Epignathus with oropharynx destruction

Pellegrini, Valerio; Colasurdo, Francesco; Guerriero, Massimiliano

doi:10.4322/acr.2021.293

Valerio Pellegrini

Ospedale Antonio Cardarelli, Regional Health Authority of Molise, Medical Department, Campobasso, Italy

http://orcid.org/0000-0002-8438-8318

Francesco Colasurdo

Ospedale Antonio Cardarelli, Regional Health Authority of Molise, Analysis Department of Transmural Services, Campobasso, Italy

http://orcid.org/0000-0001-7318-155X

Massimiliano Guerriero

Ospedale Antonio Cardarelli, Regional Health Authority of Molise, Pathology Laboratory Department of Transmural Services, Campobasso, Italy

http://orcid.org/0000-0002-7871-3655

Authors’ contributions: All authors equally contributed for the conception of the manuscript. All authors collectively proofread and approved the manuscript for the publication.

Conflict of interest: None.

Correspondence Massimiliano Guerriero Unit of Pathology, Regional Health Autority of Molise (ASReM), Antonio Cardarelli Regional Hospital Contrada Tappino s.n.c. 86100 Campobasso CB Italy Phone: +39-0874-409205 ilguerrierochecorre@gmail.com

author	Mother	Size (mm)	wgt (g)	GA (w)	IC	Grade	Outcome	OD
Our case	30 yo, 1G0P	46 × 36 × 51	38	22/3	Yes	3	Induced abortion	Yes
Kirishima et al. ¹²12 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. http://dx.doi.org/10.1186/s13000-018-077...	32 yo, 3G2P	120 × 60 × 60	270	27	Yes	NR	Stillborn	No
Huang and Pan¹⁶16 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.	28 yo, 1G0P	55 × 41 × 28	NR	18	No	NR	Induced abortion	No
Wang et al.¹³13 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.	31 yo, 1G0P	67 × 65 × 50	NR	17/6	Yes	NR	Induced abortion	No
Faghfouri et al. ¹⁷17 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. http://dx.doi.org/10.3109/15513815.2013....	33 yo, 5G4P	153 × 108	NR	24/5	No	NR	Induced abortion	Yes
Nagy et al.¹⁸18 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. http://dx.doi.org/10.1111/j.1447-0756.20...	22 yo, 2G0P	25 × 22	NR	21	No	NR	Induced abortion	Yes
Kumar et al.¹⁹19 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. http://dx.doi.org/10.4103/0974-2727.7857...	25yo, NR	95 × 75 × 60	NR	28–29	No	1	Succumbed to death immediately post-partum	No
Shertukde and Thelmo²⁰20 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. http://dx.doi.org/10.1080/15513810902772...	26 yo, 3G2P	150×90×70	645	26	No	NR	Asphyxia 45 min post-partum	Yes
Too et al.¹⁴14 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. http://dx.doi.org/10.2349/biij.4.2.e18...	32 yo, 1G0P	80×100	NR	34	Yes	NR	5 weeks of life	Yes
Witters et al.²¹21 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. http://dx.doi.org/10.1016/S0003-3995(01)...	31 yo, 7G0P	50	35	20	No	NR	Induced abortion	No
Alagappanet al.²²22 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. http://dx.doi.org/10.1159/000020862...	NR	140 × 160	810	26	Yes	NR	Perinatal death	No
Smith et al.¹⁵15 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. http://dx.doi.org/10.1002/pd.1970131008...	30 yo, 3G1P	35	NR	29	Yes	NR	Induced abortion	No
	28 yo, 1G0P	20	NR	18	Yes	NR		No
Ducket and Claireaux⁴4 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. http://dx.doi.org/10.3171/jns.1963.20.10...	26 yo, 2G1P	NR	50	31 w	Yes	NR	55 min post-partum	No

author	Mother	Size (mm)	wgt (g)	GA	IC	Grade	Outcome	OD
Izadi et al.²³23 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. http://dx.doi.org/10.1097/00001665-20030...	29 yo, 3G2P	160 × 200 × 60	371	29 w	No	NR	Successful excision	No
Prevedello et al.²⁴24 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.	NR, 3G2P	68 × 65 × 62	NR	33 w	No	NR	Successful excision	No
Sumiyoshi et al.²⁵25 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. http://dx.doi.org/10.1016/j.ijom.2010.04...	23 yo, NR	100 longest axis	246	28/5 w	No	NR	Successful excision	No
Rayudu et al.²⁶26 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. http://dx.doi.org/10.4103/0971-9261.8349...	NR	90 × 100	NR	NR, full term pregnancy, 2 days old neonate	No	NR	Successful excision	No
Chung et al.²⁷27 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. http://dx.doi.org/10.1097/AOG.0b013e3182...	29 yo, 1G0P	150 × 90 × 60	392	27/5 w	No	3	Successful excision	No
Ince et al.¹¹11 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. http://dx.doi.org/10.1016/j.anndiagpath....	24 yo, 1G1P	130 × 110 × 90	545	33/1 w	No	NR + Nephroblastoma component	NR	No

[1] Authors’ contributions: All authors equally contributed for the conception of the manuscript. All authors collectively proofread and approved the manuscript for the publication.

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Epignathus with oropharynx destruction

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