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Published on: September 19, 2015
Treatment of infants with Syndromic Robin sequence with modified palatal plates: a minimally invasive treatment
Silvia Müller-Hagedorn1,2,3, Wolfgang Buchenau1,2, Jörg Arand2
1Interdisciplinary Centre for Craniofacial Malformations, Tuebingen University Hospital, Calwerstrasse 7, 72076, Tuebingen, Germany.
Insights
A modified oral appliance effectively treated upper airway obstruction and feeding issues in infants with syndromic Robin sequence (RS). This safe treatment improved breathing and oral feeding, potentially avoiding invasive interventions for these challenging cases.
Area of Science:
- Craniofacial anomalies
- Pediatric surgery
- Sleep medicine
Background:
- Infants with Robin sequence (RS) often experience severe upper airway obstruction (UAO) and feeding difficulties.
- These challenges are particularly pronounced in syndromic RS, necessitating effective management strategies.
- Previous research demonstrated the efficacy of an oral appliance with a velar extension in isolated RS.
Purpose of the Study:
- To evaluate the effectiveness and safety of a modified oral appliance with a velar extension for treating UAO and feeding problems in infants with syndromic RS.
- To assess the impact of the treatment on obstructive sleep apnea severity and feeding modalities.
- To determine if this conservative approach can prevent the need for more invasive interventions.
Main Methods:
- A retrospective review of children with syndromic RS admitted between 2003 and 2009.
- Quantification of UAO using cardiorespiratory sleep studies (measuring mixed obstructive sleep apnea index - MOAI) before and during treatment.
- Evaluation of feeding methods (gavage vs. oral) and weight gain (standard deviation score - SDS) pre- and post-treatment.
Main Results:
- Treatment with the modified palatal plate (TPP) significantly reduced median MOAI from 8.5 to 1.1 (p < 0.001).
- The number of infants exclusively gavage fed decreased from 23 to 7, while exclusively orally fed infants increased from 18 to 30.
- Median SDS for weight showed a slight improvement, and only three children ultimately required tracheostomy, with no treatment-related deaths.
Conclusions:
- The modified palatal plate with a velar extension is an effective and safe treatment for UAO and feeding problems in syndromic RS.
- This approach led to significant improvements in airway patency and oral feeding capabilities.
- Further prospective studies are warranted to confirm these findings and support the avoidance of invasive procedures.
Background:
Infants with Robin sequence (RS) suffer from upper airway obstruction (UAO) and feeding problems. We developed an oral appliance with a velar extension in combination with functional treatment and appropriate feeding techniques, which was proven effective in isolated RS. As the above problems are particularly challenging in syndromic RS, we set out to evaluate our treatment concept also in these patients.
Methods:
We searched our electronic departmental database to identify all children admitted to our department between 01/01/2003 and 31/12/2009 because of syndromic RS. UAO was quantified by cardiorespiratory sleep studies performed before and during treatment with a modified palatal plate. This appliance consists of a palatal part, covering the hard palate as well as the alveolar ridges and the potential cleft, and a velar extension shifting the tongue in a more anterior position, thereby opening the pharyngeal airway. It is adjusted by fiberoptic nasopharyngoscopy and controlled by cardiorespiratory sleep studies. Obstructive sleep apnea was defined as a mixed obstructive sleep apnea index (MOAI) >3/h. Feeding modalities before and after treatment and weight gain, determined as standard deviation score, were also evaluated.
Results:
Of 68 children meeting inclusion criteria, 56 completed treatment (46 of these being infants). Underlying diagnoses included craniofacial dysostosis (N = 13) and synostosis syndromes (N = 5), unspecified dysmorphic syndromes (N = 23) and miscellaneous rare conditions (N = 27). Median MOAI decreased from 8.5 (range 0.3-76.0) at admission to 1.1 (0.0-5.2) at discharge (p < 0.001). 51 children received only a TPP and 5 additionally continuous positive airway pressure (CPAP) or high-flow nasal cannula during sleep for mild residual OSA. Three children ultimately required tracheostomy. The number of exclusively gavage fed infants was reduced from 23 to 7. Conversely, the number of children fed exclusively by mouth increased from 18 to 30. Median SDS for weight decreased from -1.6 (-3.5-1.7) to -1.3 (-4.1-2.5). Twelve children had their treatment prematurely discontinued, e.g. due to laryngeal collapse/laryngomalacia. No patient died during treatment.
Conclusion:
Treatment of UAO and feeding problems in these children with syndromic RS by a modified palatal plate with a velar extension was shown to be effective and safe. If confirmed in prospective studies, it may help to avoid more invasive interventions.
