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.

Endoscopic lithotripsy combined with drug lithotripsy vs. drug lithotripsy for the treatment of phytobezoars: analysis of 165 cases.

AIM: To analyze efficacy of endoscopic lithotripsy combined with drug lithotripsy as compared with drug lithotripsy for the treatment of phytobezoars. METHODS: We collected and evaluated case records of 165 patients with phytobezoars from 2014 to 2023. And we analyzed demographic and clinical characteristics, imaging features, endoscopic features, complications of phytobezoars, and compared efficacy between endoscopic lithotripsy combined with drug lithotripsy (Group A) and drug lithotripsy (sodium bicarbonate combined with proton pump inhibitor) (Group B). RESULTS: The median age of patients with phytobezoars was 67.84 ± 4.286 years old. Abdominal pain was the most common symptom and peptic ulcers (67.5%) were the most common complication. Bezoar-induced ulcers were more frequent in the gastric angle. The success rate of phytobezoars vanishing in Group A and Group B were similar (92.3% vs. 85.1% within 48 h, 98.7% vs. 97.7% within a week), while the average hospitalization period, average hospitalization cost, second endoscopy rate, and average endoscopic operation time were significantly lower in patients in Group B than in Group A. CONCLUSION: Drug lithotripsy is the preferred effective and safe treatment option for phytobezoars. We advise that an endoscopy should be completed after 48 h for drug lithotripsy.

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.

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.

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.

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.

Vesical fungal bezoars on post-TURBT scar tissue causing obstruction and mimicking urothelial carcinoma.

Fungal bezoars (fungal balls) are rarely reported in the upper or lower urinary tract. They can be the cause of severe morbidities such as urinary tract obstruction, renal failure and fungaemia. Hereby, we present a rare case of a male patient who underwent transurethral resection of bladder tumour (TURBT), and during his postoperative period, he was diagnosed with bladder fungal bezoars adherent to his resection area. The fungal bezoars were covering an extended area of the right lateral bladder wall, including the right ureteric orifice and causing right urinary tract obstruction. Those findings were manifested only after a relooked cystoscopy and histological evaluation.We aim to present a rare example of fungal bezoars mimicking other pathologies in the urinary tract and review the current literature for similar documentation. We underline the necessity of follow-up examinations for urologists performing TURBT surgeries, including urinalysis, imaging modalities and cystoscopy.

Whole-genome SNP genotyping unveils ancestral and recent introgression in wild and domestic goats.

After the domestication of goats around 10,000 years before the present (BP), humans transported goats far beyond the range of their wild ancestor, the bezoar goat. This brought domestic goats into contact with many wild goat species such as ibex and markhor, enabling introgression between domestic and wild goats. To investigate this, while shedding light on the taxonomic status of wild and domestic goats, we analysed genome-wide SNP data of 613 specimens from 14 taxonomic units, including Capra hircus, C. pyrenaica, C. ibex (from Switzerland, Austria, Germany and Slovenia), C. aegagrus aegagrus, C. a. cretica, C. h. dorcas, C. caucasica caucasica, C. c. severtzovi, C. c. cylindricornis, C. falconeri, C. sibirica sibirica, C. s. alaiana and C. nubiana, as well as Oreamnos americanus (mountain goat) as an outgroup. To trace gene flow between domestic and wild goats, we integrated genotype data of local goat breeds from the Alps as well as from countries such as Spain, Greece, Türkiye, Egypt, Sudan, Iran, Russia (Caucasus and Altai) and Pakistan. Our phylogenetic analyses displayed a clear separation between bezoar-type and ibex-type clades with wild goats from the Greek islands of Crete and Youra clustered within domestic goats, confirming their feral origin. Our analyses also revealed gene flow between the lineages of Caucasian tur and domestic goats that most likely occurred before or during early domestication. Within the clade of domestic goats, analyses inferred gene flow between African and Iberian goats. The detected events of introgression were consistent with previous reports and offered interesting insights into the historical relationships among domestic and wild goats.

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.

[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.

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.

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).

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.