Rapunzel syndrome in children: a retrospective review of ten cases combined with literature review in a tertiary referral center.

PURPOSE: Rapunzel syndrome is an uncommon condition in children, and its clinical features remain unclear. This study presents the largest single-center series of pediatric cases to date, with the objective of documenting the clinical characteristics and treatment approaches for children with Rapunzel syndrome. METHODS: A retrospective study was conducted in children with Rapunzel syndrome from 2019 to 2023. We recorded age, gender, symptoms, locations of bezoar, complications, and treatment options. RESULTS: Ten patients with Rapunzel syndrome were included. The median age was 9.1 years, with all of whom were female. The most common clinical symptoms were upper abdominal mass (90%), abdominal pain (80%), and nausea and vomiting (50%). Complications occurred in six cases (60%), including small bowel obstruction (20%), severe gastric dilatation (10%), intestinal perforation (10%), choledochodilation (10%), acute pancreatitis with cholecystitis (10%). Preoperative ultrasonography suggested low-echoic foreign bodies continuing to the jejunum or ileocecal region in five cases (50%). Preoperative gastroscopy attempted in four cases (40%) to remove the foreign bodies, all of which failed. All patients underwent surgical treatment, with nine cases undergoing gastric incision foreign body removal, and one case undergoing gastric incision foreign body removal combined with intestinal perforation repair. All patients recovered well. No recurrence was observed during follow-up. CONCLUSION: The accuracy of ultrasound diagnosis in identifying Rapunzel syndrome is high; however, it may lead to misdiagnosis if not complemented with the patient's medical history. Endoscopic presents a heightened treatment risk and a reduced success rate. The condition commonly presents with severe complications, thus making laparotomy a safe and effective option for intervention.

Computed tomographic diagnosis and clinical outcomes of small intestinal obstruction caused by trichobezoars in client-owned rabbits.

Small intestinal obstruction in rabbits, although demonstrated to be common, is still poorly characterized. The objective of this study was to report features of small intestinal obstruction caused by trichobezoars in rabbits upon CT and their clinical outcomes. Rabbits with a diagnosis of intestinal obstruction that underwent CT scanning were included in this study. From each CT scan, obstruction location, appearance, Hounsfield units, size (mm), volume (mm3), distance from the stomach, and diameter of the small intestine proximal and distal to it, were extracted. Nine rabbits were included in the study. All rabbits presented for anorexia and had a distended or enlarged stomach (7) or abdomen (2). CT scan showed a non-contrast-enhancing, ovoid, smoothly marginated structure with a hyperattenuating rim, heterogeneous center, and variable amount of centrally located gas, in the duodenum (5), the jejunum (2), or the ileum (2). The density of the trichobezoars ranged from -156 to 58 HU (median, -44 HU) and volume ranged from 86.1 to 633.8 mm3 (median, 320.6 mm3). Three rabbits underwent medical management. Two of them survived, and one of them died. Six rabbits underwent surgery, of which three were successfully discharged from the hospital. CT proved to be a valuable imaging modality in rabbits with a suspected intestinal obstruction to provide the exact location of the obstruction and the characteristics of the obstructive material. The location and size of the trichobezoar may have relevant clinical implications.

Gastrointestinal Bezoars in Paediatrics: Case Series and Literature Review.

BACKGROUND: Bezoars are indigestible lumps which are usually found in stomach. Types of bezoar include phytobezoar, trichobezoar, lithobezoar, pharmacobezoar, plasticobezoar, lactobezoar and metal bezoar. Trichobezoars mostly affect females in 20s and 30s with a rarity in paediatrics. Unexplained complaints with a palpable mass are commonly found in these patients. Treatment involves retrieval of mass with searching for others. The purpose of this study was to present data and surgical management of cases with trichobezoars. MATERIALS AND METHODS: We documented a retrospective review of trichobezoars done in our hospital between 2016 and 2022. All demographic data collected included gender and age of cases, composition and extent of bezoar, clinical presentation, imaging modalities, endoscopic trial, surgical approach and outcome. RESULTS: Five cases of gastrointestinal tract (GIT) trichobezoars underwent surgery. All cases were females between (13 and 16 years). Trichobezoars were three gastric, one ileal and one of combined gastric and colonic. Complaints were abdominal pain, vomiting, weight loss and halitosis. Three cases had a palpable abdominal mass. Different radiological modalities were performed. Endoscopic retrieval was tried in one patient and the laparoscopic approach in another one, but the first route failed. Laparotomy followed by gastrotomy, enterotomy and colotomy was done without complications. CONCLUSIONS: Trichobezoars should be suspected in any child with unexplained abdominal complaints or with a palpable abdominal mass, especially in girls. Imaging can be done in different modalities for diagnosis. Endoscopic retrieval could be tried; however, its failure is common, necessitating laparotomy, which has an excellent outcome.

Expect the unexpected: bezoar-caused gastric perforation in the 19-year-old patient, after traffic accident.

A 19-year-old female involved in a traffic accident presented to the Emergency Room (ER) with no trauma-related symptoms but a palpable mass in the epigastrium. Imaging revealed a massive trichobezoar causing gastric perforation. Urgent laparotomy was performed, and a 1.5-kilogram bezoar was removed, along with repairing coexisting gastric ulcers. The patient had a history of trichophagia, suggesting a psychiatric association. This case highlights the potential of trichobezoars to cause gastric perforation, even in patients admitted for unrelated reasons. CT-scan proves effective in diagnosing such cases. While a traffic accident might be a plausible cause, the presence of a bezoar can elevate the risk of complications. Psychiatric evaluation is recommended when trichophagia is identified. The study underscores the need for vigilance in unexpected scenarios, demonstrating the importance of multidisciplinary approaches in managing such cases.

Gastrointestinal trichobezoars in the pediatric population: a retrospective study.

BACKGROUND: Trichobezoar is an extremely rare condition characterized by a foreign body in the gastrointestinal tract (GIT) among children. The foreign body may exist in the digestive tract for several years, and it becomes evident if complications develop. The current study aimed to present 21 cases of GIT trichobezoars. METHODS: Retrospective analysis of children who were diagnosed with trichobezoars between August 2012 and December 2022. Patient demographics, clinical presentation, diagnosis, and therapy were collected and analyzed.Twenty-one patients had GIT trichobezoars. Data were collected and analyzed retrospectively. RESULTS: Twenty-one patients were identified. All patients were female. Their mean age at admission was 8.9 ± 1.9 years. Furthermore, 19 (90.5%) patients presented with abdominal pain, 16 (76.2%) with vomiting, and 13 (61.9%) with a palpable mass. Sixteen patients underwent gastroduodenoscopy. Among them, 15 had gastric trichobezoars. Moreover, 12 patients underwent computed tomography scan. Eight patients presented with gastric and small intestinal BZs, one presented with increased small intestinal contents with dilation, and one presented with abundant gastric contents. Then, 20 patients underwent surgery. Among them, five underwent laparoscopic-assisted minilaparotomy (LAML), and the rest underwent laparotomy. The results showed that 10 (50%) patients had gastric trichobezoars; 7 (35%), Rapunzel syndrome; and 3 (15%), small bowel trichobezoars. Two patients developed superficial wound infection postoperatively. One patient had a recurrent gastric trichobezoar. CONCLUSION: Trichobezoar should be considered in young girls with a history of hair eating or those with hair in the vomit or feces. Timely diagnosis and aggressive treatment are the keys to reducing complications and improving prognosis. Laparoscopic-assisted minilaparotomy is a safe, feasible, and effective surgical method for treating trichobezoars.

A case of Rapunzel syndrome.

Rapunzel syndrome is a rare disorder characterized by a trichobezoar in the gastroduodenal tract caused by trichophagia. Diagnosis was confirmed by upper endoscopy and treatment was surgical.

Point-of-Care Ultrasound of a Pediatric Gastric Trichobezoar: A Case Report.

ABSTRACT: This case report describes a previously healthy pediatric patient with acute onset of abdominal pain and distention who was found to have an epigastric mass on physical examination. Point-of-care ultrasound (POCUS) demonstrated a large gastric mass with ultrasonographic features consistent with a trichobezoar. After POCUS was performed, trichophagia was confirmed on history, and the patient went to the operating room for removal of a large trichobezoar. We conclude POCUS may be helpful for evaluation of epigastric masses and diagnosis of gastric trichobezoars. We review the ultrasound technique, sonographic findings, and literature regarding ultrasound diagnosis of trichobezoars.

A rare cause of intestinal obstruction in children trichobezoar: How to diagnose?

BACKGROUND: Trichobezoar is a rare clinical condition in children, which is caused by the accumulation of swallowed hair mass in the digestive tract. This condition is most common in young women with psychiatric histories who suffer from trichotillomania (TTM), where they have an irresistible urge to pull out their hair. Diagnosis and treatment of this pathology, which is already extremely rare, and its variable clinical presentations are challenging. The aim of this study was to increase awareness of trichobezoar in the differential diagnosis of signs of intestinal obstruction in children and to evaluate the diagnosis and management of this rare pathology. METHODS: The clinical data of six patients who were treated for trichobezoars in the pediatric surgery department of our hospital between 2009 and 2022 were retrospectively analyzed. RESULTS: Six female patients were treated with the diagnosis of trichobezoar during this period. Patients were diagnosed with the help of anamnesis, physical examination, abdominal ultrasonography (USG), and finally, endoscopy. USG can predict the intestinal wall infiltration and the tail extended to the duodenum through pylorus in the series. All patients were evaluated with contrast-enhanced abdominal radiography. Five surgical interventions were performed in four of the cases. In a case who underwent surgery twice, the distal intestinal satellite bezoar was not noticed in the first operation. Two patients were diagnosed to have trichobezoar, but surgery was not required. These patients were younger and had early-onset TTM (before 10 years old). The patients were followed for an average of 10.8 years and no recurrence was detected. CONCLUSION: Trichobezoar is a rare cause of intestinal obstruction in children with fatal complications when diagnosed late. Failure to follow an algorithm for the management of the disease causes difficulties in the diagnosis and treatment. Especially in pa-tients with a known psychiatric history, whole abdominal USG and laparoscopy performed with awareness can prevent unnecessary examinations.

Small bowel obstruction secondary to phytobezoar in a patient with myotonic dystrophy.

A male patient in his 30s, with myotonic dystrophy (DM), presented to the emergency department with abdominal pain and vomiting. CT imaging revealed a soft tissue lesion in the terminal ileum causing small bowel obstruction (SBO). The patient underwent diagnostic laparoscopy which allowed identification and removal of the obstructing lesion. This was in the form of an intact, undigested potato, a phytobezoar. Bezoars are collections of undigested material found in the gastrointestinal (GI) tract, a phytobezoar is composed of plant material and is the most common form of bezoar. DM is a multisystem disorder characterised by skeletal muscle weakness, however it often presents with GI symptoms and the muscles of mastication are often affected. DM is a known risk factor for bezoar formation and should be considered as an important differential in DM patients presenting with SBO.

[Rapunzel syndrome: Radiological diagnosis]. / Síndrome de Rapunzel: diagnóstico radiológico.

Background: Rapunzel syndrome is a rare presentation of trichobezoar, secondary to the ingestion of hair known as trichophagia. This bezoar has been found mainly in women, it invades the stomach and extends to the small intestine. Clinically, patients present weight loss and chronic obstructive symptoms at the intestinal level. A case of Rapunzel syndrome is presented. Clinical case: A 13-year-old female presented with a weight loss of 10kg in two months, chronic constipation, predominantly nocturnal vomiting, and abdominal pain of seven days' duration. Physical examination revealed decreased peristalsis and a palpable mass in the epigastrium. Laboratories taken on admission: normal blood count, kidney function tests, and liver function tests. The abdominal X-ray showed opacity in the fundus, body and gastric antrum, the abdominal ultrasound showed non-specific findings in the epigastrium, later an abdominal tomography was performed with a swallow of water-soluble contrast medium and showed occupation in the gastric lumen. She underwent exploratory laparotomy and the finding was a trichobezoar in the stomach with extension to the duodenum and part of the jejunum, which was removed without complications. The evolution of the patient was favorable. Conclusions: For the diagnosis of Rapunzel Syndrome, the use of contrast imaging studies is necessary, and the treatment of choice is surgical.

Introducción: el síndrome de Rapunzel es una presentación poco frecuente de tricobezoar, secundario a la ingesta de cabello conocida como tricofagia. Este bezoar se ha encontrado principalmente en mujeres, invade estómago y se extiende a intestino delgado. Clínicamente, los pacientes presentan pérdida de peso y síntomas crónicos de tipo obstructivo a nivel intestinal. Se presenta un caso de síndrome de Rapunzel. Caso clínico: paciente mujer de 13 años que se presenta con pérdida de peso de 10 kg en dos meses, estreñimiento crónico, vómito de predominio nocturno y dolor abdominal de siete días de evolución. A la exploración física, se encontró peristalsis disminuida y masa palpable en epigastrio. Laboratorios tomados a su ingreso: biometría hemática, pruebas de función renal y hepáticas normales. La radiografía de abdomen mostró opacidad en fundus, cuerpo y antro gástrico, la ecografía de abdomen mostró hallazgos inespecíficos en epigastrio, posteriormente se realizó tomografía abdominal con trago de medio de contraste hidrosoluble y mostró ocupación en la luz gástrica. Se sometió a laparotomía exploradora y el hallazgo fue un tricobezoar en estómago con extensión a duodeno y parte de yeyuno, fue removido sin complicaciones. La evolución de la paciente fue favorable. Conclusiones: para el diagnóstico del síndrome de Rapunzel es necesario el uso de estudios de imagen contrastados y el tratamiento de elección es quirúrgico.

A "madd"-ening confounding: fruit seeds mimicking enteral drug concealment by computed tomography.

OBJECTIVE: To highlight the similarity between madd fruit seeds and enteral drug concealment ("body packing") on computed tomography when evaluated by Hounsfield Units. CASE REPORT: A 13-year-old girl from Senegal presented to the Emergency Department with severe abdominal pain. Examination showed right lower quadrant tenderness with rebound. Computed tomography scan of the abdomen and pelvis revealed smooth, well circumscribed, multiple intraluminal foreign bodies measuring up to 2 cm in size with Hounsfield Units measuring up to 200. The emergency department radiologist reported that these were suspicious for "body packer packets" of either opioids or cocaine, based on their appearance and Hounsfield Unit characteristics. Dietary history later revealed consuption of madd fruit (Saba senegalensis) seeds, which can cause bezoar formation and intestinal obstruction. CONCLUSION: Madd fruit seeds may appear similar to drug packets on computed tomography with similar Hounsfield Unit characteristics. History and clinical context are paramount to avoid misdiagnosis.

Recurrent Rapunzel Syndrome in an Adolescent Female.

Bezoars are concrete masses found within the gastrointestinal tract that can lead to obstructions. One of the most common forms of bezoars is trichobezoar, composed of swallowed hair. Many bezoars are confined to the stomach; however, a small occurrence of trichobezoars extends past the pylorus and into the duodenum, or small bowel, termed Rapunzel syndrome. In the literature, there have been few cases of recurrent Rapunzel syndrome. Our case is a 13-year-old female with recurrent Rapunzel syndrome requiring three operative interventions.

Treatment of large gastric trichobezoar in children: Two case reports and literature review.

RATIONALE: Gastric trichobezoars are a rare form of bezoar formed from swallowed human hair as well as hair from dolls or animals, blankets, and carpets. They usually develop in young women who are emotionally disturbed, depressed, or mentally retarded, with trichotillomania and trichophagia. They can lead to abdominal pain, gastric ulceration, bleeding, obstruction, and perforation. PATIENT CONCERNS: A 13-year-old girl was admitted to our institution with complaints of nausea, vomiting, and epigastric pain for 2 days. She underwent laparoscopic removal of a large gastric trichobezoar at our institution 5 years ago. A 12-year-old girl presented at our institution due to epigastric pain, dizziness, and melena for 2 days. DIAGNOSIS: They were diagnosed with gastric trichobezoar by abdominal computed tomography scan and upper gastrointestinal endoscopy. INTERVENTIONS: Large gastric trichobezoars were removed via a mini-laparotomy. OUTCOMES: They recovered well postoperatively without complication. LESSONS: Although the recurrence of gastric trichobezoar after surgery is rare, few recurrent cases were reported in the literature. Therefore, psychiatric consultation and regular follow-up after treatment should be considered in the children and their parents to prevent the recurrence of gastric trichobezoar.