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.

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.

Small bowel obstruction caused by dehydrated apple ingestion: the challenges of preoperative radiological diagnosis and surgical management.

A man in his 60s with a virgin abdomen presented with sudden-onset generalised abdominal pain and fevers. The night prior, he snacked on supermarket purchased dehydrated apples. CT abdomen and pelvis revealed small bowel obstruction (SBO) to the mid ileum and small amount of free fluid within the pelvis. The patient underwent emergency exploratory laparotomy. High-grade SBO was identified, caused by large obstructing phytobezoars, with three further proximal large phytobezoars identified. All four phytobezoars were extracted and found to be rehydrated pieces of dehydrated apple that had increased in size in the gastrointestinal tract. The patient was later found to have further phytobezoars in the stomach which passed conservatively. The patient recovered well. This case demonstrates the challenges of preoperative radiological diagnosis in phytobezoar-related SBO and the significance of enterotomy orientation and closure to ensure a safe repair to withstand the passage of any residual rehydrating phytobezoars.

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.

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.

Histopathologic diagnosis and patient characteristics in cats with small intestinal obstructions secondary to trichobezoars.

OBJECTIVES: The aim of the present study was to describe the distribution of gastrointestinal histopathology findings associated with gastrointestinal obstructions secondary to trichobezoar formation in cats. METHODS: A total of 100 surgical gastrointestinal biopsies were obtained from 44 cats with gastrointestinal obstructions secondary to a trichobezoar. Medical records, including signalment, type and duration of clinical signs, surgical reports and histopathologic analysis, were reviewed for each cat. RESULTS: Biopsies taken near the site of the trichobezoar were more likely to show neutrophilic inflammation and mucosal erosion/ulceration compared with biopsies taken elsewhere in the small intestine. Lymphoplasmacytic and mixed lymphocytic and eosinophilic populations were the most common histopathologic findings from all biopsies followed by alimentary small cell lymphoma. Biopsy samples were more likely to represent a diagnosis of alimentary lymphoma in cats older than 10 years. CONCLUSIONS AND RELEVANCE: Gastrointestinal biopsies taken at the time of surgery in cats with trichobezoar obstructions may represent an important diagnostic tool for further evaluation of potential feline chronic enteropathy. Biopsies taken at the site of the obstruction should be interpreted cautiously as the presence of a trichobezoar may induce an acute inflammatory reaction. The resultant histologic interpretation at this site may not represent the chronic state of the intestinal mucosa, supporting the utility of obtaining multiple biopsies orad and aborad to the obstruction.

Endoscopic administration of Coca-Cola for medical management of a wedged intestinal trichobezoar in a cat.

A 16-year-old, neutered male domestic longhair feline with gastroduodenal and jejunal trichobezoars was treated with administration of Coca-Cola through endoscopic injection catheter. Examination with a Karl Storz endoscope identified a trichobezoar causing suspected partial or early complete obstruction of the jejunum; however, the length of the scope was inadequate to retrieve the trichobezoar. Consequently, 55 mL of Coca-Cola and 5 mL of iohexol were instilled into an injection catheter to disrupt the trichobezoar. No peri- or postoperative complications were reported, and the cat recovered uneventfully. Key clinical message: This case report demonstrates a minimally invasive approach to treatment of a small intestinal trichobezoar in a cat with no intra- or postoperative complications. Coca-Cola infusion through an endoscopic injection catheter may be a viable treatment in cats when a surgical approach is not an option, although further cases are needed to determine whether these results can be generalized to the larger patient population.

Administration endoscopique de Coca-Cola pour la prise en charge médicale d'un trichobézoard intestinal coincé chez un chat. Un félin domestique à poil long mâle castré de 16 ans atteint de trichobézoards gastroduodénaux et jéjunaux a été traité par l'administration de Coca-Cola via un cathéter d'injection endoscopique. L'examen avec un endoscope Karl Storz a identifié un trichobézoard suspecté d'obstruction partielle ou complète précoce du jéjunum; cependant, la longueur de l'endoscope était insuffisante pour récupérer le trichobézoard. Par conséquent, 55 ml de Coca-Cola et 5 ml d'iohexol ont été instillés dans un cathéter d'injection pour défaire le trichobézoard. Aucune complication péri- ou postopératoire n'a été signalée et le chat s'est rétabli sans incident.Message clinique clé :Ce rapport de cas démontre une approche peu invasive du traitement d'un trichobézoard de l'intestin grêle chez un chat sans complications per- ou postopératoires. L'infusion de Coca-Cola via un cathéter d'injection endoscopique peut être un traitement viable chez les chats lorsqu'une approche chirurgicale n'est pas une option, bien que d'autres cas soient nécessaires pour déterminer si ces résultats peuvent être généralisés à une population de patients plus large.(Traduit par Dr Serge Messier).

Bezoar at Jejunojejunostomy Following Liver Transplant: A Case Report.

During pediatric liver transplant, biliary reconstruction is often performed using Roux-en-Y choledoc-hojejunostomy or hepaticojejunostomy due to size mismatch, excessive tension caused by distance between donor and recipient ducts, or for transplant to treat primary biliary pathology. This method can be associated with additional small bowel-related complications compared with end-to-end ductal anastomosis. We report a case of late small bowel obstruction secondary to an impacted bezoar that formed at a patulous jejunojejunostomy portion of the biliary-enteric anastomosis. The patient was a 26-year-old male patient, who underwent deceased donor whole liver transplant for pediatric acute liver failure. Prior to his presentation to our institution with 2 days of abdominal pain, nausea, persistent burping, and intermittent vomiting, the patient reported an uneventful posttransplant course and was followed up at the institution where his transplant was performed. There were no reported changes in diet or lifestyle and no similar episodes during his follow-up. The patient was managed surgically after a brief trial of nonoperative management. At laparotomy, the anastomosis was resected and reconstructed to improve enteric drainage and prevent recurrent bezoar formation. The patient was discharged with no postoperative complications and remained asymptomatic at 11 months follow-up. We describe the clinical course and our technical approach at initial choledochojejunostomy creation and at jejunojejunostomy revision.

Phytobezoar-Induced Mechanical Ileus and Incipient Intussusception: A Case Report.

Phytobezoars constitute conglomerates of indigested plant fibers and are a rare cause of acute mechanical ileus. They exhibit an increased prevalence in the elderly population and people with specific predisposing conditions. Radiological imaging can often set a definitive diagnosis and dictate the optimal therapeutic approach, combined with the patient's clinical status. An 81-year-old male presented with deteriorating clinical symptoms of intestinal obstruction, and an exploratory laparotomy was performed following inconclusive radiological findings; multiple phytobezoars and incipient intussusception were revealed intraoperatively. A patient's medical history can often raise clinical suspicion of phytobezoars. However, a careful etiological investigation is imperative in all cases of mechanical ileus in advanced ages; early detection and dissolution of phytobezoars, when applicable, can reduce the need for surgical 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.

Laparoscopic treatment for multiple huge diospyrobezoars in the stomach.

We report a case of a patient with multiple diospyrobezoars, a phytobezoar attributed to persimmons (Diospyros kaki) consumption, in the stomach, who was treated with laparoscopic complete surgical excision. A 76-year-old man with gastric phytobezoars presented to our hospital. Abdominal contrast-enhanced computed tomography revealed three well-defined, oval, nonhomogeneous masses with a mottled appearance in the stomach. Esophagogastroduodenoscopy revealed three large brown solid phytobezoars and gastric ulcers at the gastric angle. The clinical diagnosis was diospyrobezoar, and, due to the huge masses, the patient eventually underwent laparoscopic treatment when the medical and endoscopic approaches were unsuccessful. After gastrotomy in the anterior wall of the stomach, the phytobezoar was mobile inside the stomach, which was opened beside the gastric incision. The three phytobezoars were removed through the wound protector using sponge-holding forceps; the hole in the gastrotomy was closed in the mucosal and seromuscular layers with an intracorporeal suture technique. The weight and size of the phytobezoars were 140 g and 115 × 55 × 50 mm, 70 g and 55 × 45 × 35 mm, and 60 g and 50 × 40 × 35 mm, respectively. The patient was discharged on the 8th postoperative day without any complications. Laparoscopic surgery to extract bezoar is the treatment of choice for this rare entity, because it is a safe and effective approach.

Trichobezoar-A rare cause of hyperemesis in a pregnant woman.

Bezoars consist of ingested foreign material or organic matter which forms a mass in the gastrointestinal tract, usually in the stomach. Trichobezoars formed by swallowed hair may present with vomiting, malnutrition, weight loss or abdominal pain with signs of gastrointestinal obstruction. There are limited case reports of Trichobezoar during pregnancy. We present a case of young pregnant women who was admitted on few occasions with vomiting in early pregnancy and was treated as hyperemesis gravidarum. Failure of response to conventional management prompted further investigation, revealing trichobezoar. Our case highlights the diagnostic challenge posed by trichobezoars in young pregnant women.

Duodenal diastatic perforation due to double gastric and jejunal trichobezoar in a patient with Rapunzel syndrome.

Rapunzel syndrome is a rare clinical entity in which a trichobezoar is produced by the ingestion of hair at the gastric level, extending in the form of a tail towards the duodenum. It occurs in young patients with trichotillomania and trichophagia. We present the case of a 24-year-old woman with a history of anxiety without treatment, who for 10 years presented trichotillomania and trichophagia, producing a picture of intestinal obstruction that required surgical intervention, evidencing a giant Trichobezoar throughout the gastric cavity and one jejunum associated with diastatic perforation. duodenal.

Evaluation of the location, number and diameter of bezoars in patients with a history of previous gastrointestinal surgery.

OBJECTIVE: Bezoars are foreign bodies developed due to the swallowing of indigestible substances in the stomach that accumulate in the lumen of the gastrointestinal tract. This study aimed to compare the location, size, and diameter of bezoars between patients with and without a history of previous gastrointestinal surgery and between operated and non-operated patients retrospectively. METHODS: A total of 188 patients who presented to our gastroenterology clinic and in whom bezoar was suspected on clinical examination and the diagnosis confirmed through abdominal CT scans were included in the study. The patients were divided into two groups; patients with a history of previous gastrointestinal surgery were assigned to Group 1 (n = 70), and those who had no history of previous surgery (n = 118) to Group 2. RESULTS: The mean age was found as 56.16 ± 15.75 years in Group 1 and 57.71 ± 15.95 years in Group 2. The mean bezoar width was significantly higher in Group 1 (p = 0.049). The mean bezoar length was significantly higher in Group 1 (p = 0.004). Considering localization of bezoars, the rate of patients who underwent enterotomy (80%) was statistically significantly higher than the patients who underwent gastrotomy (23.50%), gastrotomy + milking (28.60%) and milking (44.70%) in the operations performed in the jejunum. CONCLUSION: Bezoars are a rare cause of intestinal obstruction. The median width and length of the bezoars were significantly higher in patients with a history of previous gastric surgery. There was no significant difference in other parameters. The most common localization was jejunum.