Article

Telling the tale of Rapunzel syndrome

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  • University of Central Florida - College of Medicine
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... Neychev et al. reviewed 72 case reports of Rapunzel syndrome and reported that of all, three patients (4%) had died. 10 Recurrence of Rapunzel syndrome was reported in five (7%) patients. Gorter et al. found different pathologies associated along with trichobezoars in four patients. ...
Article
A bezoar is a mass of indigestible foreign material found in the gastrointestinal tract, usually in the stomach. Trichobezoars are the second most common bezoars which are usually found in young psychiatric patients. A 15-year-old female patient presented with signs of intestinal obstruction and acute abdomen. The preoperative investigations revealed a perforated appendix, and an exploratory laparotomy was performed. Intraoperative findings showed presence of a mass of hair in the stomach along with a long tail extending into the small intestine. Postoperatively, the patient's condition worsened and she died because of complications of perforation peritonitis on day 3. Early diagnosis of Rapunzel syndrome can help in making early interventions. The prognosis of incidental detection of this condition depends on the accompanying cause.
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El síndrome de Rapunzel es una enfermedad inusual y rara que se presenta en personas jóvenes, caracterizada por un acúmulo de cabello ingerido (tricobezoar gástrico) y confinado generalmente a la cámara gástrica que se extiende al intestino delgado, asociada a tricofagia. Reportamos el caso de una paciente de 25 años con antecedente de tricofagia que ingresa a un centro de salud en curso de dolor, distensión abdominal y datos de obstrucción intestinal asociado a hemorragia digestiva alta. Con documentación preoperatoria por tomografía computada abdominal de un tricobezoar con extensión duodenal, el cual se extrae por laparotomía. La paciente se remite para manejo multimodal por servicio de psiquiatría. Es una afección para tener en cuenta como diagnóstico diferencial en los cuadros de abdomen agudo quirúrgico, de la mano con obstrucción intestinal y sangrado gastrointestinal.
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Introduction. Rapunzel syndrome involves an unusual form of gastric trichobezoar located from the stomach into the pylorus and extending to the small intestine, and or right colon. The trichobezoar, one of the bezoars that most occur in clinical practice, consists of a mass formed by hair due to ingestion of it either deliberately or unintentionally. Objective. Describe the frequency of signs and symptoms of Rapunzel syndrome in published case reports and present a comparison between the condition at pediatric and adult ages. Method. A systematic review in the PubMed database, EBSCO, and Scopus, using the terms "Rapunzel syndrome" AND "case report" OR "clinical case." Results. A total of 110 cases of 98 case report articles were analyzed. Differences between sex and age were found. Trichotillomania was distributed without predominance by age group. Also, differences in the reporting of some clinical manifestations analyzed by age group, including diarrhea, constipation, vomiting, and abdominal pain, were found. Recurrent cases represented 9%. Conclusions. Rapunzel syndrome is an entity found in patients with psychiatric disorders such as trichotillomania and trichophagia. It is more common in women than in men. Endoscopy was the chosen diagnostic technique, and the treatment selected included surgical removal and psychotherapy.
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RESUMEN. Introducción: El síndrome de Rapunzel es una forma inusual de tricobezoar gástrico, se localiza desde el estómago llegando a traspasar el píloro y extendiéndose hasta el intestino delgado y/o colon derecho. Se le llama bezoar a la formación de cuerpos extraños, debido a la ingestión de uno o varios materiales no digeribles. El tricobezoar, uno de los bezoares que más se presentan en la práctica clínica consisten en una masa formada por cabello debido a la ingestión de este.Objetivo: describir la frecuencia de signos y síntomas del síndrome de Rapunzel en los reportes de caso y ofrecer una comparación en edad pediátrica y adulta.Método. Revisión sistemática en la base de datos PubMed, Google Académico, EBSCO y Scopus, empleando los términos “Rapunzel syndrome” AND “case report” OR “Clinical case”Resultados. Un total de 110 artículos de reporte de caso fueron analizados, encontrando diferencias por sexo y edad. La tricotilomanía se distribuyó sin predominancia por grupo de edad. Se identificaron diferencias en el reporte de algunas manifestaciones clínicas al analizarlas por grupo de edad, incluyendo diarrea, constipación, vómitos y dolor abdominal. Los casos recidivantes representaron un 9%.Conclusiones: El síndrome de Rapunzel es una entidad que se encuentra en pacientes con patologías psiquiátricas como la tricotilomanía y la tricofagia. Es más frecuente en mujeres que en hombres. La técnica diagnóstica por elección fue la endoscopía y el tratamiento elegido incluyó la remoción quirúrgica con psicoterapia. Palabras clave: síndrome de Rapunzel, signos y síntomas, bezoar, diagnóstico, tricotilomanía, adolescencia. Introduction: Rapunzel syndrome is an unusual type of gastric trichobezoar, which is located from the stomach through the pylorus and extends into the small intestine and/or right colon. Tricobezoar is a collection of foreign bodies that can form in the stomach, small intestine or colon, due to the ingestion of one or more indigestible materials. trichobezoar, one of the most present bezoars in Clinical practice consists of a mass formed by hair due to the ingestion of it, either consciously or unconsciously. Objective: To compile case reports to identify and analyze little-known aspects of Rapunzel syndrome, as well as patient characteristics along with the best and most documented methods of diagnosis and treatment. Method. Systematic review in the Pubmed, Google Scholar, EBSCO and Scopus databases using as search terms: "Rapunzel syndrome" AND "case report" OR "Clinical case" Results: A total of 110 case report articles were analyzed, finding differences by sex and age. Trichotillomania was distributed without predominance by age group. Differences were identified in the reporting of some clinical manifestations when analyzed by age group, including diarrhea, constipation, vomiting and abdominal pain. Recidivist cases were 9%. Conclusion: Throughout the review of reported cases, we found that part of the patient's recovery includes psychiatric treatment; Rapunzel syndrome is not a mental disorder, however, both trichotillomania and pica as factors of it. The entity is more frequent in women than in men, the technique for diagnosis by choice was the endoscopy and the chosen treatment included surgical removal with psychotherapy. Keywords: Rapunzel Syndrome, Signs and Symptoms, Bezoar, Diagnosis Trichotillomania, Adolescence.
Article
Rapunzel syndrome is an extremely rare condition associated with trichophagia (hair eating disorder) secondary to a psychiatric illness called trichotillomania (hair-pulling behaviour). It is most commonly seen in children and adolescents. Untreated cases can lead to a number of complications. We present a case of a middle-aged woman with sudden intractable vomiting and constipation associated with bilateral pedal oedema and significant weight loss. Laboratory investigations revealed low serum protein levels. Laparotomy was performed, and a hairball was removed from her stomach and ileum. The patient was managed with the help of a psychiatrist and was given nutritional support. We performed a comprehensive search and summarised data for a total of 88 cases. No time or language limit was placed. The purpose of this discussion is to highlight the clinical spectrum of Rapunzel syndrome and also to report its rare association with hypoproteinaemia.
Article
Abstract Anorexia nervosa is a medical and psychological disorder classically seen in young women who present with significant weight loss, a distorted body image, and an intense fear of gaining weight despite being underweight. A rare diagnosis that is also associated with weight loss is a trichobezoar, a collection of hair or hair-like fibers in the gastrointestinal tract, primarily in the stomach. In this report, we present the case of a 14.5-year-old girl with weight loss caused by a trichobezoar, initially thought to be due to anorexia nervosa, and we review the details of both disorders.
Article
The surgical removal of a trichobezoar is the rare end complication of the psychiatric disorders trichotillomania and trichophagia. The more severe form of the disease is termed Rapunzel syndrome, where the bezoar extends from the gastric body beyond the pylorus into the duodenum. Traditional therapy has included endoscopy, often with subsequent laparotomy, and associated psychiatric intervention. We present the largest and most recent series of patients with trichobezoars managed in a single institution. A retrospective review of all cases of trichobezoar at our institution from 2003 to 2011 was performed. Demographic data, presenting complaints, imaging, surgical treatment, and subsequent management were collected. All 7 patients were female, ages 5 to 23 years (mean, 11.5 years). Although multiple imaging modalities were necessary for preoperative diagnosis, most patients were accurately diagnosed without endoscopic evaluation (85%). All patients required an exploratory laparotomy for definitive treatment. At laparotomy, 5 patients were found to have postpyloric extension of the trichobezoar (71%). One of 7 patients had a wound infection postoperatively. There were no other surgical complications or recurrences requiring further exploration. Our series of trichobezoar patients appear to have a high rate of Rapunzel syndrome, and perhaps postpyloric extension should be considered the rule rather than the exception. Our series demonstrates that diagnosis can be established with a thorough history combined with radiography, and treatment should be a combination of laparoscopy and/or laparotomy with psychiatric consultation.
Article
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Gastric bezoars are a rare clinical entity, most commonly observed in patients with mental or emotional illness. Large bezoars can be difficult to remove laparoscopically without extending a port incision. We report the case of a large symptomatic trichobezoar with Rapunzel syndrome that occurred in a 17-year-old girl who had trichotillomania. The bezoar was removed laparoscopically, in piecemeal fashion, through a gastrotomy port. This procedure did not require an extension of any incision, nor did it require the contents of the stomach to directly touch the incision, thereby reducing the risk of infection. The patient was discharged home, on the fourth postoperative day, free of any complications. This case illustrates the safety of the laparoscopic approach in the removal of large gastric bezoars. In considering use of this approach, the potentially long operative time must be weighed against the benefits of both minimal risk of infection and minimal incisions.
Article
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Trichobezoars (hair ball) are usually located in the stomach, but may extend through the pylorus into the duodenum and small bowel (Rapunzel syndrome). They are almost always associated with trichotillomania and trichophagia or other psychiatric disorders. In the literature several treatment options are proposed, including removal by conventional laparotomy, laparoscopy and endoscopy. We present our experience with four patients and provide a review of the recent literature. According to our experience and in line with the published results, conventional laparotomy is still the treatment of choice. In addition, psychiatric consultation is necessary to prevent relapses.
Article
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Rapunzel syndrome is a rare presentation of a trichobezoar, with 24 cases having been reported in literature so far. This syndrome has been reported in 23 female and 1 male patient with a mean age of 10.8 years. The patients commonly present with abdominal pain, nausea, vomiting and signs of obstruction. The distal end of the bezoar may be in the jejunum, ileum or the colon. We evaluate the various cases reported and discuss the various modes of presentation of this uncommon syndrome, and also present a series of 3 cases, all females, aged 16, 18 and 21 years, and had a trichobezoar in the stomach with a tail extending into the ileum.
Article
Patients with trichotillomania often first present to dermatologists, as patients may be unaware of or deny hair pulling and seek an etiology for their hair loss. It therefore becomes the job of the dermatologist to correctly diagnose trichotillomania as well as offer treatment options. There appear to be three groups characterized by age of onset: preschool-age children, preadolescents to young adults, and adults. Young children often have a self-limited course of hair pulling. Adults frequently have psychiatric conditions associated with their trichotillomania. Preadolescents to young adults may benefit the most from active intervention, such as increasing awareness of hair pulling behaviors and behavior modification training. The approach of a patient by age of onset is helpful in guiding a dermatologist towards effective treatment options.
Laparoscopic intragastric re-moval of giant trichobezoar Fig 2. The trichobezoar in the form of the distal esoph-agus, stomach, and duodenal sweep
  • Dorn Hf
  • Gillick Jl
  • Stringel
Dorn HF, Gillick JL, Stringel G. Laparoscopic intragastric re-moval of giant trichobezoar. JSLS 2010;14:259-62. Fig 2. The trichobezoar in the form of the distal esoph-agus, stomach, and duodenal sweep. Surgery February 2013 298 Neychev, Famiglietti, and Saldinger
  • D E Sah
  • Koo J Price
  • V H Trichotillomania
Sah DE, Koo J, Price VH. Trichotillomania. Dermatol Ther 2008;21:13-21.