Tongue–lip adhesion and tongue repositioning for obstructive sleep apnoea in Pierre Robin sequence: A systematic review and meta-analysis

Camacho, Macario; Noller, Michael; Zaghi, Soroush; Reckley, Lauren K.; Fernandez-Salvador, Camilo; Ho, Erika; Dunn, Brandyn; Chan, Dylan K.

doi:10.1017/s0022215117000056

Cited by 24 publications

(9 citation statements)

References 15 publications

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“…This has potential significance in some infants, where division of the lingual frenulum (to improve tongue mobility) has potential to compromise resting tongue position and “stability.” This has been reported in several infants with Pierre Robin sequence, who experienced airway compromise postfrenotomy (Genther et al, ). Although uncommonly performed currently, infants with Pierre Robin sequence and airway compromise were historically considered for a surgical procedure that sutured to tip of the tongue to the lip, recognizing that anterior tongue position can alter the oropharyngeal airway, the apnoea/hypopnea index and oxygen saturation in infants with retrognathia (Camacho et al, ). As such, it should be recognized that division of the lingual frenulum may alter tongue position at rest, with potential to worsen any pre‐existing airway compromise.…”

Section: Discussionmentioning

confidence: 99%

Defining the anatomy of the neonatal lingual frenulum

et al. 2019

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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.

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Section: Discussionmentioning

confidence: 99%

Defining the anatomy of the neonatal lingual frenulum

et al. 2019

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“…Mandibular distraction osteogenesis was the most common primary procedure, followed by tongue-lip adhesion, and tracheostomy (84). A meta-analysis of 7 studies with 90 patients showed that tongue-lip adhesion and tongue repositioning can improve AHI and oxygenation parameters during sleep (85).…”

Section: Pierre-robin Sequence/syndromementioning

confidence: 99%

Sleep-disordered breathing in paediatric setting: existing and upcoming of the genetic disorders

Zaffanello¹,

Antoniazzi²,

Tenero³

et al. 2018

Ann. Transl. Med.

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Childhood obstructive sleep apnea syndrome (OSAS) is characterized by anatomical and functional upper airway abnormalities as pathophysiological determinants, and clinical symptoms are frequently clear. OSAS is widely described in rare genetic disorders, such as achondroplasia, Down syndrome, Prader-Willi syndrome, Pierre Robin sequence, and mucopolysaccharidosis. Craniofacial and upper airway involvement is frequently morbid conditions. In children with genetic diseases, the clinical symptoms of OSAS are often slight or absent, and related morbidities are usually more severe and can be observed at any age. The present review is aimed to updating the discoveries regarding OSAS on Achondroplasia, Down syndrome, Prader-Willi syndrome, Pierre Robin sequence, Sickle cell disease, or encountered in our clinical practice (Ehlers-Danlos syndrome, Ellis-van Creveld syndrome, Noonan syndrome). Two additional groups of genetic disorders will be focused (mucopolysaccharidoses and osteogenesis imperfecta). The flowing items are covered for each disease: (I) what is the pathophysiology of OSAS? (II) What is the incidence/prevalence of OSAS? (III) What result from the management and prognosis? (IV) What are the recommendations? Considering the worries of OSAS, such as inattention and behavioural problems, daytime sleepiness, failure to thrive, cardiological and metabolic complications, the benefit of a widespread screening and the treatment in children with genetic diseases is undoubtful. The goals of the further efforts can be the inclusion of various genetic diseases into guidelines for the screening of OSAS, updating the shreds of evidence based on the research progression.

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“…8 ± 22.3 to 15.4 ± 18.9, which is a 50% reduction. [ 10 ] In addition, TLA improved the lowest oxygen saturation from 75.8 ± 6.8% to 84.4 ±7.3%. In this case it was not until the patient underwent MDO and adenotonsillectomy that the OSA resolved while the patient's TLA was still in place, suggesting early workup for adenotonsillar hypertrophy should be considered in cases with severe OSA.…”

Section: Discussionmentioning

confidence: 98%

“…Glossopexy, or the tongue–lip adhesion (TLA), is associated with decreased morbidity and normalized weight gain, including an improvement in lowest oxygen saturation reading by 8.6% in one meta-analysis. [ 10 , 11 ] It is estimated that airway issues that accompany PRS are expected to improve or resolve as the mandible continues to grow by the age of one, at which time palate repair is typically planned. [ 1 , 15 ] However, this option has decreased in popularity due to its temporary effectiveness and the frequent need for nutritional support for >1 month and additional surgical procedures.…”

Section: Introductionmentioning

confidence: 99%

Resolution of obstructive sleep apnea after mandibular distraction osteogenesis in setting of delayed tongue–lip adhesion takedown

Randall¹,

Kian

Chin

et al. 2018

Medicine

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Rationale:There is a high prevalence of obstructive sleep apnea (OSA) in patients with Pierre Robin sequence (PRS), and treatment approaches are highly variable. One approach is a temporary tongue-lip adhesion (TLA) that acts as a temporizing measure while the mandible continues to grow and is usually taken down at 1 year of age.Patient concerns:Side effects of prolonged tongue-lip adhesion and optimal workup and treatment of persistent OSA in the setting of a tongue-lip adhesion.Diagnoses:Pierre Robin sequence (PRS), persistent obstructive sleep apnea (OSA), and tongue-lip adhesion (TLA).Interventions:Mandibular distraction osteogenesis (MDO), adenotonsillectomy, and tongue-lip adhesion takedown.Outcomes:Resolution of OSA.Lessons:This case puts into question the efficacy of isolated TLA in infants with Pierre Robin sequence and OSA, and places emphasis on the importance of considering an earlier workup of other potential causes of obstruction and the potential need for MDO as a primary or adjunctive approach to treatment.

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Tongue–lip adhesion and tongue repositioning for obstructive sleep apnoea in Pierre Robin sequence: A systematic review and meta-analysis

Abstract: Tongue-lip adhesion and tongue repositioning can improve apnoea/hypopnoea index and oxygenation parameters in children with Pierre Robin sequence and obstructive sleep apnoea.

Cited by 24 publications

References 15 publications

Defining the anatomy of the neonatal lingual frenulum

Defining the anatomy of the neonatal lingual frenulum

Sleep-disordered breathing in paediatric setting: existing and upcoming of the genetic disorders

Resolution of obstructive sleep apnea after mandibular distraction osteogenesis in setting of delayed tongue–lip adhesion takedown

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