Subscribe to RSS
DOI: 10.1055/s-0039-1698765
Fetus in Fetu: Lessons Learned from a Large Multicenter Cohort Study
Funding This study was financed entirely through intramural funding by the participating departments.Publication History
13 May 2019
02 September 2019
Publication Date:
26 November 2019 (online)
Abstract
Introduction Fetus in fetu (FIF) is an extremely rare condition of abnormal twinning during embryogenesis. Most publications are single case reports. We describe the combined experience of four large tertiary referral centers with FIF which were not previously reported or published, and thereby draw conclusions to establish criteria for the workup, diagnosis, and management including intraoperative risk.
Materials and Methods A survey was forwarded to a national pediatric surgery group which includes members from all pediatric surgery centers in the country enquiring about unpublished cases of FIF encountered over a 20-year interval. The cohort was analyzed for age of presentation, type of presentation, diagnostic workup, surgical management, and outcome.
Results From 1998 to 2018, a total of 10 FIF cases were included in the study. Mean age of presentation was 4 months. Computed tomography and ultrasound were the main preoperative diagnostic modality in our cohort. Resection of the mass was curative in nine cases. Two cases in which the FIF was in direct topographic proximity to the biliary tree suffered severe intraoperative or lethal postoperative complications.
Conclusion Complete excision of FIF is the treatment of choice and generally results in excellent long-term quality of life. Mortality is rare and may be associated with biliary involvement and retroperitoneal right upper quadrant location of the FIF tends to be associated with increased risk in excision, and there is also a possible association with the presence of immature elements in the pathology report.
-
References
- 1 Grant P, Pearn JH. Foetus-in-foetu. Med J Aust 1969; 1 (20) 1016-1019
- 2 Pang KK, Chao NS, Tsang TK. , et al. From observation to aetiology: a case report of a twin fetus-in-fetu and a revisit of the known rarity. Hong Kong Med J 2015; 21 (01) 80-83
- 3 Hoeffel CC, Nguyen KQ, Phan HT. , et al. Fetus in fetu: a case report and literature review. Pediatrics 2000; 105 (06) 1335-1344
- 4 Mohan H, Chhabra S, Handa U. Fetus-in-fetu: a rare entity. Fetal Diagn Ther 2007; 22 (03) 195-197
- 5 Ji Y, Chen S, Zhong L. , et al. Fetus in fetu: two case reports and literature review. BMC Pediatr 2014; 14: 88
- 6 Mohta A, Khurana N. Fetus-in-fetu or well-differentiated teratoma- a continued controversy. Indian J Surg 2011; 73 (05) 372-374
- 7 de Lagausie P, de Napoli Cocci S, Stempfle N. , et al. Highly differentiated teratoma and fetus-in-fetu: a single pathology?. J Pediatr Surg 1997; 32 (01) 115-116
- 8 Gerber RE, Kamaya A, Miller SS. , et al. Fetus in fetu: 11 fetoid forms in a single fetus: review of the literature and imaging. J Ultrasound Med 2008; 27 (09) 1381-1387
- 9 Lewis RH. Foetus in foetu and the retroperitoneal teratoma. Arch Dis Child 1961; 36: 220-226
- 10 Willis RA. The Borderland of Embryology and Pathology, 2nd ed. Washington, DC: Butterworth & Co Publishers Ltd; 1962: 442-462
- 11 Gonzalez-Crussi F. Extragonadal Teratomas (Atlas of Tumor Pathology), 2nd ed. Washington, DC: Armed Forces Institute of Pathology; 1982: 20-24
- 12 Spencer R. Parasitic conjoined twins: external, internal (fetuses in fetu and teratomas), and detached (acardiacs). Clin Anat 2001; 14 (06) 428-444
- 13 Kimmel DL, Moyer EK, Peale AR. , et al. A cerebral tumor containing five human fetuses; a case of fetus in fetu. Anat Rec 1950; 106 (02) 141-165
- 14 Sathe PA, Ghodke RK, Kandalkar BM. Fetus in fetu: an institutional experience. Pediatr Dev Pathol 2014; 17 (04) 243-249
- 15 Hopkins KL, Dickson PK, Ball TI, Ricketts RR, O'Shea PA, Abramowsky CR. Fetus-in-fetu with malignant recurrence. J Pediatr Surg 1997; 32 (10) 1476-1479
- 16 Tsai CH, Lin JS, Tsai FJ. Intraventricular fetus in fetu: report of one case. Zhonghua Min Guo Xiao Er Ke Yi Xue Hui Za Zhi 1993; 34 (02) 143-150
- 17 Sanal M, Küçükçelebi A, Abasiyanik F, Erdoğan S, Kocabaşoğlu U. Fetus in fetu and cystic rectal duplication in a newborn. Eur J Pediatr Surg 1997; 7 (02) 120-121
- 18 Kim YJ, Sohn SH, Lee JY. , et al. Misdiagnosis of fetus-in-fetu as meconium peritonitis. Korean J Pediatr 2011; 54 (03) 133-136
- 19 Higgins KR, Coley BD. Fetus in fetu and fetaform teratoma in 2 neonates: an embryologic spectrum?. J Ultrasound Med 2006; 25 (02) 259-263
- 20 Khalifa NM, Maximous DW, Abd-Elsayed AA. Fetus in fetu: a case report. J Med Case Reports 2008; 2: 2
- 21 Goyal RB, Gupta R, Prabhakar G, Dagla R. Fetus in fetu: report of two cases. APSP J Case Rep 2014; 5 (03) 28
- 22 Joshi M, Parelkar S, Shah H, Agrawal A, Mishra P. Foetus in fetu with common bile duct injury: a case report and review of literature. ANZ J Surg 2009; 79 (09) 651-652
- 23 Keshava Murthy M, , Hanumanthaiah, Adarsh E. , et al. Fetus in fetu: a case report and review of literature. JIMSA 2014; 27 (01) 26