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.

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.

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.

[Nasogastric tube obstruction due to enteral feeding bezoar]. / Obstrucción de sonda nasogástrica por molde de nutrición enteral.

Introduction: Introduction: the administration of enteral nutrition through a nasogastric tube can cause potentially serious complications. We present a case of esophageal obstruction due to an enteral nutrition bezoar. Case report: the 77-year-old patient was admitted to our center in the intensive care unit for COVID pneumonia. The patient received enteral nutrition through a nasogastric tube (NGT), presenting difficulty passing it after a month of follow-up. After removal of the tube and attached to it, an esophageal mold for enteral nutrition was extracted. Later, a solvent solution is administered through a new SNG and the formula is modified for a hydrolyzed one. Discussion: enteral nutrition bezoars are a rare but can be a life-threatening complication.

Introducción: Introducción: la administración de nutrición enteral por sonda nasogástrica puede presentar complicaciones potencialmente graves. Presentamos un caso de obstrucción esofágica por un bezoar de nutrición enteral. Caso clínico: el paciente de 77 años ingresó en nuestro centro en la unidad de cuidados intensivos por neumonía COVID. El paciente recibía nutrición enteral por sonda nasogástrica (SNG) presentando al mes del seguimiento dificultad para el paso de la misma. Tras retirada de la sonda y unida a ella se extrajo un molde esofágico de nutrición enteral. Posteriormente se administra a través de una nueva SNG una solución disolvente y se modifica la fórmula por una hidrolizada. Discusión: los bezoar de nutrición enteral son una complicación rara pero potencialmente mortal.

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.

[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.

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.

Bezoar types in children and aetiological factors affecting bezoar formation: A single-centre retrospective study.

Background: Bezoar is formed as a result of the accumulation of undigested food or foreign substances in the gastrointestinal tract (GIS). The present study aims to investigate the bezoar types seen in children and the aetiological factors involved in bezoar formation. Methods: A total of 16 patients who underwent an endoscopy and/or surgical treatment for bezoar at Harran University, Faculty of Medicine Pediatric Surgery Clinic between 2011 and 2019 were included in the study. The demographic information, laboratory and radiological findings were obtained from the patients' file records. Results: A total of ten patients (62.5%) were female with a mean age of 7.8 ± 4.9 years. Phytobezoars were detected in ten patients, trichobezoars in two patients, lactobezoar in one patient, and other types in three patients. The aetiological factors were determined to be congenital GIS anomaly in 6 (37.5%) patients; trichotillomania in 2 (12.5%) patients; mental retardation in 2 (12.5%) patients; ingestion of a foreign body during infancy in 2 (12.5%) patients; high intake of high-fibre fruit in 3 (18.5%) patients; and postoperative dysmotility in 2 (12.5%) patients. Conclusions: Congenital GIS anomalies are mostly responsible for bezoar etiology in children and phytobezoar is the most common type of bezoar.

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.

An Unusual Small Bowel Phytobezoar Successfully Resolved by Double-balloon Enteroscopy.

We herein report a very unusual case of small bowel obstruction caused by phytobezoar in a 69-year-old woman who consumed a large amount of bracken. The patient presented with nausea and vomiting. Computed tomography revealed an air-filled foreign body in the jejunum that had likely caused the small bowel obstruction. A fibrous foreign body diagnosed as a phytobezoar was detected using double-balloon enteroscopy. The obstruction was successfully resolved by crushing the phytobezoar repeatedly using a snare. Small bowel obstructions caused by phytobezoars are often treated with surgical interventions. However, endoscopic fragmentation using a snare is a minimally invasive treatment alternative.

Colonic Obstruction Secondary to Phytobezoar Caused by Vitex Agnus-Castus Seeds: A Case Report.

Plant seeds of Vitex agnus-castus (chaste berry) are known for benefits in reproductive health and also reduce bloating, depression, headache, and constipation. Plant seeds are a rare cause of phytobezoars. Bezoar is a formation of mass by piling up of material that is ingested but not digested or partially digested at any level of the gastrointestinal tract. While swallowed material mostly forms a bezoar in the stomach, it can rarely reach the colon and cause obstruction or perforation. Bezoars causing colonic obstruction can be treated by medical observation, endoscopic intervention, or surgical procedure. Herein, we present a case of colonic obstruction secondary to bezoar composed of plant seeds of Vitex agnus-castus and our approach to this condition. Key Words: Chaste berry, Vitex agnus-castus, Phytobezoar, Colonic obstruction.