Universität zu Köln

Poets, Christian F., Maas, Christoph, Buchenau, Wolfgang, Arand, Joerg, Vierzig, Anne, Braumann, Bert and Mueller-Hagedorn, Silvia (2017). Multicenter study on the effectiveness of the pre-epiglottic baton plate for airway obstruction and feeding problems in Robin sequence. Orphanet J. Rare Dis., 12. LONDON: BIOMED CENTRAL LTD. ISSN 1750-1172

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Abstract

Background: Treatment of Robin sequence is often either invasive or of unproven effectiveness. The pre-epiglottic baton plate (PEBP) is a well-studied alternative, yet is not widely applied internationally. We report on a prospective 3-center cohort study investigating this treatment. Based on an agreed protocol, parents of infants with Robin sequence referred to participating centers were offered enrollment, which involved taking a maxillary cast followed by endoscopy to fit the plate and sleep studies to monitor its effectiveness. Recordings were centrally analyzed by sleep specialists blinded to timing and center. Primary outcome was the mixed-obstructive apnea index, defined as the number of such apneas/h of sleep; secondary outcomes included the desaturation index to < 80% pulse oximeter saturation and weight gain. Results: Of 75 infants referred, 49 could be included; 1 center failed to perform appropriate sleep studies. Within a mean hospitalization of 3 weeks, the mixed-obstructive apnea index decreased (median; interquartile range) from 15.9 (6.3-31.5) to 2.3 (1.2-5.4); it decreased further to 0.7 (0.1-2.4) in the 32 infants who had a successful 3-month follow-up sleep study performed. The desaturation index normalized (from 0.38 (0-2.7) to 0.0 (0-0.1)). Mean standard deviation score for weight remained unchanged between admission and follow-up, while the proportion of tube-fed infants decreased from 74 to 14%. Conclusions: This prospective multi-center cohort study confirms retrospective audits on the effectiveness of PEBP treatment in improving upper airway obstruction and feeding problems, the main clinical problems of infants with Robin sequence. International collaboration is required to compare this with other treatment approaches.

Item Type:	Journal Article
Creators:	Creators Email ORCID ORCID Put Code Poets, Christian F. UNSPECIFIED UNSPECIFIED UNSPECIFIED Maas, Christoph UNSPECIFIED UNSPECIFIED UNSPECIFIED Buchenau, Wolfgang UNSPECIFIED UNSPECIFIED UNSPECIFIED Arand, Joerg UNSPECIFIED UNSPECIFIED UNSPECIFIED Vierzig, Anne UNSPECIFIED UNSPECIFIED UNSPECIFIED Braumann, Bert UNSPECIFIED UNSPECIFIED UNSPECIFIED Mueller-Hagedorn, Silvia UNSPECIFIED UNSPECIFIED UNSPECIFIED
URN:	urn:nbn:de:hbz:38-236719
DOI:	10.1186/s13023-017-0602-8
Journal or Publication Title:	Orphanet J. Rare Dis.
Volume:	12
Date:	2017
Publisher:	BIOMED CENTRAL LTD
Place of Publication:	LONDON
ISSN:	1750-1172
Language:	English
Faculty:	Unspecified
Divisions:	Unspecified
Subjects:	no entry
Uncontrolled Keywords:	Keywords Language INFANTS; SLEEP Multiple languages Genetics & Heredity; Medicine, Research & Experimental Multiple languages
Refereed:	Yes
URI:	http://kups.ub.uni-koeln.de/id/eprint/23671