Modern Pediatrics. Ukraine. (2024). 4(140): 73-82. doi: 10.15574/SP.2024.140.73
Konoplitskyi V. S.¹, Honcharuk V. B.², Shyshkovsky O. M.², Korobko Yu. Ye.¹, Sapriga I. V.¹, Pasichnyk O. V.^1
1National Pirogov Memorial Medical University, Vinnytsia, Ukraine
²СEI «Vinnytsia Regional Children’s Clinical Hospital of Vinnytsia Regional Council», Ukraine

For citation: Konoplitskyi VS, Honcharuk VB, Shyshkovsky OM, Korobko YuYe, Sapriga IV, Pasichnyk OV. (2024). Presacral cysts in children. Modern Pediatrics. Ukraine. 4(140): 73–82. doi: 10.15574/SP.2024.140.73.
Article received: Mar 09, 2024. Accepted for publication: May 14, 2024.

Presacral epidermal cyst is a rare congenital formation in pediatric practice of ectodermal origin, from ectodermal remnants of tissues that are displaced during embryogenesis due to the development of surrounding structures. In general, presacral dermoid cysts make up less than 5% of all tumor-like formations of the presacral space. Presacral formations can have a nervous, vascular/lymphatic, mesenchymal or mixed (like medulloepithelioma) origin, they can be primary (in focal diseases) or systemic (in multifocal diseases). Presacral cysts are often encountered as incidental findings, due to their unusual localization and slow growth, which ensures their long asymptomatic course. The painful symptom is usually associated with secondary infection and malignant transformation.
The aim is to highlight the experience of diagnosis and treatment of presacral cysts based on literature sources and own experience.
Presacral cysts in children are a rare pathology that requires significant diagnostic efforts in terms of visualization and differential diagnosis. Treatment of presacral cysts in childhood should be only operative, consisting in extirpation of the formation with mandatory morphological verification of the final diagnosis. The choice of operative approach for the extirpation of presacral cysts should take into account the data of visualization methods of the localization of formations in the presacral space, taking into account the height of their proximal pole to prevent possible perioperative complications. As far as possible, extirpation of cysts should take place as sparingly as possible, in order to prevent damage to their walls and the possibility of probable insemination by totipotent cells of the presacral space and prevention of recurrences of the pathology.
The research was carried out in accordance with the principles of the Declaration of Helsinki. Informed consent of the child's parents was obtained for the research.
The authors declare no conflict of interest.
Keywords: intestinal obstruction, children, surgical access, perioperative complications, presacral cyst, perforation of the rectum, tumor.
REFERENCES

1. Abe K, Nakamura S, Ikeda R, Wada M. (2008). Gastrointestinal infections. children [Review]. Current World Literature. Current Opinion in Gastroenterology. 26(1): 69-84. https://doi.org/10.1097/MOG.0b013e328334fef1; PMid:19996885

2. Aihole JS, Aruna G, Deepak J, Supriya S. (2018). Precoccygeal epidermoid cyst in a child – A unique case report. African Journal of Urology. 24(4): 336-338. https://doi.org/10.1016/j.afju.2018.07.002

3. Alvi MI, Mubarak F, Khandwala K, Barakzai MD, Memon A, Alvi MI, Barakzai MDD. (2018). A rare case of presacral epidermoid cyst in an adult male: emphasis on diffusion weighted magnetic resonance sequences in preoperative imaging. Cureus. 10(1). https://doi.org/10.7759/cureus.2050

4. Ammar S, Cheikhrouhou T, Jallouli M, Chtourou R, Sellami S, Zitouni H, Mhiri R. (2021). Pediatric case of presacral ganglioneuroma: diagnostic considerations and therapeutic strategy. Annals of Pediatric Surgeryю 17(1). https://doi.org/10.1186/s43159-021-00100-z

5. Aranda-Narváez JM, González-Sánchez AJ, Montiel-Casado C, Sánchez-Pérez B, Jiménez-Mazure C et al. (2012). Posterior approach (Kraske procedure) for surgical treatment of presacral tumors. World journal of gastrointestinal surgery. 4(5): 126. https://doi.org/10.4240/wjgs.v4.i5.126; PMid:22655127 PMCid:PMC3364338

6. Coco C, Manno A, Mattana C, Verbo A, Sermoneta D, Franceschini G et al. (2008). Congenital tumors of the retrorectal space in the adult: report of two cases and review of the literature. Tumori Journal. 94(4): 602-607. https://doi.org/10.1177/030089160809400428; PMid:18822703

7. Dahan H, Arrivé L, Wendum D, le Pointe HD, Djouhri H, Tubiana JM. (2001). Retrorectal developmental cysts in adults: clinical and radiologic-histopathologic review, differential diagnosis, and treatment. Radiographics. 21(3): 575-584. https://doi.org/10.1148/radiographics.21.3.g01ma13575; PMid:11353107

8. De Bruijn H, Maeda Y, Murphy J, Warusavitarne J, Vaizey CJ. (2018). Combined laparoscopic and perineal approach to omental interposition repair of complex rectovaginal fistula. Diseases of the Colon & Rectum. 61(1): 140-143. https://doi.org/10.1097/DCR.0000000000000980; PMid:29219924

9. Duclos J, Maggiori L, Zappa M, Ferron M, Panis Y. (2014). Laparoscopic resection of retrorectal tumors: a feasibility study in 12 consecutive patients. Surgical endoscopy. 28: 1223-1229. https://doi.org/10.1007/s00464-013-3312-x; PMid:24263459

10. Dziki Ł, Włodarczyk M, Sobolewska-Włodarczyk A, Saliński A, Salińska M, Tchórzewski M et al. (2019). Presacral tumors: diagnosis and treatment-a challenge for a surgeon. Archives of Medical Science. 15(3): 722-729. https://doi.org/10.5114/aoms.2016.61441; PMid:31110540 PMCid:PMC6524179

11. Ebinesh A. Challenges in Diagnosis and Management of Pediatric Presacral Tumors: A Radiologist's Perspective. Asian and oceanic forum of pediatric radiology. 7(6): 31-36.

12. Gu L, Berkowitz CL, Stratigis JD, Collins LK, Mostyka M, Spigland NA. (2021). Presacral epidermoid cyst in a pediatric patient. Journal of Pediatric Surgery Case Reportsю 71: 101904. https://doi.org/10.1016/j.epsc.2021.101904

13. Hassan I, Wietfeldt ED. (2009). Presacral tumors: diagnosis and management. Clinics in colon and rectal surgery. 22(02): 084-093. https://doi.org/10.1055/s-0029-1223839; PMid:20436832 PMCid:PMC2780241

14. Hokenstad ED, Hammoudeh ZS, Tran NV, Chua HK, Occhino JA. (2016). Rectovaginal fistula repair using a gracilis muscle flap. International urogynecology journal. 27: 965-967. https://doi.org/10.1007/s00192-015-2942-z; PMid:26811111

15. Huddart S, Mann J, Robinson K, Raafat F, Imeson J, Gornall P et al. (2003). Sacrococcygeal teratomas: the UK children's cancer study group's experience. I. Neonatal. Pediatric surgery international. 19: 47-51. https://doi.org/10.1007/s00383-002-0884-2; PMid:12721723

16. Ikegami M, Takahashi T, Shiojima S, Yoshizawa Y, Kimata M, Konno H et al. (2023). A novel surgical intervention using transvaginal endoscopic ultrasonography for the children with OHVIRA syndrome. Journal of Pediatric Surgery Case Reports. 92: 102622. https://doi.org/10.1016/j.epsc.2023.102622

17. Jadeja D, Shah B, Shah J, Kotak S. (2021, Dec 1). Pelvic retroperitoneal masses in children: what does the radiologist need to know? GAIMS Journal of Medical Sciences. 1 (1): 18-27.

18. Jao SW, Beart Jr RW, Spencer RJ, Reiman HM, Ilstrup DM. (1985). Retrorectal tumors: mayo clinic experience, 1960-1979. Diseases of the colon & rectum. 28(9): 644-652. https://doi.org/10.1007/BF02553440; PMid:2996861

19. Jones M, Khosa J. (2013). Presacral tumours: a rare case of a dermoid cyst in a paediatric patient. Case Reports. 2013: bcr2013008783. https://doi.org/10.1136/bcr-2013-008783; PMid:23682084 PMCid:PMC3669780

20. Kesici U, Sakman G, Mataraci E. (2013). Retrorectal/Presacral epidermoid cyst: report of a case. The Eurasian journal of medicine. 45(3): 207. https://doi.org/10.5152/eajm.2013.40; PMid:25610280 PMCid:PMC4261424

21. Keslar PJ, Buck JL, Suarez ES. (1994). Germ cell tumors of the sacrococcygeal region: radiologic-pathologic correlation. Radiographics. 14(3): 607-620. https://doi.org/10.1148/radiographics.14.3.8066275; PMid:8066275

22. Kim HC, Lee HL, Lee SH, Kim GY. (2008). Squamous cell carcinoma arising from a presacral epidermoid cyst: CT and MR findings. Abdominal Radiology. 33(4): 498. https://doi.org/10.1007/s00261-007-9287-0; PMid:17680300

23. Kim HJ, Ho IG, Ihn K, Han SJ, Oh JT. (2020). Clinical Characteristics and Treatment of Currarino Syndrome: A Single Institutional Experience. Advances in Pediatric Surgery. 26(2): 46-53. https://doi.org/10.13029/aps.2020.26.2.46

24. Kocaoglu M, Frush DP. (2006). Pediatric presacral masses. Radiographics. 26(3): 833-857. https://doi.org/10.1148/rg.263055102; PMid:16702458

25. Kouyate M, Diakite A, Magassa M, Traore IL, Dicko B, Toure SM et al. (2022). Sacrococcygeal Teratoma in A Newborn in Kay ES (Mali) about 3 Cases. SAS J Surg. 2: 60-62.

26. Kuroyanagi H, Oya M, Ueno M, Fujimoto Y, Yamaguchi T, Muto T. (2008). Standardized technique of laparoscopic intracorporeal rectal transection and anastomosis for low anterior resection. Surgical endoscopy. 22: 557-561. https://doi.org/10.1007/s00464-007-9626-9; PMid:18193475

27. Kwon YS, Lee N, Lee HS, Youn EJ, Lee SK, Kim Y, Lee JJ. (2020). Risk of rectal puncture due to needle entry into the presacral space: importance of measuring the distance between the rectum and sacrococcyx, and the thickness of the sacrococcyx. Medicine. 99(28): e20935. https://doi.org/10.1097/MD.0000000000020935; PMid:32664091 PMCid:PMC7360314

28. Li Z, Lu M. (2021). Presacral tumor: insights from a decade's experience of this rare and diverse disease. Frontiers in Oncology. 11: 639028. https://doi.org/10.3389/fonc.2021.639028; PMid:33796466 PMCid:PMC8008122

29. Mendonça GS, Artiles CB, Malheiros GC, Amorim VB. (2021). Presacral medulloepithelioma with peritoneal carcinomatosis in an 11-year-old boy: An extremely rare association. https://doi.org/10.1016/j.radcr.2020.12.060; PMid:33747332 PMCid:PMC7960498

30. Messick CA, Londono JMR, Hull T. (2013). Presacral tumors: how do they compare in pediatric and adult patients? Polish Journal of Surgery. 85(5): 253-261. https://doi.org/10.2478/pjs-2013-0039; PMid:23770525

31. Nedelcu M, Andreica A, Skalli M, Pirlet I, Guillon F, Nocca D, Fabre JM. (2013). Laparoscopic approach for retrorectal tumors. Surgical endoscopy. 27: 4177-4183. https://doi.org/10.1007/s00464-013-3017-1; PMid:23728916

32. NM BS, JM SG, MI ER. (2020, Jan). Chronic constipation due to Currarino syndrome. Anales de Pediatria, 93; 5: 349-351. https://doi.org/10.1016/j.anpedi.2019.11.009; PMid:31928948

33. Pappalardo G, Frattaroli FM, Casciani E, Moles N, Mascagni D, Spoletini D et al. (2009). Retrorectal tumors: the choice of surgical approach based on a new classification. The American Surgeon. 75(3): 240-248. https://doi.org/10.1177/000313480907500311; PMid:19350861

34. Pena A, Hong A. (2003). The posterior sagittal trans-sphincteric and trans-rectal approaches. Techniques in Coloproctology 7: 35-44. https://doi.org/10.1007/s101510300006; PMid:12750953

35. Pescatori M, Brusciano L, Binda GA, Serventi A. (2005). A novel approach for perirectal tumours: the perianal intersphincteric excision. International Journal of Colorectal Disease. 20: 72-75. https://doi.org/10.1007/s00384-004-0623-3; PMid:15338167

36. Poudel D, Shrestha BM, Kandel BP, Shrestha S, Kansal A, Joshi Lakhey P. (2021). Presacral dermoid cyst in a young female patient: A case report. Clinical Case Reports. 9(11): e05062. https://doi.org/10.1002/ccr3.5062; PMid:34795897 PMCid:PMC8582024

37. Prakash A, Ashta A, Garg A, Verma A, Padaliya P. (2023). Pediatric presacral tumors with intraspinal extension: a rare entity with diagnostic challenges. Acta Radiologica. 64(12). https://doi.org/10.1177/02841851231202688; PMid:37753549

38. Reiter MJ, Schwope RB, Bui-Mansfield LT, Lisanti CJ, Glasgow SC. (2015). Surgical management of retrorectal lesions: what the radiologist needs to know. American Journal of Roentgenology. 204(2): 386-395. https://doi.org/10.2214/AJR.14.12791; PMid:25615762

39. Riojas CM, Hahn CD, Johnson EK. (2010). Presacral epidermoid cyst in a male: a case report and literature review. Journal of Surgical Education. 67(4): 227-232. https://doi.org/10.1016/j.jsurg.2010.06.005; PMid:20816358

40. Singh GP. (2017). Neuroembryology. In Essentials of neuroanesthesia: 41-50. Academic Press. https://doi.org/10.1016/B978-0-12-805299-0.00002-6

41. Vanni AJ, Buckley JC, Zinman LN. (2010). Management of surgical and radiation induced rectourethral fistulas with an interposition muscle flap and selective buccal mucosal onlay graft. The Journal of urology. 184(6): 2400-2404. https://doi.org/10.1016/j.juro.2010.08.004; PMid:20952036

42. Wang GC, Liu LB, Han GS, Ren YK. (2012). Application of an arc-shaped transperineal incision in front of the apex of coccyx during the resection of pelvic retroperitoneal tumors. Zhonghua Zhong liu za zhi. [Chinese Journal of Oncology]. 34(1): 65-67.

43. Wang G, Miao C. (2023). Chinese expert consensus on standardized treatment for presacral cysts. Gastroenterology Report. 11: goac079. https://doi.org/10.1093/gastro/goac079; PMid:37655176 PMCid:PMC10468046

44. Wang G, Han G, Ren Y, Cheng Y. (2013). Clinical effects of pedicled omentum covering the intestinal anastomotic stoma in preventing anastomotic fistula. Chinese Journal of Digestive Surgery, (12): 508-511.

45. Wells RG, Sty JR. (1990). Imaging of sacrococcygeal germ cell tumors. Radiographics. 10(4): 701-713. https://doi.org/10.1148/radiographics.10.4.2165626; PMid:2165626

46. Wolpert A, Beer-Gabel M, Lifschitz O, Zbar AP. (2002). The management of presacral masses in the adult. Techniques in coloproctology. 6: 43-49. https://doi.org/10.1007/s101510200008; PMid:12077641

47. Yang BL, Gu YF, Shao WJ, Chen HJ, Sun GD, Jin HY, Zhu X. (2010). Retrorectal tumors in adults: magnetic resonance imaging findings. World Journal of Gastroenterology: WJG. 16(46): 5822. https://doi.org/10.3748/wjg.v16.i46.5822; PMid:21155003 PMCid:PMC3001973

48. Yoon HM, Byeon SJ, Hwang JY, Kim JR, Jung AY, Lee JS et al. (2018). Sacrococcygeal teratomas in newborns: a comprehensive review for the radiologists. Acta Radiologica. 59(2): 236-246. https://doi.org/10.1177/0284185117710680; PMid:28530139

49. Zang YW, Xiang JB. (2019). Pathologic and functional anatomy basis of intersphincteric resection. Zhonghua wei Chang wai ke za zhi = Chinese Journal of Gastrointestinal Surgery. 22(10): 937-942.

50. Zhou JL, Wu B, Xiao Y, Lin GL, Wang WZ, Zhang GN, Qiu HZ. (2014). A laparoscopic approach to benign retrorectal tumors. Techniques in coloproctology. 18: 825-833. https://doi.org/10.1007/s10151-014-1146-8; PMid:24718777

51. Zhu XL, Su WW, Tang JL, Yao LD, Lu LJ, Sun XJ. (2019). Primary alveolar rhabdomyosarcoma of retrorectal-presacral space in an adult patient: A case report of an uncommon tumor with rare presentation. Medicine. 98(10): e13416. https://doi.org/10.1097/MD.0000000000013416; PMid:30855432 PMCid:PMC6417633