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.

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.

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.

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).

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.

Uncommon cause of respiratory failure due to a bezoar in the hypopharynx: a case report.

BACKGROUND: Trichotillomania and trichophagia cause trichobezoars, which are masses made of hair. The main presentation of this condition is abdominal pain. However, other complications include gastric outlet obstruction, nausea, vomiting, weight loss, malnutrition, hematemesis, diarrhea, and constipation. CASE PRESENTATION: A 57-year-old woman with trichotillomania was admitted to the Emergency Department with the chief complaints of dyspnea on exertion, shortness of breath, dysphagia, generalized weakness, and hoarseness. Spiral chest computed tomography (CT) scan did not reveal any parenchymal lesions Pulmonary CT angiography did not reveal pulmonary embolism. The patient was admitted to the Surgery Department for hand fasciotomy due to contrast leakage, and during laryngoscopy, a trichobezoar was detected that was removed with Magill forceps. CONCLUSIONS: Rare cases of trichobezoars can be observed in humans with gastrointestinal and respiratory symptoms. Precise and timely diagnosis are key for the prevention of more invasive diagnostic procedures.

Multiple bezoars causing small bowel obstruction: a unique case of resolution with upper and low GI endoscopy.

Reports of obstructing foreign bodies in the small bowel are rare and the majority describe surgical resolution. We report a case of successful endoscopic treatment of a small bowel obstruction (SBO) caused by multiple bezoars. A 92-year-old female presented to the Emergency Department with a history of persistent vomiting. Computed tomography (CT) showed gastric distension and an intraluminal ovaloid foreign body with a heterogenous density in the second portion of the duodenum, suggestive of bezoar.

Bezoar: an uncommon cause of upper gastrointestinal bleeding.

Bezoars are aggregates of non-digestible material that accumulate in the gastrointestinal tract. They can be classified according to their composition, and phytobezoars (composed of vegetable or fruit fiber) is the most common type. They most often occur in patients with some risk factors, namely prior gastric surgery, neuropsychiatric, endocrine or other disorders that lead to abnormal gastric function, or poor gastric peristalsis. Bezoars may be asymptomatic but most commonly cause abdominal discomfort or pain, nausea, vomiting, fullness, difficulty in swallowing, or anorexia and weight loss. Upper GI endoscopy for both diagnostic and therapeutic purposes is key in the management of bezoars.

STOMACH BEZOARIS, CAUSES OF DEVELOPMENT, DIAGNOSIS AND METHODS OF TREATMENT.

OBJECTIVE: The aim: Is to determine the tactics and methods of treatment of bezoars of the gastrointestinal tract. PATIENTS AND METHODS: Materials and methods: From 2001 to 2019, 17 patients were diagnosed with "bezoar". RESULTS: Results: Due to the "weariness" of the clinic, the diagnosis was made in the first 3 days only for 3 (17,6 %) patients. On the basis of the obtained average pH values for 4 (23,5 %) patients established moderate hypoacid, for 4 (23,5 %) - pronounced hypoacid, for 6 (35,3 %) - anacid. For 5 (29,4 %) patients, the bezoars were withdrawn on the first attempt, while the other 5 (29,4 %) were "lumped". In the course of fibrogastroscopic examination, all patients were diagnosed with impaired motor-evacuation function of the stomach: gastroesophageal and duodenogastric refluxes, presence of passive discharge of the contents of the stomach into the esophagus. CONCLUSION: Conclusions: Therefore, the preconditions for the development of bezoars may be: hypo- and anacid, impaired motor-evacuation function of the stomach, chronic gastric ulcer,cognitive impairment. Endoscopic method should be preferred in the treatment, on condition of its failure - laparoscopic gastrotomy with bezoar extraction.

Esophagogastric polyurethane bezoar complicated by stomach wall microperforation and acute peritonitis: case report.

BACKGROUND: Bezoars are collections of indigestible material in the gastrointestinal tract, mostly described in children. Polyurethane "plastobezoars" consisting of composites used in the construction industry are rarely described bezoars formed in the esophagus and stomach, causing gastrointestinal obstruction, usually necessitating gastrectomy. We describe an unusual presentation of polyurethane bezoar with a volcanic rock consistency, that caused gastrointestinal obstruction and perforation of the stomach wall. CASE PRESENTATION: A 39-year-old man, a construction worker, was referred with signs and symptoms of high gastrointestinal obstruction and abdominal pain. Esophagoscopy revealed a foreign body in the esophagus, 20 cm from the incisor line, causing its obstruction. The attempt to collect the material with forceps failed as the material was too hard. Spiral computed tomography visualized a wide, gas-filled esophagus and a large stomach. The patient with symptoms of acute peritonitis was operated. There were several microperforations of the stomach wall, caused by sharp bezoar fragments that filled the upper one-third of the stomach and lower part of the esophagus. After a longitudinal stomach incision, the bezoar was bluntly dissected from the wall and removed, and the stomach microperforations were closed by wall duplication. After the operation, the patient confessed to drinking, of his own free will, a two-component building foam used to seal pipes. The patient started normal feeding on the 4th day and was discharge home. CONCLUSIONS: Polyurethane bezoars may cause stomach wall perforation and acute peritonitis. Computed tomography has limited usefulness in patients with polyurethane bezoars due to their low specific weight.

What Is That? Innumerable Mysterious Densities Identified on Abdominal Imaging.

Radiopaque densities can be observed on imaging after the ingestion of either foreign bodies or some medications. Our case report discusses an 11-year-old boy with autism spectrum disorder and attention deficient disorder who presented to the emergency department because of concerns for constipation and dehydration. Incidentally, an abdominal x-ray showed numerous radiopaque densities throughout his intestines in addition to his constipation. He was admitted, and his home regimen was reviewed to attempt to identify a potential source for these radiopaque densities. This case presented an interesting teaching opportunity in the identification of the radiopaque densities and review of pharmacokinetics.

Bezoar in incarcerated and complicated hiatal hernia.

INTRODUCTION: the identification of a 10 cm bezoar in a hiatal hernia is unusual. Hiatal hernias are usually asymptomatic. When signs of strangulation are present the prognosis is unlucky if urgent surgical intervention is not performed. Although sometimes the prognosis is bad because of the advanced state of gastric ischemia. CASE REPORT: a 67-year-old female patient undergoing Nissen fundoplication with recurrence of hiatal hernia, volvulated and strangulated with giant bezoar inside the hhital hernia sac. DISCUSSION: despite of poor results, surgical treatment in these cases must be urgent.

[Giant Rapunzel syndrome with atypical complication. Report of a case]. / Síndrome de Rapunzel gigante con complicación atípica. Reporte de un caso.

Rapunzel syndrome is an unusual and rare form of gastric trichobezoar that extends into the small intestine. The formation is due to the continuous ingestion of hair which leads to the impaction of the hair along with mucus and food, but it is rare that gastric perforation occurs. We report the case of a female patient of 16 years of age admitted to our institution with a history of depression and clinical symptoms characterized by nausea, severe epigastralgia and pathological loss of weight. The physical examination showed a tense abdomen with a peritoneal reaction. Contrast-enhanced tomography showed a distended stomach with a heterogeneous content that extended to the duodenum, hydro pneumoperitoneum, Douglas sack bottom liquid, and it was decided to perform an exploratory laparotomy. Showed perforation in the anterior face of the stomach which is infrequent, so we proceeded to the extraction of giant trichobezoar with duodenal extension; Evolving favorably, the patient being referred to outpatient controls with psychiatry; Finally, a high endoscopy was performed at 6 months, which evidenced the scar of the surgical procedure.

Fungaemia caused by obstructive renal candida bezoars leads to bilateral chorioretinitis: a case report.

BACKGROUND: Renal fungal bezoars are remarkably rare and mostly occur in immunodeficient patients. Only a small number of cases with immunocompetent patients have been published so far. The published treatment approaches comprised systemic antimycotic therapy and surgical or minimal invasive removal of the fungal balls. In some cases irrigation of the renal duct system with amphotericin B was performed. By obstruction of the urinary tract bezoars can lead to infected hydronephrosis and severe urosepsis with high lethality. Fungaemia can cause fungal colonization in different distant organs. Fulminant chorioretinitis and irreversible visual impairment can be the consequence of ocular fundus colonization. The following report highlights that a co-operation between urologists and ophthalmologists is absolutely indispensible in case of fungaemia. CASE PRESENTATION: Hereinafter we describe a case of an immunocompetent 56 years old woman, presenting with flank pain and shivering. The diagnosis turned out to be difficult due to initially negative urine culture. The fungaemia caused by obstructive nephropathy led to bilateral candida chorioretinitis. The patient was treated with intravenous amphotericin b and the bezoar was removed by percutaneous "nephrolitholapaxy". After two months, a follow up revealed the patient felt well, chorioretinal lesions regressed and urine culture did not show any fungal growth. CONCLUSION: To the best of our knowledge, this is the first case reporting on obstructive renal bezoars, which lead to haematogenous fungus spread and bilateral chorioretinitis. It points out that extensive ophthalmologic examination should be performed in case of fungaemia even if the patient is not suffering from any visual impairment.

Rapunzel syndrome: an infrequent cause of severe iron deficiency anemia and abdominal pain presenting to the pediatric emergency department.

BACKGROUND: Iron deficiency anemia (IDA) and abdominal pain are commonly seen in a pediatric emergency department (8 and 18% incidence respectively in our center). They are manifestations of a wide variety of diseases ranging from benign to immediately life-threatening. Trichobezoar is an under-diagnosed entity that has to be considered in children and adolescents, expecially female, suffering from trichotillomania (compulsion to pull hair) and trichophagy (compulsion to swallow hair). When undiagnosed, gastric bezoars may cause gastric ulceration, perforation, haemorrhage and obstruction. CASE PRESENTATION: To underline the importance of including this pathology in the differential diagnosis of IDA and abdominal pain, we present the case of a 14 year-old girl with a huge trichobezoar which completely filled the stomach and extended into the small bowel. Since trichobezoar has an extension to the small bowel, it is classified as Rapunzel syndrome. As the bezoar couldn't be removed by endoscopy, the girl underwent surgical intervention. The patient passed through a gradual re-feeding, with iron and vitamins supplementation, and through a psychiatric counselling. CONCLUSION: The Rapunzel syndrome is a rare entity that may be complicated by life-threatening events. A prompt diagnosis and an appropriate therapy can reduce comorbidities. Gradual re-feeding with supplementation of micronutrients allows adequate catch-up weight with normalization of haematochemical nutritional parameters. Since many of these patients suffer from psychiatric pathology such as PICA with emotional problems and mental retardation, psychological/psychiatric counselling plays an important role in order to prevent bezoar recurrence.

A Rare Case of Fatal Bowel Obstruction Secondary to a Colonic Bezoar.

A bezoar is a mass of undigested, or partially digested, material forming in the lumen of the gastroenteric tract, causing occlusive or subocclusive events. The most frequent types of bezoars are those composed of vegetable fibers, also called phytobezoars, which, by virtue of their high content in cellulose, hemicellulose, and lignin, remain undigested in the stomach and intestines and, from there, can migrate and occlude the narrowest portions of the bowel. The areas that are most frequently affected by occlusive phenomena related to the presence of bezoars are the stomach and the small intestine, although colic localizations are extremely rare. In this article, we have studied the case of a fatal colic obstruction caused by a phytobezoar in an 84-year-old woman who was found dead at her home. The autopsy revealed that the cause of the obstruction was a large artichoke fragment occluding the central part of the descending colon. Additional histological examinations confirmed that the death was attributable to bowel obstruction resulting in acute peritonitis.

Catastrophic gastric rupture in a horse secondary to psyllium pharmacobezoars.

A mare was euthanized because of gastric rupture secondary to complete duodenal obstruction by 2 bezoars located in the pylorus and proximal duodenum. Infrared spectroscopy showed that the bezoars were composed of psyllium. The mare had been receiving treatment with a pelleted psyllium product at 4 times the recommended dosage. Veterinarians should be aware that treatment of colic in horses with pelleted psyllium products could be associated with gastric impaction.

Rupture gastrique catastrophique secondaire à des pharmacobézoards de psyllium en boulettes chez un cheval. Une jument a été euthanasiée en raison d'une rupture gastrique secondaire à une obstruction duodénale complète par deux bézoards situés dans le pylore et le duodénum proximal. La spectroscopie infrarouge a montré que les bézoards se composaient de psyllium. La jument avait reçu un traitement composé d'un produit de psyllium en boulettes à quatre fois la dose recommandée. Les vétérinaires devraient être au courant que le traitement des coliques chez les chevaux avec des produits de psyllium en boulettes pourrait être associé à une impaction gastrique.(Traduit par Isabelle Vallières).