Trichobezoar: A Rare Cause of Protein-losing Enteropathy

**Background:** Protein-losing enteropathy (PLE) is a condition characterized by excessive protein loss through the gastrointestinal tract, leading to hypoalbuminemia. Common pediatric causes include cow milk protein allergy, celiac disease, inflammatory bowel disease, hypertrophic gastritis, intestinal lymphangiectasia, and right-sided heart dysfunction. Trichobezoar—a mass of ingested hair—is a rare cause of PLE. Bezoars most commonly form in the stomach and can extend into the small intestine (Rapunzel syndrome). This report presents a case of trichobezoar presenting as PLE with edema and anemia.

**Methods:** This is a single case report of a 12-year-old previously healthy male who presented with a 3-month history of bloating and fatigue and a 2-week history of bilateral lower extremity and scrotal edema. Physical examination revealed abdominal distention and bilateral lower extremity edema to the knees. Laboratory evaluation showed hemoglobin 11.5 g/dL (reference 12.1–16.1 g/dL), hematocrit 34.9% (36–47%), mean corpuscular volume 87.5 fL (78–95 fL), and albumin 1.5 g/dL (3.8–5.4 g/dL). Urinalysis was negative for protein. Iron studies were consistent with iron deficiency: serum iron 40 µg/dL (44–142 µg/dL), total iron binding capacity 155 mg/dL (224–435 mg/dL), and ferritin 9 ng/mL (10–300 ng/mL). Liver and kidney function were normal. Stool alpha-1-antitrypsin was elevated at 51.5 mg/g (<2.1 mg/g), confirming PLE. Abdominal ultrasound showed small bowel-to-small bowel intussusception without ischemia. CT of the abdomen revealed a mass filling and distending the stomach and extending through the proximal small bowel to the proximal jejunum, with diffuse wall thickening and mucosal hyperenhancement along the duodenum and jejunum. Small ascites and diffuse subcutaneous edema were present.

**Key Results:** The patient underwent exploratory laparotomy with gastrotomy for bezoar removal. The gastric portion of the trichobezoar measured 16 × 5 × 5 cm and retained the shape of the stomach. The small bowel extension measured 141 cm in length. Psychology was consulted during admission. The patient was discharged on ferrous sulfate. At follow-up, he remained asymptomatic without recurrence of edema, and hypoalbuminemia resolved. The authors also summarize 9 previously reported cases of trichobezoar presenting with edema and hypoalbuminemia (Table 1), all of which were treated surgically. Additionally, 10 cases of successful endoscopic removal are reviewed (Table 2), though the authors note that endoscopic removal is often prolonged, requires expert endoscopists, and is outnumbered by cases where endoscopic removal was unsuccessful.

**Clinical Implications:** Trichobezoar should be considered in the differential diagnosis of PLE, particularly in children and adolescents presenting with unexplained hypoalbuminemia, edema, and iron deficiency anemia. The authors speculate that the mechanism of PLE in this case involved mucosal irritation from the bezoar tail extending into the small bowel, early satiety and decreased food intake from the space-occupying gastric mass, and possible lymphatic obstruction. CT is the imaging modality of choice for diagnosis. Surgical removal via laparotomy remains the definitive treatment for large or extended trichobezoars, while endoscopic removal may be considered for small, intragastric bezoars in select cases. Underlying psychiatric disorders (e.g., trichotillomania) should be evaluated.