Rapunzel Syndrome – A Case Report: Trichobezoar as a Cause of Gastric Outlet Obstruction and Intestinal Perforation

Dubai Medical Journal ◽

10.1159/000520733 ◽

2021 ◽

pp. 1-3

Author(s):

Nazia Ishaq ◽

Tahir Khaleeq

Keyword(s):

Case Report ◽

Gastric Outlet Obstruction ◽

Intestinal Perforation ◽

Unusual Case ◽

Outlet Obstruction ◽

Adolescent Females ◽

Rapunzel Syndrome ◽

Gastric Trichobezoar ◽

Rare Manifestation

Rapunzel syndrome is described as a rare manifestation of gastric trichobezoar extending throughout the bowel and is associated with psychiatric disorders. Its incidence in Pakistan is very rare and is found characteristically in adolescent females. Large or complicated trichobezoars are best managed by surgery. Psychiatric follow-up is essential to diminish the frequency of recurrence. We report an unusual case of Rapunzel syndrome causing gastric outlet obstruction and intestinal perforation.

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Gastric trichobezoar and Rapunzel syndrome: a case report

International Surgery Journal ◽

10.18203/2349-2902.isj20201710 ◽

2020 ◽

Vol 7 (5) ◽

pp. 1634

Author(s):

Abhishek Gupta ◽

Panna Lal Gupta

Keyword(s):

Case Report ◽

Epigastric Pain ◽

Exploratory Laparotomy ◽

Adolescent Females ◽

Young Adolescent ◽

Adolescent Female ◽

Rapunzel Syndrome ◽

Gastric Trichobezoar ◽

History Of

Giant gastric trichobezoars are unusual form of foreign body found in stomach of mostly young adolescent females which may lead to morbidity and high mortality 30%, if goes unnoticed. These females have history of trichophagia or trichotillomania. This report is of a 12- year old young adolescent female presented with epigastric pain and mass. An exploratory laparotomy with anterior gastrotomy was performed and a giant trichobezoar with a very large tail of 2.5 ft was removed, weighing 2.52 kg. She recovered well and was discharged on 7th post-operative day. Patient was advised for psychiatric follow up.

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Rapunzel syndrome causing partial gastric outlet obstruction requiring emergency laparotomy

BMJ Case Reports ◽

10.1136/bcr-2019-232904 ◽

2020 ◽

Vol 13 (1) ◽

pp. e232904

Author(s):

Robert Lyons ◽

Granit Ismaili ◽

Michael Devine ◽

Haroon Malik

Keyword(s):

Ct Scan ◽

Gastric Outlet Obstruction ◽

Epigastric Pain ◽

Outlet Obstruction ◽

Postoperative Recovery ◽

Emergency Laparotomy ◽

Rapunzel Syndrome ◽

History Of

A 16-year-old girl with a background of childhood trichophagia presented with a 2-day history of epigastric pain and associated anorexia with vomiting. An epigastric mass was palpable on examination. A CT scan revealed an intragastric trichobezoar, extending into the duodenum consistent with Rapunzel syndrome with evidence of partial gastric outlet obstruction and a possible perforation. The patient underwent an urgent laparotomy and extraction of the trichobezoar. The bezoar was removed without complication and no intraoperative evidence of perforation was detected. After an uncomplicated postoperative recovery, she was discharged home with psychiatric follow-up.

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A181 ENDOSCOPIC BALLOON DILATION IS EFFECTIVE AND SAFE IN THE MANAGEMENT OF NSAID-INDUCED NON-ULCERATIVE GASTRIC OUTLET OBSTRUCTION

Journal of the Canadian Association of Gastroenterology ◽

10.1093/jcag/gwz047.180 ◽

2020 ◽

Vol 3 (Supplement_1) ◽

pp. 48-50

Author(s):

D R Lim ◽

D Farina ◽

W Huang ◽

J Zhu

Keyword(s):

Case Report ◽

Pyloric Stenosis ◽

Gastric Outlet Obstruction ◽

Unusual Case ◽

Surgical Intervention ◽

Balloon Dilation ◽

Outlet Obstruction ◽

Gi Bleeding ◽

Endoscopic Balloon Dilation ◽

Endoscopic Balloon

Abstract Background We describe an unusual case of NSAID-induced gastric outlet obstruction (GOO) in the absence of malignancy or ulcers. This was successfully managed conservatively with drug withdrawal and serial endoscopic balloon dilation (EBD). Aims Case Report Methods Case Report and Literature Review Results A 58-year-old woman with 20-year history of daily Ketorolac use for osteoarthritis presented with iron deficiency anemia (IDA) of 58 g/L and post-prandial emesis for 2 months. There was no overt GI bleeding. Gastroscopy revealed severe erosive esophagitis and GOO with no ulcers. After a negative CT imaging ruled out extrinsic malignant compression, GOO was managed with EBD. Examination of the duodenum post-EBD revealed complete atrophy and scalloping. Focused biopsies reveal chronic gastritis, complete villous atrophy in the duodenum; ruled out H. Pylori, celiac disease, amyloidosis and dysplasia. EUS negative for infiltrative disease or regional adenopathy. Vitamin B12, anti-TTG I IgA, HLA DQ2/8 were normal. These findings made drug-induced enteropathy the top contender. PPI therapy was initiated and NSAID discontinued. Five serial EBD were performed over 4 months to 18mm. A pureed diet was tolerated after 2 dilations. Follow-up at 3 months showed partial recovery of enteropathy and pyloric stenosis. No adverse events were seen. The severe esophagitis was likely an erosive process secondary to reflux from GOO. Her IDA is likely multifactorial: Severe enteropathy and GOO may have led to chronic malabsorption; occult GI bleeding from erosions or ulcers that have healed may further contribute. Ketorolac could explain the enteropathy. COX-1 inhibition leads to decreased gastric cytoprotection. In rat models, COX-2 inhibition has been suggested to delay gastric healing and dysregulated immune response to food antigens in the small bowel [4–6]. NSAID-induced GOO almost always occur in the context of peptic ulcer disease [1,2]. A similar case [3] found pyloric stenosis in a 75-year old woman with esophagitis and ulcer-induced pyloric stenosis. They postulate that post ulcerative healing led to benign pyloric stenosis and explained the absence of ulcers. Historically, surgical intervention and stent placement have played a major role in the management of benign mechanical GOO. EBD has replaced surgical intervention as first line therapy [7] showing promising results beyond 3 months post EBD[8], sparing patients from surgery related morbidity. An algorithm has been suggested by us [Img 1] for the management of benign GOO. Conclusions We present an unusual case of NSAID-induced mechanical GOO and enteropathy. This case highlights these entities as rare but serious complications of chronic NSAID use. Management of benign mechanical GOO should be individualized. Prudence with prescribing NSAIDs to at risk populations is recommended. Funding Agencies None

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Unusual cause of gastric outlet obstruction: giant gastric trichobezoar: a case report

Cases Journal ◽

10.1186/1757-1626-1-399 ◽

2008 ◽

Vol 1 (1) ◽

pp. 399 ◽

Cited By ~ 6

Author(s):

Ibrahim Yetim ◽

Orhan Ozkan ◽

Ersan Semerci ◽

Recep Abanoz

Keyword(s):

Case Report ◽

Gastric Outlet Obstruction ◽

Outlet Obstruction ◽

Gastric Trichobezoar

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Closed-perforation of gastric fundus and gastric outlet obstruction caused by a giant gastric trichobezoar: A case report

Turkish Journal of Surgery ◽

10.5152/ucd.2015.2994 ◽

2017 ◽

Vol 33 (3) ◽

pp. 230-232 ◽

Cited By ~ 1

Author(s):

Bunyamin Gurbulak ◽

Ozgur Segmen ◽

Taskin Rakici ◽

Kenan Buyukasik ◽

Mazlum Yavas

Keyword(s):

Case Report ◽

Gastric Outlet Obstruction ◽

Outlet Obstruction ◽

Gastric Fundus ◽

Gastric Trichobezoar

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Unusual Tissue – Unusual Issue: Pancreatic Heterotopia Presenting as Gastric Outlet Obstruction

Case Reports in Gastroenterology ◽

10.1159/000512427 ◽

2021 ◽

pp. 338-343

Author(s):

Thu L. Nguyen ◽

Shivani Kapur ◽

Stephen C. Schlack-Haerer ◽

Grzegorz T. Gurda ◽

Milan E. Folkers

Keyword(s):

Gastric Outlet Obstruction ◽

Epigastric Pain ◽

Outlet Obstruction ◽

Needle Aspiration ◽

Pancreatic Tissue ◽

Cystic Mass ◽

Pancreatic Heterotopia ◽

History Of

Pancreatic heterotopia (PH) is a common, but typically small (<1 cm), incidental and asymptomatic finding; however, PH should be considered even for large and symptomatic upper gastrointestinal masses. A 27-year-old white woman presented with a 3-week history of burning epigastric pain, nausea, early satiety, and constipation. Physical examination revealed epigastric and right upper quadrant tenderness with normal laboratory workup, but imaging revealed a 5-cm, partly cystic mass arising from the gastric antrum with resulting pyloric stenosis and partial gastric outlet obstruction. Endoscopic ultrasound-guided fine needle aspiration revealed PH – an anomalous pancreatic tissue lying in a nonphysiological site. The patient ultimately underwent a resection and recovered uneventfully, with a complete pathologic examination revealing normal exocrine pancreatic tissue (PH type 2) without malignant transformation. We report a case of heterotopic pancreas manifesting as severe gastric outlet obstruction, in addition to a thorough diagnostic workup and surgical follow-up, in a young adult. Differential diagnoses and features that speak to benignity of a large, symptomatic mass lesion (PH in particular) are discussed.

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