Heering G, Lin Z, Rosman M
… +3 more, Hara N, Shakil F, Georgostathis D
Case Rep Gastroenterol
· 2024 · PMID 38872730
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INTRODUCTION: AL amyloidosis can involve the gastrointestinal (GI) tract in a sporadic manner, affecting certain anatomical areas while sparing others. CASE PRESENTATION: Our patient with AL amyloidosis and confirmed col...INTRODUCTION: AL amyloidosis can involve the gastrointestinal (GI) tract in a sporadic manner, affecting certain anatomical areas while sparing others. CASE PRESENTATION: Our patient with AL amyloidosis and confirmed colonic involvement was found to have new odynophagia, GI bleeding, and imaging findings that might suggest AL amyloidosis. However, negative pathology results from esophageal biopsies suggested the patient's new ulcerations were more likely a side effect of her autologous stem cell transplant (SCT) and chemotherapy meant to target amyloidosis, as opposed to an effect of amyloid infiltration itself. CONCLUSION: GI involvement of amyloidosis requires a high degree of clinical suspicion and should be considered in patients with systemic diseases affecting the kidney, heart, and GI tract; however, when satisfactory biopsies obtained from endoscopy results are negative, other causes should be considered.
Bhatt SK, Ashcherkin N, Fanous J
… +2 more, Pai RK, Athale J
Case Rep Gastroenterol
· 2024 · PMID 38868155
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INTRODUCTION: Langerhans cell histiocytosis (LCH) is a rare hematologic condition which can affect multiple organ systems and has variable presentation. LCH is more commonly seen as a malignancy of childhood. LCH in adul...INTRODUCTION: Langerhans cell histiocytosis (LCH) is a rare hematologic condition which can affect multiple organ systems and has variable presentation. LCH is more commonly seen as a malignancy of childhood. LCH in adulthood can have poor outcomes depending on the involvement of critical organs. CASE PRESENTATION: We report a case of a 71-year-old female who presented with progressive weakness, weight loss, diarrhea, and jaundice, and had been undergoing outpatient workup for elevated liver enzymes for the last 2 years. She required admission to the intensive care unit for vasodilatory shock, requiring vasopressor and chronotropic support. Imaging showed an underlying multiorgan process involving the gastrointestinal tract, liver, spleen, and central nervous system. A repeat liver biopsy after a prior inconclusive one revealed the diagnosis of multisystem LCH presenting as secondary sclerosing cholangitis. CONCLUSION: The uniqueness of this multisystem LCH case lies not only in its rarity but also in the diagnostic journey that necessitated a repeat biopsy for a conclusive diagnosis. Early identification and targeted intervention can help in ensuring better patient outcomes, especially when the presentation can overlap with various other possible conditions.
Bagger-Jörgensen H, Abdulrasak M, Sandeman K
… +2 more, Binsalman M, Sjöberg K
Case Rep Gastroenterol
· 2024 · PMID 38751992
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INTRODUCTION: Lichen planus is a relatively common inflammatory condition of the nails, skin, and mucosal surfaces. Oesophageal involvement of lichen planus is thought to be very rare, mainly described in case reports, b...INTRODUCTION: Lichen planus is a relatively common inflammatory condition of the nails, skin, and mucosal surfaces. Oesophageal involvement of lichen planus is thought to be very rare, mainly described in case reports, but is associated with a high risk of oesophageal stenosis as well as squamous cell carcinoma. No evidence-based treatment recommendations exist, with the majority of described treatment regimens involving systemic immunosuppression. CASE REPORT: In this case report, we describe a novel approach in treating oesophageal lichen planus in a patient with budesonide orodispersible tablets, a treatment normally reserved for eosinophilic oesophagitis. The patient achieved complete relief of dysphagia, with a follow-up oesophagogastroduodenoscopy 2 months after treatment commencement being macroscopically and microscopically free of inflammatory activity. This case report is to our knowledge the first to report this treatment regimen in oesophageal lichen planus. CONCLUSION: We consider a trial of budesonide orodispersible tablets a reasonable initial management as it's a local therapy specific to the oesophagus with a more benign side effect profile than systemic immunosuppression, but further studies need to be undertaken to corroborate our findings. Also, based on the severity and malignant potential of oesophageal lichen planus, we suggest that physicians be liberal in ordering oesophagogastroduodenoscopy with biopsy taking as part of the workup of dysphagia in a patient with known lichen planus.
Alkurdi A, Herrmann J, Bikmukhametov D
… +1 more, Tschöpe R
Case Rep Gastroenterol
· 2024 · PMID 38737441
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INTRODUCTION: Secondary sclerosing cholangitis in critically ill patients (SSC-CIP) is a rare but underdiagnosed entity that occurs after life-threatening events and treatment in the intensive care unit (ICU). The etiolo...INTRODUCTION: Secondary sclerosing cholangitis in critically ill patients (SSC-CIP) is a rare but underdiagnosed entity that occurs after life-threatening events and treatment in the intensive care unit (ICU). The etiology of SSC-CIP is not fully understood but may be caused by ischemic bile duct injury. SSC-CIP is a cholestatic liver disease that rapidly progresses to liver cirrhosis, with a high mortality rate in the first year of 50%. Endoscopic retrograde cholangiopancreatography (ERCP), which is the gold standard for diagnosing SSC-CIP, shows primary SC-like changes, usually in the intrahepatic bile ducts. Biliary cast formation is pathognomonic for SSC-CIP. No proven effective conservative treatment is available for SSC-CIP, and liver transplantation is the only curative therapy when liver cirrhosis or recurrent cholangitis occurs. CASE PRESENTATION: We report the case of a 47-year-old male patient who developed cholestasis after a long treatment in the ICU for severe pneumonia. ERCP showed characteristic findings with rarefication and multiple segmental stenosis in the intrahepatic bile ducts. We removed multiple biliary casts from the bile ducts. CONCLUSION: SSC-CIP should be considered for ICU patients with unclear cholestasis, especially when the cholestasis persists after recovery from the underlying disease. Early diagnosis is important to achieve better outcomes; without liver transplantation, the prognosis is generally poor.
Brakenhoff SM, Chi H, Friederich P
… +5 more, Doukas M, den Hoed C, Flink HJ, de Knegt RJ, de Man RA
Case Rep Gastroenterol
· 2024 · PMID 38681803
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INTRODUCTION: Emerging evidence suggests that long-term nucleos(t)ide analogue (NA) therapy can be ceased in a selective group of chronic hepatitis B (CHB). This is being gradually implemented in clinical practice. CASE...INTRODUCTION: Emerging evidence suggests that long-term nucleos(t)ide analogue (NA) therapy can be ceased in a selective group of chronic hepatitis B (CHB). This is being gradually implemented in clinical practice. CASE PRESENTATION: A 68-year-old man known with a chronic hepatitis B e antigen-positive hepatitis B infection without signs of advanced liver fibrosis or cirrhosis was admitted with acute liver failure. Two months prior to his admission, he ceased his NA therapy. During the admission, NA therapy was restarted, but the liver function worsened. The patient was put on the high-urgency liver transplantation waiting list, and the next day, he was successfully transplanted. However, the patient died 17 days later due to hemorrhagic shock that resulted from intra-abdominal bleeding and acute pancreatitis. CONCLUSION: Current guidelines suggest that NA therapy can be discontinued in a selective group of CHB patients. However, these guidelines suggest different stopping and follow-up criteria. This case illustrates that NA withdrawal is not without risks and that these differences in recommendations may lead to inadequate management and eventually a fatal outcome.
Seki T, Osone K, Ogawa H
… +5 more, Okada T, Shiraishi T, Sohda M, Shirabe K, Saeki H
Case Rep Gastroenterol
· 2024 · PMID 38665146
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INTRODUCTION: Crohn's disease (CD) is complicated by intestinal strictures and fistula formation; however, intestinal perforation is relatively rare. CASE PRESENTATION: Following a traffic accident in the evening, a 39-y...INTRODUCTION: Crohn's disease (CD) is complicated by intestinal strictures and fistula formation; however, intestinal perforation is relatively rare. CASE PRESENTATION: Following a traffic accident in the evening, a 39-year-old woman experienced abdominal pain that worsened the following morning and was taken to the emergency department. She had a 17-year history of CD and eight endoscopic balloon dilations for descending colonic strictures. She presented with a high fever of 40.0°C, along with tenderness and rebound pain throughout her abdomen, with the most substantial point being in the lower left abdomen. Computed tomography showed thickening of the descending colon wall, increased fat concentration around the wall, and a slight presence of air in the mesentery near the intestinal wall. We diagnosed the patient with generalized peritonitis due to traumatic penetration of the mesentery of the descending colon and performed emergency surgery. Intraoperative observation of the abdominal cavity with a laparoscope revealed purulent ascites but no apparent perforation or edematous mesentery, with white moss and redness in the descending colon. This prompted the decision to perform peritoneal lavage drainage and a transverse colonic double colostomy. The postoperative course was favorable, and the patient was discharged from the hospital on the postoperative day 14. Four months after discharge, colostomy closure was performed. CONCLUSION: Relatively minor trauma in patients with CD can result in colon injury. An injured bowel is usually accompanied by active lesions due to CD; however, caution is required, as endoscopic balloon dilatation without accompaniment may be a background factor.
Tan R, Abasszade JH, Dhillon H
… +3 more, Kuan CC, Worland T, Tabatabai S
Case Rep Gastroenterol
· 2024 · PMID 38665145
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INTRODUCTION: Olmesartan, an angiotensin II receptor antagonist, is associated with an uncommon complication of enteropathy that presents insidiously, usually months to years after initial commencement of anti-hypertensi...INTRODUCTION: Olmesartan, an angiotensin II receptor antagonist, is associated with an uncommon complication of enteropathy that presents insidiously, usually months to years after initial commencement of anti-hypertensive therapy which can be dose-dependent. It has a variable spectrum of clinical presentation but commonly presents as a moderate to severe malabsorptive process with potential severe complications related to poor end-organ perfusion. Lymphocytic gastritis and microscopic colitis are often noted in patients presenting with olmesartan-induced enteropathy; however, hepatic involvement has been less frequently observed. CASE PRESENTATION: We illustrate a case of a 43-year-old female presenting with 2 weeks of profuse non-bloody diarrhea in the context of olmesartan enteropathy which was complicated by an acute severe ischemic and enteropathic hepatopathy. CONCLUSION: Our case prompts clinicians to maintain a high index of suspicion in cases presenting with a seronegative enteropathy and concurrent acute liver injury while on olmesartan therapy. Cessation of olmesartan therapy resulted in prompt resolution of diarrheal symptoms and normalization of the acute transaminitis on subsequent three-week follow-up.
Arzivian A, Jones B, Joshua F
… +4 more, Paul M, Lynch T, Brown M, Gasiorowski R
Case Rep Gastroenterol
· 2024 · PMID 38645407
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INTRODUCTION: Whipple's disease is a rare condition that can present with atypical and non-specific features requiring a high index of suspicion for diagnosis. CASE PRESENTATION: We present a case of a man in his 40s wit...INTRODUCTION: Whipple's disease is a rare condition that can present with atypical and non-specific features requiring a high index of suspicion for diagnosis. CASE PRESENTATION: We present a case of a man in his 40s with peripheral arthritis and bilateral sacro-ileitis for 4-5 years that was treated with an anti-tumour necrosis factor therapy, which led to worsening of his symptoms, elevation of the inflammatory markers, and the development of fever, night sweats, anorexia, and a significant weight loss. The patient had no abdominal pain, diarrhoea, or other gastrointestinal symptoms. An FDG-PET scan showed increased uptake in the stomach and caecum. Endoscopic examination showed inflammatory changes in the stomach and normal mucosa of the duodenum, jejunum, terminal ileum, caecum, and colon. Histopathology was inconclusive, but the diagnosis was confirmed with PCR testing. He had no neurological symptoms, but cerebrospinal fluid PCR was positive. He was treated with intravenous ceftriaxone 2 g daily for 4 weeks, followed by trimethoprim/sulfamethoxazole 160/800 mg twice daily for 1 year with close monitoring and follow-up. CONCLUSION: This case presents an atypical and challenging presentation of Whipple's disease and the importance of proactive testing for neurological involvement.
Huang S, Shah JM, Quintero E
… +3 more, Xiao P, Asarian A, Reddy M
Case Rep Gastroenterol
· 2024 · PMID 38645406
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INTRODUCTION: Mesenteric fibromatosis (intra-abdominal desmoid tumor) is rare, with only a few cases reported in the literature. Clinical symptoms range from asymptomatic, nausea, early satiety, abdominal pain, and gastr...INTRODUCTION: Mesenteric fibromatosis (intra-abdominal desmoid tumor) is rare, with only a few cases reported in the literature. Clinical symptoms range from asymptomatic, nausea, early satiety, abdominal pain, and gastrointestinal bleeding. Although histologically benign, such a tumor may become locally invasive, and aggressive forms contribute to significant morbidity and mortality. CASE PRESENTATION: We report the case of a 52-year-old West African male with a 1-year history of intermittent hematochezia and intermittent bloating. Colonoscopy revealed a 4-mm rectal polyp and internal hemorrhoids. Esophagogastroduodenoscopy revealed a severe duodenal stricture 4-5 cm distal to the ampulla. Further work-up with contrast-enhanced computed tomography of the abdomen and pelvis revealed a 5.0 × 3.7 × 4.3-cm mass within the mesentery, encasing the distal portion of the duodenum. Exploratory laparotomy was performed, and the mass was excised from the jejunum. Histopathology findings and immunohistochemical analysis revealed the diagnosis to be mesenteric fibromatosis (desmoid tumor), positive for nuclear β-catenin and SMA, and negative expression of STAT6, desmin, caldesmon, pan-cytokeratin, or c-KIT. The Ki67 index is <1%. CONCLUSION: This case report highlights the diagnostic challenges of mesenteric fibromatosis due to its nonspecific clinical presentation. Recognizing uncommon presentations of mesenteric fibromatosis and risk factors aids in early diagnosis, management, and treatment. Importantly, this also aids in the prevention of complications such as intestinal obstruction, bowel ischemia, and fistula formation.
Ohta M, Fukushima K, Nishimaki H
… +3 more, Morita T, Ito Y, Namiki K
Case Rep Gastroenterol
· 2024 · PMID 38628808
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INTRODUCTION: Portal annular pancreas (PAP) is a congenital anomaly resulting from aberrant fusion of the ventral and dorsal pancreatic buds around the portal vein (PV). PAP was classified into three types by Joseph et a...INTRODUCTION: Portal annular pancreas (PAP) is a congenital anomaly resulting from aberrant fusion of the ventral and dorsal pancreatic buds around the portal vein (PV). PAP was classified into three types by Joseph et al., based on the location of the main pancreatic duct around the PV. The presence of PAP is important for the surgical procedure because it is associated with the postoperative pancreatic fistula. There are no standardized surgical procedures of resection and reconstruction for PAP. CASE PRESENTATION: We report 2 cases of subtotal stomach-preserving pancreatoduodenectomy in patients with PAP. One case of PAP was discovered coincidentally intraoperatively, and the other case was diagnosed before surgery. The first case was an 84-year-old male patient who underwent surgery for distal bile duct cancer. PAP was noticed intraoperatively when the uncinate process of the pancreas was detached from behind the PV. The second case was an 84-year-old female patient who also underwent surgery for distal bile duct cancer. We recognized PAP from preoperative computed tomography images. In both cases, the ductal anatomy was consistent with type IIIA PAP, and the dorsal pancreas was resected using a stapling device. During the postoperative period, there was no clinically relevant postoperative pancreatic fistula. CONCLUSION: PAP is rarely encountered intraoperatively; however, it is important to recognize it before surgery and take it into consideration when deciding upon the procedures for resection and reconstruction.
Alsalman J, Alqourain A, Alyousef M
… +2 more, Aldandan O, Alzahrani I
Case Rep Gastroenterol
· 2024 · PMID 38590835
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INTRODUCTION: Ampullary neuroendocrine tumors (NETs) are usually diagnosed in the 5th-6th decades of life, and no cases were reported in <20 years of age. We report a rare case, presenting at a very young age, of well-di...INTRODUCTION: Ampullary neuroendocrine tumors (NETs) are usually diagnosed in the 5th-6th decades of life, and no cases were reported in <20 years of age. We report a rare case, presenting at a very young age, of well-differentiated NET involving the ampulla of Vater with lymph node metastasis. CASE PRESENTATION: An 18-year-old man presented with a 3-month history of upper abdominal pain and jaundice. Abdominal ultrasound showed a dilated common bile duct, and endoscopic retrograde cholangiopancreatography revealed two duodenal polypoid lesions, one of them overlying the ampulla of Vater, with an erythematous and ulcerated surface. Histopathological examination confirmed the diagnosis of NET grade 1. Octreotide scan revealed 2 para-aortic lymph nodes with intense radiotracer uptake. The patient had undergone Whipple surgery with para-aortic lymph node dissection. Histopathological examination of the surgical specimens was confirmatory of NET grade 2 and paraganglioma in a few of the dissected lymph nodes. Postoperatively, the patient was kept on monthly intramuscular octreotide. Follow-up gallium-68 DOTATATE is unremarkable apart from an avid left para-aortic lymph node which is showing stability over 12 months of follow-up. CONCLUSION: This case demonstrates that NETs of the ampulla of Vater can present at a very young age. Radical surgical excision with extended lymph node dissection and postoperative octreotide is associated with better patient outcomes and survival.
Case Rep Gastroenterol
· 2024 · PMID 38585022
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INTRODUCTION: Caustic injuries remain a major public health concern. Nutritional status plays a pivotal role in determining the outcome. Unfortunately, nutritional care guidelines are not widely implemented in clinical p...INTRODUCTION: Caustic injuries remain a major public health concern. Nutritional status plays a pivotal role in determining the outcome. Unfortunately, nutritional care guidelines are not widely implemented in clinical practice, and decisions are often based on prior experience and local policies. CASE PRESENTATION: We present the case of an 83-year-old man who accidentally ingested alkali, resulting in severe caustic injury and subsequent complications that further deteriorated his nutritional status. The management of esophageal strictures necessitated constant adjustments to the nutritional strategies employed. The clinical evaluation revealed protein and energy malnutrition, accompanied by type 2 intestinal failure. However, with individually tailored parenteral nutritional therapy, a significant improvement in the patient's nutritional status was observed. CONCLUSION: Recognizing that caustic injuries increase metabolic demands, a comprehensive and active nutritional assessment is crucial, focusing on the need for adequate energy, high protein intake, and an appropriate feeding route. In cases of acute or prolonged type 2 intestinal failure with insufficient oral or enteral nutrition, parenteral feeding should be the primary therapy. Effective management of caustic injuries requires a multidisciplinary and multicenter approach, integrating nutritional evaluation, including body composition measurements, into the clinical algorithm. Early initiation of nutritional therapy is vital to prevent chronic intestinal failure.
Yoneda A, Ogata R, Ryu S
… +9 more, Yoshino K, Fukui S, Ikeda T, Kitasato A, Sugiyama N, Takeshita H, Minami S, Ito M, Kuroki T
Case Rep Gastroenterol
· 2024 · PMID 38550658
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INTRODUCTION: Primary malignant melanoma of the esophagus is a very rare disease with a poor prognosis. We herein report a patient with primary malignant melanoma of the esophagus who underwent surgical resection. CASE P...INTRODUCTION: Primary malignant melanoma of the esophagus is a very rare disease with a poor prognosis. We herein report a patient with primary malignant melanoma of the esophagus who underwent surgical resection. CASE PRESENTATION: A 73-year-old female underwent an upper gastrointestinal endoscopy during follow-up for colonic diverticulitis. An endoscopic examination and constructed radiography revealed a slightly elevated black pigmented lesion in the upper esophagus and a black pigmented area in the esophagogastric junction. Through a preoperative endoscopic biopsy, she was diagnosed with malignant melanoma of the esophagus. We performed thoracoscopy-assisted and laparoscopy-assisted subtotal esophagectomy with lymphadenectomy. The surgical specimens were subjected to immunohistochemical analysis, resulting in a diagnosis of malignant melanoma. The tumor cells were positive for Melan-A and HMB-45 diffusely, supporting that diagnosis. We performed surgical resection in a case of primary malignant melanoma of the esophagus, and the patient has remained disease free for 2 years since the surgery. CONCLUSION: Early diagnosis and radical resection may be essential for long-term survival in patients with malignant melanoma of the esophagus.
Case Rep Gastroenterol
· 2024 · PMID 38545369
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INTRODUCTION: Polyethylene glycol 3,350 and electrolytes is a commonly prescribed bowel regimen for colonoscopy preparation with an overall excellent safety profile, though prior reports have demonstrated risk of volume...INTRODUCTION: Polyethylene glycol 3,350 and electrolytes is a commonly prescribed bowel regimen for colonoscopy preparation with an overall excellent safety profile, though prior reports have demonstrated risk of volume overload. CASE PRESENTATION: A 55-year-old man with significant cardiopulmonary co-morbidities was admitted for acute hypoxic respiratory failure and subsequent evaluation for lung transplant. As part of his pretransplant evaluation, colon cancer screening was advised. Despite multiple days of bowel preparation, his stools contained sediment. Unfortunately, he developed pulmonary edema due to prolonged bowel preparation. CONCLUSION: While bowel preparation is considered generally safe, our case report highlights the importance of judicious use with monitoring in high-risk individuals.
Ando T, Nitta H, Umemura A
… +7 more, Katagiri H, Kanno S, Takeda D, Nishiya M, Uesugi N, Sugai T, Sasaki A
Case Rep Gastroenterol
· 2024 · PMID 38545368
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INTRODUCTION: Autoimmune pancreatitis (AIP) is recognized as a disease with a good prognosis that responds well to steroids, but the complication of pancreatic ductal adenocarcinoma (PDAC) in AIP is a rare condition. We...INTRODUCTION: Autoimmune pancreatitis (AIP) is recognized as a disease with a good prognosis that responds well to steroids, but the complication of pancreatic ductal adenocarcinoma (PDAC) in AIP is a rare condition. We report a case of PDAC encapsulated by tumor-forming type 1 AIP. CASE PRESENTATION: The patient, a 65-year-old female, was found to have high CA19-9 levels and a pancreatic mass with a diameter of 30 mm on abdominal ultrasonography. Contrast-enhanced computed tomography revealed a 40-mm mass in the tail of the pancreas that had a 27-mm oligemic mass inside it. From these work-up examinations, this tumor was diagnosed as PDAC, with evidence of colonic invasion. As curative resection for PDAC, a distal pancreatectomy with splenectomy and combined colon resection were performed. Histopathological examination showed invasive PDAC surrounded by IgG4-positive plasma cell infiltration. Based on these findings, a diagnosis was made of PDAC located in the pancreatic tail capsulized by type 1 AIP. The postoperative course was uneventful, and the patient was discharged on postoperative day 15. She underwent postoperative adjuvant chemotherapy with S-1 for 6 months, and no recurrence was noted for 2 years after operation. CONCLUSION: Currently, there are two hypothetical mechanisms of PDAC induction by AIP: (1) carcinogenic stimulation due to chronic inflammation and (2) paraneoplastic syndrome caused by AIP. Further study of the relationship between AIP and pancreatic cancer is needed, and follow-up should be conducted while keeping in mind the possibility of complications.
Case Rep Gastroenterol
· 2024 · PMID 38532800
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INTRODUCTION: We report the first case of a choledocholithiasis migrating into the main pancreatic duct (MPD) due to catheter manipulation during endoscopic retrograde cholangiopancreatography (ERCP). CASE PRESENTATION:...INTRODUCTION: We report the first case of a choledocholithiasis migrating into the main pancreatic duct (MPD) due to catheter manipulation during endoscopic retrograde cholangiopancreatography (ERCP). CASE PRESENTATION: A 57-year-old woman complaining of vomiting was diagnosed with acute cholangitis and pancreatitis due to choledocholithiasis. During ERCP, the stone migrated from the papilla into the MPD due to the pushing motion of the catheter. However, the ERCP session was completed after biliary sphincterotomy without intervention in the MPD because the migration was not noticed. The migrated stone became apparent on computed tomography the following day. The second ERCP revealed the stone measuring 5 mm in the MPD. After pancreatic sphincterotomy, a pancreatic stent was placed, which improved the obstructive pancreatitis. CONCLUSION: Endoscopists performing ERCP should be aware of this rare but serious complication.
Fiske HW, Saeed F, Ward C
… +5 more, Sinayuk B, Ulici V, Curry M, Feller E, Shah SA
Case Rep Gastroenterol
· 2024 · PMID 38532799
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INTRODUCTION: Hepatobiliary overlap syndromes describe the coinciding presentation of more than one immune-mediated biliary and liver disease in a single patient and present complex challenges in diagnosis and treatment....INTRODUCTION: Hepatobiliary overlap syndromes describe the coinciding presentation of more than one immune-mediated biliary and liver disease in a single patient and present complex challenges in diagnosis and treatment. We report a case of ulcerative colitis with primary sclerosing cholangitis and autoimmune hepatitis overlap syndrome responsive to vancomycin. CASE PRESENTATION: The patient is a 30-year-old female with known ulcerative pancolitis and autoimmune hepatitis. She presented to the emergency department with a constellation of gastrointestinal symptoms, including diffuse lower abdominal pain, bloody diarrhea, and nausea with bilious vomiting. Subsequent imaging revealed the additional diagnosis of primary sclerosing cholangitis, and she was diagnosed with overlap syndrome. Multiple treatment regimens were trialed with minimal improvement. She eventually achieved normalization of both clinical status and biochemical markers after the addition of vancomycin. CONCLUSION: Vancomycin is an underutilized therapy; its potential role in primary sclerosing cholangitis and autoimmune hepatitis overlap syndrome has not been previously reported.
Bapir R, Qader D, Gharib D
… +9 more, Tahir S, Abdullah A, Asaad H, Ahmed S, Ali H, Abdullah H, Ahmed S, Hussein Kakamad F, Aghaways I
Case Rep Gastroenterol
· 2024 · PMID 38524872
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INTRODUCTION: Gastric duplication cyst (GDC) is a rare congenital anomaly of the gastrointestinal tract. Though GDC is often misdiagnosed, misidentification as an adrenal cyst has rarely been reported. Herein, we report...INTRODUCTION: Gastric duplication cyst (GDC) is a rare congenital anomaly of the gastrointestinal tract. Though GDC is often misdiagnosed, misidentification as an adrenal cyst has rarely been reported. Herein, we report a case of GDC in a young female mimicking an adrenal cyst. CASE PRESENTATION: A 17-year-old female presented with chronic epigastric pain, nausea, and intermittent vomiting. Physical examinations revealed mild tenderness in the epigastric region. Esophagogastroduodenoscopy showed no abnormality. Ultrasound, contrast-enhanced computed tomography scan, and MRI of the abdomen and pelvis showed an oval-shaped left adrenal cystic lesion measuring 33 × 26 mm. Preoperative blood investigations and hormonal assessments were normal. Laparoscopy showed that the cyst originated from the greater curvature of the stomach. The left adrenal gland was normal. After an intraoperative consultation with a gastrointestinal surgeon, a wedge resection of the cyst was performed. Histopathology confirmed the gastric duplication cyst. CONCLUSION: GDCs are rare congenital malformations that may become symptomatic during adulthood. They can mimic adrenal cysts and lead to misdiagnosis.
Salam R, Verma A, Noeske M
… +3 more, Alnimer L, Sieloff EM, Piper MS
Case Rep Gastroenterol
· 2024 · PMID 38501151
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INTRODUCTION: Pyogenic liver abscess is a noteworthy health concern in North America, characterized by a mortality rate ranging from 2 to 12%. This condition is often polymicrobial, with species and as the predominant...INTRODUCTION: Pyogenic liver abscess is a noteworthy health concern in North America, characterized by a mortality rate ranging from 2 to 12%. This condition is often polymicrobial, with species and as the predominant causal pathogens in Western countries. species, typically commensals of gastrointestinal, genital, and oral flora, have been implicated in the rare formation of tonsillar abscesses and Lemierre syndrome, including its gastrointestinal variant known as pylephlebitis. CASE PRESENTATION: We present the case of an immunocompetent male with a 2-week history of abdominal distention and pain. Abdominal magnetic resonance imaging revealed multiseptated cystic hepatic masses and portal vein thrombosis. A subsequent liver biopsy confirmed etiology. The patient was initiated on intravenous cefepime and oral metronidazole antibiotics. Unfortunately, the patient succumbed to cardiac arrest before a final diagnosis could be established. CONCLUSION: species-associated liver abscess, coupled with the rare gastrointestinal variant of Lemierre syndrome (pylephlebitis), poses a significant mortality risk. This case underscores the rarity and clinical challenges associated with these conditions. Increased awareness among clinicians is crucial for early diagnosis and prompt intervention, potentially improving outcomes in such cases.
Hakoda H, Kawaguchi Y, Miyata Y
… +4 more, Togashi J, Nagai M, Suzuki Y, Nomura Y
Case Rep Gastroenterol
· 2024 · PMID 38501150
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INTRODUCTION: Adenosquamous carcinoma (ASC) of the ampulla of Vater (AmV) is rare. The prognosis is generally worse in patients undergoing resection of ASC of the AmV than in those undergoing resection of adenocarcinoma...INTRODUCTION: Adenosquamous carcinoma (ASC) of the ampulla of Vater (AmV) is rare. The prognosis is generally worse in patients undergoing resection of ASC of the AmV than in those undergoing resection of adenocarcinoma of the AmV because the former shows early recurrence after surgery. A treatment strategy for ASC of the AmV has not been established, and the efficacy of adjuvant chemotherapy after curative resection is unclear. Given the paucity of data, we report a case of ASC of the AmV that was curatively resected and treated with adjuvant chemotherapy. CASE PRESENTATION: A 66-year-old man presented with pruritus and anorexia. Contrast-enhanced computed tomography revealed a tumor measuring 1.6 cm in diameter located at the AmV and distal bile duct. Biopsy revealed adenocarcinoma of the AmV. The patient underwent subtotal stomach-preserving pancreaticoduodenectomy. Histopathological examination contradictorily revealed ASC of the AmV and lymph node metastases. The postoperative course of the patient was uneventful, and he was discharged on day 25. The patient underwent S-1 adjuvant chemotherapy for 6 months and did not exhibit any postoperative recurrence for a follow-up duration of 28 months. CONCLUSION: Although treatment strategy for ASC of the AmV has not been established, our case shows that surgery followed by S-1 adjuvant chemotherapy could improve prognosis of patients with such tumors. However, further research is required to determine the efficacy of adjuvant chemotherapy and treatment strategies for resectable ASC of the AmV.