Functional treatment of airway obstruction and feeding problems in infants with Robin sequence

Buchenau, W; Wenzel, Sarah G.; Bacher, M.; Müller-Hagedorn, Silvia; Arand, Joerg; Poets, Christian F.

doi:10.1136/archdischild-2016-311407

Cited by 49 publications

(41 citation statements)

References 29 publications

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“…An orthodontic appliance with a velar extension that moves the tongue anteriorly may relieve upper airway obstruction in infants with isolated Pierre Robin sequence and mixed obstructive apnoea index >3 episodes•h -1 [147]. This effect persists at 3 months of device use and is associated with an increase in growth [148]. Tongue-to-lip adhesion is another method to prevent airway obstruction by the tongue, but it is technically difficult and may be complicated by wound rupture (up to 57% of cases) or aspiration pneumonia [114,146].…”

Section: Literature Reviewmentioning

confidence: 99%

ERS statement on obstructive sleep disordered breathing in 1- to 23-month-old children

et al. 2017

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The present statement was produced by a European Respiratory Society Task Force to summarise the evidence and current practice on the diagnosis and management of obstructive sleep disordered breathing (SDB) in children aged 1-23 months. A systematic literature search was completed and 159 articles were summarised to answer clinically relevant questions. SDB is suspected when symptoms or abnormalities related to upper airway obstruction are identified. Morbidity (pulmonary hypertension, growth delay, behavioural problems) and coexisting conditions (feeding difficulties, recurrent otitis media) may be present. SDB severity is measured objectively, preferably by polysomnography, or alternatively polygraphy or nocturnal oximetry. Children with apparent upper airway obstruction during wakefulness, those with abnormal sleep study in combination with SDB symptoms ( snoring) and/or conditions predisposing to SDB ( mandibular hypoplasia) as well as children with SDB and complex conditions ( Down syndrome, Prader-Willi syndrome) will benefit from treatment. Adenotonsillectomy and continuous positive airway pressure are the most frequently used treatment measures along with interventions targeting specific conditions ( supraglottoplasty for laryngomalacia or nasopharyngeal airway for mandibular hypoplasia). Hence, obstructive SDB in children aged 1-23 months is a multifactorial disorder that requires objective assessment and treatment of all underlying abnormalities that contribute to upper airway obstruction during sleep.

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Section: Literature Reviewmentioning

confidence: 99%

ERS statement on obstructive sleep disordered breathing in 1- to 23-month-old children

et al. 2017

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“…Our treatment concept consists of palatal plates with a tubular structure bridging the narrow airway. This treatment modality has been successfully evaluated in infants with isolated and syndromic Robin sequence [22,25]. While it may be considered a causal treatment in the former, providing a growth incentive to the hypoplastic mandible, it may be considered only a symptomatic treatment in patients with SCS, being similar in effect to a nasopharyngeal airway, CPAP or tracheostomy.…”

Section: Discussionmentioning

confidence: 99%

“…All recordings were manually analyzed for the presence of respiratory events using slightly modified standard criteria [26] as used in our previous work [22][23][24][25]. In brief, total sleep time (TST) was determined from the first 10-min epoch without movement artifact or a distorted pulse waveform to the last such 10-min epoch; recordings comprising less than 3 h of TST were excluded.…”

Section: Sleep Studiesmentioning

confidence: 99%

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Less invasive treatment of sleep-disordered breathing in children with syndromic craniosynostosis

Mueller-Hagedorn

Wiechers

Arand

et al. 2019

International Journal of Oral and Maxillofacial Surgery

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Background: Infants and children with syndromic craniosynostosis (SCS), such as Apert-, Crouzon-or Pfeiffer syndrome, are prone to sleep disordered breathing (SDB) including obstructive sleep apnea and upper airway resistance syndrome (OSAS, UARS), potentially leading to tracheostomy. We modified the Tübingen Palatal Plate (TPP), an oral appliance with a velar extension effectively treating airway obstruction in Robin sequence, by attaching a tube to its velar extension to bridge the narrow pharyngeal airway in SCS patients. Here, we evaluated this treatment concept. Methods: Our hospital's electronic patient files were searched for all children with a diagnosis of SCS admitted between 01/01/2004 and 31/12/2016. Children with isolated craniosynostosis were excluded. OSAS was defined as a mixed-obstructive apnea-hypopnea index (MOAHI) > 1, and UARS as more than 1 episode with nasal flow limitation/h, but absent OSAS. Children with a diagnosis of OSAS received the TPP and fiberoptic nasopharyngoscopy to assess the type of obstruction and to adjust the plate. Growth and weight gain, determined as standard deviation scores, were also evaluated before and during treatment. Results: Of 34 patients included, 24 presented with SDB (19 OSAS, 5 UARS) and 27 had midface hypoplasia. Proportions of SDB were 78% in those with, and 22% in those without midface hypoplasia. In the OSAS group (n = 19), 13 patients were treated with palatal plates, with the remaining receiving continuous positive airway pressure, midface surgery or tracheal intubation. The MOAHI decreased across all children receiving palatal plate treatment from 14.6 (range 0.0-50.7) at admission to 0.9 (range 0.0-3.5) at discharge (p = 0.002). SDS for weight and body length also improved (p < 0.05 for weight and p = 0.05 for body length). Only one child required tracheostomy. Conclusion: Treatment of upper airway obstruction by a modified TPP in these children with SCS was shown to be mostly effective and safe. If confirmed in larger prospective studies, it may help to avoid more invasive interventions.

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“…Mild cases are often treated by prone positioning, but this is fraught with an increased risk of sudden infant death syndrome [6]. In Germany, according to a recent survey [7], 37% of infants with RS are treated with a pre-epiglottic baton plate that is comparable to the Tuebingen Palatal Plate (TPP) and is a less invasive, yet effective and comparatively well studied alternative for patients with isolated [8][9][10][11] and syndromic RS [12]. This treatment consists of the standard or modified TPP in combination with Manual Orofacial Therapy according to Castillo Morales [13] and Brondo [14].…”

Section: Introductionmentioning

confidence: 99%

An innovative method for manufacturing the Tuebingen palatal plate for infants with Robin sequence

et al. 2020

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Background: Robin sequence (RS) is characterized by mandibular micro-and retrognathia, glossoptosis, upper airway obstruction and optionally a cleft palate. With an incidence of 1:8000, it belongs to the so-called rare diseases; 30-50% of patients have RS as part of a syndrome. A comparatively well-studied treatment option is the Tuebingen Palatal Plate (TPP), which has proven effective in both, isolated and syndromic RS, but often requires multiple endoscopies for perfect fit and effectiveness. We report on a new method for fitting the TPP with only one session of nasopharyngeal endoscopy resulting in the plate being finished in one day. Methods and results: First, a prototype is produced, consisting of a traditional acrylic palatal part and a velar extension made of thermoplastic resin, usually measuring 10x40mm. Using polymerization, a scale is added to the posterior part of the extension to help with determining its optimal length during endoscopic evaluation. The extension is pre-bent in the dental laboratory to achieve an approximate shape. During endoscopy, the prototype can be adjusted to the infant's anatomy: first, the angulation is customized by controlled heating, bending and cooling of the thermoplastic spur. Second, the length of the spur is adapted by grinding its tip. Then the prototype is returned to the dental laboratory for completion; the final plate can be delivered to the patient on the same day. It acts by shifting the tongue into a more anterior position, thereby opening the airway and releasing upper airway obstruction, as well as by acting as a functional orthodontic appliance that stimulates mandibular growth through exerting pressure on the base of the tongue. Conclusions:With the thermoplastic spur presented here, a TPP can be produced within one day, requiring only one endoscopy. This approach may facilitate fabricating the TPP.

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Functional treatment of airway obstruction and feeding problems in infants with Robin sequence

Abstract: These longitudinal cohort data suggest that this functional approach may be an option to treat both, upper airway obstruction and feeding problems, in infants with isolated RS.

Cited by 49 publications

References 29 publications

ERS statement on obstructive sleep disordered breathing in 1- to 23-month-old children

ERS statement on obstructive sleep disordered breathing in 1- to 23-month-old children

Less invasive treatment of sleep-disordered breathing in children with syndromic craniosynostosis

An innovative method for manufacturing the Tuebingen palatal plate for infants with Robin sequence

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