Bezoars of gastro-intestinal tract in children
DOI:
https://doi.org/10.15574/PS.2024.4(85).136142Keywords:
children, bezoar, Rapunzel syndrome, diagnostic, treatmentAbstract
Bezoars are indigestible substances that accumulated in the lumen of digestive canal. Among various types of bezoars, trichobezoar is the most frequent form, which revealed in stomach and often associated with psychiatric disorders. Rapunzel syndrome is a rare form of bezoar that included the presence of bezoar in stomach that extending through the pylorus, with the potential to extend up to the cecum. Due to the relatively rarity of this entity, there are no generally established guidelines for its diagnostic and management.
Аim - summarizing the own experience of the treating children with bezoars, including with Rapunzel syndrome.
Clinical cases. Five patients (4 girls and one boy) that were treated at Lviv reginal children’s clinical hospital «OHMATDYT» for 1997-2024 years were included in this study. Pain syndrome and presence of palpable mass at the upper parts of abdomen was noted in all patients, by that the weight loss was revealed only in one patient. Four patients were hospitalized for the emergent condition – signs of intestinal obstruction. For the diagnosis establishment, the endoscopy was applied in two patients, ultrasonography – in two patients, and computed tomography – in one patient. All patients were operated with the applying of laparotomic approach: in three patients were revealed trichobezoar, including two cases of Rapunzel syndrome, in one patient bezoar consisted of a cluster of chewing gum, and in one patient – undigested pieces of cotton tissue, which were not only in the stomach, but also in the small intestine. Endoscopy and computed tomography are the main instrumental methods for the confirmation of bezoar diagnosis. The endoscopic or laparoscopic removing of bezoar can be applied in selected patients, but the main method of treatment is open surgery. The consultation and treatment of psychiatric is required at the follow-up.
Conclusions. Clinical course of bezoars during long time may be asymptomatic, by that the main clinical appearance are the pain or discomfort in epigastric region and the presence of the dense mass at the projection of stomach.
The research was carried out in accordance with the principles of the Declaration of Helsinki. Informed consent of the patients was obtained for the research.
No conflict of interests was declared by the authors.
References
Antunes H, Barroso C, Faria C, Correia-Pinto J. (2018) Images in paediatrics: Rapunzel syndrome: the pathway for a prompt diagnosis. Arch Dis Child. 105(3): 298. https://doi.org/10.1136/archdischild-2018-315910; PMid:30509953
Awawda A, Al Ashhab H, Shammas I, et al. (2022) Endoscopic management of unusual bezoar in a Prader-Willi syndrome patient. Cureus. 14(10): e29900. https://doi.org/10.7759/cureus.29900; PMid:36348828 PMCid:PMC9632933
Belhadj A, Touati MD, Othmane MRB, et al. (2023) Revelation of Rapunzel syndrome: A rare case report of gastric trichobezoar-induced acute purulent peritonitis. Int J Surg Case Rep. 111: 108860. https://doi.org/10.1016/j.ijscr.2023.108860; PMid:37748384 PMCid:PMC10520814
Cintolo J, Telem DA, Divino CM, et al. (2009) Laparoscopic removal of a large gastric trichobezoar in a 4-year-old girl. JSLS 13(4): 608-611. https://doi.org/10.4293/108680809X12589999538110; PMid:20202404 PMCid:PMC3030801
Coufal NG, Kansagra AP, Doucet J, et al. (2011) Gastric trichobezoar causing intermittent small bowel obstruction: report of a case and review of the literature. Case Rep Med. 2011: 217570. https://doi.org/10.1155/2011/217570; PMid:21687591 PMCid:PMC3114431
Dogra S, Kulkarni AK, Rao PP. (2012) Rapunzel syndrome - a case report. Med J Armed Forces India. 68(3): 249-251. https://doi.org/10.1016/j.mjafi.2011.11.005; PMid:24532879 PMCid:PMC3862928
Elghazeery MA, Hassan AM. (2024). Gastrointestinal bezoars in paediatrics: Case series and literature review. Afr J Paediatr Surg. 21(2): 101-106. https://doi.org/10.4103/ajps.ajps_104_22 ; PMid:38546247 PMCid:PMC11003570
England R, Patel R, Marven S. (2013) Laparoscopic management of primary intestinal trichobezoar. J Pediatr Surg Case Reports. 1(5): 108-110. https://doi.org/10.1016/j.epsc.2013.03.014
Ezziti M, Haddad F, Tahiri M, et al. (2017) Gastric trichobezoard: A case report. Pan Afr Med J. 26: 74. [Article in French]. https://doi.org/10.11604/pamj.2017.26.74.11826; PMid:28451051 PMCid:PMC5398866
Frey AS, McKee M, King RA, Martin A. (2005) Hair apparent: Rapunzel syndrome. Am J Psychiatry. 162(2): 242-248. https://doi.org/10.1176/appi.ajp.162.2.242; PMid:15677585
Gonuguntla V, Joshi D-D. (2009) Rapunzel syndrome: a comprehensive review of an unusual case of trichobezoar. Clin Med Res. 7(3): 99-102. https://doi.org/10.3121/cmr.2009.822; PMid:19625498 PMCid:PMC2757434
Gupta A, Mishra SC, Upadhyay VD, Kannegant P. (2022) Rapunzel syndrome in a seven-year-old girl. Indian J Surg. 84(1): 236-237. https://doi.org/10.1007/s12262-021-02788-0
Hamidi H, Muhammadi M, Saberi B, Sarwari MA. (2016) A rare clinic entity: huge trichobezoar. Int J Surg Case Rep. 28: 127-130. https://doi.org/10.1016/j.ijscr.2016.09.039; PMid:27701002 PMCid:PMC5048628
Hernández-Peredo-Rezk G, Escárcega-Fujigaki P, Campillo-Ojeda ZV, et al. (2009) Trichobezoar can be treated laparoscopically. J Laparoendosc Adv Surg Tech. 19(1):111-113. https://doi.org/10.1089/lap.2008.0068; PMid:18991522
Honda H, Ikeya T, Kashiwagi E, et al. (2017) Successful emergency endoscopic treatment of gastric outlet obstruction due to gastric bezoar with gastric pneumatosis. Case Rep. Gastroenterol. 11(3): 718-723. https://doi.org/10.1159/000484130; PMid:29430223 PMCid:PMC5803725
Jinushi R, Yano T, Imamura N, Ishii N. (2021) Endoscopic treatment for a giant gastric bezoar: Sequential use of electrohydraulic lithotripsy, alligator forceps, and snares. JGH Open. 5(4): 522-524. https://doi.org/10.1002/jgh3.12491; PMid:33860105 PMCid:PMC8035430
Khanna S, Barua A, Choudhury S. (2023) Laparoscopic management of rare smoked dry pork bezoar in the stomach. J Min Access Surg. 19(4): 552-554. https://doi.org/10.4103/jmas.jmas_266_22; PMid:37706408 PMCid:PMC10695313
Kim SC, Kim SH, Kim SJ. (2016) A case report: Large trichobezoar causing Rapunzel syndrome. Medicine (Baltimore). 95(22): e3745. https://doi.org/10.1097/MD.0000000000003745; PMid:27258502 PMCid:PMC4900710
Kyin C, Patel P, Casas-Melley A, et al. (2023) Acute case of trichobezoar diagnosed from computed tomography and 3D images: Rapunzel syndrome re-examined. Cureus. 15(2): e35597. https://doi.org/10.7759/cureus.35597; PMid:37007333 PMCid:PMC10063164
Ladas SD, Kamberoglou D, Karamanolis G, et al. (2013) Systematic review: Coca-Cola can effectively dissolve gastric phytobezoars as a first-line treatment. Aliment Pharmacol Ther. 37(2): 169-173. https://doi.org/10.1111/apt.12141; PMid:23252775
Madhusudhan KS, Vaishnav M, Joy D. (2022) Rapunzel syndrome. J Gastrointest Surg. 26(5): 1111-1112. https://doi.org/10.1007/s11605-021-05239-z; PMid:34997470
Marginean CO, Melit LE, Sasaran MO, et al. (2021) Rapunzel syndrome - an extremely rare cause of digestive symptoms in children: A case report and a review of the literature. Front Pediatr. 9: 684379. https://doi.org/10.3389/fped.2021.684379; PMid:34178901 PMCid:PMC8219913
Matsuoka R, Masuda S, Fujita S, Akiyama N. (2023) Trichobezoar effectively treated with direct endoscopic injection of Coca-Cola: A case report. DEN Open. 4(1):e283. https://doi.org/10.1002/deo2.283; PMid:37753229 PMCid:PMC10518563
Mirza MB, Talat N, Saleem M. (2020) Gastrointestinal trichobezoar: An experience with 17 cases. J Pediatr Surg. 55(11): 2504-2509. https://doi.org/10.1016/j.jpedsurg.2020.04.020; PMid:32467033
Mohanta P, Mukhopadhyay M, Maiti S, Mukhopadhyay B. (2004) Trichobezoar in children - an uncommon problem. J Indian Assoc Pediatr Surg. 9(1): 30-32.
Mouffak A, Salam S, Fahl A, et al. (2021) Rare cause of mechanical occlusion in children: A case report. Pan Afr Med J. 38: 71. [Article in French]. doi: 10.11604/pamj.2021.38.71.27634.
Naik S, Gupta V, Naik S, et al. (2007) Rapunzel syndrome reviewed and redefined. Dig Surg. 24(3): 157-161. https://doi.org/10.1159/000102098; PMid:17476105
Narra R, Guntamukkala A, Rao CB, Begum T. (2022) Rapunzel syndrome: A case of trichobezoar with small bowel complications. Surg J (N Y). 8(4): e293-e296. https://doi.org/10.1055/s-0042-1757777; PMid:36349084 PMCid:PMC9637412
Nirasawa Y, Mori T, Ito Y, et al. (1998) Laparoscopic removal of a large gastric trichobezoar. J Pediatr Surg. 33(4): 663-665. https://doi.org/10.1016/S0022-3468(98)90342-6; PMid:9574777
Ripollés T, García-Aguayo J, Martínez MJ, Gil P. (2001) Gastrointestinal bezoars: sonographic and CT characteristics. AJR Am J Roentgenol. 177(1): 65-69. https://doi.org/10.2214/ajr.177.1.1770065; PMid:11418400
Rybalchenko VF, Rusak PS, Rinzberg BS, et al. (2023). Small intestinal obstruction by food in young children: a view on the problem, literature data and own clinical observations. Paediatric Surgery (Ukraine). 2 (79): 92-98. https://doi.org/10.15574/PS.2023.79.92
Schuler L, Hodel M, Stieger C. (2023) The Rapunzel syndrome: a hairy tale. Surg Case Rep. 9(1): 49. https://doi.org/10.1186/s40792-023-01631-w; PMid:36976388 PMCid:PMC10050508
Tudor ECG, Clark MC (2013) Laparoscopic-assisted removal of gastric trichobezoar; a novel technique to reduce operative complications and time. J Pediatr Surg. 48(3): E13-E15. https://doi.org/10.1016/j.jpedsurg.2012.12.028; PMid:23480941
Vaughan ED Jr., Sawyers JL, Scott HW Jr. (1968) The Rapunzel syndrome. An unusual complication of intestinal bezoar. Surgery. 63(2): 339-343.
Vepakomma D, Alladi A. (2014) Complete laparoscopic removal of a gastric trichobezoar. J Min Access Surg. 10(3): 154-156. https://doi.org/10.4103/0972-9941.134880; PMid:25013333 PMCid:PMC4083549
Wang C, Zhao X, Mao S, et al. (2006) Management of SAH with traditional Chinese medicine in China. Neurol Res. 28(4): 436-444. https://doi.org/10.1179/016164106X115044; PMid:16759447
Western C, Bokhari S, Gould S. (2008) Rapunzel syndrome: a case report and review. J Gastrointest Surg. 12(9): 1612-1614. https://doi.org/10.1007/s11605-007-0408-4; PMid:18027058
Wolski M, Gawłowska-Sawosz M, Gogolewski M, et al. (2016) Trichotillomania, trichophagia, trichobezoar - summary of three cases. Endoscopic follow up scheme in trichotillomania. Psychiatr Pol. 50(1): 145-152. https://doi.org/10.12740/PP/43636; PMid:27086334
Zecevic M, Saxena AK. (2022) Laparoscopic approach to trichobezoars in the pediatric age group. J Pediatr Endosc Surg. (2022) 4(4): 143-147. https://doi.org/10.1007/s42804-022-00161-w
Downloads
Published
Issue
Section
License
Copyright (c) 2024 Paediatric Surgery (Ukraine)
This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
The policy of the Journal “PAEDIATRIC SURGERY. UKRAINE” is compatible with the vast majority of funders' of open access and self-archiving policies. The journal provides immediate open access route being convinced that everyone – not only scientists - can benefit from research results, and publishes articles exclusively under open access distribution, with a Creative Commons Attribution-Noncommercial 4.0 international license(СС BY-NC).
Authors transfer the copyright to the Journal “PAEDIATRIC SURGERY.UKRAINE” when the manuscript is accepted for publication. Authors declare that this manuscript has not been published nor is under simultaneous consideration for publication elsewhere. After publication, the articles become freely available on-line to the public.
Readers have the right to use, distribute, and reproduce articles in any medium, provided the articles and the journal are properly cited.
The use of published materials for commercial purposes is strongly prohibited.
