Abstract
Purpose
Sacrococcygeal teratoma (SCT) is a rare congenital tumor originating from a variant of extragonadal germ cell neoplasm. Herein, we present three cases of neonatal SCTs undergoing surgical resection and reconstruction. We also review the literature to discuss the importance of proper perinatal management and timely surgical intervention depending on the tumor type and maturity to prevent malignant transformation and recurrence and ensure functional outcomes.
Methods and results
Three cases of SCT were retrospectively analyzed. All infants underwent complete surgical resection of the SCT and coccygectomy during their neonatal/infancy period, followed by pelvic floor and buttock reconstruction while minimizing buttock contour deformity and undesirable skin scar. Two of the cases were histopathologically diagnosed as predominantly cystic mature teratomas of Altman types I and IV, and the third was a mature teratoma of Altman type II. There were no complications or tumor recurrence during the average follow-up period of 5.3 years.
Conclusions
This case-based review highlights the role of multidisciplinary team approaches, including prenatal monitoring, oncologic resection, and adequate reconstruction according to the type of tumor and anomaly. Optimal perinatal evaluation allows promising oncologic and functional outcomes in terms of timely intervention to eliminate tumor recurrence and malignant transformation. Complete oncologic surgical resection of SCTs should also include proper functional preservation strategies, such as the reconstruction of the pelvic floor, cosmetic buttock contouring, and preservation of bladder and bowel sphincter function.
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References
Yoon HM, Byeon SJ, Hwang JY, Kim JR, Jung AY, Lee JS, Yoon HK, Cho YA (2018) Sacrococcygeal teratomas in newborns: a comprehensive review for the radiologists. Acta Radiol 59:236–246
Phi JH (2021) Sacrococcygeal teratoma : a tumor at the center of embryogenesis. J Korean Neurosurg Soc 64:406–413
Savitri QM, Prihartono S, Harahap A (2019) Giant sacrococcygeal teratoma in newborn: a rare case. J Pediatr Surg Case Rep 47:101223
Altman RP, Randolph JG, Lilly JR (1974) Sacrococcygeal teratoma: American Academy of Pediatrics Surgical Section Survey-1973. J Pediatr Surg 9:389–398
Hu Q, Yan Y, Liao H, Liu H, Yu H, Zhao F (2020) Sacrococcygeal teratoma in one twin: a case report and literature review. BMC Pregnancy Childbirth 20:751
Gabra HO, Jesudason EC, McDowell HP, Pizer BL, Losty PD (2006) Sacrococcygeal teratoma—a 25-year experience in a UK regional center. J Pediatr Surg 41:1513–1516
Fishman SJ, Jennings RW, Johnson SM, Kim HB (2004) Contouring buttock reconstruction after sacrococcygeal teratoma resection. J Pediatr Surg 39:439–441
O’Shea KM, Sanders E, Farrelly PJ, Wilkinson DJ, Minshull K, Craigie RJ (2022) Sacrococcygeal teratomas: midline reconstruction improves cosmesis without compromising outcomes. Pediatr Surg Int 38:617–621
Rattan KN, Singh J (2021) Neonatal sacrococcygeal teratoma: our 20-year experience from a tertiary care center in North India. Trop Doct 51:209–212
Holcomb GW, Murphy JP, St Peter SD (2019) Holcomb and Ashcraft’s pediatric surgery E-Book. Elsevier Health Sciences
Und Aspang JS, Burnand KM, Ong H, Cross K, Thompson D, Giuliani S (2020) Sacrococcygeal teratoma with intraspinal extension: a case series and review of literature. J Pediatr Surg 55:2022–2025
Yu JA, Sohaey R, Kennedy AM, Selden NR (2007) Terminal myelocystocele and sacrococcygeal teratoma: a comparison of fetal ultrasound presentation and perinatal risk. AJNR Am J Neuroradiol 28:1058–1060
Ferraro S, Panzeri A, Braga F, Panteghini M (2019) Serum α-fetoprotein in pediatric oncology: not a children’s tale. Clin Chem Lab Med (CCLM) 57:783–797
van Heurn LJ, Knipscheer MM, Derikx JPM, van Heurn LWE (2020) Diagnostic accuracy of serum alpha-fetoprotein levels in diagnosing recurrent sacrococcygeal teratoma: a systematic review. J Pediatr Surg 55:1732–1739
De Backer A, Madern GC, Hakvoort-Cammel FG, Haentjens P, Oosterhuis JW, Hazebroek FW (2006) Study of the factors associated with recurrence in children with sacrococcygeal teratoma. J Pediatr Surg 41:173–181
Derikx JP, De Backer A, van de Schoot L, Aronson DC, de Langen ZJ, van den Hoonaard TL, Bax NM, van der Staak F, van Heurn LW (2006) Factors associated with recurrence and metastasis in sacrococcygeal teratoma. Br J Surg 93:1543–1548
Wang Y, Wu Y, Wang L, Yuan X, Jiang M, Li Y (2017) Analysis of recurrent sacrococcygeal teratoma in children: clinical features, relapse risks, and anorectal functional sequelae. Med Sci Monit 23:17–23
Yoshida M, Matsuoka K, Nakazawa A, Yoshida M, Inoue T, Kishimoto H, Nakayama M, Takaba E, Hamazaki M, Yokoyama S, Horie H, Tanaka M, Gomi K, Ohama Y, Kigasawa H, Kitano Y, Uchida H, Kanamori Y, Iwanaka T, Tanaka Y (2013) Sacrococcygeal yolk sac tumor developing after teratoma: a clinicopathological study of pediatric sacrococcygeal germ cell tumors and a proposal of the pathogenesis of sacrococcygeal yolk sac tumors. J Pediatr Surg 48:776–781
Wilson RD, Hedrick H, Flake AW, Johnson MP, Bebbington MW, Mann S, Rychik J, Liechty K, Adzick NS (2009) Sacrococcygeal teratomas: prenatal surveillance, growth and pregnancy outcome. Fetal Diagn Ther 25:15–20
Barakat MI, Abdelaal SM, Saleh AM (2011) Sacrococcygeal teratoma in infants and children. Acta Neurochir (Wien) 153:1781–1786
Guo W, Ji T, Tang X, Yang Y (2009) Outcome of conservative surgery for giant cell tumor of the sacrum. Spine (Phila Pa 1976) 34:1025–1031
Emoto S, Kaneko M, Murono K, Sasaki K, Otani K, Nishikawa T, Tanaka T, Hata K, Kawai K, Imai H, Saito N, Kobayashi H, Tanaka S, Ikemura M, Ushiku T, Nozawa H (2018) Surgical management for a huge presacral teratoma and a meningocele in an adult with Currarino triad: a case report. Surg Case Rep 4(1):9
Yoneda A, Usui N, Taguchi T, Kitano Y, Sago H, Kanamori Y, Nakamura T, Nosaka S, Oba MS (2013) Impact of the histological type on the prognosis of patients with prenatally diagnosed sacrococcygeal teratomas: the results of a nationwide Japanese survey. Pediatr Surg Int 29:1119–1125
Hambraeus M, Al-Mashhadi A, Wester T, Svensson PJ, Stenström P, Lilja HE (2019) Functional outcome and health-related quality of life in patients with sacrococcygeal teratoma — a Swedish multicenter study. J Pediatr Surg 54:1638–1643
Heerema-McKenney A, Harrison MR, Bratton B, Farrell J, Zaloudek C (2005) Congenital teratoma: a clinicopathologic study of 22 fetal and neonatal tumors. Am J Surg Pathol 29:29–38
Funding
This research was supported by the Basic Science Research Program of the National Research Foundation of Korea (NRF) funded by the Ministry of Education, Korea (grant no. NRF‐2020R1I1A3073930).
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M-JS and TGH conceptualized the study design. MMM and TGH reviewed the medical records for three patients and the existing literature. MMM and HBS prepared the first draft of the manuscript. All authors reviewed the draft and approved the final manuscript for publication.
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This study was reviewed and approved by the Institutional Review Board of Inje University Ilsan Paik Hospital (No. 2022–06-012), and informed consent was obtained from all participants.
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Parental consent was obtained for all patients included in the study regarding the publication of personal medical data and pictures.
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Mosleh, M.M., Heo, T.G., Shin, H.B. et al. Sacrococcygeal teratomas: a case-based review from the perspective of individual multidisciplinary experts. Childs Nerv Syst 39, 57–64 (2023). https://doi.org/10.1007/s00381-022-05768-1
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DOI: https://doi.org/10.1007/s00381-022-05768-1