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Congenital Maxillomandibular Synechia with Multiple Malformations in a Very-Low-Birth-Weight Infant: A Case Report

Authors :
Ryosuke Uchi
Mayu Koto
Atsushi Nakao
Miku Hosokawa
Toshiko Ukawa
Chisa Tsurisawa
Yoshiya Hisaeda
Shusuke Amagata
Source :
American Journal of Perinatology Reports, Vol 13, Iss 02, Pp e35-e39 (2023)
Publication Year :
2023
Publisher :
Thieme Medical Publishers, Inc., 2023.

Abstract

Congenital maxillomandibular synechia is a rare malformation that is characterized by a fusion of the maxilla and mandible. The fusion is fibrous or bony and prevents mouth opening, which causes difficulties in feeding and occasionally in breathing. Although extremely rare, neonatologists must understand the disease because it can be fatal and require emergency treatment after birth. We report the case of a very-low-birth-weight (VLBW) infant with congenital maxillomandibular synechia and other malformations, including cleft palate, syndactyly, and cryptorchidism. The patient presented with extremely limited mouth opening, and endotracheal intubation seemed impossible; fortunately, the patient did not have respiratory distress syndrome. The patient underwent surgical release of the fibrous bands on days 10 and 17, and good mouth opening was achieved. The patient was able to consume breast milk orally and was discharged home at a corrected gestational age of 1 month without recurrence of difficulty in mouth opening or any sequelae. This is the first reported case of a VLBW infant with congenital maxillomandibular synechia who required more complicated management of feeding, surgical intervention, and anesthesia.

Details

Language :
English
ISSN :
21576998 and 21577005
Volume :
13
Issue :
02
Database :
Directory of Open Access Journals
Journal :
American Journal of Perinatology Reports
Publication Type :
Academic Journal
Accession number :
edsdoj.8aa6b6a0c52a47bd8b1d510791efedba
Document Type :
article
Full Text :
https://doi.org/10.1055/a-2070-8590