Immature nasopharyngeal teratoma with prenatal diagnosis: Case report and review of the literature

Authors

DOI:

https://doi.org/10.18597/rcog.3906

Keywords:

Mouth neoplasms, teratoma, prenatal diagnosis, fetus, prognosis

Abstract

Objectives: To report the case of a pregnant woman with prenatal diagnosis of fetal immature nasopharyngeal teratoma, and to conduct a review of the literature describing the prognosis of this condition.

Materials and methods: We report the case of a 27-year-old pregnant woman who received care at the Obstetrics and Gynecology Unit of a reference hospital in Bogotá (Colombia) because of a finding during a prenatal visit of evidence of polyhydramnios, secondary to a nasopharyngeal teratoma. A literature search was conducted in the Medline vía PubMed, Scopus, SciELO and ScienceDirect databases, restricted by language (English and Spanish) and date of publication (January 2001 to January 2021). Case reports and case series covering the prognosis of this condition were included.

Results: Overall, 168 titles were retrieved, 55 of which met the inclusion criteria. Perinatal outcomes for a total of 58 fetuses with a diagnosis of immature nasopharyngeal teratoma detected during the prenatal stage were reported. In the identified cases, perinatal mortality was 25.4 % and the percentage of fetal demise was close to 3.6 %.

Conclusions: Immature nasopharyngeal teratoma is an infrequent condition. The available literature suggests that fetal prognosis depends on the degree of compromise of intracranial structures and the possibility of resecting the lesion. Further studies are needed to assess the prognosis of fetuses with immature nasopharyngeal teratoma.

Author Biographies

Angy Lorena Meneses-Parra, Hospital Militar Central, Bogotá (Colombia).

Médico residente ginecología y obstetricia, Universidad Militar Nueva Granada, Hospital Militar Central, Bogotá (Colombia).

Rafael Eduardo Tarazona-Bueno, Hospital Militar Central, Bogotá (Colombia).

Médico residente ginecología y obstetricia, Universidad Militar Nueva Granada, Hospital Militar Central, Bogotá (Colombia).

Rafael Leonardo Aragón-Mendoza, Hospital Militar Central, Bogotá (Colombia).

Médico ginecoobstetra, especialista en medicina materno fetal, Hospital Militar Central, Bogotá (Colombia).

Marcela Altman-Restrepo, Hospital Militar Central, Bogotá (Colombia).

Médico ginecoobstetra, especialista en medicina materno fetal, Hospital Militar Central, Bogotá (Colombia).

References

Tenorio SJ, García MA, Muñoz BJA, Monroy HVM, Delgado SBE. Teratoma nasofaríngeo con extensión temporal y fosa subtemporal izquierda. Rev Mex Pediatr. 2015;82(5):165-70.

Noguchi T, Jinbu Y, Itoh H, Matsumoto K, Sakai O, Kusama M. Epignathus combined with cleft palate, lobulated tongue, and lingual hamartoma: Report of a case. Oral Surg, Oral Med Oral Pathol Oral Radiol Endodontol. 2006;101(4):481-6. https://doi.org/10.1016/j.tripleo.2005.06.025

Makki FM, Al-Mazrou KA. Nasopharyngeal teratoma associated with cleft palate in a newborn. Eur Arch Otorhinolaryngol. 2008;265(11):1413-5. https://doi.org/10.1007/s00405-008-0611-2

Liang CC, Lai JP, Lui CC. Cleft palate with congenital midline teratoma. Ann Plast Surg. 2003;50(5):550-4. https://doi.org/10.1097/01.SAP.0000037462.83232.1E

Fotopoulou C, Toennies H, Guschmann M, Henrich W. Prenatal sonographic diagnosis of an oropharyngeal teratoma (epignathus) on a stillborn infant: A case report. Z Geburtshilfe Neonatol. 2007;211(4):165-8. https://doi.org/10.1055/s-2007-960682

Benson RE, Fabbroni G, Russell JL. A large teratoma of the hard palate: A case report. Br J Oral Maxillofac Surg. 2009;47(1):46-9. https://doi.org/10.1016/j.bjoms.2007.12.015

Tonni G, De Felice C, Centini G, Ginanneschi C. Cervical and oral teratoma in the fetus: A systematic review of etiology, pathology, diagnosis, treatment and prognosis. Arch Gynecol Obstet. 2010;282(4):355-61. https://doi.org/10.1007/s00404-010-1500-7

Zhu P, Li XY. Management of oropharyngeal teratoma: Two case reports and a literature review. J Int Med Res. 2021;49(2):2-6. https://doi.org/10.1177/0300060521996873

Kaido Y, Kikuchi A, Oyama R, Kanasugi T, Fukushima A, Sugiyama T. Prenatal ultrasound and magnetic resonance imaging findings of a hypovascular epignathus with a favorable prognosis. J Med Ultrason. 2013;40(1):61-4. https://doi.org/10.1007/s10396-012-0381-8

Gonzalez CM, Moreno PJ, Salazar MG, García PPF, Montes-Tapia FF, Cervantes VH, et al. Surgical management of palatal teratoma (Epignathus) with the use of virtual reconstruction and 3D models: A case report and literature review. Arch Plast Surg. 2021;48(5):518-23. https://doi.org/10.5999/aps.2021.00318

Halterman SM, Igulada KN, Stelnicki EJ. Epignathus: Large obstructive teratoma arising from the palate. Cleft Palate-Craniofacial J. 2006;43(2):244-6. https://doi.org/10.1597/04-166.1

Tonni G, Centini G, Inaudi P, Rosignoli L, Ginanneschi C, De Felice C. Prenatal diagnosis of severe epignathus in a twin: Case report and review of the literature. Cleft Palate-Craniofacial J. 2010;47(4):421-5. https://doi.org/10.1597/08-224.1

Araujo E, Guimaraes HA, Saito M, Pires AB, Pontes AL, Nardozza LM, Moron AF. Prenatal diagnosis of a large fetal cervical teratoma by three-dimensional ultrasonography: A case report. Arch Gynecol Obstet. 2007; 275:141-4 https://doi.org/10.1007/s00404-006-0180-9

Lionel J, Miloslav V, Khaled AA-A. Giant epignathus. A case report. Kwait Med J. 2004;36(3):217-20.

Pinho A, Teixeira P, de Barros L, Dutra A, Daltro P, Fazecas T, et al. Prenatal diagnosis of cervical masses by magnetic resonance imaging and 3D virtual models: Perinatal and long-term follow-up outcomes. J Matern Neonatal Med. 2020;33(13):2181-9. https://doi.org/10.1080/14767058.2018.1543393

Manchali M, Sharabu C, Latha M, Kumar L. A rare case of oropharyngeal teratoma diagnosed antenatally with MRI. J Clin Imaging Sci. 2014;4(1):1-5. https://doi.org/10.4103/2156-7514.129261

Wannemuehler TJ, Deig CR, Brown BP, Morgenstein SA. Obstructing in utero oropharyngeal mass: Case report of a lymphatic malformation arising within an oropharyngeal teratoma. Ear, Nose Throat J. 2017;96(1):37-9. https://doi.org/10.1177/014556131709600106

Pellegrini V, Colasurdo F, Guerriero M. Epignathus with oropharynx destruction. Autops Case Reports. 2021;11:1-8. https://doi.org/10.4322/acr.2021.293

Takagi MM, Bussamra LCS, Araujo E, Drummond CL, Herbst SRS, Nardozza LMM, et al. Prenatal diagnosis of a large epignathus teratoma using two-dimensional and three-dimensional ultrasound: Correlation with pathological findings. Cleft Palate-Craniofacial J. 2014;51(3):350-3. https://doi.org/10.1597/12-222

Nagy GR, Neducsin BP, Lázár L, Stenczer B, Csapó Z, Rigó J. Early prenatal detection of a fast-growing fetal epignathus. J Obstet Gynaecol Res. 2012;38(11):1328-30. https://doi.org/10.1111/j.1447-0756.2012.01865.x

Kontopoulos EV, Gualtieri M, Quintero RA. Successful in utero treatment of an oral teratoma via operative fetoscopy: Case report and review of the literature. Am J Obstet Gynecol. 2012;207(1):1-4. https://doi.org/10.1016/j.ajog.2012.04.008

Faghfouri F, Bucourt M, Garel C, Benchimol M, Amarenco B, Soupre V, et al. Prenatal assessment of a fast-growing giant epignathus. Fetal Pediatr Pathol. 2014;33(1):55-9. https://doi.org/10.3109/15513815.2013.850134

Wang AC, Gu YQ, Zhou XY. Congenital giant epignathus with intracranial extension in a fetal. Chin Med J (Engl). 2017;130(19):2386-7. https://doi.org/10.4103/0366-6999.215343

Calda P, Novotná M, Čutka D, Břešt’ák M, Hašlík L, Goldová B, 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. https://doi.org/10.1002/jcu.20762

Naleini F, Farshchian N, Mehrbakhsh M, Kamangar PB. A case report of a massive epignathus. J Med Life. 2020;13(3):435-8. https://doi.org/10.25122/jml-2019-0164

Kirishima M, Yamada S, Shinya M, Onishi S, Goto Y, Kitazono I, 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):1-8. https://doi.org/10.1186/s13000-018-0776-y

Tunes RS, Cavalcanti GZ, Squarisi JMO, Patrocinio LG. Oral epignathus with maxilla duplication: Report of a rare case. Craniomaxillofac Trauma Reconstr. 2019;12(1):62-6. https://doi.org/10.1055/s-0038-1649497

Arora V, Bijarnia Mahay S, Rao S, Dimri N, Manocha A, Mansukhani C, et al. The fatal fetal tumor: A geneticist’s perspective. J Matern Neonatal Med. 2021;34(6):1006-8. https://doi.org/10.1080/14767058.2019.1622671

Aydemir F, Mutaf M, Eryılmaz MA. Giant epignathus (teratoma of palatine tonsil): A case report. Turkish Arch Otorhinolaryngol. 2021;59(2):158-61. https://doi.org/10.4274/tao.2021.2021-4-7

Too SC, Sarji SA, Yik YI, Ramanujam TM. Malignant epignathus teratoma. Biomed Imaging Interv J [Internet]. 2008;4(2). Disponible en: https://pubmed.ncbi.nlm.nih.gov/21614323/

Kumar K, Setty J, Sitaram A. Epignathus with Fetiform Features. J Lab Physicians. 2011;3(01):056-8. https://doi.org/10.4103/0974-2727.78571

Morlino S, Castori M, Servadei F, Laino L, Silvestri E, Polimeni A, et al. Oropharyngeal teratoma, oral duplication, cervical diplomyelia and anencephaly in a 22-week fetus: A review of the craniofacial teratoma syndrome. Birth Defects Res Part A - Clin Mol Teratol. 2015;103(6):554-66. https://doi.org/10.1002/bdra.23327

Miesnik SR, Jones T, Spinner SS. Cesarean to immediate neonatal intervention: A multidisciplinary approach to the perinatal care of a pregnancy complicated by a fetal airway obstruction. JOGNN - J Obstet Gynecol Neonatal Nurs. 2014;43:S99.

Dakpé S, Demeer B, Cordonnier C, Devauchelle B. Emergency management of a congenital teratoma of the oral cavity at birth and three-year follow-up. Int J Oral Maxillofac Surg. 2014;43(4):433-6. https://doi.org/10.1016/j.ijom.2013.09.004

Sumiyoshi S, MacHida J, Yamamoto T, Fukano H, Shimozato K, Fujimoto Y, et al. Massive immature teratoma in a neonate. Int J Oral Maxillofac Surg. 2010;39(10):1020-3. https://doi.org/10.1016/j.ijom.2010.04.008

Dar P, Rosenthal J, Factor S, Dubiosso R, Murthy AS. First-trimester diagnosis of fetal epignathus with 2- and 3-dimensional sonography. J Ultrasound Med. 2009;28(12):1743-6. https://doi.org/10.7863/jum.2009.28.12.1743

Calvo MA, Kline BM, Jones BB, Care MM, Koch BL. Brain malformations associated with epignathus: A clue for the correct prenatal diagnosis. Pediatr Radiol. 2009;39(12):1369-72. https://doi.org/10.1007/s00247-009-1399-y

Staboulidou I, Miller K, Göhring G, Hillemanns P, Wüstemann M. Prenatal diagnosis of an epignathus associated with a 49,XXXXY karyotype - A case report. Fetal Diagn Ther. 2008;24(3):313-7. https://doi.org/10.1159/000160219

Saha SP, Hobson E, Joss S. Nasopharyngeal teratoma associated with a complex congenital cardiac anomaly. Clin Dysmorphol. 2007;16(2):113-4. https://doi.org/10.1097/MCD.0b013e328054c547

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(3):159-63. https://doi.org/10.1002/jcu.20300

Sherer DM, Zigalo A, Abulafia O. Prenatal 3-dimensional sonographic diagnosis of a massive fetal epignathus occluding the oral orifice and both nostrils. J Ultrasound Med. 2006;25:1503-5. https://doi.org/10.7863/jum.2006.25.11.1503

Tamura T, Yamataka A, Okazaki T, Hosoda Y, Lane GJ, Miyano T. Management of a prenatally diagnosed huge teratoma arising from the soft palate. Asian J Surg. 2006;29(3):212-5. http://doi.org/10.1016/S1015-9584(09)60090-7

Ruano R, Benachi A, Aubry MC, Parat S, Dommergues M, Manach Y. The impact of 3-dimensional ultrasonography on perinatal management of a large epignathus teratoma without ex utero intrapartum treatment. J Pediatr Surg. 2005;40(11):31-4. https://doi.org/10.1016/j.jpedsurg.2005.07.059

Takeuchi K, Masuda Y, Narita F, Kiyoshi K, Mizutori M, Maruo T. Prenatal evaluation of bidirectional epignathus: Comparison of ultrasonography and magnetic resonance imaging. Fetal Diagn Ther. 2003;18(1):26-8. https://doi.org/10.1159/000066379

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. 2003:468-72. https://doi.org/10.1097/00001665-200307000-00012

Hamed ME, El-Din M, Abdelazim I, Shikanova S, Karimova B, Kanshaiym S. Prenatal diagnosis and immediate successful management of isolated fetal epignathus. J Med Ultrasound. 2019;27(4):198-201. https://doi.org/10.4103/JMU.JMU_125_18

Santana EFM, Helfer TM, Passos JP, Junior EA. Prenatal diagnosis of a giant epignathus teratoma in the third trimester of pregnancy using three-dimensional ultrasound and magnetic resonance imaging: Case report. Med Ultrason. 2014;16(2):168-71. https://doi.org/10.11152/mu.201.3.2066.162.efms1

Dray G, Olivier C, Teissier N, Vuillard E, Michel J, Farnoux C, et al. Epignathus teratoma: Diagnostic and neonatal management. A case report. J Gynécologie Obs Biol Reprod. 2013;42:596-601. https://doi.org/10.1016/j.jgyn.2012.12.004

Vranic S, Caughron SK, Djuricic S, Bilalovic N, Zaman S, Suljevic I, et al. Hamartomas, teratomas and teratocarcinosarcomas of the head and neck: Report of 3 new cases with clinico-pathologic correlation, cytogenetic analysis, and review of the literature. BMC Ear, Nose Throat Disord. 2008;8(1):1-10. https://doi.org/10.1186/1472-6815-8-8

Barthod G, Teissier N, Bellarbi N, Viala P, Oury JF, Dray G, et al. Fetal airway management on placental support: Limitations and ethical considerations in seven cases. J Obstet Gynaecol (Lahore). 2013;33(8):787-94. https://doi.org/10.3109/01443615.2013.823924

Laje P, Howell LJ, Johnson MP, Hedrick HL, Flake AW, Adzick NS. Perinatal management of congenital oropharyngeal tumors: The ex utero intrapartum treatment (EXIT) approach. J Pediatr Surg. 2013;48(10):2005-10. https://doi.org/10.1016/j.jpedsurg.2013.02.031

Tsitouridis I, Sidiropoulos D, Michaelides M. Sonographic evaluation of epignathus. Hippokratia [Internet]. 2009;13(1):55-7. Disponible en: https://pubmed.ncbi.nlm.nih.gov/19240824/

Clement K, Chamberlain, Boyd P, Molyneux A. Prenatal diagnosis of an epignathus: A case report and review. Ultrasound Obs Gynecol. 2001;18:178-81. https://doi.org/10.1046/j.1469-0705.2001.00456.x

Moon NR, Min JY, Kim YH, Choi SK, Shin JC, Park IY. Prenatal diagnosis of epignathus with multiple malformations in one fetus of a twin pregnancy using three-dimensional ultrasonography and magnetic resonance imaging. Obstet Gynecol Sci. 2015;58(1):65. https://doi.org/10.5468/ogs.2015.58.1.65

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, Part 2):466-70. https://doi.org/10.1097/AOG.0b013e318242b3f1

Antiñolo G, de Agustín JC, Losada A, Ontanilla A, Perla AG. Diagnosis and management of a large oropharyngeal teratoma (epignathus) in a twin pregnancy. Int J Gynecol Obstet. 2009;104(2):143-4. https://doi.org/10.1016/j.ijgo.2008.09.021

Ince EZ, Cekmez F, Yildirim Ş, Demirel A, Bilgic B, Kilicaslan I, et al. Malignant epignathus including a nephroblastoma component and successful management. Ann Diagn Pathol. 2013;17(3):288-90. https://doi.org/10.1016/j.anndiagpath.2012.01.008

Prevedello DM, Kassam AB, Carrau RL, Snyderman CH, Thomas A, Gardner P, 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. https://doi.org/10.3171/PED-07/09/266

Agarwal P, Chandran S, Sriram B, Chang KTE. Ex-utero intrapartum treatment (EXIT) procedure for giant fetal epignathus. Indian Pediatr. 2015;52(10):893-5. https://doi.org/10.1007/s13312-015-0740-9

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. https://doi.org/10.1016/S0003-3995(01)01090-5

Sarioglu N, Wegner RD, Gasiorek-Wiens A, Entezami M, Schmock J, Hagen A, et al. Epignathus: Always a simple teratoma? Report of an exceptional case with two additional fetiforme bodies. Ultrasound Obstet Gynecol. 2003;21(4):397-403. https://doi.org/10.1002/uog.92

Harar RPS, Pratap R, Chadha N, Tolley N. Bilateral tension pneumothorax following rigid bronchoscopy: A report of an epignathus in a newborn delivered by the EXIT procedure with a fatal outcome. J Laryngol Otol. 2005;119(5):400-2. https://doi.org/10.1258/0022215053945813

Rugolotto S, Bertolini A, Cogo I, Pietrobelli A, Pecori S, Furlani M, et al. Epignato, sindrome del cuore sinistro ipoplasico e trisomia 18 in una gemella piccola per età gestazionale. Pediatr Medica Chir. 2013;35:191-3. https://doi.org/10.4081/pmc.2013.41

Chen P, Shin J, Huang J. Two- and three-dimensional ultrasound demonstration of a giant epignathus. Ultrasound Obs Gynecol. 2003;21:407-12. https://doi.org/10.1002/uog.70

Allen LM. Prenatal 3-dimensional imaging techniques in the sonographic evaluation of an oral mass: Comparison with postnatal imaging modalities. J Ultrasound Med. 2011;30(4):561-8. https://doi.org/10.7863/jum.2011.30.4.561

Hodges MM, Crombleholme TM, Marwan AI, Mirsky D, Meyers M, Behrendt N, et al. 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 Reports. 2017;17:15-9. https://doi.org/10.1016/j.epsc.2016.11.013

Nogales FF, Gasca L, Llamas R, Blanco A, Sanz JF. Case report: Epignathus. Clinical, radiologic, and pathologic considerations. Int J Gynecol Obstet. 1977;15(1):41-3. https://doi.org/10.1002/j.1879-3479.1977.tb00641.x

Benouaiche L, Couly G, Michel B, Devauchelle B. Diagnosis and management of cervicofacial congenital teratomas: About 4 cases, literature review and restatement. Ann Chir Plast Esthet. 2007;52(2):114-23. https://doi.org/10.1016/j.anplas.2006.08.007

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Meneses-Parra AL, Tarazona-Bueno RE, Aragón-Mendoza RL, Altman-Restrepo M. Immature nasopharyngeal teratoma with prenatal diagnosis: Case report and review of the literature . Rev. colomb. obstet. ginecol. [Internet]. 2023 Mar. 30 [cited 2024 May 18];74(1):68-86. Available from: https://revista.fecolsog.org/index.php/rcog/article/view/3906

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2023-03-30
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