Surg Today DOI 10.1007/s00595-014-0922-4
CASE REPORT
Double and synchronous trichobezoars causing small-bowel obstruction and detected by multidetector computed tomography: report of two cases Shigeyoshi Aoi • Kouseki Kimura • Tomoki Tsuda
Received: 18 September 2013 / Accepted: 26 December 2013 Ó Springer Japan 2014
Abstract A trichobezoar is a rare mass formed by the ingestion and accumulation of hair within the gastrointestinal tract, especially the stomach. Cases of an isolated gastric trichobezoar with extension into the duodenum or the jejunum have been reported; however, synchronous gastric and intestinal trichobezoars causing a small-bowel obstruction is very unusual. We report our experience of two such cases to demonstrate the efficiency of preoperative multidetector computed tomography in locating the double bezoars and assisting us in surgical decision making. Open surgery is inevitable for symptomatic bezoars, because the masses are too hard and large to break up with endoscopic devices. Keywords Trichobezoar Diagnosis Small-bowel obstruction
Introduction Trichobezoars often form in patients who suffer from trichotillomania combined with trichophagia. Trichobezoars typically form in the stomach and may cause abdominal pain, anorexia, and eventually, vomiting. If they enlarge, gastric outlet obstruction or bleeding and perforation will occur. Trichobezoars rarely cause small-bowel obstruction (SBO); however, we recently treated two children who presented with an SBO, found to be caused by double and synchronous trichobezoars in the stomach
S. Aoi (&) K. Kimura T. Tsuda Department of Pediatric Surgery, Omihachiman Community Medical Center, 1379 Tsuchida-cho, Omihachiman, Shiga 523-0082, Japan e-mail: [email protected]
and ileum. Preoperative multidetector computed tomography (MDCT) images showed the trichobezoars clearly in the small intestine, which assisted us in choosing the optimal surgical approach.
Case report Case 1 A 13-year-old girl with a 3-week history of epigastralgia and appetite loss with non-bloody or bilious vomiting over the past few days was taken to a local hospital. A hard mass was detected in her epigastrium, and she was transferred to our hospital for investigation and treatment. On admission, she complained of crampy abdominal pain in the epigastric region. She had suffered from trichophagia since the age of 9 and had been absent from school for 1 year. There was no obvious hair loss from her scalp. Mild abdominal distention and a firm mass were evident in her epigastrium and the left lower abdominal quadrant, with moderate tenderness. CT images from the previous hospital showed two heterogeneous air-containing intraluminal mottled masses in the stomach and small intestine, without ascites. Laboratory findings were unremarkable. Based on her history and the CT images, we diagnosed double trichobezoars in the stomach and ileum. As her abdominal condition did not appear to be urgent, we initially planned endoscopic removal of the gastric trichobezoar and expected that the ileal trichobezoar would be excreted fecally with the aid of glycerin enemas. However, after two glycerin enemas, her abdominal pain worsened dramatically. MDCT was performed as we suspected the development of an SBO with impending intestinal perforation. The MDCT showed new ascites in her pelvic cavity with a markedly dilated small
123
Surg Today Fig. 1 Case 1: coronal view of the abdominal computed tomography scan of a 13-yearold girl on admission, showing two trichobezoars: one in the stomach (asterisk) and one in the ileum (arrow), causing small-bowel obstruction and ascites, with collapse of the large intestine
lumen. We pulled out the trichobezoars through a transverse gastrotomy and ileotomy, respectively. The bezoars were too hard and tight to be grasped or divided, even with large scissors and forceps. The mass extracted from the stomach weighed 370 g and measured 19 9 8 9 6.5 cm (Fig. 2), and that extracted from the ileum weighed 170 g and measured 20 9 4.5 9 4 cm. The patient was discharged uneventfully 2 weeks later and provided with ongoing psychiatric support. Case 2
Fig. 2 The large trichobezoar from this patient (Case 1) weighed 370 g, measured 19 9 8 9 6.5 cm, and conformed to the shape of the stomach
intestine (Fig. 1). The patient underwent emergency laparotomy via a 5-cm upper abdominal midline incision, which was the smallest diameter of the bezoars. Fortunately, no perforation had occurred. One trichobezoar was identified in her stomach and another in her ileum, 90 cm proximal to the ileocecal valve. The trichobezoar in the ileum was completely impacted and immovable in the
123
A 4-year-old girl was taken to a local hospital with a 5-day history of non-bilious vomiting without diarrhea. A plain abdominal X-ray showed an SBO and she was transferred to our hospital for surgery. An interview with her mother disclosed that the child had exhibited episodes of trichophagia for about 1 year when she was 2 years old, just after her younger brother was born. The girl had apparently stopped the habit after a year without any abdominal symptoms. On admission, there was no obvious hair loss from her scalp. There were two palpable masses in her epigastrium and right lower quadrant of the abdomen, with moderate tenderness. As we suspected that the trichobezoars had caused the SBO, MDCT was performed to obtain a definitive diagnosis. The MDCT images showed two aircontaining intraluminal masses in her stomach and small intestine as well as a dilated small intestine with ascites (Fig. 3). She underwent emergency surgery for the SBO through a 3-cm upper transverse incision, according to the
Surg Today Fig. 3 Case 2: coronal view plain abdominal computed tomography scan of a 4-year-old girl, showing two trichobezoars: one in the stomach (asterisk) and one in the ileum (arrow), with a markedly dilated small bowel and ascites
CT images. Trichobezoars were found in the stomach and the ileum, 50 cm proximal to the ileocecal valve. The ileal trichobezoar was completely impacted in the lumen and immovable. We removed the two masses through the transverse gastrotomy and ileotomy, respectively. The mass in the stomach weighed 75 g and measured 8.2 9 3.5 9 3.2 cm, and that in the ileum weighed 25 g and measured 4.0 9 3.0 9 2.8 cm. The patient was discharged uneventfully 12 days later. As she no longer suffered from trichophagia, the psychiatric support provided to her was kept to a minimum.
Discussion Trichobezoars form after the long-term ingestion of hair. In a large-scale review, DeBakey and Ochsner [1] reported that about 80 % of individuals in whom trichobezoars developed were younger than 30 years, and 90 % were female. Almost all patients with a trichobezoar suffer from trichotillomania. About 1 % of the population has trichotillomania and about one-third of these individuals exhibit trichophagia. Approximately 1 % of these patients eat so much hair that need they need to be operated upon [2]. Gastric trichobezoars cause epigastric pain, nausea, vomiting, and anorexia and, as they grow larger, the gastric outlet becomes affected [3–5]. However, SBO occurs in fewer than 10 % of patients with trichobezoars [6, 7], although simultaneous trichobezoars in the stomach and small intestine have been reported previously [3, 8, 9].
Before any examinations are performed, it is essential to interview the patient’s family. Neither of our patients had any evidence of scalp hair loss on their initial presentation. In case 1, the trichobezoars were considered to be composed of pubic hair, which was confirmed in the operation room. In case 2, the trichobezoars might have affected the patient even 2 years after the trichophagia had stopped. As a diagnostic modality, MDCT clearly shows the characteristic findings of a trichobezoar; namely, a large heterogeneous, mottled, mesh-like, air-trapping mass at the site of obstruction [10, 11]. MDCT can provide us with more information than conventional radiography, so that spontaneously fragmented and impacted pieces of the trichobezoars will not be overlooked, whereby re-operation is avoided, as reported by Hoover [9]. When one of synchronous trichobezoars has obstructed the small bowel, the gastric trichobezoar might conceal clinical symptoms, such as bilious emesis, and delay diagnosis. If whole abdominal MDCT is performed, it will show SBO and pinpoint the exact location of the bezoars. In both our patients, MDCT clearly identified the pathogenic trichobezoars, which allowed us to initiate appropriate surgical intervention. Although the incidence of double trichobezoars as a cause of SBO is low, we stress the importance of preoperative examinations of the whole abdominal cavity before removing gastric trichobezoars. MDCT clearly shows the size and density of the hair in the mass and guides us as to the correct skin incision site. By defining the extreme density and size of the bezoars, the MDCT allows us to avoid unsuccessful attempts at endoscopic or laparoscopic extraction.
123
Surg Today
In the course of trichophagia, gastric trichobezoars can tear and become impacted in the small intestine, resulting in SBO. However, the mechanisms responsible for pieces tearing off the gastric trichobezoars are unclear. In case 2, the mother of the patient told us after the operation that the patient’s 2-year-old brother had accidentally hit the patient in her abdomen 1 week before her admission. This might explain how some ileal trichobezoars develop; however, the bezoars we removed were very hard and would not have broken with such weak impact. In a report by Coufal et al. [12], a gastric bezoar in a patient presenting with SBO moved between the ileum and the stomach, so extension of a gastric bezoar into the duodenum, like the ‘‘Rapunzel syndrome’’ may not cause SBO, unless it continues to move away from the stomach and separates. As trichobezoars cannot be dissolved by enzymatic or chemical methods, small gastric trichobezoars may be able to be removed using mechanical fragmentation endoscopically [13]. However, large and symptomatic trichobezoars must be removed surgically via an open or laparoscopic approach [14, 15]. As demonstrated in the two cases we reported, symptomatic trichobezoars that cause intestinal obstruction are too hard and large to cut, even with surgical scissors, and need to be removed by an open surgical approach. Obviously, it is important to prevent the further formation of trichobezoars through psychiatric support. Conflict of interest Shigeyoshi Aoi and his co-authors have no conflicts of interest to declare.
References 1. DeBakey M, Ochsner A. Bezoars and concretions: a comprehensive review of the literature with analysis of 303 collected
123
2. 3.
4. 5.
6.
7.
8.
9.
10.
11.
12.
13.
14.
15.
cases and a presentation of 8 additional cases. Surgery. 1939;5:132–60. Frey AS, McKee M, King RA, Martin A. Hair apparent: rapunzel syndrome. Am J Psychiatry. 2005;162:242–8. Alsafwah S, Alzein M. Small bowel obstruction due to trichobezoar: role of upper endoscopy in diagnosis. Gastrointest Endosc. 2000;52:784–6. O’Sullivan MJ, McGreal G, Walsh JG, Redmond HP. Trichobezoar. J R Soc Med. 2001;94:68–70. Jensen AR, Trankiem CT, Lebovitch S, Grewal H. Gastric outlet obstruction secondary to a large trichobezoar. J Pediatr Surg. 2005;40:1364–5. Vaughan ED, Sawyers JL, Scott HW. The Rapunzel syndrome. an unusual complication of intestinal bezoar. Surgery. 1968;63:339–43. Krol CM, Karol I, Khalil H, Shah S. Small-bowel obstruction due to migratory trichobezoar in a child: CT diagnosis. Am J Roentgenol. 2001;177:255–6. Klipfel AA, Kessler E, Schein M. Rapunzel syndrome causing gastric emphysema and small bowel obstruction. Surgery. 2003;133:120–1. Hoover K, Piotrowski J, Pierre K, Katz A, Goldstein AM. Simultaneous gastric and small intestinal trichobezoars—a hairy problem. J Pediatr Surg. 2006;41:1495–7. West WM, Duncan ND. CT appearances of the Rapunzel syndrome: an unusual form of bezoar and gastrointestinal obstruction. Pediatr Radiol. 1998;28:315–6. Newman B, Girdany BR. Gastric trichobezoars—sonographic and computed tomographic appearance. Pediatr Radiol. 1990;20:526–7. Coufal NG, Kansagra AP, Doucet J, Lee J, Coimbra R, Bansal V. Gastric trichobezoar causing intermittent small bowel obstruction: report of a case and review of the literature. Case Rep Med. 2011;. doi:10.1155/2011/217570. De Backer 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. Nirasawa Y, Mori T, Ito Y, Tanaka H, Seki N, Atomi Y. Laparoscopic removal of a large gastric trichobezoar. J Pediatr Surg. 1998;33:663–5. Hussain AA, Geddoa E, Abood M, Alazzawy MT. Trichobezoar causing small-bowel obstruction. S Afr Med J. 2007;97:343–4.
CASE REPORT
Double and synchronous trichobezoars causing small-bowel obstruction and detected by multidetector computed tomography: report of two cases Shigeyoshi Aoi • Kouseki Kimura • Tomoki Tsuda
Received: 18 September 2013 / Accepted: 26 December 2013 Ó Springer Japan 2014
Abstract A trichobezoar is a rare mass formed by the ingestion and accumulation of hair within the gastrointestinal tract, especially the stomach. Cases of an isolated gastric trichobezoar with extension into the duodenum or the jejunum have been reported; however, synchronous gastric and intestinal trichobezoars causing a small-bowel obstruction is very unusual. We report our experience of two such cases to demonstrate the efficiency of preoperative multidetector computed tomography in locating the double bezoars and assisting us in surgical decision making. Open surgery is inevitable for symptomatic bezoars, because the masses are too hard and large to break up with endoscopic devices. Keywords Trichobezoar Diagnosis Small-bowel obstruction
Introduction Trichobezoars often form in patients who suffer from trichotillomania combined with trichophagia. Trichobezoars typically form in the stomach and may cause abdominal pain, anorexia, and eventually, vomiting. If they enlarge, gastric outlet obstruction or bleeding and perforation will occur. Trichobezoars rarely cause small-bowel obstruction (SBO); however, we recently treated two children who presented with an SBO, found to be caused by double and synchronous trichobezoars in the stomach
S. Aoi (&) K. Kimura T. Tsuda Department of Pediatric Surgery, Omihachiman Community Medical Center, 1379 Tsuchida-cho, Omihachiman, Shiga 523-0082, Japan e-mail: [email protected]
and ileum. Preoperative multidetector computed tomography (MDCT) images showed the trichobezoars clearly in the small intestine, which assisted us in choosing the optimal surgical approach.
Case report Case 1 A 13-year-old girl with a 3-week history of epigastralgia and appetite loss with non-bloody or bilious vomiting over the past few days was taken to a local hospital. A hard mass was detected in her epigastrium, and she was transferred to our hospital for investigation and treatment. On admission, she complained of crampy abdominal pain in the epigastric region. She had suffered from trichophagia since the age of 9 and had been absent from school for 1 year. There was no obvious hair loss from her scalp. Mild abdominal distention and a firm mass were evident in her epigastrium and the left lower abdominal quadrant, with moderate tenderness. CT images from the previous hospital showed two heterogeneous air-containing intraluminal mottled masses in the stomach and small intestine, without ascites. Laboratory findings were unremarkable. Based on her history and the CT images, we diagnosed double trichobezoars in the stomach and ileum. As her abdominal condition did not appear to be urgent, we initially planned endoscopic removal of the gastric trichobezoar and expected that the ileal trichobezoar would be excreted fecally with the aid of glycerin enemas. However, after two glycerin enemas, her abdominal pain worsened dramatically. MDCT was performed as we suspected the development of an SBO with impending intestinal perforation. The MDCT showed new ascites in her pelvic cavity with a markedly dilated small
123
Surg Today Fig. 1 Case 1: coronal view of the abdominal computed tomography scan of a 13-yearold girl on admission, showing two trichobezoars: one in the stomach (asterisk) and one in the ileum (arrow), causing small-bowel obstruction and ascites, with collapse of the large intestine
lumen. We pulled out the trichobezoars through a transverse gastrotomy and ileotomy, respectively. The bezoars were too hard and tight to be grasped or divided, even with large scissors and forceps. The mass extracted from the stomach weighed 370 g and measured 19 9 8 9 6.5 cm (Fig. 2), and that extracted from the ileum weighed 170 g and measured 20 9 4.5 9 4 cm. The patient was discharged uneventfully 2 weeks later and provided with ongoing psychiatric support. Case 2
Fig. 2 The large trichobezoar from this patient (Case 1) weighed 370 g, measured 19 9 8 9 6.5 cm, and conformed to the shape of the stomach
intestine (Fig. 1). The patient underwent emergency laparotomy via a 5-cm upper abdominal midline incision, which was the smallest diameter of the bezoars. Fortunately, no perforation had occurred. One trichobezoar was identified in her stomach and another in her ileum, 90 cm proximal to the ileocecal valve. The trichobezoar in the ileum was completely impacted and immovable in the
123
A 4-year-old girl was taken to a local hospital with a 5-day history of non-bilious vomiting without diarrhea. A plain abdominal X-ray showed an SBO and she was transferred to our hospital for surgery. An interview with her mother disclosed that the child had exhibited episodes of trichophagia for about 1 year when she was 2 years old, just after her younger brother was born. The girl had apparently stopped the habit after a year without any abdominal symptoms. On admission, there was no obvious hair loss from her scalp. There were two palpable masses in her epigastrium and right lower quadrant of the abdomen, with moderate tenderness. As we suspected that the trichobezoars had caused the SBO, MDCT was performed to obtain a definitive diagnosis. The MDCT images showed two aircontaining intraluminal masses in her stomach and small intestine as well as a dilated small intestine with ascites (Fig. 3). She underwent emergency surgery for the SBO through a 3-cm upper transverse incision, according to the
Surg Today Fig. 3 Case 2: coronal view plain abdominal computed tomography scan of a 4-year-old girl, showing two trichobezoars: one in the stomach (asterisk) and one in the ileum (arrow), with a markedly dilated small bowel and ascites
CT images. Trichobezoars were found in the stomach and the ileum, 50 cm proximal to the ileocecal valve. The ileal trichobezoar was completely impacted in the lumen and immovable. We removed the two masses through the transverse gastrotomy and ileotomy, respectively. The mass in the stomach weighed 75 g and measured 8.2 9 3.5 9 3.2 cm, and that in the ileum weighed 25 g and measured 4.0 9 3.0 9 2.8 cm. The patient was discharged uneventfully 12 days later. As she no longer suffered from trichophagia, the psychiatric support provided to her was kept to a minimum.
Discussion Trichobezoars form after the long-term ingestion of hair. In a large-scale review, DeBakey and Ochsner [1] reported that about 80 % of individuals in whom trichobezoars developed were younger than 30 years, and 90 % were female. Almost all patients with a trichobezoar suffer from trichotillomania. About 1 % of the population has trichotillomania and about one-third of these individuals exhibit trichophagia. Approximately 1 % of these patients eat so much hair that need they need to be operated upon [2]. Gastric trichobezoars cause epigastric pain, nausea, vomiting, and anorexia and, as they grow larger, the gastric outlet becomes affected [3–5]. However, SBO occurs in fewer than 10 % of patients with trichobezoars [6, 7], although simultaneous trichobezoars in the stomach and small intestine have been reported previously [3, 8, 9].
Before any examinations are performed, it is essential to interview the patient’s family. Neither of our patients had any evidence of scalp hair loss on their initial presentation. In case 1, the trichobezoars were considered to be composed of pubic hair, which was confirmed in the operation room. In case 2, the trichobezoars might have affected the patient even 2 years after the trichophagia had stopped. As a diagnostic modality, MDCT clearly shows the characteristic findings of a trichobezoar; namely, a large heterogeneous, mottled, mesh-like, air-trapping mass at the site of obstruction [10, 11]. MDCT can provide us with more information than conventional radiography, so that spontaneously fragmented and impacted pieces of the trichobezoars will not be overlooked, whereby re-operation is avoided, as reported by Hoover [9]. When one of synchronous trichobezoars has obstructed the small bowel, the gastric trichobezoar might conceal clinical symptoms, such as bilious emesis, and delay diagnosis. If whole abdominal MDCT is performed, it will show SBO and pinpoint the exact location of the bezoars. In both our patients, MDCT clearly identified the pathogenic trichobezoars, which allowed us to initiate appropriate surgical intervention. Although the incidence of double trichobezoars as a cause of SBO is low, we stress the importance of preoperative examinations of the whole abdominal cavity before removing gastric trichobezoars. MDCT clearly shows the size and density of the hair in the mass and guides us as to the correct skin incision site. By defining the extreme density and size of the bezoars, the MDCT allows us to avoid unsuccessful attempts at endoscopic or laparoscopic extraction.
123
Surg Today
In the course of trichophagia, gastric trichobezoars can tear and become impacted in the small intestine, resulting in SBO. However, the mechanisms responsible for pieces tearing off the gastric trichobezoars are unclear. In case 2, the mother of the patient told us after the operation that the patient’s 2-year-old brother had accidentally hit the patient in her abdomen 1 week before her admission. This might explain how some ileal trichobezoars develop; however, the bezoars we removed were very hard and would not have broken with such weak impact. In a report by Coufal et al. [12], a gastric bezoar in a patient presenting with SBO moved between the ileum and the stomach, so extension of a gastric bezoar into the duodenum, like the ‘‘Rapunzel syndrome’’ may not cause SBO, unless it continues to move away from the stomach and separates. As trichobezoars cannot be dissolved by enzymatic or chemical methods, small gastric trichobezoars may be able to be removed using mechanical fragmentation endoscopically [13]. However, large and symptomatic trichobezoars must be removed surgically via an open or laparoscopic approach [14, 15]. As demonstrated in the two cases we reported, symptomatic trichobezoars that cause intestinal obstruction are too hard and large to cut, even with surgical scissors, and need to be removed by an open surgical approach. Obviously, it is important to prevent the further formation of trichobezoars through psychiatric support. Conflict of interest Shigeyoshi Aoi and his co-authors have no conflicts of interest to declare.
References 1. DeBakey M, Ochsner A. Bezoars and concretions: a comprehensive review of the literature with analysis of 303 collected
123
2. 3.
4. 5.
6.
7.
8.
9.
10.
11.
12.
13.
14.
15.
cases and a presentation of 8 additional cases. Surgery. 1939;5:132–60. Frey AS, McKee M, King RA, Martin A. Hair apparent: rapunzel syndrome. Am J Psychiatry. 2005;162:242–8. Alsafwah S, Alzein M. Small bowel obstruction due to trichobezoar: role of upper endoscopy in diagnosis. Gastrointest Endosc. 2000;52:784–6. O’Sullivan MJ, McGreal G, Walsh JG, Redmond HP. Trichobezoar. J R Soc Med. 2001;94:68–70. Jensen AR, Trankiem CT, Lebovitch S, Grewal H. Gastric outlet obstruction secondary to a large trichobezoar. J Pediatr Surg. 2005;40:1364–5. Vaughan ED, Sawyers JL, Scott HW. The Rapunzel syndrome. an unusual complication of intestinal bezoar. Surgery. 1968;63:339–43. Krol CM, Karol I, Khalil H, Shah S. Small-bowel obstruction due to migratory trichobezoar in a child: CT diagnosis. Am J Roentgenol. 2001;177:255–6. Klipfel AA, Kessler E, Schein M. Rapunzel syndrome causing gastric emphysema and small bowel obstruction. Surgery. 2003;133:120–1. Hoover K, Piotrowski J, Pierre K, Katz A, Goldstein AM. Simultaneous gastric and small intestinal trichobezoars—a hairy problem. J Pediatr Surg. 2006;41:1495–7. West WM, Duncan ND. CT appearances of the Rapunzel syndrome: an unusual form of bezoar and gastrointestinal obstruction. Pediatr Radiol. 1998;28:315–6. Newman B, Girdany BR. Gastric trichobezoars—sonographic and computed tomographic appearance. Pediatr Radiol. 1990;20:526–7. Coufal NG, Kansagra AP, Doucet J, Lee J, Coimbra R, Bansal V. Gastric trichobezoar causing intermittent small bowel obstruction: report of a case and review of the literature. Case Rep Med. 2011;. doi:10.1155/2011/217570. De Backer 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. Nirasawa Y, Mori T, Ito Y, Tanaka H, Seki N, Atomi Y. Laparoscopic removal of a large gastric trichobezoar. J Pediatr Surg. 1998;33:663–5. Hussain AA, Geddoa E, Abood M, Alazzawy MT. Trichobezoar causing small-bowel obstruction. S Afr Med J. 2007;97:343–4.
