Case Report: Trichobezoar in a 25 year old Autistic Female

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A 25 year old female with autism was brought to the ER with worsening abdominal pain and decreased appetite. The primary historian was the patient’s father.  He described that his daughter began to have intermittent, cramping, non-radiating, upper abdominal pain three weeks prior to admission. It worsened in severity and duration over the last week. No nausea or vomiting occurred.  The pain was associated with a 10-pound weight loss over three weeks. Due to her decreased appetite, she was brought to the ER for evaluation.  Her medications included ferrous sulfate and buspirone. She had no history of prior surgery.

In the ER, her vitals were stable and she appeared in no distress. Her physical exam was significant for moderate diffuse abdominal tenderness without guarding or rigidity. A firm nontender well-defined mass in the region of her stomach was palpated.

Labs showed mild anemia with a hematocrit of 33%.  White blood cell count and basic metabolic chemistry panel were normal.  Chest x-ray was unremarkable as well. An abdominal x-ray showed no evidence of obstruction or pneumoperitoneum. However, a soft tissue density was visible in the left upper quadrant.

The patient was kept NPO and treated with intravenous fluid hydration.  A CT Scan of her abdomen showed a markedly distended stomach with a centrally located soft tissue abnormality, suggestive of a large gastric bezoar.

Upper endoscopy the following day revealed a large gastric trichobezoar that filled the gastric lumen. The pylorus was obstructed and therapeutic endoscopy was deemed inappropriate due to the large size of the trichobezoar.

The patient underwent exploratory laparotomy and gastrotomy. The trichobezoar was removed intact as a firm black mass. The patient’s post-operative course was uneventful. She received psychiatric counseling and was discharged home.

Discussion

Bezoars are rare masses formed of indigestible materials found in the gastrointestinal tract. The term Bezoar is believed to originate from the Arabic ‘Badzehr’, Persian ‘Padzahr’ or Turkish word ‘Panzehir’, all of which stand for a substance that acts as an antidote or counterpoison. ¹

Bezoar formation is rare in healthy subjects. They are believed to be a physical manifestation of an underlying psychological disorder. The first case of human trichobezoar was described in 1779, by Baudamant, ² while the first surgical removal of a trichobezoar was performed in 1883, by Schonborn. Incidence of bezoars peaks in the second decade. While 80% of cases occur before the age of 30 years, more than 90% of bezoars occur in adolescent girls. Bezoars, though benign may cause serious complications. Early detection and surgical removal is associated with less than 4% mortality.

Of the different types of bezoars, those formed of fruit and vegetable fibers, called phytobezoars, are the most common. Trichobezoars arise from aggregation of ingested hair with other indigestible organic fibers. Trichotillomania, the practice of habitually pulling hair out, in association with habitual ingestion of hair (trichophagia) can predispose to the formation of trichobezoars.  Other concretions may arise from ingestion of various indigestible foreign materials; for example, certain medicines can form pharmacobezoars.

Delay in gastric emptying due to diabetes mellitus, mixed connective tissue disease, prior gastric surgery with decreased acidity or hypothyroidism, can predispose to the formation of bezoars. ³  Trichobezoars are formed over time by recurrent ingestion of hair.  The hairball enlarges over time as it enmeshes within itself all other indigestible fibrous material.  If left untreated, it may grow large enough to occupy the entire stomach and extend into the duodenum and small intestine.

The most common presenting symptom is upper abdominal discomfort or pain and weight loss. Patients also experience stomach fullness, a “dragging” sensation in the upper abdomen, and periodic episodes of nausea and vomiting. If large in size; a uniformly firm, mobile, non tender abdominal mass may be palpable. A history of habitual ingestion of foreign substance like hair, or pica can usually be elicited and patients may be found have focal alopecia. ³

Bezoars are known to cause gastric ulcers, intestinal obstruction or perforation, hemorrhage, peritonitis, anemia, and malabsorption syndromes. Rarely, jaundice, pancreatitis, or colonic obstruction may result if large bezoars develop. ^{3 4}  Rapunzel syndrome is a rare clinical scenario that arises when a large stomach trichobezoar mass extends through the duodenum and into the small intestine with or without mechanical intestinal obstruction. ⁵  It derives its name from the long – haired heroine in the fairy tale by the Grimm brothers.

Patient may be found to have mild anemia or leukocytosis. An abdominal x-ray and upper gastrointestinal contrast study usually shows a heterogenous density in the stomach. An abdominal ultrasound would confirm an intraluminal mass with a hyper- echoic arc-like surface and a marked acoustic shadow suggestive of a bezoar. An abdominal CT scan can delineate a well-defined oval intraluminal mass with air bubbles retained within the interstice or a homogenous mottled appearance in the region of the stomach or intestine. ⁶ Upper gastrointestinal contrast study would delineate a filling defect outlined by the barium, but is usually not necessary.

Presence of a ball of hair or a mobile mass within the stomach cavity directly visualized by upper endoscopy is confirmatory. A dark greenish brown or black mass with a slimy surface and a strong odor secondary to decomposition of various organic residue interspersed with hair is typically identified on upper endoscopy. ⁷ Upper endoscopy enables one not only to classify the type of bezoar depending upon its composition but also helps in deciding on the further mode of treatment.

Removal of the bezoar is the primary treatment goal. Small bezoars may be fragmented into smaller pieces and aspirated endoscopically. Dissolution with papain saline, acetylcysteine, and cellulase are generally used for small phytobezoars. ^{8 9} Various new techniques have been developed to tackle the issue of bezoars with variable successes like water jet, bedside coca-cola lavage, ³ direct large channel endoscopic aspiration, ¹⁰ Dormia basket, ¹¹ forceps, polypectomy snare, ¹² Nd: YAG laser therapy, use of modified needle – knife [bezotome] and modified lithotripter [bezotriptor]. ¹³

However, endoscopic modalities are of limited use because the bezoar must be small and soft. Gastrotomy is the modality of choice for removal of recurrent or large solid bezoars. ⁸  Enterotomy may be required when bezoars extend into the small intestine. Recurrence cannot be avoided unless the underlying behavior is corrected. Behavioral training, in addition to serotonin re-uptake inhibitors or neuroleptic agents helps to treat mood disorders and prevent recurrence. Prognosis is excellent after removal of the mass, psychiatric counseling, and regular follow up.