CC BY-NC 4.0 · Arch Plast Surg 2021; 48(05): 518-523
DOI: 10.5999/aps.2021.00318
Pediatric/Craniomaxillofacial/Head&Neck
Case Report

Surgical management of palatal teratoma (epignathus) with the use of virtual reconstruction and 3D models: a case report and literature review

Plastic, Aesthetic and Reconstructive Surgery Service, University Hospital “Dr. José Eleuterio González”, Faculty of Medicine, Autonomous University of Nuevo Leon, Monterrey, Mexico
,
Faculty of Medicine, Autonomous University of Nuevo Leon, Monterrey, Mexico
,
Faculty of Medicine, Autonomous University of Nuevo Leon, Monterrey, Mexico
,
Faculty of Medicine, Autonomous University of Nuevo Leon, Monterrey, Mexico
,
Pediatric Surgery Service, University Hospital “Dr. José Eleuterio González”, Faculty of Medicine, Autonomous University of Nuevo Leon, Monterrey, Mexico
,
Laboratory of Tissue Engineering, Faculty of Medicine, University of Colima, Colima, Mexico
,
Plastic, Aesthetic and Reconstructive Surgery Service, University Hospital “Dr. José Eleuterio González”, Faculty of Medicine, Autonomous University of Nuevo Leon, Monterrey, Mexico
› Author Affiliations
We thank Sergio Lozano MD (Scientific Publication Support Coordinator, University Hospital “Dr. José Eleuterio González”, Autonomous University of Nuevo Leon, Monterrey, Nuevo Leon, Mexico) for his review of the manuscript, and the patient, the patient´s mother, and the hospital staff for their help. Also, we thank Hernan Chacon-Moreno MD (Professor of Plastic, Aesthetic and Reconstructive Surgery Service, University Hospital “Dr. José Eleuterio González”, and Faculty of Medicine, Autonomous University of Nuevo Leon, Monterrey, Nuevo Leon, Mexico) for all the facilities provided to carry out this work.

Epignathus is a rare congenital orofacial teratoma that arises from the sphenoid region of the palate or the pharynx. It occurs in approximately 1:35,000 to 1:200,000 live births representing 2% to 9% of all teratomas. We present the case of a newborn of 39.4 weeks of gestation with a tumor that occupied the entire oral cavity. The patient was delivered by cesarean section. Oral resection was managed by pediatric surgery. Plastic surgery used virtual 3-dimensional models to establish the extension, and depth of the tumor. Bloc resection and reconstruction of the epignathus were performed. The mass was diagnosed as a mature teratoma associated with cleft lip and palate, nasoethmoidal meningocele that conditions hypertelorism, and a pseudomacrostoma. Tridimensional technology was applied to plan the surgical intervention. It contributed to a better understanding of the relationships between the tumor and the adjacent structures. This optimized the surgical approach and outcome.



Publication History

Received: 10 February 2021

Accepted: 27 May 2021

Article published online:
19 March 2022

© 2021. The Korean Society of Plastic and Reconstructive Surgeons. This is an open access article published by Thieme under the terms of the Creative Commons Attribution-NonCommercial License, permitting unrestricted noncommercial use, distribution, and reproduction so long as the original work is given appropriate credit. Contents may not be used for commercial purposes. (https://creativecommons.org/licenses/by-nc/4.0/)

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  • REFERENCES

  • 1 Jadhav SS, Korday CS, Malik S. Epignathus leading to fatal airway obstruction in a neonate. J Clin Diagn Res 2017; 11: SD04-5
  • 2 Herath M, Siriwardena BSMS, Jayasinghe RD. Oropharyngeal epignathus with partial facial duplication: report of a rare case. Int J Oral Maxillofac Surg 2018; 47: 442-4
  • 3 Tunes RS, Cavalcanti GZ, Squarisi JMO. Oral epignathus with maxilla duplication: report of a rare case. Craniomaxillofac Trauma Reconstr 2019; 12: 62-6
  • 4 Kirishima M, Yamada S, Shinya M. An autopsy case of epignathus (immature teratoma of the soft palate) with intracranial extension but without brain invasion: case report and literature review. Diagn Pathol 2018; 13: 99
  • 5 ElSherbiny Hamed M, El-Din MHN, Abdelazim IA. Prenatal diagnosis and immediate successful management of isolated fetal epignathus. J Med Ultrasound 2019; 27: 198-201
  • 6 Bauermeister AJ, Zuriarrain A, Newman MI. Three-dimensional printing in plastic and reconstructive surgery: a systematic review. Ann Plast Surg 2016; 77: 569-76
  • 7 Jacobs CA, Lin AY. A new classification of three-dimensional printing technologies: systematic review of three-dimensional printing for patient-specific craniomaxillofacial surgery. Plast Reconstr Surg 2017; 139: 1211-20
  • 8 Halterman SM, Igulada KN, Stelnicki EJ. Epignathus: large obstructive teratoma arising from the palate. Cleft Palate Craniofac J 2006; 43: 244-6
  • 9 Daskalakis G, Efthimiou T, Pilalis A. Prenatal diagnosis and management of fetal pharyngeal teratoma: a case report and review of the literature. J Clin Ultrasound 2007; 35: 159-63
  • 10 Tonni G, Centini G, Inaudi P. Prenatal diagnosis of severe epignathus in a twin: case report and review of the literature. Cleft Palate Craniofac J 2010; 47: 421-5
  • 11 Kaido Y, Kikuchi A, Oyama R. Prenatal ultrasound and magnetic resonance imaging findings of a hypovascular epignathus with a favorable prognosis. J Med Ultrason (2001) 2013; 40: 61-4
  • 12 Kumar KM, Veligandla I, Lakshmi AR. Congenital giant teratoma arising from the hard palate: a rare clinical presentation. J Clin Diagn Res 2016; 10: ED03-4
  • 13 Paula Pinho Matos A, Teixeira Castro P, de Barros Duarte L. Prenatal diagnosis of cervical masses by magnetic resonance imaging and 3D virtual models: perinatal and longterm follow-up outcomes. J Matern Fetal Neonatal Med 2020; 33: 2181-9
  • 14 Kumar SY, Shrikrishna U, Shetty J. Epignathus with fetiform features. J Lab Physicians 2011; 3: 56-8
  • 15 Kadlub N, Touma J, Leboulanger N. Head and neck teratoma: from diagnosis to treatment. J Craniomaxillofac Surg 2014; 42: 1598-603
  • 16 Hodges MM, Crombleholme TM, Marwan AI. Massive facial teratoma managed with the ex utero intrapartum treatment (EXIT) procedure and use of a 3-dimensional printed model for planning of staged debulking. J Pediatr Surg Case Rep 2017; 17: 15-9
  • 17 Bence CM, Wagner AJ. Ex utero intrapartum treatment (EXIT) procedures. Semin Pediatr Surg 2019; 28: 150820
  • 18 Hsieh TY, Dedhia R, Cervenka B. 3D printing: current use in facial plastic and reconstructive surgery. Curr Opin Otolaryngol Head Neck Surg 2017; 25: 291-9
  • 19 Aldaadaa A, Owji N, Knowles J. Three-dimensional printing in maxillofacial surgery: hype versus reality. J Tissue Eng 2018; 9: 2041731418770909
  • 20 Chae MP, Rozen WM, McMenamin PG. Emerging applications of bedside 3D printing in plastic surgery. Front Surg 2015; 2: 25