and readily managed when compared to posterior ankyloglossia. Posterior ankyloglossia is a poorly recognized condition that may contribute to breastfeeding difficulties. The diagnosis is difficult due to the subtle clinical findings but relevant health care providers should be aware of this condition. Frenotomy is a simple, safe, and effective intervention for ankyloglossia which improves breastfeeding.