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先天性悬垂腭并腭裂：病例报告。

Congenital epignathus associated with a cleft palate: a case report.

机构信息

Surgery Department, Heal Africa Hospital, Goma, Democratic Republic of Congo.

ENT Department, GOMA Provincial Referral Hospital, Goma, Democratic Republic of Congo.

出版信息

J Med Case Rep. 2021 Aug 3;15(1):422. doi: 10.1186/s13256-021-03007-w.

DOI:10.1186/s13256-021-03007-w
PMID:34340710
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8330006/
Abstract

BACKGROUND

Epignathus is a rare, benign, congenital teratoma of the hard palate with an estimated incidence of 1 in 200,000 live births. Epignathus frequently leads to a high mortality (80-100%) due to airway obstruction in the neonatal period.

CASE PRESENTATION

We report a case of successful management of a rare oropharyngeal teratoma in a African newborn girl who was referred to our institute with a large protruding intraoral mass, combined with cleft palate, causing some respiratory compromise. The palatal mass was removed on the fifth day after birth, and a palatoplasty performed on day 30.

CONCLUSION

Epignathus is a life-threatening condition at the time of delivery. Appropriate management begins with securing the airway, followed by complete resection of the tumor.

摘要

背景

颅荐部畸胎瘤是一种罕见的、良性的硬腭先天性畸胎瘤，估计每 20 万例活产中就有 1 例。颅荐部畸胎瘤常导致新生儿期气道阻塞，死亡率高（80-100%）。

病例介绍

我们报告了一例罕见的非洲新生儿女孩口咽畸胎瘤的成功治疗。该女孩因口腔内有一巨大的突出肿块，伴有腭裂，导致呼吸有些受限而被转诊至我们医院。腭部肿块在出生后第 5 天切除，并在第 30 天行腭裂修复术。

结论

颅荐部畸胎瘤在分娩时是一种危及生命的情况。适当的治疗始于确保气道通畅，然后是肿瘤的完全切除。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/5bda4875c686/13256_2021_3007_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/0cfcd783abf2/13256_2021_3007_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/15f06b5a3ea7/13256_2021_3007_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/f79d91de9c8f/13256_2021_3007_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/54dc4c973430/13256_2021_3007_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/5bda4875c686/13256_2021_3007_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/0cfcd783abf2/13256_2021_3007_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/15f06b5a3ea7/13256_2021_3007_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/f79d91de9c8f/13256_2021_3007_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/54dc4c973430/13256_2021_3007_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fef3/8330006/5bda4875c686/13256_2021_3007_Fig5_HTML.jpg

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本文引用的文献

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An autopsy case of epignathus (immature teratoma of the soft palate) with intracranial extension but without brain invasion: case report and literature review.1例伴有颅内扩展但无脑侵犯的上颌咽囊肿（软腭未成熟畸胎瘤）尸检病例：病例报告及文献复习
Diagn Pathol. 2018 Dec 22;13(1):99. doi: 10.1186/s13000-018-0776-y.
2
Intraoral teratoma in a newborn presenting as severe respiratory distress.一名表现为严重呼吸窘迫的新生儿口腔内畸胎瘤。
BMJ Case Rep. 2013 Mar 25;2013:bcr2013008735. doi: 10.1136/bcr-2013-008735.
3
Giant epignathus teratoma involving the palate, tongue, and floor of the mouth.
巨大的上颌部畸胎瘤累及腭部、舌部及口腔底部。
J Craniofac Surg. 2013 Jan;24(1):e97-9. doi: 10.1097/SCS.0b013e3182798f25.
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Oral teratomas: a report of 5 cases.口腔畸胎瘤：5例报告。
J Oral Maxillofac Surg. 2012 Dec;70(12):2809-13. doi: 10.1016/j.joms.2012.01.028. Epub 2012 Aug 9.
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Epignathus with fetiform features.具有胎儿样特征的上颌寄生胎。
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A large teratoma of the hard palate: a case report.硬腭巨大畸胎瘤：一例报告
Br J Oral Maxillofac Surg. 2009 Jan;47(1):46-9. doi: 10.1016/j.bjoms.2007.12.015. Epub 2008 Jul 3.
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A newborn baby with a tumor protruding from the mouth. Diagnosis: congenital gingival granular cell tumor.一名新生儿口腔内有肿瘤突出。诊断：先天性牙龈颗粒细胞瘤。
Acta Dermatovenerol Alp Pannonica Adriat. 2007 Sep;16(3):128-30.
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[Diagnosis and management of cervicofacial congenital teratomas: about 4 cases, literature review and restatement].
Ann Chir Plast Esthet. 2007 Apr;52(2):114-23. doi: 10.1016/j.anplas.2006.08.007. Epub 2006 Oct 6.
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