Abstract
Trichobezoars are relatively uncommon problems with a known female predominance. We report two female children with gastric bezoars. Main presenting symptoms were abdominal distension, weight loss and anaemia. Upper abdominal mass was palpable in both. Diagnosis was suspected on initial abdominal radiograph and ultrasound scan then confirmed by upper endoscopy. No bowel extension was recorded in either case. We report here a modification of the surgical technique in which the gastrostomy cut edges were anchored to the laparotomy skin. This modification aided easy and complete delivery of hair balls avoiding any spillage or wound contamination.
Keywords: bezoar, children
Background
Gastric trichobezoars are a relatively infrequent pathology.1 Rapunzel syndrome is so called due to the extension of a bezoar beyond the gastric body into the duodenum.2 Mobilising the stomach up into a small wound and operative delivery, particularly of such extensive bezoars, can be difficult in young children. In addition, postoperative wound infections in the region of 15% have been reported due to contamination with gastric contents.3 We are reporting here a modification of the usual technique, aiming for decreasing the incidence of wound infection and facilitating the removal of the bezoar.
Case presentation
Case 1
A 10-year-old girl with 22 kg body weight was transferred from the local hospital with history of malaise, weight loss and anaemia. Mass was felt in the epigastrium and in the left upper quadrant. Abdominal X-ray and ultrasound (US) scan showed a heterogeneous mass in the stomach suggestive of a bezoar. She had an upper gastrointestinal (GI) endoscopy that confirmed the presence of the bezoar. This was followed by a laparotomy and gastrostomy with the technique mentioned below. Histopathological specimen confirmed the presence of matted hair in the specimen. She had a smooth postoperative recovery without any evidence of wound infection and was discharged home 7 days after the procedure. She was reviewed 3 months and 1 year postoperatively with very good outcome.
Case 2
A 3-year-old girl with 15 kg body weight, presenting with progressive abdominal distension of 6-month duration. This was not associated with any vomiting, constipation or bleeding per rectum. There was a 1 year history of pulling her hair and eating it, in addition to eating carpet threads and any hair that she can find on the floor. Abdominal examination showed marked upper abdominal distension with a lump in her left upper quadrant.
Abdominal US showed a mass with increased echogenicity inside but separate from the stomach wall. She had an upper GI endoscopy (figure 1) that confirmed the diagnosis of trichobezoar. This was followed by laparotomy and removal of the bezoar with the technique mentioned below. Histopathological specimen showed matted elongated bezoar with a collection of hair measuring 40 cm in length and up to 5 cm in width. She had a smooth postoperative recovery and was discharged home 6 days postoperatively without any evidence of wound infection. Three months postoperatively, she was doing very well.
Figure 1.
Upper gastrointestinal endoscopy showed trichobezoar in the stomach.
Treatment
Technique
Single dose of Co-amoxiclav 30 mg/kg intravenous (maximum 1.2 g) is given on induction. A transverse full thickness incision is made in the left upper quadrant. The stomach is mobilised into the wound and opened longitudinally. The cut edges of the stomach are secured to the skin with a circumferential full thickness 4/0 prolene running suture (polypropylene non-absorbable monofilament, Ethicon) (figure 2). The bezoar was then being safely and easily delivered in stages without the need to maintain active traction on the stomach (figures 3 and 4). In addition, the stomach may be easily suctioned to reduce wound contamination. After careful inspection of the stomach to ensure complete removal of the bezoar, the securing prolene suture may be cut and easily removed to allow full thickness closure of the stomach in the usual fashion.
Figure 2.
Gastrostomy performed with suturing the edges of the stomach to the skin (temporary gastrostomy).
Figure 3.
Delivery of the hair ball without contact with the edges of the wound.
Figure 4.
Hair ball after removal.
Outcome and follow-up
Both patients were reviewed 3 and 12 months (case 1) postoperatively and they were doing very well with excellent cosmetic results and no evidence of any wound infection.
Discussion
We are reporting our experience with two patients, who were diagnosed and treated for trichobezoar in our centre. This rare entity is known to happen mostly in women and this was the case in our two patients. Young women below the age of 30 are the most susceptible.4 A recent case series of seven patients with trichobezoars, all of whom were girls, found a mean age of 11.5 years at presentation,4 which is comparable to our first case age (10 years) while our second case age of 3 years falls slightly below the age range in the same study (range 5–23 years). The youngest reported case with this problem was 6 months of age.5 Trichophagia for hair and hair-like fibres as a predisposing factor was evident in the second case.
Trichobezoars are most commonly found in the stomach, often extending into the first portion of the duodenum which happened in our two cases. Less common presentations include extension of hair to involve the small bowel, a condition known as Rapunzel syndrome.
Abdominal pain, distension, unexplained weight loss and anaemia were the main presenting symptoms and upper abdominal mass was easily felt in both cases. The condition could be asymptomatic or often present with additional anorexia, nausea, vomiting or less commonly partial or complete bowel obstruction. Severe halitosis and patchy alopecia may provide clues on physical examination. Trichobezoars can be a rare cause of melena, gastric or bowel perforation, intussusception, appendicitis, pancreatitis, nephrotic syndrome and biliary obstruction.6–11 None of the later complication was reported in either of the cases.
As patients occasionally remain asymptomatic for many years and the clinical findings are non-specific, imaging plays an important role in diagnosis. In our cases, the diagnosis was suspected on the initial abdominal X-ray and ultrasonography showing a significant intraluminal mass separate from the stomach wall, so we proceeded with upper endoscopy that was diagnostic in both cases. In equivocal cases, CT can be another helpful option and may give an idea about small bowel extension including more distal satellite lesions.12 MRI and contrast studies are less commonly needed.
Trichbezoar as a topic gained more popularity and became in focus with the recent reports on feasibility of minimally invasive techniques for its removal. These include mainly endoscopic fragmentation and/or retrieval and laparoscopic or laparoscopic assisted removal. The reported success rates of endoscopy are very low (5%) and restricted to small trichobezoars. Laparoscopy is a more viable option with a relatively higher success rates of around 75% and less complication rate according to a recent overview of literature that collected eight patients treated by laparoscopic approach, of whom six were successful but the operative time remains quite long (2–5 hours). Laparotomy, on the other hand, has a success rate of almost 100% and reasonable complication rate of 12%–14% in most reviews. Furthermore, the shorter operative time and the ability to examine the entire GI tract for satellites during open laparotomy stay behind its worldwide acceptance as the treatment of choice for trichobezoar in children.3 6
The first surgical removal was performed by Schonbern in 1883. The mainstay of current surgical treatment is adequate exposure via an upper abdominal laparotomy, removal of the bezoar without spillage into the peritoneal cavity and wound protection through packing off with laparotomy pads to avoid contact with the foul putrid nature of the bezoar. With all current precautions, intra-abdominal spillage still occurs with a relatively high wound infection rate of 14% being evident in most recent work.3
The described new surgical modification in this case report fully achieves the main goals of surgery including easy and complete delivery of the bezoar avoiding any spillage or wound contamination. We did not have wound infection issues with our two patients but the small number makes it impossible to draw any conclusion. On the other hand, we feel that this modification is simple, efficient and easy to adopt in similar future case reports or prospective studies to test our hypothesis on its role in decreasing the relatively high current intra-abdominal and wound infection rates. It needs to be emphasised that this technique was used in gastric trichobezoar and we are unable to recommend or comment on its feasibility in Rapunzel syndrome or when an enterotomy is needed for whatever surgical reason. The postoperative recovery was excellent and our hospital stay is comparable to similar recent reports.3
Learning points.
Trichobezoar is a rare condition; laparotomy is the widely accepted treatment.
Wound infection still represents a main concern.
The technique of temporary anchoring the longitudinal gastrostomy edges to the laparotomy wound before trichbezoar delivery (temporary gastrostomy technique) can help reduce the wound infection and peritoneal soiling.
This technique represents a simple and efficient adaptation to the trans-gastric removal of a bezoar while minimising gastric trauma.
Footnotes
Contributors: All the authors of this manuscript have worked actively in drafting and revision of the article. AAD: drafting the work and revising the article. Final approval of the version published. AEMA: drafting and revision of the article. EP: involved in data collection and drafting the article. RG-C: final approval of the version published.
Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Competing interests: None declared.
Provenance and peer review: Not commissioned; externally peer reviewed.
Patient consent for publication: Parental/guardian consent obtained.
References
- 1. Iwamuro M, Okada H, Matsueda K, et al. Review of the diagnosis and management of gastrointestinal bezoars. World J Gastrointest Endosc 2015;7:336–45. 10.4253/wjge.v7.i4.336 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 2. Vaughan ED, Sawyers JL, Scott HW. The Rapunzel syndrome. An unusual complication of intestinal bezoar. Surgery 1968;63:339–43. [PubMed] [Google Scholar]
- 3. Fallon SC, Slater BJ, Larimer EL, et al. The surgical management of Rapunzel syndrome: a case series and literature review. J Pediatr Surg 2013;48:830–4. 10.1016/j.jpedsurg.2012.07.046 [DOI] [PubMed] [Google Scholar]
- 4. Duke DC, Keeley ML, Geffken GR, et al. Trichotillomania: a current review. Clin Psychol Rev 2010;30:181–93. 10.1016/j.cpr.2009.10.008 [DOI] [PubMed] [Google Scholar]
- 5. Schreiber H, Filston HC. Obstructive jaundice due to gastric trichobezoar. J Pediatr Surg 1976;11:103–4. 10.1016/0022-3468(76)90182-2 [DOI] [PubMed] [Google Scholar]
- 6. Gorter RR, Kneepkens CM, Mattens EC, et al. Management of trichobezoar: case report and literature review. Pediatr Surg Int 2010;26:457–63. 10.1007/s00383-010-2570-0 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 7. Kibria R, Michail S, Ali SA. Rapunzel Syndrome—A Rare Cause of Multiple Jejunal Intussusception. South. Med. J 2009;Apr102(4:416–8. [DOI] [PubMed] [Google Scholar]
- 8. Dogra S, Yadav YK, Sharma U, et al. Rapunzel Syndrome Causing Appendicitis in an 8-year-old Girl. Int. J. Trichol 2012;4:278–9. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 9. Umbetalina NS, Turgunov EM, Turgunova LG, et al. Nephrotic syndrome in a female patient with Rapunzel syndrome, Ter . Arkhiv 2014;86:78–80. [DOI] [PubMed] [Google Scholar]
- 10. Chogle A, Bonilla S, Browne M, et al. Rapunzel syndrome: a rare cause of biliary obstruction. J Pediatr Gastroenterol Nutr 2010;51:522–3. 10.1097/MPG.0b013e3181cb950a [DOI] [PubMed] [Google Scholar]
- 11. Tayyem R, Ilyas I, Smith I, et al. Rapunzel syndrome and gastric perforation. Ann R Coll Surg Engl 2010;92:e27–e28. 10.1308/147870810X476700 [DOI] [PMC free article] [PubMed] [Google Scholar]
- 12. Gayer G, Jonas T, Apter S, et al. Bezoars in the stomach and small bowel CT appearance. Clin Radiol 1999;54:228–32. 10.1016/S0009-9260(99)91156-X [DOI] [PubMed] [Google Scholar]




