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

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 under...

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Autores principales: Uchi, Ryosuke, Koto, Mayu, Nakao, Atsushi, Hosokawa, Miku, Ukawa, Toshiko, Tsurisawa, Chisa, Hisaeda, Yoshiya, Amagata, Shusuke
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Thieme Medical Publishers, Inc. 2023
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10198758/
https://www.ncbi.nlm.nih.gov/pubmed/37215368
http://dx.doi.org/10.1055/a-2070-8590
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author Uchi, Ryosuke
Koto, Mayu
Nakao, Atsushi
Hosokawa, Miku
Ukawa, Toshiko
Tsurisawa, Chisa
Hisaeda, Yoshiya
Amagata, Shusuke
author_facet Uchi, Ryosuke
Koto, Mayu
Nakao, Atsushi
Hosokawa, Miku
Ukawa, Toshiko
Tsurisawa, Chisa
Hisaeda, Yoshiya
Amagata, Shusuke
author_sort Uchi, Ryosuke
collection PubMed
description 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.
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spelling pubmed-101987582023-05-20 Congenital Maxillomandibular Synechia with Multiple Malformations in a Very-Low-Birth-Weight Infant: A Case Report Uchi, Ryosuke Koto, Mayu Nakao, Atsushi Hosokawa, Miku Ukawa, Toshiko Tsurisawa, Chisa Hisaeda, Yoshiya Amagata, Shusuke AJP Rep 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. Thieme Medical Publishers, Inc. 2023-05-19 /pmc/articles/PMC10198758/ /pubmed/37215368 http://dx.doi.org/10.1055/a-2070-8590 Text en The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonDerivative-NonCommercial License, permitting copying and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes, or adapted, remixed, transformed or built upon. ( https://creativecommons.org/licenses/by-nc-nd/4.0/ ) https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives License, which permits unrestricted reproduction and distribution, for non-commercial purposes only; and use and reproduction, but not distribution, of adapted material for non-commercial purposes only, provided the original work is properly cited.
spellingShingle Uchi, Ryosuke
Koto, Mayu
Nakao, Atsushi
Hosokawa, Miku
Ukawa, Toshiko
Tsurisawa, Chisa
Hisaeda, Yoshiya
Amagata, Shusuke
Congenital Maxillomandibular Synechia with Multiple Malformations in a Very-Low-Birth-Weight Infant: A Case Report
title Congenital Maxillomandibular Synechia with Multiple Malformations in a Very-Low-Birth-Weight Infant: A Case Report
title_full Congenital Maxillomandibular Synechia with Multiple Malformations in a Very-Low-Birth-Weight Infant: A Case Report
title_fullStr Congenital Maxillomandibular Synechia with Multiple Malformations in a Very-Low-Birth-Weight Infant: A Case Report
title_full_unstemmed Congenital Maxillomandibular Synechia with Multiple Malformations in a Very-Low-Birth-Weight Infant: A Case Report
title_short Congenital Maxillomandibular Synechia with Multiple Malformations in a Very-Low-Birth-Weight Infant: A Case Report
title_sort congenital maxillomandibular synechia with multiple malformations in a very-low-birth-weight infant: a case report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10198758/
https://www.ncbi.nlm.nih.gov/pubmed/37215368
http://dx.doi.org/10.1055/a-2070-8590
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