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Int J Surg Case Rep
Int J Surg Case Rep
International Journal of Surgery Case Reports
2210-2612
Elsevier

S2210-2612(24)01064-2
10.1016/j.ijscr.2024.110283
110283
Case Report
A giant trichobezoar in a child with attention deficit hyperactivity disorder: A case report
Forooghi M. a
Shahrokhi R. b
Yousufzai Sh. Yousefzaishayan@sums.ac.ir
b⁎
a Pediatric Surgery Department, Shiraz University of Medical Sciences, Shiraz 7138433608, Iran
b Student Research Committee, Shiraz University of Medical Sciences, Shiraz 7138433608, Iran
⁎ Corresponding author. Yousefzaishayan@sums.ac.ir
11 9 2024
10 2024
11 9 2024
123 1102838 8 2024
5 9 2024
9 9 2024
© 2024 The Authors
2024
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Introduction and importance

Trichobezoars are uncommon gastrointestinal concretions that develop due to the accumulation of ingested hair, predominantly affecting young females with concurrent psychiatric conditions like trichotillomania and Trichophagia. These masses typically form in the stomach but may also protrude into the intestines, potentially leading to severe complications if not promptly identified and treated. The present case study outlines the clinical manifestation, diagnostic complexities, surgical intervention, and postoperative care of a 10-year-old female patient with a substantial gastric trichobezoar, underscoring the significance of considering this disorder in pediatric individuals with gastrointestinal manifestations and a psychiatric history.

Case presentation

The research article details the case of a 10-year-old female patient with a multifaceted medical history, which includes attention-deficit/hyperactivity disorder (ADHD), chronic constipation, growth retardation, and a background of neonatal seizures. She was admitted to the emergency department presenting with epigastric pain, nausea, and vomiting. Notably, the patient displayed pica-like behaviors, specifically the ingestion of hair, which ultimately led to the diagnosis of a trichobezoar, a form of gastric obstruction resulting from the accumulation of ingested hair. Diagnostic imaging techniques, including abdominal X-ray and computed tomography (CT) scan, confirmed the presence of a substantial trichobezoar within the stomach, necessitating surgical intervention via exploratory laparotomy for removal. The pathological examination of the excised mass revealed a considerable quantity of hair intermingled with fecal matter. Following the surgical procedure, the patient exhibited a positive recovery trajectory and was subsequently referred for psychiatric assessment and behavioral therapy to address the underlying issues associated with trichotillomania, with the objective of preventing future occurrences. This case underscores the critical need for the identification and management of behavioral concerns in pediatric patients exhibiting similar clinical presentations.

Discussion

The patient exhibited symptoms such as epigastric pain, nausea, and vomiting, in addition to a medical history of Attention Deficit Hyperactivity Disorder (ADHD) and pica-like behaviors. Diagnostic imaging confirmed the presence of a large trichobezoar, leading to the necessity of an exploratory laparotomy for its surgical removal. This case highlights the crucial role of comprehensive clinical assessment and imaging in the diagnosis of trichobezoars, especially in children with behavioral concerns. Managing such cases typically involves a multidisciplinary approach that combines surgical procedures with psychiatric assessment and behavioral therapy to reduce the likelihood of recurrence.

Conclusion

This case underscores the importance of considering trichobezoars in the list of potential diagnoses for pediatric patients exhibiting gastrointestinal symptoms, particularly in individuals with psychiatric or neurodevelopmental histories. Timely identification and management are crucial in averting serious complications linked to trichobezoars. The favorable surgical result in this particular patient underscores the significance of holistic care that attends to both the physical and psychological dimensions of the condition, ultimately enhancing the long-term outlook. Additional research is required to explore the relationship between underlying psychiatric conditions, particularly ADHD, and the formation of trichobezoars. Furthermore, novel therapeutic strategies need to be developed.

Highlights

• Trichobezoars are rare but serious clinical entities in pediatric patients, often linked to psychiatric disorders like trichotillomania and Trichophagia

• This case describes a 10-year-old girl with ADHD who presented with epigastric pain, nausea, vomiting, and a palpable abdominal mass. Imaging confirmed a large trichobezoar after which our patient underwent successful surgical removal of the trichobezoar via laparotomy and gastrostomy.

• Psychiatric evaluation and long-term follow-up are essential to address underlying behavioral issues and prevent recurrence, which can occur in up to 20% of cases

• Further research is needed to investigate the relationship between ADHD and trichobezoar formation, as well as explore novel minimally invasive techniques for removal

Keywords

Trichophagia
Trichobezoar
Trichotillomania
Pediatrics surgery
Psychiatry
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pmc1 Introduction

Trichobezoars, rare but significant clinical entities, are composed of ingested hair, usually accumulating in the stomach and potentially extending into the jejunum, ileum, or colon. They are most common in young females and often linked to psychiatric disorders like trichotillomania and Trichophagia, where patients compulsively pull out and ingest their hair [1]. Trichobezoars, although rare, can cause serious gastrointestinal issues like obstruction, perforation, and bleeding if not promptly identified and treated. If left untreated, trichobezoars can grow larger and heavier due to the continuous ingestion of hair, raising the risk of severe complications such as gastric mucosal erosion, ulcers, and perforation of the stomach or small intestine. Other reported complications include intussusception, volvulus, cholangitis, pancreatitis, and even death [2,3]. The term “bezoar” comes from the Persian word “pādzahr,” meaning “antidote,” historically referring to any mass in the gastrointestinal tract believed to have medicinal properties. Modern medicine recognizes various types of bezoars, such as phytobezoars (plant material), pharmacobezoars (medications), and trichobezoars (hair) [4,5]. This case report discusses the clinical presentation, diagnostic workup, surgical management, and postoperative care of a 10-year-old girl with a large gastric trichobezoar.

2 Case presentation

A 10-year-old girl with a history of ADHD, chronic constipation, growth retardation, and a neonatal seizure presented to the emergency department with a three-day history of epigastric pain, nausea, and vomiting. Her parents reported that she often does not chew her food properly and has exhibited pica-like behaviors, including hair ingestion when stressed. The patient was asymptomatic until three days prior to admission when she developed epigastric pain, which was intermittently relieved by defecation and was associated with early satiety. Accompanying symptoms included nausea and vomiting. Her parents reported a decreased appetite but no objective measurement of weight was available. There was no history of rectal bleeding, diarrhea, or urinary symptoms. As her parents noticed a bulging in her upper abdomen, she was taken to a pediatrician who referred them to a pediatric surgeon with the impression of a fecal mass on examination. On examination, a mass was palpated in her epigastric area. An abdominal X-ray was requested, which showed a mass in her stomach. Considering the patient's previous history of hair ingestion, she was admitted to the pediatric surgery ward with the impression of a trichobezoar.

The patient was delivered by C-section and had a medical history of a seizure in infancy, likely due to asphyxia, chronic constipation, growth retardation, and ADHD. She was taking Risperidone and Ritalin for ADHD. There were no previous surgeries, and her immunizations were current. There was no significant family history of medical or psychiatric illnesses. On physical examination, the patient appeared well, sitting comfortably on the bed. She weighed 20 kg and was 130 cm tall, below the 5th and 10th percentiles for her age. Her weight z-score for height was >−3. Vital signs were stable. No bald patches or thinning of hair or eyebrows were apparent. Abdominal examination showed a firm, non-tender mass in the epigastric region. Cardiopulmonary examination was normal, with regular heart and lung sounds.

3 Patient workup

The abdominal X-ray showed a large, radiopaque mass in the epigastric region with a heterogeneous shadow (Fig. 1). No signs of free air under the diaphragm were found, indicating no gastrointestinal perforation. The absence of an air-fluid level suggested no obstruction. An abdominopelvic CT scan with IV contrast revealed a well-defined, heterogeneous mass in the stomach with mixed density, consistent with a trichobezoar (Fig. 2). The mass occupied a significant portion of the gastric lumen, causing stomach distension. A rim of fluid was observed in the pelvic cavity. Laboratory tests indicated a hemoglobin level of 10.2 g/dL, with an MCV of 59.2 and MCH of 18. The white blood cell count was normal. Kidney function tests and serum electrolytes were within normal ranges. Urinalysis showed 3+ glucose and ketones. The clinical presentation and physical examination findings suggested a gastric bezoar. Subsequent diagnostic imaging confirmed a large trichobezoar in the stomach. The patient underwent exploratory laparotomy under general anesthesia. A large trichobezoar was found in the stomach and removed through gastrostomy after a high midline incision (Fig. 3). The incision site was repaired using the Lambert method with PDS. The pathology report confirmed the trichobezoar as a 180 × 50 × 50 mm fragment of hair mixed with fecal matter. The patient recovered without any issues after surgery and was monitored for possible complications. She was referred for psychiatric evaluation and behavioral therapy to address trichotillomania and prevent it from happening again. Studies suggest that recurrence of bezoars has been observed following their initial removal, which is why ongoing psychiatric follow-up is recommended [6]. Nonetheless, the patient's willingness to participate in psychiatric or psychological treatment, such as cognitive-behavioral therapy aimed at decreasing obsessive-compulsive behaviors, is crucial for effectively preventing the return of trichobezoars. When this motivation is present, the long-term outlook for these patients is generally positive [7].Fig. 1 Abdominal radiography (supine): an epigastric mass.

Fig. 1

Fig. 2 Transverse section of the patients' CT of the abdomen and pelvis with intravenous contrast: A well-defined heterogeneous mass is seen in the stomach.

Fig. 2

Fig. 3 Trichobezoar after removal. The mass had taken the shape of stomach.

Fig. 3

4 Discussion

The gastrointestinal tract frequently serves as the ultimate site for foreign objects prior to the manifestation of clinical symptoms. Foreign objects may enter the body via the oral cavity or the anus. Commonly ingested items encompass coins, fish bones, chicken bones, dental appliances, and food masses. The majority of ingested foreign bodies traverse the gastrointestinal system without resulting in significant complications. Less than 1 % of ingested foreign bodies result in perforation, a serious and potentially life-threatening condition. Perforation is associated with a high risk of complications and typically necessitates emergency surgical intervention [[8], [9], [10]]. Trichobezoars are a rare but noteworthy clinical entity in pediatric surgery, with potentially severe complications if left undiagnosed or untreated. The case of our 10-year-old patient with ADHD illustrates several key points in the presentation, diagnosis, and management of trichobezoars. Trichobezoars predominantly affect young females, with a peak incidence in the second decade of life [11,12]. Our patient, at 10 years old, represents a slightly younger demographic than typically reported. The association between trichobezoars and psychiatric disorders is well-established, with trichotillomania and Trichophagia frequently observed in patients with anxiety disorders, depression, and obsessive-compulsive disorder [13]. The link between ADHD and trichobezoars, as seen in our patient, is less documented and demands further investigation. However, There are limited published articles that explore the relationship between Attention Deficit Hyperactivity Disorder (ADHD) and Trichotillomania within the adult population [14]. It's likely that the impulsivity and inattention characteristic of ADHD may contribute to Trichophagia behavior. The nonspecific nature of trichobezoar symptoms often leads to delayed diagnosis. Common presentations include abdominal pain, nausea, vomiting, and early satiety, all of which were observed in our patient [15]. Chronic malnutrition and failure to thrive, as evidenced by our patient's growth retardation, can be important diagnostic clues [16,17]. The presence of a palpable epigastric mass, as in our case, is a significant physical finding that should encourage further investigation [18]. Imaging plays a fundamental role in diagnosis. While abdominal X-rays can reveal a gastric mass, CT scans provide more detailed information about the extent and composition of the bezoar [5,11]. The CT findings in our case, showing gastric dilatation with impacted material, are typical of large trichobezoars [19,20]. Upper gastrointestinal endoscopy remains the gold standard for diagnosis, offering both visual confirmation and the potential for therapeutic intervention in smaller bezoars [21]. The management of trichobezoars primarily involves surgical removal, as demonstrated in our case. While endoscopic removal has been reported successful in some cases, particularly for smaller bezoars, it is often challenging and potentially risky for large trichobezoars due to their size and density. Attempts at endoscopic fragmentation and removal carry the risk of distal migration of fragments, potentially causing intestinal obstruction [22]. Laparoscopic approaches have gained attention in recent years, as they offer the benefits of minimally invasive surgery. However, the technical challenges of manipulating large, dense trichobezoars laparoscopically, together with the need for thorough examination of the entire gastrointestinal tract for satellite lesions, often necessitate conversion to open surgery. In our case, and in many reported in the literature, laparotomy with gastrostomy remains the most common and effective method for large trichobezoars [3]. It is recommended in the literature that large trichobezoars in pediatric patients be effectively managed through a combination of endoscopic and laparoscopic surgical techniques. The combined approach of endoscopy, laparoscopy, and gastrostomy offers several benefits, including reduced leakage, minimized injury, decreased risk of gastric motility disorders, diminished thermal injury, and the facilitation of earlier enteral feeding. Additional advantages of this integrated technique include minimal trauma, accurate evaluation of endoscopic remnants, a procedural duration of less than one hour, improved cosmetic outcomes, and a reduced risk associated with anesthesia [[23], [24], [25]].

Psychiatric evaluation and long-term follow-up are essential to address underlying behavioral issues. In patients with ADHD, like our case, this may involve adjusting medication regimens and implementing behavioral interventions to reduce impulsive behaviors.

The over-diagnosis of Attention Deficit Hyperactivity Disorder (ADHD) in children raises significant concerns, primarily due to the complexities inherent in the diagnostic process and the potential for misdiagnosis. ADHD is characterized by symptoms of inattention, hyperactivity, and impulsivity, which can adversely affect a child's functioning across various environments. The subjective nature of these symptoms complicates the accuracy of diagnosis. Several factors contribute to the inflated rates of ADHD diagnoses, including cultural perceptions, educational pressures, and insufficient evaluation processes. The utilization of standardized diagnostic criteria, such as those outlined in the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition (DSM-5), is crucial for ensuring accurate diagnosis. It is imperative that differential diagnoses take into account other conditions, such as anxiety disorders, mood disorders, learning disabilities, and sleep disturbances, which may present symptoms similar to those of ADHD (“National Institute for Health and Care Excellence”).

Untreated trichobezoars can lead to severe complications, including gastric perforation, intestinal obstruction, and intussusception. Our patient's presentation before the onset of such complications highlights the importance of early recognition and intervention. The long-term prognosis for patients with trichobezoars is generally good following successful removal, as long as underlying psychiatric issues are addressed. However, reported recurrence rates of up to 20 % in patients without adequate psychiatric follow-up emphasizes the need for a multidisciplinary approach to treatment [26,27]. Further research is needed to reveal that how ADHD in children can result in trichotillomania and trichobezoar formation. Prospective studies examining the prevalence of Trichophagia in ADHD patients could inform screening practices. Additionally, investigation into novel minimally invasive techniques for trichobezoar removal, such as combined endoscopic and laparoscopic approaches, may offer improved outcomes with reduced morbidity.

5 Methods

The current study has been reported in line with Scare criteria [28].

6 Conclusion

Our case highlights the importance of considering trichobezoars in the differential diagnosis of pediatric patients presenting with gastrointestinal symptoms, particularly those with a history of psychiatric or neurodevelopmental disorders. A high index of suspicion, prompt diagnosis, and a multidisciplinary approach involving surgical intervention and comprehensive psychiatric care are essential for optimal outcomes in these challenging cases.

Approval of the research protocol by an Institutional Reviewer Board

Non applicable.

Informed consent

Written informed consent was obtained from the patient's parents/legal guardian for publication and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Consent for publication

Yes.

Ethical approval

The Ethics Committee of Shiraz University of Medical Sciences does not mandate the acquisition of an ethical approval code for the publication of case reports. Consequently, our study was granted an exemption from the requirement of obtaining an ethical code.

Funding

No funding.

Author contribution

Mehdi Forooghi: Diagnosis, Surgery, and Conceptualization; Raha Shahrokhi: Initial draft composition; Shayan Yousufzai: Final Revision of the manuscript, Supervision, data curation, and submission.

Guarantor

Shayan Yousufzai.

Research registration number

Non applicable.

Declaration of generative AI and AI-assisted technologies in the writing process

During the preparation of this work the author used Wordvice.AI/Paraphrasing and proofreading tool in order to avoid either plagiarism or grammar errors. After using this tool/service, the author reviewed and edited the content as needed and takes full responsibility for the content of the publication.

Conflict of interest statement

The authors declare that they have no competing interests.

Data availability

The corresponding author can provide the editorial in chief with data and material upon request.

Acknowledgements

None.
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