A rare giant gastric trichobezoar in a young female patient: Case report and review of the literature

Abstract A bezoar is an aggregate of undigested foreign materials that accumulate in the gastrointestinal tract and may cause serious symptoms or even life‐threatening complications. Trichobezoars, a subtype of bezoars, are a rare condition usually occurring in females with psychiatric disorders, with Rapunzel syndrome being an uncommon form of trichobezoar.

Temperature = 36.9°C, Heart Rate = 83, Respiratory Rate = 21, and Blood Pressure = 98/59 mmHg. The patient was taking no medication and had no other underlying medical condition or intellectual disability. Furthermore, she had no history of previous surgical interventions.
The clinical examination revealed halitosis, a mobile palpable mass occupying the region between the umbilicus and the xiphoid process with size 25 × 9 cm, normal bowel sounds, and no muscle contraction of the abdominal wall or any other signs indicative of peritonitis. In addition, there was no blood or palpable mass on digital rectal examination. No abnormal signs were found during physical examination of the cardiopulmonary and urogenital systems.
The blood tests revealed anemia (Hemoglobin concentration = 7.1 g/dl with normal values ranging between 11.9 and 14.7 g/dl and Hematocrit = 25.1% with normal values ranging between 36.8% and 45.6%). All other blood test results were falling within normal range (Table 1).
On admission, an abdominal X-ray was performed, but it was not diagnostic, followed by an abdominal ultrasound (U/S) which revealed a well-defined mass with dimensions 26cm × 9cm × 9cm in the epigastric region. To determine its origin and composition, an abdominal computerized tomography (CT) was ordered. The imaging results were in accordance with those of U/S, demonstrating the presence of a large, well-circumscribed, non-homogeneous mass, which lacked blood supply and occupied the whole stomach. The dimensions of that mass were 26,6cm × 9,7cm × 9,8cm. The images were strongly suggestive of a bezoar ( Figure 1). An upper GI endoscopy was performed on the second day after admission, confirming the diagnosis of trichobezoar, but failing to extract the mass of hairs. The patient underwent an exploratory laparotomy with an upper midline incision on the same day. Intraoperatively, the diagnosis of trichobezoar was confirmed. An intraluminal mass was seen and felt inside the stomach. A 10 cm incision was made in the anterior wall of the gastric antrum, and a massive trichobezoar in the shape of the stomach with size 27cm × 10cm × 10cm was extracted (Figures 2 and 3).
The patient had an uneventful recovery and was discharged on the 4th postoperative day. Intraoperatively she received a single dose of Cefoxitin 1g and Metronidazole 500 mg. Postoperatively, she received the same antibiotic combination for 3 consecutive days. Paracetamol and Tramadol were administered intravenously as analgesics, along with intravenous fluids, for the same number of days. Oral feeding was initiated on the 3rd postoperative day.
Despite the patient and her parents initially denying a history of trichophagia, it was later revealed, upon the first psychiatric consultation, that the patient was exhibiting both trichotillomania and trichophagia. Today, five years after the surgical procedure, she has been attending regular meetings with a psychiatrist.

| DISCUSSION
Bezoars of the GI tract are a rare pathological entity which presents mainly in the stomach. A recent study, extending over a period of more than 20 years, reports a total of 40 cases among 58.000 upper GI endoscopies, representing an incidence of 0.068%. 4 Interestingly, bezoars can form in any segment of the GI tract. 3 However, the stomach is the most common organ to be affected. 3 Bezoars can occasionally occur in individuals with normal GI anatomy and physiology. 1,5 Nevertheless, patients with altered anatomy or motility of the GI tract are at increased risk of developing them. 1,5 Several factors can contribute to the formation of bezoars. 3 Previous gastric surgery appears to be the most common risk factor, as it decreases the size of the stomach, interferes with the passage of gastric contents, and leads to a reduced secretion of gastric acid. 3 Other predisposing risk factors include diabetes mellitus, autoimmune diseases, peptic ulcer disease, Crohn's disease, carcinoma of the GI tract, hypothyroidism, and excessive fiber intake. 1 These pathologies can affect the pyloric sphincter's function, normal gastric pH, and gastric motility, leading to delayed stomach emptying. 1 A trichobezoar is a hair ball trapped in the gastrointestinal tract, affecting mostly females (in approximately 90% of cases) between the ages of 10 and 19 years, but only in half of them, a history of trichophagia is found. 6,7 The term trichobezoar was first described by Swain in 1854 following postmortem findings, in a patient presenting with acute abdomen. 7 Human hairs cannot be digested and are retained in the gastric folds, escaping from the peristaltic propulsion 2,7,8 ; thus, accumulating in the stomach where they are denatured and oxidized by gastric acid, and combined with food, form an entangled mass. 2,7,8 The accurate incidence of trichobezoars is unknown. From our review of the literature, we have concluded that this medical condition is not as uncommon as it is referred by several authors. It is difficult to establish how many distinct cases of trichobezoars have been reported, since most of them are published in medical journals which are found in small search platforms. Furthermore, some of these cases are repeated in different publications as case reports, case series, clinical studies, or review articles. The actual number of reported cases must be several hundred.
The diagnosis of a trichobezoar involves, firstly, a detailed patient's medical history, focusing primarily on their dietary habits. 2,7,8 Trichotillomania and trichophagia may be observed in female patients with concurrent psychological or behavioral conditions such as depression and personality disorders. 6,7 The physical examination, when a trichobezoar is suspected, may reveal severe halitosis and patchy hair loss. 7 Symptoms may include abdominal pain, a feeling of fullness in the epigastric region, nausea, vomiting, weight loss, dysphagia, or hematemesis; additional symptoms such as hypotension, altered mental state, and shock may develop due to life-threatening complications such as GI bleeding and obstruction, pancreatitis, necrosis of the visceral wall, and subsequent perforation. 1-3 Blood tests may be indicative of anemia, electrolyte disorders, the presence of trace element deficiencies, hypoproteinemia, and malabsorption of iron and vitamins. 2,7,8 A well-defined abdominal mass in the epigastric region is palpable in 85% of patients. 4 In our patient, the most prominent clinical signs and symptoms were vomiting, abdominal pain, nausea, anemia, and a palpable abdominal mass. The differential diagnosis of a trichobezoar should include other pathological entities such as different types of other bezoars, pancreatic and mesenteric cysts, pancreatic pseudocysts, gastric or pancreatic neoplasms, F I G U R E 1 CT scan showing a low-density intragastric mass which contains air bubbles and exhibits a characteristic mottled appearance. The arrow is demonstrating the intragastric mass F I G U R E 2 Intraoperative photograph of trichobezoar's subtraction through the gastrotomy F I G U R E 3 Trichobezoar after its removal from the stomach, with a 15 cm length ruler hydatid disease, volvulus (either cecal or intestinal), and, rarely, ovarian cysts and pregnancy. 14 An abdominal X-ray may give rise to clinical suspicion that a trichobezoar is present, while an abdominal U/S depicts the trichobezoar as a non-vascularized mass of the stomach or other hollow viscus. 1,2,4,7 In addition, an abdominal CT scan can reveal the presence of a mass and is very helpful in detecting small intestinal trichobezoars. 1,2,4,7 However, upper gastrointestinal endoscopy remains the diagnostic gold standard and can also be used for treatment in selected cases with small-sized trichobezoars. 1,2,4,6,7 Rapunzel syndrome is a rare form of a trichobezoar, with varying definitions. Some authors define the syndrome as a gastric trichobezoar with a tail extending into the jejunum or beyond it, whereas others describe it as a bezoar of any size which may cause intestinal obstruction. 2,8,14,15 It is a medical condition found mostly in females and from our extensive literature review, only seven cases of males with Rapunzel syndrome have been reported. [16][17][18][19][20][21] In a systematic review published in January 2020, a total of 110 cases with Rapunzel syndrome were reported by Janssen-Aguilar et al. 22 The treatment of a trichobezoar depends on its size, position, and consistency. Endoscopy usually fails to extract the mass of hair, but that depends on its size and density. 1,7,23 In contrast to phytobezoars, for which Coca-Cola and other enzymes such as cellulase, pancreatin, papain, and ursodeoxycholic acid have been used successfully for dissolution, trichobezoars are resistant to enzymatic degradation. [24][25][26] Surgery remains the preferred method of treatment as it offers the possibility of examining the entire gastrointestinal tract and is associated with higher success rates and a low percentage of complications. 9,27 In small-or moderate-sized trichobezoars, laparoscopy may be used. 28,29 The laparoscopic removal of giant gastric trichobezoars has been reported only by a few authors. 28,29 In up to 20% of patients, who have undergone removal of a trichobezoar either surgically or endoscopically, the condition may recur. [30][31][32] Recurrence may be anticipated when the patient's emotional or psychiatric disorder is not given adequate attention and properly managed through counseling and medication. [30][31][32]

| CONCLUSION
Trichobezoars of the GI tract are a rare medical condition but may pose a potentially serious health risk. It is crucial for physicians to be aware of the risk factors that lead to the development of trichobezoars as well as the subtle clinical findings which may facilitate early clinical investigation and diagnosis. Endoscopy can help reaching the final diagnosis and, in some cases, with the fragmentation and removal of the trichobezoar. Nevertheless, surgery remains the gold standard intervention for the treatment of this medical condition. For patients presenting with trichobezoars, psychiatric surveillance is advised as part of the treatment plan to prevent recurrence by targeting the underlying physical or emotional causes.