Epignathus with oropharynx destruction
DOI:
https://doi.org/10.4322/acr.2021.293Keywords:
Fetus, Pathology, Oral TeratomaAbstract
Epignathus, is a rare oropharyngeal teratoma arising from the head and neck region. Sporadic cases have been described with associated intracerebral teratoma. Even more infrequent and extraordinary is the circumstance of a teratoma with oropharynx destruction. We describe the case of a fetus with pharyngeal mass that completely destroyed the oral cavity. The histological examination revealed an immature teratoma (G3); only one other G3 case has been described.
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Levine AB, Alvarez M, Wedgwood J, Berkowitz RL, Holzman I. Contemporary management of a potentially lethal fetal anomaly: a successful perinatal approach to epignathus, fibstet. Obstet Gynecol. 1990;76(5 Pt 2):962-6. PMid:2216267.
Holt GR, Holt JE, Weaver RG. Dermoids and teratomas of the head and neck. Ear Nose Throat J. 1979;58(12):520-31. PMid:540545.
Ang AT, Ho NK, Ong CL. Giant epignathus with intracranial teratoma in a newborn infant. Australas Radiol. 1990;34(4):358-60. http://dx.doi.org/10.1111/j.1440-1673.1990.tb02676.x. PMid:2092670.
Duckett S, Claireaux AE. Cerebral teratoma associated with epignathus in a newborn infant. J Neurosurg. 1963;20(10):888-91. http://dx.doi.org/10.3171/jns.1963.20.10.0888. PMid:14186084.
Ehrich WE. Teratoid parasites of the mouth (episphenoids, epipalati, epignathi). Am J Orthodont. Oral Surg. 1945;31:650-9.
Becker S, Schon R, Gutwald R, et al. A congenital teratoma with a cleft palate: report of a case. Br J Oral Maxillofac Surg. 2007;45(4):326-7. http://dx.doi.org/10.1016/j.bjoms.2005.11.007. PMid:16377040.
Barksdale EM Jr, Obokhare I. Teratomas in infants and children. Curr Opin Pediatr. 2009;21(3):344-9. http://dx.doi.org/10.1097/MOP.0b013e32832b41ee. PMid:19417664.
Bianchi B, Ferri A, Silini EM, Magnani C, Sesenna E. Congenital cervical teratoma: a case report. J Oral Maxillofac Surg. 2010;68(3):667-70. http://dx.doi.org/10.1016/j.joms.2009.04.054. PMid:19926379.
He J, Wang Y, Zhu H, Qiu W, He Y. Nasopharyngeal teratoma associated with cleft palate in newborn: report of 2 cases. Oral Maxillofac Surg. 2010;109(2):211-6. PMid:19969490.
Calda P, Novotná M, Cutka D, et al. A case of an epignathus with intracranial extension appearing as a persistently open mouth at 16 weeks and subsequently diagnosed at 20 weeks of gestation. J Clin Ultrasound. 2011;39(3):164-8. http://dx.doi.org/10.1002/jcu.20762. PMid:21387329.
Ince EZ, Cekmez F, Yıldırım Ş, et al. Malignant epignathus including a nephroblastoma component and successful management. Ann Diagn Pathol. 2013;17(3):288-90. http://dx.doi.org/10.1016/j.anndiagpath.2012.01.008. PMid:22405521.
Kirishima M, Yamada S, Shinya M, et al. 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(1):99. http://dx.doi.org/10.1186/s13000-018-0776-y. PMid:30579363.
Wang AC, Gu YQ, Zhou XY. Congenital giant epignathus with intracranial extension in a fetal. Chin Med J. 2017;130(19):2386-7. PMid:28937049.
Too S, Ahmad Sarji S, Yik Y, Ramanujam T. Malignant epignathus teratoma. Biomed Imaging Interv J. 2008;4(2):e18. http://dx.doi.org/10.2349/biij.4.2.e18. PMid:21614323.
Smith NM, Chambers SE, Billson VR, Laing I, West CP, Bell JE. Oral teratoma (epignathus) with intracranial extension: a report of two cases. Prenat Diagn. 1993;13(10):945-52. http://dx.doi.org/10.1002/pd.1970131008. PMid:8309901.
Huang Y, Pan H. A fetus with a mass in the oral cavity: a rare case of oral immature teratoma. Int J Clin Exp Pathol. 2017;10(7):7890-2. PMid:31966637.
Faghfouri F, Bucourt M, Garel C, et al. Prenatal assessment of a fast-growing giant epignathus. Fetal Pediatr Pathol. 2014;33(1):55-9. http://dx.doi.org/10.3109/15513815.2013.850134. PMid:24164281.
Nagy GR, Neducsin BP, Lázár L, Stenczer B, Csapó Z, Rigó J Jr. Early prenatal detection of a fast-growing fetal epignathus. J Obstet Gynaecol Res. 2012;38(11):1328-30. http://dx.doi.org/10.1111/j.1447-0756.2012.01865.x. PMid:22564030.
Kumar SY, Shrikrishna U, Shetty J, Sitaram A. Epignathus with fetiform features. J Lab Physicians. 2011;3(1):56-8. http://dx.doi.org/10.4103/0974-2727.78571. PMid:21701667.
Shertukde SP, Thelmo ML. Epignathus (a clinicopathologic case report). Fetal Pediatr Pathol. 2009;28(3):132-8. http://dx.doi.org/10.1080/15513810902772532. PMid:19365741.
Witters I, Moerman P, Louwagie D, Van Assche FA, Migeon BR, Fryns JP. Second trimester prenatal diagnosis of epignathus teratoma in ring X chromosome mosaicism with inactive ring X chromosome. Ann Genet. 2001;44(4):179-82. http://dx.doi.org/10.1016/S0003-3995(01)01090-5. PMid:11755101.
Alagappan A, Shattuck KE, Rowe T, Hawkins H. Massive intracranial immature teratoma with extracranial extension into oral cavity, nose, and neck. Fetal Diagn Ther. 1998;13(5):321-4. http://dx.doi.org/10.1159/000020862. PMid:9813428.
Izadi K, Smith M, Askari M, Hackam D, Hameed AA, Bradley JP. A patient with an epignathus: management of a large oropharyngeal teratoma in a newborn. J Craniofac Surg. 2003;14(4):468-72. http://dx.doi.org/10.1097/00001665-200307000-00012. PMid:12867858.
Prevedello DM, Kassam AB, Carrau RL, et al. Transpalatal endoscopic endonasal resection of a giant epignathus skull base teratoma in a newborn. Case report. J Neurosurg. 2007;107(3, Suppl):266-71. PMid:17918540.
Sumiyoshi S, Machida J, Yamamoto T, et al. Massive immature teratoma in a neonate. Int J Oral Maxillofac Surg. 2010;39(10):1020-3. http://dx.doi.org/10.1016/j.ijom.2010.04.008. PMid:20483564.
Rayudu HH, Lakshmi K, Reddy K, Varma S. Malignant epignathus. J Indian Assoc Pediatr Surg. 2011;16(3):104-5. http://dx.doi.org/10.4103/0971-9261.83490. PMid:21897571.
Chung JH, Farinelli CK, Porto M, Major CA. Fetal epignathus: the case of an early EXIT (ex utero intrapartum treatment). Obstet Gynecol. 2012;119(2 Pt 2):466-70. http://dx.doi.org/10.1097/AOG.0b013e318242b3f1. PMid:22270441.
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