What Is Known/What Is New
What Is Known
- Rapunzel syndrome is a trichobezoar that extends into the small bowel, having surgery as the treatment of choice.
- A previous extensive review published by Gorter in 2010 reported 5% successful rate of trichobezoar endoscopic retrieval.
What Is New
- A trichobezoar is made of a patient‘s own hair and doll's hair. The ninth reported case of a trichobezoar in a coeliac patient.
- Review of literature found 16 successful endoscopic removals versus 36 unsuccessful attempts; a rate of success of 30.7%.
- Out of the total endoscopic removals of trichobezoars, more than 70% have been performed during the last 10 years.
See “MacGyver and Rapunzel in the Pediatric Endoscopy Suite" by Lightdale on page 147.
Trichobezoars are collections of swallowed hair that together with undigested foods form a mass. Typically, a patient pulls out their own hair (also known as trichotillomania) and eats it (trichophagia). It has been estimated that only 5% to 10% of patients with trichotillomania engage in trichophagia (1) and of these only 1% develop a trichobezoar.
Whenever the trichobezoar extends into the small bowel, the condition is termed Rapunzel syndrome from the fairy tale princess Rapunzel. She was locked into a tower and made a rope of her hair to allow the Prince to climb the tower to her room to rescue her.
The gold standard for diagnosis is endoscopy, whereas the treatment is either endoscopic removal or surgical intervention. Endoscopic removal may be less invasive and more cost effective than surgical removal, but its feasibility may depend upon the shape and dimensions of the trichobezoar.
Sometimes, endoscopic treatment is, however, not successful because of the extension of trichobezoar tail into the small bowel, the size of the entire bezoar or its embedding into gastric mucosa. Procedural success is defined as uncomplicated endoscopic removal without the need for surgical laparoscopy or laparotomy. In this review of endoscopic approaches to Rapunzel syndrome, the authors describe an attempted endoscopic removal of a gastric trichobezoar that subsequently required multiple surgical interventions because of unforeseen and unpredictable complications.
We report the case of a 9-year-old girl with a 5 months’ history of intermittent central abdominal pain, occasional vomiting and halitosis, and significant worsening over a 10-day period. At the age of 5 years she started pulling out her hair and she was known to suffer from anxiety. No other remarkable medical history was noted and specifically no gastrointestinal illness. Physical examination revealed upper abdominal tenderness and a mass was palpable in the left upper quadrant and epigastrium. No evidence of peritonism was noted. Alopaecia with the impression of telogen effluvium was noted. Her vital signs were stable. Her weight was <0.4th percentile and her height <2nd percentile. Abdominal ultrasound showed an echogenic mass in the stomach. On MRI, there was a large signal void within the stomach extending into the duodenum to the level of the junction of D2 and D3 and fluid filled dilated loops of small bowel (Fig. 1A1 and A2). Laboratory investigations showed an Hb of 94 g/L and an MCV of 59 fL, serum iron 1 micromol/L (range 9–30), ferritin 3 ng/mL (range 22–322), normal transferrin and low transferrin saturation of 2% (range 15–50). IgA level was within normal range and IgA anti-tissue transglutaminase level was 128 U/mL (normal range 0–7).
Upper endoscopy was performed under general anaesthesia with an XQ260 gastroscope (Olympus Optical Co., Ltd., Tokyo, Japan) and found a large trichobezoar occupying two-thirds of stomach, extending into the duodenum and a large ulcer in D1, without active bleeding (Fig. 1B1). The approximate size of the trichobezoar was 25 cm x 30 cm. About 70% of the trichobezoar was fragmented and removed piecemeal using APC and electrocautery, 30 and 50 mm polyp snares and hexagonal and oblique 30 mm snares. The entire procedure took 3 hours. An amount of gas/smoke was generated during the electrical dissolution of the trichobezoar and was immediately removed by suction. No thermal damage to the gastric mucosa was identified at any point. A decision to remove the remaining 20% of the mass 48 hours later was made.
Postprocedure she remained well but approximately 24 hours later, her clinical status deteriorated and there was imaging evidence of perforation, therefore, laparotomy was performed revealing 18 small bowel perforations. The most proximal was situated at 5 cm from duodeno-jejunal flexure. A 1 cm posterior gastric perforation into the lesser sac was also noted. Gastrotomy was performed with removal of the residual trichobezoar and closure of the gastric perforation. One hundred and seven centimetres of small bowel were resected with perforation sites along the mesenteric border, measuring up to 1 cm, with thin blonde and also dark hair emerging from some of the perforation sites. After 5 days, because of bile leak from perforation, a jejunostomy was created.
Due to significant blood loss via the surgical drains requiring blood transfusion, a second endoscopy was performed and identified 2 nonbleeding ulcers, the largest one situated in D1-D2 and a 1 cm perforation of the lesser curvature, which was clipped with Boston Scientific Resolution Clips but not completely closed. A subsequent closure was achieved with the Over-The-Scope-Clip (OTSC) endoscopic clipping system. The 12 mm Over-The-Scope Clip could not traverse the upper oesophageal sphincter and after oesophageal dilatation, a 11 mm Over-The-Scope Clip was passed with the XQ260 scope, the perforation was centralized and the clip deployed while suction was applied (Fig. 1B2). As no leak into the stoma bag was seen during endoscopic gastric inflation after clipping, unlike previously, it was presumed that the perforation was successfully closed. After 3 more days, because of bleeding per rectum a re-look laparotomy was performed and a large hole in the posterior wall of the stomach was identified, together with the opening of the old ulcer site and a gastro-colic fistula at the splenic flexure. Both perforations and fistula were closed and a corrugated drain was placed at the splenic flexure. The patient recovered slowly and she was discharged 3 months later on home parenteral nutrition. Bowel reconnection and stoma closure were carried out after 2 months. Psychiatric input was helpful. On follow-up she was well, tolerating a normal but gluten-free diet.
It is interesting to speculate on the clinical lead up to this situation. The duodenal biopsies taken confirmed coeliac disease, and this is likely to have been responsible for low iron indices and resultant anaemia. It is well known that iron deficiency leads to pica, and trichophagia is one of the manifestations of this. Unusually, the patient was swallowing not only her own hair but, it transpired subsequently, that of her dolls. This may have been significant in the pathogenesis of the subsequent gastric and small bowel perforations because of the mechanical and chemical composition of the doll hair, as will be discussed further.
The first endoscopic removal of a trichobezoar was attempted 20 years ago (2) and the last review addressing the treatment options for trichobezoars was published over 10 years ago (3).
We performed an extensive literature review, which was restricted to case reports of trichobezoars in humans (children and adults) in which endoscopic removal was attempted. Publications on other types of bezoars were excluded as were those where surgery was the method of removal without an endoscopic attempt. Another review on reported cases of trichobezoars in coeliac disease patients was also carried out.
Databases PubMed, Scopus, and Semantic scholar were searched from their beginning until December 31, 2018. The search terms included “trichobezoar” AND “endoscopy” OR “endoscopic,” “trichobezoar” AND “coeliac.” Titles and abstracts were evaluated and after exclusion, the publications which qualified were read in full.
Informed consent from the caregivers of the child was obtained.
Results of Literature Review
The 3 databases searched for “trichobezoar” and “endoscopy” have returned different numbers of articles. All the articles have been evaluated by title/abstract; this resulted in the inclusion of 31 articles on unsuccessful endoscopic removal of trichobezoars (selection criteria: the endoscopic removal was attempted and proved unsuccessful), together with 16 articles describing successful endoscopic retrieval of trichobezoars. Therefore, because of the fact that some articles were case series reports, a total number of 52 cases were identified of which 16 were successfully removed by endoscopy alone, that is, a success rate of 30.7% (16/52). Details are provided in Table 1.
In the unsuccessful cases, there were 26 (72.2%) children and 10 adults, whereas in the successful cases, there were 14 (87.5%) children and 2 adults.
The majority of the endoscopic removal attempts occurred in the last 10 years—72.2% of the unsuccessful ones and 75% of the successful ones.
Among the successful cases, there were 3 patients with oesophageal localization of the trichobezoar and 5 with extension in to the duodenum. In 2 cases, the endotracheal tube was dislodged during removal of the trichobezoar resulting in respiratory compromise and necessitating its extraction from the airway.
Regarding trichobezoars in coeliac disease patients, 6 cases were identified. This is a disproportionate amount compared with the general population—iron-deficiency associated pica may be postulated as a reason. It could be proposed that the presence of celiac disease may have led to a weakened gastrointestinal wall. Whether it contributed to predisposition to perforation in our case is, however, open to debate. Perforation is not reported as a complication in other cases of attempted endoscopic removal.
Endoscopic Removal of Trichobezoars
Although surgery was the treatment of choice for bezoars in the past, endoscopy is now the preferred treatment in 66% to 77% of bezoar cases (4). Trichobezoars are, however, the most difficult type of bezoar, as fragmentation before endoscopy by dissolution with cola or proteolytic enzymes, which is effective in phytobezoars, lactobezoars, pharmacobezoars, and trychophytobezoars, does not work for trichobezoars (4).
Over a decade ago, Gorter et al (3) reviewed approaches to trichobezoar removal and reported 5% success rate for endoscopic retrieval. Since then, there has been an acceleration in success rate, which might be attributable to the increase in skill mix of the operators and the sophistication of endoscopic techniques at the disposal of the endoscopist in these cases. More recently, however, there have been increasing reports of endoscopic success, most of them being during the last 10 years.
The increased success rate of over 30% that we describe may be attributed to the combination of an increased willingness of endoscopists to try this approach alongside the development of a wider range of therapeutic endoscopic accessories now available to the operator.
Patient demographics, location, and size of the trichobezoars and details of the endoscopic tools used for removal and details about the duration of the procedures are outlined in Table 2. The majority were fragmented using either Nd : YAG laser, hot biopsy forceps, polypectomy snare or APC.
The first attempt was by Van Gossum in 1989 using Nd : YAG laser and extracorporeal shock-wave lithotripsy in multiple endoscopic sessions, which failed (2). In the same year, Soehendra (5) successfully removed a 15 cm × 7 cm trichobezoar using Nd : YAG laser in 3 sessions of 2 to 3 hours. Saeed grasped the bezoar with “pelican-type” forceps and partly engaged it into an overtube, removing a 12 cm trichobezoar together with the overtube as a single unit, without fragmenting it (6).
Wang et al used a bezotome—modified needle knife and monopolar current—in order to fragment and remove a 10 cm trichobezoar (7). Aybar and Safta (9) fragmented an 8 cm × 7 cm trichobezoar into 13 pieces by using hot biopsy forceps and an electrocautery snare (ERBE APC230; settings: effect 2–4; 30–40 W) and managed to remove it after 25 passes over 3 hours. Konuma et al(10) successfully retrieved a gastric trichobezoar without fragmentation using only a grasper and a net due to the favourable shape (longitudinal) and diameter of the trichobezoar 1.8 cm × 3.2 cm × 34 cm in length.
Electrocautery/APC as a method of trichobezoar fragmentation have been employed 4 times. Iwamuro used APC and electrosurgical endo-knives in order to cut the hairball into pieces and remove it (14). Benatta (17) used a polypectomy snare and APC (ERBE, VIO, 200 D; settings: effect 2; 40 W) to fragment an 8 cm × 4 cm trichobezoar and successfully removed it in 15 passes over 50 minutes. Zhao et al. (18) applied APC to a 10.5 × 3.5 cm trichophytobezoar and divided it using endoscopic scissors removing it in 2 sessions.
A combined method using laparoscopy-assisted fragmentation (laparoscopic scissors used through a 1 cm gastric incision) and endoscopic retrieval of fragments was used recently in some cases with favourable outcomes.
Reports of unsuccessful attempts of trichobezoars endoscopic removal are listed in Table 3. There are no case reports of associated complications, such as perforation—in all likelihood because of the inert noncombustible properties of human hair versus artificial hair.
Coeliac Disease and Trichobezoars
Pica is defined by the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition, Text Revision(50), as an appetite for nonfood substances that are nonnutritious, lasting more than 1 month, in children over 5 years of age. Pica itself is associated with iron deficiency anaemia and is reported in coeliac disease.
Unlike other bezoars, trichobezoars are not associated with gastrointestinal motility disorders but with psychiatric disorders. The underlying mechanisms for trichotillomania and trichophagia are not yet understood, but the actions of pulling the hair out, playing and swallowing it are associated with a sense of gratification and relief resulting in anxiety reduction.
The comprehensive literature research focusing on trichobezoar in coeliac disease, which we have performed revealed only 8 cases (35,51–57). All were young girls, age range 5 to 16 years, and 5 had Rapunzel syndrome for which laparotomy was performed, failed endoscopic removal being attempted in 1 case. Four of the cases had associated iron deficiency anaemia.
Complications of Trichobezoars
The first few hair meals get trapped within the gastric folds escaping peristalsis because of the hair's lack of friction and as the ingestion continues, the mass assumes the shape of the stomach. The patient may remain asymptomatic for years. As the size of the trichobezoar increases, so does the risk of mucosal ulceration.
Among the trichobezoar complications not because of attempted endoscopic removal are: perforation of the stomach or intestine (10.1%); intussusception (1.85%); acute pancreatitis (0.92%); cholangitis (0.92%); biliary perforation; obstructive jaundice; and death (37).
Complications Specific to This Case
The trichobezoar's weight may exert pressure on the mucosa, exerting a mechanical effect, which may lead to ischemia. The lesser curvature of the stomach is more vulnerable to perforation as its vascularization comes from an arterial watershed formed by anastomoses of the right and left gastric artery branches—this may make some of the gastric wall susceptible to vascular compromise with resultant ischaemic necrosis and perforation.
In our case, several clumps of hair had detached from the main trichobezoar, either spontaneously or in the process of fragmentation travelling to multiple areas of the small intestine. Luminal occlusion leading to distention and stretching of the intestinal wall with resultant vascular compromise is 1 possible contributory cause of the perforations. In addition, we postulate, that repetitive mucosal microtrauma, because of the contact of multiple individual hair wires, combined with a “cheese-wire effect” between 2 adjacent areas of small bowel when peristalsis occurred (the doll hair is strong and does not stretch easily) and that these factors created “a perfect storm” for the evolution of perforation when added to the noxious gas presence. Another putative mechanism may have been that the weight of the trichobezoar pieces caused a pressure effect on the very thin small bowel wall and led to mechanical perforation.
Proprietary composition of doll hair as a contributing factor
Electrocautery may have harmful consequences when used in circumstances that favour combustible gas production in a closed space. Four factors are important: gastrointestinal gases; an oxygen-rich environment; electrocautery; and an enclosed space. Obstruction as a complication of trichobezoar is the first step of a possible inflammable scenario. Following the obstruction, bacterial proliferation may lead to an increase in intestinal combustible gas production, involving a combination of methane, oxygen, and hydrogen. Face mask ventilation using 100% oxygen may lead to air trapping in the stomach, and therefore, to a high-oxygen environment, although all children in this scenario undergoing therapeutic endoscopy for prolonged periods should be intubated.
Unusually in this case, part of the trichobezoar was formed by doll's hair ingested by the patient. Her mother reported later that an entire collection of almost 20 dolls were found without hair.
The authors have contacted the manufacturer of the doll with enquiries on the composition of the doll hair. The material used to make the artificial hair was resin, specifically a vinyl polymer called vinylidene chloride. At temperatures above 180 °C the melting point is reached and gases containing carbon monoxide, volatile resin gas, and hydrogen chloride gas are generated because of incomplete combustion of thermal decomposition. Among the material safety measures, the firefighting instructions state that those who may be exposed to vapours or products of decomposition because of fire should wear full bunker gear including a positive pressure self-contained breathing apparatus and full protective clothes in any closed space.
Hydrogen chloride upon contact with water forms hydrochloric acid, both substances being corrosive, leading to necrosis, ulceration, and perforation. We can, therefore hypothesize that APC/electrocautery when applied to this artificial hair, trichobezoar melted the doll hair, which produced hydrogen chloride gas, which both as a vapour and liquid, following the combination with water/gastric juice in the stomach may have led to chemical corrosion of the GI wall—this could explain the very unusual and repeated late complication of multiple perforations—some of which were distant from the actual stomach where APC/electrocautery were applied, that is, the noxious gas may have travelled through the small bowel, possibly promoted by air insufflation during the endoscopy, thereby causing multiple late perforations in the small bowel, some days later. This may have occurred despite suction of gases during the procedure. The endoscopist in this case (M.T.) has had experience of human hair endoscopic retrieval previously and has not seen gas/smoke production in those cases—presumably, as this trichobezoar was predominantly artificial hair.
The normal hair shaft is covered by a cuticle made of overlapping layers of elongated cells that slant outward. A sample of doll hair has been analysed using scanning electron microscopy (SEM) (Department of Biomedical Sciences, Sheffield University) and revealed that its structure is perfectly round and straight without any slanting layers, as seen in human hair. Therefore, compared with human hair, we postulate that it could have produced a more powerful “cheese-wire” effect on the thin wall of the small intestine (Fig. 1C1 and C2) (58).
CONCLUSIONS AND LEARNING POINTS TAKEN FROM THIS CASE
This case offers some important learning points for colleagues who may consider attempting endoscopic retrieval of trichobezoars in children. In fact, despite the unforeseen complications described in our case, we believe it remains reasonable to attempt endoscopic retrieval of both oesophageal and gastric trichobezoars assuming they appear to occupy less than two-thirds of the stomach and have limited and no extension into the small bowel. In addition, we would recommend pre-procedural coordination of care with a surgical team, particularly if a trichobezoar appears large on radiological imaging, or if it extends into the small bowel. Pre-operative planning with the anaesthetist is also critical, as endoscopic removal of a trichobezoar may pose airway management risks. Our team also believes that the skill mix of the endoscopist and the potential complexity of the case should be carefully assessed to ensure that they are matched, and that the endoscopist should have familiarity and access to a full complement of endoscopic accessories and tools.
Upon reflection of this case, our endoscopy team would also propose a number of standard steps that may be taken in order to avoid complications. First, an endoscopist should always seek to determine what substance(s) might be involved in a trichobezoar before attempting removal. Second, an endoscopist should take measures to ensure a well-prepared multi-disciplinary team that includes surgical colleagues. In regards to the patient and family, an endoscopist attempting an endoscopic approach to Rapunzel syndrome should consider patient-specific vulnerabilities and engage in a thoughtful informed consent process that covers multiple possible complications, even if not previously reported in the literature. Finally, the authors would note that a lesson learned in this case is that neither reports of success in the literature of endoscopic trichobezoar removal, nor prior successful experiences by any specific endoscopist with this procedure, should currently be interpreted as highly suggestive of repeated success.
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