INVITED REVIEW
Human and Doll’s Hair in a Gastric Trichobezoar,
Endoscopic Retrieval Hazards

Andreia F. Niţă, yChris J. Hill, zRichard M. Lindley, zSean S. Marven, and Mike A. Thomson
See ‘‘MacGyver and Rapunzel in the Pediatric Endoscopy
Suite’’ by Lightdale on page 147.
ABSTRACT
Trichobezoars are masses of ingested hair, usually the individual’s own hair,
that accumulate in the gastrointestinal tract, most commonly in the stomach.
When extending into the small intestine, this is termed ‘‘Rapunzel syn-
drome.’’ Removal has traditionally been by laparotomy; however, success-
ful endoscopic removal has also been described. We report the case of a 9-
Downloaded from http://journals.lww.com/jpgn by BhDMf5ePHKbH4TTImqenVAPwFBsBoeDVA+qMomVsl6nP6vsEO/bhqsOTjyg+HwE+ on 07/28/2020
year-old-girl with undiagnosed coeliac disease and Rapunzel syndrome who
underwent endoscopic removal of a large trichobezoar, which was followed
by unexpected multiple perforations of the small bowel and stomach. Argon
plasma coagulation (APC) and snare electrocautery were employed during
endoscopy to remove the trichobezoar piecemeal, and approximately 70%
was removed without any clear signs of damage to the mucosa. It was
discovered subsequently that about 20 of her dolls were found without hair.
On investigating the composition of a specific doll hair from the manufac-
turer, it was discovered that it could be hazardous if burned. It was, therefore,
hypothesized that a constellation of factors had conspired to lead to
perforation, that is, the potentially hazardous gas produced from the
electrical energy applied to the synthetic hair and possible mucosal damage
by the physical abrasion of this hair. A review of the literature on endoscopic
attempts to remove trichobezoars irrespective of the result reveals a success
rate of 30.7%.
Key Words: celiac, doll, endoscopy, perforations, trichobezoar
(JPGN 2020;71: 163–170)
T richobezoars 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
Received March 8, 2019; accepted March 1, 2020.
From the Centre for Paediatric Gastroenterology, International Academy
for Paediatric Endoscopy Sheffield Children‘s NHS Trust, Western
Bank, the yElectron Microscopy Service, Department of Biomedical
Science, Sheffield University, and the zDepartment of Paediatric Sur-
gery, Sheffield Children’s NHS Trust, Western Bank, Sheffield, UK.
Address correspondence and reprint requests to Mike A. Thomson, Center
for Paediatric Gastroenterology, International Academy for Paediatric
Endoscopy, Sheffield Children’s NHS Trust, Western Bank, Sheffield
S10 2TH, UK (e-mail: mike.thomson@sch.nhs.uk).
The authors report no conflicts of interest.
Copyright # 2020 by European Society for Pediatric Gastroenterology,
Hepatology, and Nutrition and North American Society for Pediatric
Gastroenterology, Hepatology, and Nutrition
DOI: 10.1097/MPG.0000000000002779
JPGN  Volume 71, Number 2, August 2020
Copyright © ESPGHAN and NASPGHAN. All rights reserved.
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 trichobe-
zoars, more than 70% have been performed during
the last 10 years.
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 success-
ful 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 endo-
scopic 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.
CASE REPORT
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
163
Nit2ă et al
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 evi-
dence of perforation, therefore, laparotomy was performed reveal-
ing 18 small bowel perforations. The most proximal was situated at
5 cm from duodeno-jejunal flexure. A 1 cm posterior gastric perfo-
ration 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 identi-
fied 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. Unusu-
ally, the patient was swallowing not only her own hair but, it
transpired subsequently, that of her dolls. This may have been
164
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JPGN  Volume 71, Number 2, August 2020
significant in the pathogenesis of the subsequent gastric and small
bowel perforations because of the mechanical and chemical com-
position of the doll hair, as will be discussed further.
DISCUSSION
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).
Methods
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 ‘‘endo-
scopic,’’ ‘‘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 ‘‘endos-
copy’’ 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 success-
ful 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 compro-
mise 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
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JPGN  Volume 71, Number 2, August 2020 Human and Doll’s Hair in a Gastric Trichobezoar

FIGURE 1. Trichobezoar on MRI—coronal (A1) and transeverse views (A2). Arrows pointing to the trichobezoar taking the shape of stomach.
Trichobezoar—endoscopic view (B1) and over-the-scope clip closing the gastric perforation (B2). Scanning electron microscopy of doll hair (C1)
performed in Sheffield Childrens versus human hair (C2) (58).

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Nit2ă et al
TABLE 1. Results of literature review using databases
Articles on
trichobezoars endoscopic
removal attempted
and unsuccessful
Trichobezoar
‘‘Trichobezoar’’ and articles included Common
‘‘endoscopy’’ search (evaluation by articles,
Database results, n title/abstract), n n ¼ 13
PubMed 404 101 18
SCOPUS 136 126 18
Semantic scholar 350 165 21
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 phytobe-
zoars, 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 endosco-
pists 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 mul-
tiple 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 fragmenta-
tion have been employed 4 times. Iwamuro used APC and electro-
surgical 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 remov-
ing it in 2 sessions.
166
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JPGN  Volume 71, Number 2, August 2020
Articles on
successful Trichobezoar
endoscopic removal and Coeliac
of trichobezoars disease papers
Total unique Common Total unique Common
articles, articles, articles, articles, Total unique
n ¼ 31 n¼8 n ¼ 16 n¼2 articles, n ¼ 6
12 2
11 6
9 2
A combined method using laparoscopy-assisted fragmenta-
tion (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 endo-
scopic 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 gratifica-
tion and relief resulting in anxiety reduction.
The comprehensive literature research focusing on tricho-
bezoar 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 jaun-
dice; and death (37).
Complications Specific to This Case
Mechanical considerations
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
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JPGN  Volume 71, Number 2, August 2020 Human and Doll’s Hair in a Gastric Trichobezoar

TABLE 2. Successful endoscopic retrieval of trichobezoars in the literature

Year of Patient’s age Trichobezoar No of No of Duration of

No Authors publication and sex Extension dimensions Endoscopic instruments passages sessions session Comments

1 Soehendra 1989 17/F Stomach 15 cm  7 cm Nd: YAG laser, Dormia >100 3 2 to 3 hours
(5) 120 g basket, Celestin tube,
3-arm polyp retrieval
forceps
2 Saeed 1993 53/M Stomach 12 cm Two-channel endoscope, 1 1 30 minutes
et al (6) 55 g overtube, pelican-type
forceps
3 Wang et al (7) 1998 15/F Stomach 10 cm  8 cm Bezotome, modified N/A 2 2 to 3 hours
 8 cm needle-knife monopolar
8 cm  6 cm coagulation current
 4 cm
4 Michail 2007 7/M Lower N/A Roth net 1.8 mm N/A 1 N/A Trichobezoar was
et al (8) oesophagus attached to the
suture from
fundoplication
performed during
neonatal period
5 Aybar and 2011 5/F Stomach, 8 cm  7 cm Hot bipsy forceps, 25 1 3 hours
Safta (9) duodenal bulb electrocautery snare
6 Konuma 2011 9/F Stomach 1.8 cm  3.2 Gasper with 2 1 15 minutes
et al (10) cm  34 cm 100 g 5 prolongs, net
7 Esmaili 2011 17/F stomach N/A endoscopic forceps N/A 1 N/A The patient suffered
et al (11) Emergency rigid respiratory arrest
esophagoscopy during removal of
trichobezoar,
therefore, the
hairball was
removed by the
otolaryngologist
using a rigid
esophagoscope
8 Renji et al (12) 2013 12/F Lower 6 cm  2 cm?? N/A N/A 1 N/A
oesophagus Retained G tube
inside
trichobezoar
9 Malhotra 2013 16/F Stomach N/A N/A Multiple 1 N/A
et al (13)
10 Iwamuro 2014 10/F Stomach N/A Argon plasma coagulation, N/A N/A N/A
et al (14) polypectomy snare
electrosurgical knives for
submucosal dissection:
insulation-tipped
diathermic knife (Olympus
Optical Co., Tokyo,
Japan), ClutchCutter
(Fujifilm Medical Co.,
Tokyo, Japan) and
Mucosectom (Pentax
Medical Co., Tokyo,
Japan)
11 Boussaadni 2014 3/F Oesophagus N/A N/A N/A N/A N/A
et al (15)
12 Kao 2015 5/F Stomach 4 cm N/A N/A 1 N/A The patient was
et al (16) detubated during
the retrieval of the
trichobezoar; the
otolaryngologist
removed the
hairball using
Magill forceps
13 Benatta (17) 2016 6/F Stomach, 8 cm  4 cm Polypectomy snare 15 1 50 minutes
duodenal bulb APC
14 Zhao 2017 12/F Stomach, 10.5 cm  3.5 cm Endoscopic scissors, N/A 2 1 hour The mass was a
et al (18) duodenal bulb polypectomy snare, argon trichophytobezoar
plasma coagulation,
grasping forceps
15 Gremida 2017 21/F Stomach, N/A Raptor forceps, endoscopic N/A N/A N/A
et al (19) duodenum overtube, Roth net
16 Iwama 2018 13/F Stomach, 13 cm  3 cm 5-prong grasper, basket 2 1 2 hours
et al (20) duodenum 9 cm  3.5 cm forceps
30 g

Data from (5–20).

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 TABLE 3. Unsuccessful attempts of trichobezoars endoscopic retrieval in the literature

168
Number of cases for which
Nit2ă et al

Publication endoscopic removal Patient‘s age

No. Authors year Type of article was attempted and sex Title of the article

1 Van Gossum et al (2) 1989 Case report 1 14/F Failure of nonsurgical procedures to treat gastric trichobezoar
2 De Baker et al (21) 1999 Case report 1 10/F Huge gastric trichobezoar in a 10-year-old girl: case report with emphasis on
endoscopy in diagnosis and therapy
3 Zamir et al (22) 2004 Retrospective review 1 14/F Phytobezoars and trichobezoars. A 10-year experience
of cases
4 Quraishi and 2005 Case report 1 5/F Rapunzel syndrome
Kamath (23)
5 Ruiz et al (24) 2005 Case series 4 22 to 73 years/ Tricobezoares gastroduodenales: una causa poco frecuente de obstrucción del
3F and 1M tracto de salida
6 Silva et al (25) 2007 Case report 1 10/F Giant gastric and duodenal trichobezoar
7 Al Wadan et al (26) 2008 Case report 1 7/F ’Rapunzel syndrome’ trichobezoar in a 7-year-old girl: a case report
8 Bounaim et al (27) 2009 Case report 1 13/F Trichobézoard gastrique géant
9 Kansagra et al (28) 2010 Case report 1 37/F Rapunzel’s Syndrome
10 Gorter et al (3) 2010 Case report and 2 9/F Management of trichobezoar: case report and literature review
literature review 15/F
11 Hassani et al (29) 2010 Case report 2 9/F Trichobezoar gastrique—à propos de deux cas
12 Bege et al (30) 2011 Case report 1 27F The Rapunzel syndrome: a hard-to-swallow tale
13 Nieto et al (31) 2011 Case report and 1 20/F Rapunzel syndrome. A case report and literature review
literature review
14 Phavicitr and 2012 Case report 1 10/F Rapunzel syndrome in a Thai girl with an asymptomatic abdominal mass: a case
Vathanasanti (32) report
15 Veloso et al (33) 2013 Case report 1 7/F Trichotillomania and trichophagia: the causes of Rapunzel syndrome
16 El-Mazary et al (34) 2014 Case report and 1 7/F Rapunzel syndrome in a 7-year-old Egyptian girl with primary biliary cirrhosis: a
literature review case report and literature review
17 Irastroza et al (35) 2014 Case report 1 8/F A trichobezoar in a child with undiagnosed celiac disease: a case report
18 Mariotto et al (36) 2014 Case report 1 10/F Trichobezoars in children: therapeutic complications
19 Altonbary and 2015 Case report 1 15/F Rapunzel syndrome
Bahgat (37)
20 Castle et al (38) 2015 Retrospective review In 2 out of 6 cases 12/F Management of complicated gastric bezoars in children and adolescents
of cases endoscopic retrieval 4/F
was attempted
21 Yik et al (39) 2015 Case report 1 13/F Stomach trichobezoar (Rapunzel syndrome) with iatrogenic intussusception
JPGN 

22 Kim et al (40) 2016 Case report 1 8/F Large trichobezoar causing Rapunzel syndrome
23 Hamidi et al (41) 2016 Case report 1 18/F A rare clinic entity: huge trichobezoar
24 Paparoupa and 2016 Case report 1 21/F Trichobezoar
Schippert (42)
25 Yik and How (43) 2016 Case report 1 16/F A ‘‘hairy’’ problem: trichotillomania, trichophagia and trichobezoars

26 Wang et al (44) 2016 Case report 1 13/F The diagnosis and treatment of Rapunzel syndrome

Copyright © ESPGHAN and NASPGHAN. All rights reserved.

27 Nour et al (45) 2017 Case report and 1 4/F Rapunzel syndrome (gastric trichobezoar), a rare presentation with generalized
literature review oedema: case report and review of the literature
28 Sailer et al (46) 2017 Case report 1 8/F Trichobezoar (Rapunzel syndrome) causing severe anaemia and cardiac failure
29 Sun et al (47) 2017 Case report 1 12/F Trichobezoar and Rapunzel syndrome
30 Al-Osail et al (48) 2018 Case report 1 17/F Best management modality of trichobezoar: a case report
31 Akbar et al (49) 2018 Case report 1 18/F Trichobezoar-induced heartburn in a teenage girl: a case report

Data from (2,3,21–49).

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JPGN  Volume 71, Number 2, August 2020
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 frag-
mentation travelling to multiple areas of the small intestine. Lumi-
nal 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 com-
bustible 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 under-
going 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 8C 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 hydro-
chloric 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
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Human and Doll’s Hair in a Gastric Trichobezoar
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 collea-
gues who may consider attempting endoscopic retrieval of tricho-
bezoars 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 endo-
scopic 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 endo-
scopic 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 syn-
drome 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.
REFERENCES
1. Schlosser S, Black DW, Blum N, et al. The demography, phenomen-
ology, and family history of 22 persons with compulsive hair pulling.
Ann Clin Psychiatry 1994;6:147–52.
2. Van Gossum A, Delhaye M, Cremer M. Failure of nonsurgical proce-
dures to treat gastric trichobezoar. Endoscopy 1989;21:113.
3. Gorter RR, Kneepkens CM, Mattens EC, et al. Management of tricho-
bezoar: case report and literature review. Pediatr Surg Int 2010;26:457–
63.
4. Park SE, Ahn JY, Jung HY, et al. Clinical outcomes associated with
treatment modalities for gastrointestinal bezoars. Gut Liver
2014;8:400–7.
5. Soehendra N. Endoscopic removal of a trichobezoar. Endoscopy
1989;21:201.
6. Saeed ZA, Ramirez FC, Hepps KS, et al. A method for the endoscopic
retrieval of trichobezoars. Gastrointest Endosc 1993;39:698–700.
169
Nit2ă et al
7. Wang YG, Seitz U, Li ZL, et al. Endoscopic management of huge
bezoars. Endoscopy 1998;30:371–4.
8. Michail S, Nanagas V, Mezoff AG. An unusual cause of postfundopli-
cation vomiting. J Pediatr Surg 2008;43:E45–7.
9. Aybar A, Safta AM. Endoscopic removal of a gastric trichobezoar in a
pediatric patient. Gastrointest Endosc 2011;74:435–7.
10. Konuma H, Fu K, Morimoto T, et al. Endoscopic retrieval of a gastric
trichobezoar. World J Gastrointest Endosc 2011;3:20–2.
11. Esmaili MR, Abbasi HR, Baradaranfar MH. Raspiratory arrest due to
airway obstruction following endoscopic removal of trichobezoar. J Pak
Med Assoc 2011;61:700–1.
12. Renji E, Nathan AK, Dalzell MA. Hidden treasure in an endoscopically
retrieved oesophageal trichobezoar. BMJ Case Rep 2013:pii:
bcr2012007858.
13. Malhotra P, Malhotra V, Kaur M, et al. Trichobezoar and amenorrhea: a
rare association. J Gynec Surgery 2013;29:210–2.
14. Iwamuro M, Tanaka S, Shiode J, et al. Clinical characteristics and
treatment outcomes of nineteen Japanese patients with gastrointestinal
bezoars. Intern Med 2014;53:1099–105.
15. El Boussaadni Y, El Mahjoubi S, El Ouali A, et al. Une cause rare de
dysphagie chez l’enfant: le bézoard [a rare cause of dysphagia in
children: the bezoar]. Pan Afr Med J 2014;18:109.
16. Kao EY, Scalzitti NJ, Dion GR, et al. Trichobezoar causing airway
compromise during esophagogastroduodenoscopy. Case Rep Med
2015;2015:806857.
17. Benatta MA. Endoscopic retrieval of gastric trichobezoar after frag-
mentation with electrocautery using polypectomy snare and argon
plasma coagulation in a pediatrci patient. Gastroenterol Rep (Oxf)
2016;4:251–3.
18. Zhao JL, Zhao WC, Wang YS. Endoscopic retrieval of gastric tricho-
phytobezoar: case report of a 12-year-old-girl with trichophagia. Med-
icine (Baltimore) 2017;96:e5969.
19. Gremida A, Mojtahed A, Petty L, et al. A fairy tale with a hairy tail. Dig
Dis Sci 2017;62:3321–4.
20. Iwama I, Nambu R, Hara T. A novel finding of Rapunzel syndrome. Clin
J Gastroenterol 2018;11:19–22.
21. De Baker A, Van Nooten V, Vandenplas Y. Huge gastric trichobezoar in
a 10-year-old girl: case report with emphasis on endoscopy in diagnosis
and therapy. J Pediatr Gastroenterol Nutr 1999;28:513–5.
22. Zamir D, Golblum C, Linova L, et al. Phytobezoars and trichobezoars: a
10-year experience. J Clin Gastroenterol 2004;38:873–6.
23. Quraishi AH, Kamath BS. Rapunzel syndrome. Gastrointest Endosc
2005;62:611.
24. Ruiz HD, Palermo M, Ritondale O, et al. Tricobezoares gastroduode-
nales: una causa poco frecuente de obstrucción del tracto de salida. Acta
Gastroenterol Latinoam 2005;35:24–7.
25. Silva AP, Carvalho J, Pinho R, et al. Giant gastric and duodenal
trichobezoar. Endoscopy 2007;39 Suppl 1:E12–3.
26. Al Wadan AH, Al Kaff H, Al Senabani J, et al. Rapunzel syndrome’
trichobezoar in a 7-year-old girl: a case report. Cases J 2008;1:205.
27. Bounaim A, Sabir A, Mouhadi K, et al. Trichobézoard gastrique géant. J
Afr Hepato Gastroenterol 2009;3:91.
28. Kansagra A, Nagaria N, Ahlawat S. Rapunzel‘s syndrome. Dig Dis Sci
2010;55:3284–5.
29. Hassani KI, El Bouhaddouti H, Benamar Y, et al. Trichobezoar gas-
trique–à propos de deux cas [Gastric trichobezoar–about two cases].
Pan Afr Med J 2010;6:19.
30. Bege T, Desjeux A, Coquet-Reinier B, et al. The Rapunzel syndrome: a
hard-to-swallow tale. J Gastrointest Surg 2011;15:1486–7.
31. Nieto JO, Suarez JV, Vergara JV, et al. Rapunzel syndrome. A case
report and literature review. Rev Col Gastroenterol 2011;26:70–3.
32. Phavicitr N, Vathanasanti C. Rapunzel syndrome in a Thai girl with an
asymptomatic abdominal mass: a case report. J Med Assoc Thai 2012;95
Suppl 5:S177–80.
170
Copyright © ESPGHAN and NASPGHAN. All rights reserved.
JPGN  Volume 71, Number 2, August 2020
33. Veloso N, Silva JD, Goncalves L, et al. Trichotillomania and tricho-
phagia: the causes of Rapunzel syndrome. Rev Esp Enfrem Dig
2013;105:103–4.
34. El-Mazary AM, Ahmed FM, Hasanin A, et al. Rapunzel Syndrome in a
7-year-old Egyptian girl with primary biliary cirrhosis: a case report and
literature review. J Psychiatry 2014;17:10000155.
35. Irastroza I, Tutau C, Vitoria JC. A trichobezoar in a child with
undiagnosed celiac disease: a case report. World J Gastroenterol
2014;20:1357–60.
36. Mariotto A, Peretti M, Scire G, et al. Trichobezoars in children:
therapeutic complications. Pediatr Med Chir 2014;36:101.
37. Altonbary AY, Bahgat MH. Rapunzel syndrome. J Transl Int Med
2015;3:79–81.
38. Castle SL, Zmora O, Papillon S, et al. Management of complicated
gastric bezoars in children and adolescents. Isr Med Assoc J
2015;17:541–4.
39. Yik YI, How AK. Stomach trichobezoar (rapunzel syndrome) with
iatrogenic intussusception. Med J Malaysia 2016;71:74–6.
40. Kim SC, Kim SH, Kim SJ. A case report: Large trichobezoar causing
Rapunzel syndrome. Medicine (Baltimore) 2016;95:e3745.
41. Hamidi H, Muhammadi M, Saberi B, et al. A rare clinic entity: huge
trichobezoar. Int J Surg Case Rep 2016;28:127–30.
42. Paparoupa M, Schippert F. Trichobezoar. Mayo Clin Proc 2016;9:275–6.
43. Yik YI, How AK. A ‘‘hairy’’ problem: trichotillomania, trichophagia
and trichobezoars. Singapore Med J 2016;57:411.
44. Wang Z, Cao F, Liu D, et al. The diagnosis and treatment of Rapunzel
syndrome. Acta Radiol Open 2016;5:2058460115627660.
45. Nour I, Abd Alatef M, Megahed A, et al. Rapunzel syndrome (gastric
trichobezoar), a rare presentation with generalised oedema: case report
and review of the literature. Paediatr Int Child Health 2019;39:76–8.
46. Sailer S, de Carlos Vicente JC, Garcia AA, et al. Trichobezoar (Ra-
punzel syndrome) causing severe anemia and cardiac failure. Klin
Padiatr 2017;229:88–9.
47. Sun ZN, Hu DL, Chen ZM. Trichobezoar and Rapunzel syndrome.
World J Pediatr 2017;13:91.
48. Al-Osail EM, Zakary NY, Abdelhadi Y. Best management modality of
trichobezoar: a case report. Int J Surg Case Rep 2018;53:458–60.
49. Akbar S, Akbar L, Akbar N, et al. Trichobezoar-induced heartburn in a
teenage girl: a case report. J Investig Med High Impact Case Rep
2018;6:2124709618776345.
50. American Psychiatric Association. Diagnostic and Statistical Manual of
Mental Disorders (5th edition). American Psychiatric Association:
Washington, DC; 2013.
51. Larsson LT, Nivenius K, Wettrell G, et al. Trichobezoar in a child with
concomitant coeliac disease: a case report. Acta Paediatr 2004;93:278–80.
52. Marcos Alonso S, Bravo Mata M, Bautista Casasnova A, et al. Gastric
trichobezoar as an atypical form of presentatopn of celiac disease. An
Pediatr (Barc) 2005;62:601–2.
53. Carr JR, Sholevar EH, Baron DA. Trichotillomania and trichobezoar: a
clinical practice insight with report of illustrative case. J Am Osteopath
Assoc 2006;106:647–52.
54. McCallum IJ, Van zanten C, Inam IZ, et al. Trichobezoar in a child with
undiagnosed coeliac disease. J Paediatr Child Health 2008;44:524–5.
55. Kalyoncu T, Cildir DA, Ozabaran B. Trichotillomania in celiac disease
patient refractory to iron replacement. Int j Adolesc Med Health 2017.
56. Appak YC, Ertan D, Karakoyun M, et al. The cause of abdominal mass
in a child with celiac disease: Rapunzel syndrome. A case report. Sao
Paulo Med J 2018;137:292–4.
57. Benameur S, Bbabakhouya A, Es-Seddiki A, et al. Gastric trichobezoar
and celiac disease: a rare association in children. A case report and
review of literature. Med Ther 2018;24:101–4.
58. Adav SS, Subbaiaih RS, Kerk SK, et al. Studies on the proteome of
human hair - identifcation of histones and deamidated keratins. Sci Rep
2018;8:1599.
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