Tongue-based Airway Obstruction Research Articles

Congenital Deformities of the Face, Head, and Neck.

Preface Preface Congenital Deformities of the Face, Head, and Neck Travis T. Tollefson, MD, MPH, FACS 1 1 Department of Otolaryngology, University of California, Davis, California Facial Plast Surg 2016;32:121–122. This issue is dedicated to the surgical reconstruction of facial deformities, which have occurred due to abnormalities of development. We revere the delicate balance of craniofacial development. The phenomena of organized fusion and com- munication between embryonal growth centers lead to a perfectly beautiful spectrum of facial structures. In this issue, leaders from several surgical specialties have brought forth the state of the science in the management of craniosynostosis and congenital nasal and eyelid deformities. The management strategies for children with cleft lip and palate are outlined in relation to treatment in the early years of speech development and secondary speech surgery. An evidence-based approach to vascular malformations of the head and neck is presented with rigorous inspection of the available supportive research for the treatment of these varied and difficult facial deformities. In addition, the management options for newborns with tongue-based airway obstruction have expanded with broad application of the principles of distraction osteogenesis. The algorithm for treating these children is presented with de- tailed surgical technique and images. Microtia reconstruction is one of the most challenging and often humbling endeavors, due in part to the complex ear shape, unpredictable healing, and the need for staged procedures. The authors have pro- duced a synthesis of decades of experience in microtia repair, which is a testament to teamwork and streamlining an efficient surgical paradigm. The area of facial nerve reanimation has grown signifi- cantly for children with developmental or congenital facial paralysis. The evolution of the transplanted neuromuscular flap for reanimating a paralyzed face is most commonly performed with the gracilis muscle. The authors’ current approach is detailed with at- tention to the techniques of the colleagues who popular- ized the procedures. As the field of global sur- gery emerges, traveling surgi- Travis T. Tollefson, MD, MPH, FACS cal (missions) teams are transitioning from solely short-term humanitarian care de- livery to hybrid models, which incorporate host country health care professionals, academic–private partnerships, a broader view of education, and support. These novel models and the research in the burden of surgical disease are summarized. I hope you will enjoy reading this issue as much as I have. As Mark Twain suggests, these authors contribute to the surgical literature with recapitulation of work from others, while innovating with modern technologies. Address for correspondence Travis T. Tollefson, MD, MPH, FACS, Facial Plastic and Reconstructive Surgery, Department of Otolaryngology- Head and Neck Surgery, University of California, Davis, 2521 Stockton Blvd., Suite 7200, Sacramento, CA 95817 (e-mail: tttollefson@ucdavis. edu). Copyright © 2016 by Thieme Medical Publishers, Inc., 333 Seventh Avenue, New York, NY 10001, USA. Tel: +1(212) 584-4662. Issue Theme Congenital Deformities of the Face, Head, and Neck; Guest Editor, Travis T. Tollefson, MD, MPH, FACS We simply take a lot of old ideas & put them into a sort of mental kaleidoscope… We give them a turn and they make new and curious combinations… indefinitely. —Mark Twain, a Biography 1 Reference 1 Paine AB. Mark Twain, a Biography. Vol III, Part 1: 1900–1907. ECHO LIB, 2006:103. ISBN: 1847029833 DOI http://dx.doi.org/ 10.1055/s-0036-1581139. ISSN 0736-6825.

Mandibular distraction osteogenesis for the treatment of neonatal tongue-based airway obstruction.

Early postnatal tracheostomy for airway compromise is associated with high morbidity and cost. In certain patients with tongue-base airway obstruction (TBAO), mandibular distraction osteogenesis may be preferred. We present a comprehensive analysis of surgical, airway, and cephalometric outcomes in a large series of neonatal patients with TBAO. A retrospective review was performed of patients with laryngoscopically proven TBAO who underwent mandibular distraction osteogenesis before 1 year of age at our institution. Demographic, operative, postoperative, polysomnographic, and radiographic data were analyzed with the appropriate statistical test. Between 2010 and 2013, 28 patients younger than 1 year underwent mandibular distraction for TBAO. Distraction was performed for documented TBAO and failure to thrive at an average age of 58 days (range, 11-312) days with distractor removal after an average of 90 days. Preoperative polysomnograms were obtained on 20 patients with an average apnea-hypopnea index of 39.3 ± 22.0/h; the apnea-hypopnea index on postoperative polysomnograms obtained after distraction completion was significantly reduced in all 14 patients in whom it was measured (mean, 3.0 ± 1.5/h; P < 0.0001). Twenty patients transitioned to oral feeding, and cephalometric and airway diameters were improved (P < 0.0001). Distraction was successful in all but 4 patients including all patients with GILLS scores of 2 or less and 66% of patients with GILLS scores of 3 or greater. Neonatal mandibular distraction is a powerful tool to treat critical obstructive apnea in patients with TBAO. Appropriate patient selection remains a challenge; however, mandibular distraction represents a compelling treatment modality.

Neonates with Tongue‐Based Airway Obstruction

In this systematic review, the authors summarize the current evidence in the literature regarding diagnosis, treatment, and long-term outcomes in neonates with tongue-based airway obstruction (TBAO) and assess the level of evidence of included studies. The terms Pierre Robin syndrome/sequence, micrognathia, retrognathia, and cleft palate were combined with airway obstruction, treatment, tongue-lip plication, and osteogenesis distraction to perform an Ovid literature search, yielding 341 references. The authors excluded references containing patients with isolated choanal/nasal obstruction, patients older than 12 months, and expert opinion papers, yielding 126 articles. The authors searched 3 electronic databases and reference lists of existing reviews from 1980 to October 2010 for articles pertaining to the diagnosis, treatment, and outcomes of TBAO. Reviewers assigned a level of evidence score based on Oxford's Centre for Evidence Based Medicine scoring system and recorded relevant information. Most studies were case studies and single-center findings. The lack of standardization of diagnostic and treatment protocols and the heterogeneity of cohorts both within and between studies precluded a meta-analysis. There was little evidence beyond expert opinion and single-center evaluation regarding diagnosis, treatment, and long-term outcomes of neonates with TBAO. The variability in the phenotype of the cohorts studied and the absence of standardized indications for intervention preclude deriving any definitive conclusions regarding diagnostic tools to evaluate this patient population, treatment choices, or long-term outcomes. A coordinated multicenter study with a standardized diagnostic and treatment algorithm is recommended to develop evidence for the diagnosis and treatment of neonates with TBAO.

Mandibular Distraction Osteogenesis Used to Treat Upper Airway Obstruction

To evaluate whether mandibular distraction osteogenesis relieves tongue-based airway obstruction in patients with severe micrognathia. Retrospective medical review spanning a 7-year period in a tertiary care hospital. The inclusion criterion was defined as micrognathia associated with a severe tongue-based obstruction. The patients included 11 neonates and infants (mean age, 4.3 months) and 2 pediatric patients (mean age, 5.4 years). Two patients had already received tracheotomies, 11 had not. The intervention was bilateral mandibular osteotomies with distraction osteogenesis. The outcome measures were avoidance of tracheotomy and decannulation. Ten of 11 patients avoided tracheotomy. Two of 2 patients who had already undergone tracheotomies were successfully decannulated. Mandibular distraction osteogenesis is an acceptable treatment alternative to tracheotomy in select pediatric patients with micrognathia and severe tongue-based obstruction.

Tongue-Lip Adhesion in the Treatment of Pierre Robin Sequence

Pierre Robin sequence (PRS) is a congenital malformation in which micrognathia causes retroposition of the tongue. This results in feeding and breathing difficulties that can lead to severe complications. If conservative treatment is not sufficient, a surgical procedure such as a tongue-lip adhesion can be performed. The objective of this study was to evaluate our benefits and complications with this operation. All patients under the care of the Department of Plastic and Reconstructive Surgery of the VU University Medical Center, Amsterdam, The Netherlands, in the period 1993 to 2002. A consecutive series of 22 patients who needed operative intervention for PRS. All underwent a tongue-lip adhesion. Retrospective analysis of the patient charts. Follow-up has been performed for a minimum of 1 year. In 16 (73%) of the patients operated on, feeding and breathing have improved. In 6 children, the results were limited because of concomitant congenital anomalies. Complications, all without lasting effect, were observed in 12 (55%) of the patients: 4 of 5 children with dehiscence of the adhesion needed reoperation, 6 developed small chin abscess that resolved after removal of the supporting suture, and 1 patient was reintubated for bronchospasm. Tongue-lip adhesion is a good surgical treatment for most children with PRS who have an isolated tongue-base airway obstruction. More invasive procedures such as mandibular distraction can be reserved for patients where a tongue-lip adhesion has not been successful.

To Distract or Not to Distract: An Algorithm for Airway Management in Isolated Pierre Robin Sequence

Approaches advocated for treatment of airway obstruction among neonates with Pierre Robin sequence include positioning, tongue-lip adhesion, mandibular distraction, and tracheostomy, with no established guidelines regarding which modality is appropriate for a specific patient. This report proposes an algorithm for the management of neonatal upper airway obstruction among patients with isolated Pierre Robin sequence. Data for 21 patients with isolated Pierre Robin sequence who were treated by one surgeon during a 9-year period were reviewed. Eighteen patients presented during the first 1 week of life and three patients presented late, between 12 and 33 months of age. Follow-up periods ranged from 9 to 70 months (median, 33 months). Successful airway management was achieved with positioning alone for 10 patients, with tongue-lip adhesion for seven of nine patients, with tracheostomy for two patients, and with mandibular distraction for three patients. Changes in the maxillary-mandibular discrepancy were significant with natural mandibular growth during the first 1 year of life (p < 0.0001). Oromotor studies performed 3 months or more after tongue-lip adhesion reversal (n = 9) demonstrated no appreciable deficits in tongue function, relative to other children with cleft lips/palates. A multidisciplinary team should evaluate all patients with isolated Pierre Robin sequence, to fully assess the maxillary-mandibular relationship, anatomically define the site of airway obstruction, and identify feeding difficulties. Patients should be evaluated for episodes of desaturation occurring spontaneously, during feeding, or during sleeping. Patients with desaturation should be further evaluated with double endoscopy (nasoendoscopy and bronchoscopy). If the airway obstruction is localized to the tongue base alone and cannot be controlled with positioning, then tongue-lip adhesion is the initial treatment of choice, because such patients demonstrate significant mandibular growth during the first 1 year of life. Mandibular distraction among neonates is reserved for failures of tongue-lip adhesion in which isolated tongue-base airway obstruction is documented. Neither of the patients who experienced failure of tongue-lip adhesion in this series would have been a candidate for distraction with the algorithm presented. Avoiding routine neonatal distraction serves to avoid facial scarring, nerve and tooth bud injury, and potential disturbances of intrinsic mandibular growth. Patients with persistent respiratory difficulties beyond age 9 months require reevaluation for multiple sites of airway obstruction. Mandibular distraction may be one of several modalities required to avoid tracheostomy for such patients.

Mandibular distraction in neonates: a strategy to avoid tracheostomy.

Over the past 5 years, the authors developed an application of mandibular distraction osteogenesis to eliminate existing tracheostomy. That experience led the authors to attempt mandibular distraction osteogenesis in neonates as an alternative before tracheostomy. Success with this approach using supporting objective airway measurements has been reported previously. This report includes six neonates diagnosed with Pierre Robin sequence. Of the six, five neonates ranging in age from 6 to 26 days (mean, 14.5 days) were treated by the authors with mandibular distraction over a 22-month period. The sixth neonate was treated with tracheostomy, because of other airway abnormalities. Findings included retrognathia, glossoptosis, incomplete cleft palate, and airway obstruction in each patient. Birth weights ranged from 2.8 to 3.2 kg. All patients were unable to control their airway during feeding, as evidenced by repeated episodes of choking and obstruction. Resting oxygen saturations were in the 70 to 80 percent range in all patients, with further deterioration during attempted feeding. Bronchoscopy was performed in all patients under anesthesia before distraction. Recurrent near-complete and intermittent complete airway obstruction were present in all patients at the level of the tongue base. There was a consensus by a pediatric intensivist, a pediatric anesthesiologist, and a pediatric otolaryngologist in all cases. Each patient met all criteria requiring ventilation for life support. Tracheostomy would be required if mandibular distraction osteogenesis was not performed, or if it failed. Patients with other airway abnormalities were not considered for treatment. Maxillomandibular disharmony measured at the midline ranged from 8 to 15 mm (mean, 11.2 mm). Active distraction was performed at the rate of 1 to 2 mm a day, with a consolidation period of 4 weeks. Total time of treatment was less than 6 weeks in all cases. All patients were extubated by the completion of active distraction. Distraction distance ranged from 8 to 15 mm (mean, 12.4 mm). All patients were discharged to home on apnea monitors, the use of which was discontinued after 90 days with no further apneic events. Weight gains met or exceeded the average 500 g a month after distraction. Bronchoscopy at the time of distractor removal showed correction of airway obstruction at the tongue base. Radiographs showed bilateral ossification of the distraction sites. Tracheostomy was avoided in all cases selected for treatment by distraction. Patient follow-up range was 9 to 22 months. In selected Pierre Robin sequence patients with tongue base airway obstruction, mandibular distraction osteogenesis can successfully avoid the need for and the associated mortality and morbidity of indwelling tracheostomy.

Distraction Osteogenesis of the Mandible for Airway Obstruction in Children

Objectives of the study were to determine the effectiveness of distraction osteogenesis of the mandible to relieve airway obstruction in children with tongue-base airway obstruction and to describe the new surgical techniques developed for use in infants and young children. Prospective, nonrandomized study. Analysis of 11 children with severe airway obstruction secondary to tongue-base obstruction was performed. Patients were between 2 weeks and 5.5 years of age. All patients underwent distraction osteogenesis of the mandible. Twelve distraction procedures in the 11 children in the study were accomplished; decannulation or extubation was successful in all children. Distraction osteogenesis of the mandible can be used to treat tongue-base airway obstruction in appropriately selected children.