Yang CC, Chen JY, Chang HY
… +4 more, Sheu MJ, Feng IC, Wang SH, Kuo HT
Case Rep Gastroenterol
· 2024 · PMID 38501149
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INTRODUCTION: Syphilis, an ancient sexually transmitted disease, is recognized as a systemic infection disease manifesting with diverse symptoms and variations. Secondary syphilis characterized by systemic symptoms resul...INTRODUCTION: Syphilis, an ancient sexually transmitted disease, is recognized as a systemic infection disease manifesting with diverse symptoms and variations. Secondary syphilis characterized by systemic symptoms resulted from hematogenous and lymphatic dissemination of the infection, may include manifestations such as hepatitis and nephrotic syndrome. However, the simultaneous occurrence of hepatitis and nephrotic syndrome in secondary syphilis is rare. CASE PRESENTATION: A young man presented with fatigue, abnormal liver function tests, and hyperbilirubinemia and had history of men who have sex with men (MSM). Serological tests confirmed the diagnosis of secondary syphilis, and kidney biopsy indicated membranous nephritis. After antibiotic treatment, the patient experienced resolution of proteinuria, and liver enzyme levels returned to normal. CONCLUSION: Syphilis should be considered in the differential diagnosis of simultaneous liver and kidney dysfunction, particularly in patients engaging in high-risk sexual behavior. This case highlights the importance of considering syphilis in young patients with MSM and presenting with unexplained nephrotic syndrome and liver abnormalities.
Hirata T, Kawaguchi S, Akamatsu T
… +8 more, Inagawa A, Hikichi T, Ohkawa K, Asahara K, Satoh T, Endo S, Suzuki M, Ohno K
Case Rep Gastroenterol
· 2024 · PMID 38476646
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INTRODUCTION: Solid organ malignancies rarely metastasize to the duodenal papilla. We describe a case of primary lung cancer with duodenal papillary metastasis in a patient who presented with melena. To the best of our k...INTRODUCTION: Solid organ malignancies rarely metastasize to the duodenal papilla. We describe a case of primary lung cancer with duodenal papillary metastasis in a patient who presented with melena. To the best of our knowledge, this is only the second report of duodenal papillary metastasis from lung cancer. CASE PRESENTATION: A 65-year-old woman presented with complaints of anorexia, weight loss, and black stool. Imaging studies led to a clinical diagnosis of stage IVB lung cancer, and anticoagulants were initiated to treat pulmonary artery thrombosis. However, endoscopic hemostasis was challenging because of bleeding from a duodenal papillary tumor. Fortunately, the patient was positive for the plasma epidermal growth factor receptor (EGFR) gene mutation, and osimertinib, an EGFR tyrosine kinase inhibitor, was administered, successfully achieving hemostasis. Subsequently, endoscopic ultrasonography-guided transbronchial needle aspiration of an enlarged mediastinal lymph node and duodenal papillary tumor biopsy confirmed duodenal papillary metastasis of the primary lung adenocarcinoma. CONCLUSION: Although duodenal papillary metastasis is extremely rare, a good clinical outcome was achieved in this case by considering duodenal papillary metastasis from lung cancer as the differential diagnosis and administering systemic osimertinib therapy.
Case Rep Gastroenterol
· 2024 · PMID 38464456
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INTRODUCTION: Biliopancreatic diversion with duodenal switch (BPD/DS) is an uncommon type of bariatric surgery that can rarely lead to bleeding in the biliopancreatic limb. The altered anatomy poses significant diagnosti...INTRODUCTION: Biliopancreatic diversion with duodenal switch (BPD/DS) is an uncommon type of bariatric surgery that can rarely lead to bleeding in the biliopancreatic limb. The altered anatomy poses significant diagnostic and therapeutic challenges. CASE PRESENTATION: We present an unusual case of a woman status post-BPD/DS nearly a decade ago who presented with gastrointestinal bleeding in the duodenum of the biliopancreatic limb, a rare phenomenon given the unique surgery. CONCLUSION: We illustrate a promising minimally invasive option of successfully treating the bleeding by interventional radiology (IR) embolization as an alternative to more invasive and challenging options of balloon-assisted enteroscopy, lumen-apposing metal stent placement and surgical intraoperative enteroscopy.
Kimura K, Ichita C, Sumida C
… +5 more, Nishino T, Nagayama M, Kubota J, Hirose H, Sasaki A
Case Rep Gastroenterol
· 2024 · PMID 38455226
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INTRODUCTION: Cold snare polypectomy (CSP) is a procedure with a low risk of complications. Here, we present our experience of a rare case of submucosal abscess following CSP in an immunosuppressed patient. CASE PRESENTA...INTRODUCTION: Cold snare polypectomy (CSP) is a procedure with a low risk of complications. Here, we present our experience of a rare case of submucosal abscess following CSP in an immunosuppressed patient. CASE PRESENTATION: Seventy-eight-year-old man underwent CSP, developing a fever, chills, and right lower abdominal pain 8 days later. Ultrasound and computed tomography revealed wall thickening of the ascending colon, presenting as whitening and thickening of the same region, and excretion of pus was observed after biopsy. The diagnosis was made as phlegmonous colitis, for which antibiotic therapy was commenced. The patient was diagnosed with chronic myelomonocytic leukemia (CMML) during admission. We considered the following reasons as possible causes of infectious complications after CSP: (1) the patient had a highly immunosuppressed state with comorbidities such as CMML as well as diabetes mellitus and (2) disruption of the mucosal barrier occurred during endoscopic resection. CONCLUSION: Although CSP is generally considered safe, our case highlights the potential for serious complications in immunosuppressed patients. Therefore, the decision to perform CSP in such patients should be made with caution to avoid unnecessary interventions. In instances where treatment is essential, thorough bowel preparation and prophylactic antibiotic use may be necessary to mitigate the risks.
Yoshiaki M, Minagawa N, Kato T
… +5 more, Okada N, Suzuki T, Ishizuka C, Fukuda A, Mori Y
Case Rep Gastroenterol
· 2024 · PMID 38439819
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INTRODUCTION: We encountered a colon cancer case with a very rare anomaly of the middle colic artery (MCA) originating from the splenic artery (SA). CASE PRESENTATION: A woman was referred to our hospital for transverse...INTRODUCTION: We encountered a colon cancer case with a very rare anomaly of the middle colic artery (MCA) originating from the splenic artery (SA). CASE PRESENTATION: A woman was referred to our hospital for transverse colon cancer. Three-dimensional computed tomography (3D-CT) angiography showed an anomalous MCA originating from the SA rather than from the superior mesenteric artery (SMA) as is typical. Laparoscopic left hemicolectomy with D3 lymph node dissection was performed. The lymph nodes around the SMA were dissected from the caudal view, confirming the absence of a typical MCA. An anomalous SA-originating MCA was identified just below the pancreas, where it was clipped and ligated; subsequently, total mesenteric excision was achieved. CONCLUSION: As D3 lymph node dissection for transverse colon cancer is technically difficult, 3D-CT angiography is useful for identifying vascular anomalies preoperatively, thereby avoiding intraoperative injury. This is the first case report of laparoscopic colectomy associated with a SA-originating MCA anomaly.
Case Rep Gastroenterol
· 2024 · PMID 38439818
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INTRODUCTION: Precut fistulotomy is of interest as one of the salvage techniques for selective bile duct cannulation using endoscopic retrograde cholangiopancreatography. Of the various endoscopic treatments reported to...INTRODUCTION: Precut fistulotomy is of interest as one of the salvage techniques for selective bile duct cannulation using endoscopic retrograde cholangiopancreatography. Of the various endoscopic treatments reported to date for bleeding associated with papillotomy incision, endoscopic hemostasis treatment with a novel self-assembling peptide (SAP) matrix-forming gel (TDM-621) (3-D Matrix Ltd., Tokyo, Japan) remains only insufficiently reported in the literature. CASE PRESENTATION: We herein report 6 cases of precut fistulotomy-related bleeding successfully treated with endoscopic hemostasis treatment with TDM-621, i.e., 5 and 1 cases during and after precut fistulotomy, respectively, in 2 males and 4 females aged 68-96 years (mean age, 85 years), 3 of whom had been on antithrombotic drugs. Types of bleeding treated included oozing bleeding ( = 5) and oozing bleeding from a visible vessel ( = 1). In all cases, complete hemostasis was achieved with TDM-621 without causing rebleeding. CONCLUSION: Endoscopic hemostasis with TDM-621 may prove effective for precut fistulotomy-related bleeding and represent a potential modality of first choice in hemostasis. In addition, endoscopic hemostasis with combined modality therapy using TDM-621 and endoscopic hemoclips may prove effective for bleeding from visible vessels.
Cmarková K, Nosáková L, Pindura M
… +3 more, Vojtko M, Bánovčin P, Miklušica J
Case Rep Gastroenterol
· 2024 · PMID 38439817
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INTRODUCTION: Benign lesions of the liver are very common findings, usually randomly discovered, especially during examinations for other indications. The frequent use of imaging modalities may be responsible for the sta...INTRODUCTION: Benign lesions of the liver are very common findings, usually randomly discovered, especially during examinations for other indications. The frequent use of imaging modalities may be responsible for the statistical increase in the incidence of these findings. CASE PRESENTATION: In this publication, we present the cases of 2 female patients with benign liver lesions, the occurrence of which is considered rare, and only a few dozen cases have been described worldwide. In both cases, clinical symptoms, diagnostic approach, and surgical treatment are presented. CONCLUSION: Due to increasing availability of imaging methods, the occurrence of previously considered rare benign liver lesions increases as well. In many cases, the malignant potential of these findings remains unclear. Decision-making process should include a multidisciplinary board.
Case Rep Gastroenterol
· 2024 · PMID 38410687
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INTRODUCTION: Hypophosphatemia occurs commonly in inflammatory bowel disease (IBD) patients and can cause considerable morbidity. The differential diagnoses in IBD include nutritional causes and hypophosphatemia induced...INTRODUCTION: Hypophosphatemia occurs commonly in inflammatory bowel disease (IBD) patients and can cause considerable morbidity. The differential diagnoses in IBD include nutritional causes and hypophosphatemia induced by some formulations of intravenous iron infusions. CASE PRESENTATION: We present the case of a 37-year-old man with active Crohn's disease, presenting with difficulty walking and fractures of the vertebrae and calcaneus. He had long-standing hypophosphatemia. Nutritional causes for hypophosphatemia were considered in the first instance given the presence of chronic diarrhea and vitamin D deficiency; however, there was minimal response to appropriate supplementation with oral phosphorous and vitamin D. Iron infusion-induced hypophosphatemia was then considered, but the nadir phosphate level preceded any iron infusion. Therefore, work-up was undertaken for less common causes. He was ultimately diagnosed with tumor-induced osteomalacia, caused by excess fibroblast growth factor 23 (FGF23) secretion from a phosphaturic mesenchymal tumor about the knee. He had complete resolution of symptoms and biochemical abnormalities following successful resection of the tumor. CONCLUSION: This case illustrates the approach to investigation of hypophosphatemia in IBD patients. If the time course and response to phosphate supplementation are not as expected for nutritional or iron infusion-induced hypophosphatemia, less common causes should be considered.
Higashi S, Nakabori T, Mukai K
… +11 more, Seiki Y, Watsuji K, Hirao T, Kawamoto Y, Urabe M, Kai Y, Takada R, Yamai T, Ikezawa K, Uehara H, Ohkawa K
Case Rep Gastroenterol
· 2024 · PMID 38371742
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INTRODUCTION: Portal vein aneurysm (PVA) is a rare saccular or fusiform portal vein dilatation. The management and optimal treatment of PVA remain unknown. CASE PRESENTATION: A 53-year-old man with hepatitis C virus (HCV...INTRODUCTION: Portal vein aneurysm (PVA) is a rare saccular or fusiform portal vein dilatation. The management and optimal treatment of PVA remain unknown. CASE PRESENTATION: A 53-year-old man with hepatitis C virus (HCV) infection was diagnosed with PVA measuring 28 mm in diameter. Under observation, his liver fibrosis progressed, and the PVA diameter gradually increased to 52 mm. The patient was treated with elbasvir-grazoprevir for 12 weeks, and HCV disappeared. After achieving sustained virological response, liver fibrosis improved and the PVA progression ceased. CONCLUSION: HCV clearance by direct-acting antiviral treatment not only regressed liver fibrosis but may have also restrained the progression of PVA in a patient with cirrhosis type C and PVA.
Case Rep Gastroenterol
· 2024 · PMID 38333765
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INTRODUCTION: Gastric adenocarcinoma with enteroblastic differentiation (GAED) is a rare histological type of gastric adenocarcinoma that occurs in the stomach and is known for its aggressive behavior. GAED is diagnosed...INTRODUCTION: Gastric adenocarcinoma with enteroblastic differentiation (GAED) is a rare histological type of gastric adenocarcinoma that occurs in the stomach and is known for its aggressive behavior. GAED is diagnosed histopathologically and is often advanced at the time of diagnosis. CASE PRESENTATION: We report the case of a 70-year-old male with a 20-mm superficial depressed lesion on the anterior wall of the antrum. Histological examination of the endoscopic submucosal dissection specimen revealed that the tumor was composed of dilated or slit-like branching tubules; additionally, the tumor cells had clear cytoplasm resembling that of the fetal digestive tract. Immunohistochemically, the tumor cells were positive for Glypican-3 and alpha-fetoprotein. A pathological diagnosis of GAEDs was established. GAED was found in approximately 30% of all the tumor cells and showed lymphatic invasion. The patient has been under recurrence-free follow-up for approximately 1 year after the endoscopic submucosal dissection. CONCLUSION: In order to detect a large number of cases, immunostaining should be aggressively performed if morphological findings are suspicious for GAED.
Ito H, Omura Y, Makuuchi T
… +12 more, Chou T, Ito A, Fujimoto R, Yokota M, Tsuda S, Nagata J, Hirose S, Yoshii H, Izumi H, Sugiyama T, Tajiri T, Suzuki T
Case Rep Gastroenterol
· 2024 · PMID 38322757
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INTRODUCTION: Hemobilia, which refers to bleeding from the bile duct, is rare and difficult to treat. We report a case of successful hemostasis of a pancreatic tumor complicated by hemobilia. CASE PRESENTATION: A 76-year...INTRODUCTION: Hemobilia, which refers to bleeding from the bile duct, is rare and difficult to treat. We report a case of successful hemostasis of a pancreatic tumor complicated by hemobilia. CASE PRESENTATION: A 76-year-old man was referred to our hospital with a pancreatic head tumor. Endoscopic retrograde cholangiopancreatography (ERCP) and endoscopic ultrasonography-FNA were performed, and the patient was diagnosed with pancreatic metastasis of renal cell carcinoma. After discharge, the patient noted worsening jaundice and progressive anemia and was readmitted. ERCP reveals active bleeding from the duodenal papillae. The patient was placed on a fully covered metallic stent and discharged after confirming hemostasis. CONCLUSION: Renal cell carcinoma is a tumor with abundant blood flow. If hemobilia occurs, bleeding from pancreatic metastatic tumors should be considered. Additionally, hemostasis using a fully covered metallic stent is useful for treating hemobilia in tumors.
Gopalakrishna H, Asif B, Rai A
… +5 more, Conjeevaram HS, Mironova M, Kleiner DE, Freeman AF, Heller T
Case Rep Gastroenterol
· 2024 · PMID 38304571
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INTRODUCTION: Prolidase deficiency is a rare autosomal recessive disorder caused by variants in the gene. Patients usually have multi-organ involvement and a wide range of clinical features including recurrent skin ulce...INTRODUCTION: Prolidase deficiency is a rare autosomal recessive disorder caused by variants in the gene. Patients usually have multi-organ involvement and a wide range of clinical features including recurrent skin ulcers, dysmorphic facial features, recurrent infections, intellectual disability, and splenomegaly. Studies have shown that patients with prolidase deficiency may have hepatic manifestations including hepatomegaly and abnormal liver enzymes. However, there is no detailed description of liver disease in this patient population. CASE PRESENTATION: Here, we present 3 patients with prolidase deficiency with varying extents of hepatic involvement. CONCLUSION: Prolidase deficiency patients with liver disease should be followed up long term to understand more about the pathophysiology and the impact of liver disease on long-term outcomes.
Watanabe A, Nishida T, Osugi N
… +11 more, Kitanaka T, Minoura Y, Okabe S, Sakamoto N, Fujii Y, Sugimoto A, Nakamatsu D, Matsumoto K, Yamamoto M, Adachi S, Fukui K
Case Rep Gastroenterol
· 2024 · PMID 38288397
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INTRODUCTION: Drug-induced liver injury (DILI) associated with 5-aminosalicylic acid (5-ASA) is a rare but potentially life-threatening adverse event. CASE PRESENTATION: We report the case of a 58-year-old woman with ulc...INTRODUCTION: Drug-induced liver injury (DILI) associated with 5-aminosalicylic acid (5-ASA) is a rare but potentially life-threatening adverse event. CASE PRESENTATION: We report the case of a 58-year-old woman with ulcerative colitis who developed DILI after initiating maintenance therapy with the multimatrix system 5-ASA. The patient presented with grade 4 liver enzyme elevation on day 98 after initiating 5-ASA and was admitted to the hospital. Blood tests revealed the mixed liver injury, and imaging studies showed no abnormalities except for mild lymph node enlargement. Liver biopsy revealed acute lobular hepatitis with interfacial activity. The patient's score on the International Autoimmune Hepatitis Group 1999 revised scoring system was a total score of 10, causing a suspicion for the diagnosis of autoimmune hepatitis. The DDW-J 2004 scale calculated a total score of six, indicating a high probability of DILI. We suspected DILI due to 5-ASA, and the 5-ASA formulations were discontinued. The patient was treated with ursodeoxycholic acid and neominophagen C, and her liver function gradually improved without steroid treatment. Finally, we definitively diagnosed DILI based on the pathological findings and clinical course after discontinuation of 5-ASA. CONCLUSION: This case highlights the importance of monitoring liver function in patients receiving 5-ASA therapy.
Abe K, Yamai D, Katsumi C
… +2 more, Oyamatsu M, Sato K
Case Rep Gastroenterol
· 2024 · PMID 38249996
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INTRODUCTION: A stoma prolapse is easy to diagnose by visual examination, and it rarely incarcerates. Therefore, manual reduction is usually performed as soon as the diagnosis is made. In this report, we describe a case...INTRODUCTION: A stoma prolapse is easy to diagnose by visual examination, and it rarely incarcerates. Therefore, manual reduction is usually performed as soon as the diagnosis is made. In this report, we describe a case of stoma prolapse that could not be reduced manually and ruptured because an incarcerated parastomal hernia occurred in the stoma, mimicking stoma prolapse. CASE PRESENTATION: A 66-year-old woman underwent total hysterectomy, bilateral salpingo-oophorectomy, pelvic and para-aortic lymphadenectomy, omentectomy, resection of dissemination, and low anterior resection with formation of a sigmoid end colostomy for endometrial cancer with infiltration of the rectum. Fourteen months after the initial operation, she presented with stoma prolapse and multiple episodes of vomiting. The prolapsed stoma was 20 cm in length, appeared swollen and edematous, and was somewhat firm. Although it looked viable, some of the mucosa was darkish red, indicating congestion. Therefore, the diagnosis was sigmoid end colostomy prolapse with an ischemic component. An attempt at manual reduction resulted in rupture, so an emergency laparotomy was performed. Intraoperatively, we found that the ileum was incarcerated in the aperture created where the colostomy had been formed. When the incarcerated ileum was released, the stoma prolapse could be reduced easily. The end colostomy was refashioned in the left upper quadrant of the abdomen. CONCLUSION: An incarcerated parastomal hernia can mimic stoma prolapse. If the findings differ from those of typical stoma prolapse, imaging should be performed to confirm whether another clinical entity is involved in the stoma prolapse.
Szopińska K, Tracz K, Malczyk Ż
… +3 more, Jarzumbek A, Grabowski A, Bąk-Drabik K
Case Rep Gastroenterol
· 2024 · PMID 38249995
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INTRODUCTION: Bezoars are masses of indigestible foreign material in the gastrointestinal tract, usually in the stomach. These materials could be indigestible fruits, hair, milk products, or tablets. In children, the mos...INTRODUCTION: Bezoars are masses of indigestible foreign material in the gastrointestinal tract, usually in the stomach. These materials could be indigestible fruits, hair, milk products, or tablets. In children, the most common type of bezoar is trichobezoar (formed from hair). CASE PRESENTATION: We describe a female patient who has been complaining about deterioration of mood, collapse without losing consciousness, scotomas, and cardiac arrhythmia for 2 years. Based on the results of thyroid hormone, resistance to thyroid hormone (RTH) was suspected. Physical examination during hospitalization revealed a palpable upper abdominal mass. Several diagnostic examinations were performed. The abdominal ultrasound showed acoustic shadowing caused by a pathological structure in the upper abdomen. Therefore, the contrast X-ray of the digestive tract revealed a deficit of contrast with an irregular shape in the stomach body and the pylorus region. Due to these results, a gastroscopy was performed, which revealed a large trichobezoar of the stomach. The trichobezoar was surgically removed without complications. CONCLUSION: The case presented shows that these nonspecific symptoms and laboratory test suggesting RTH require multi-path diagnostics and the cooperation of many specialists, ultimately giving a surprising diagnosis. It is crucial to interpret diagnostic examinations with regard to the patient's physical condition. Diagnosis of trichobezoar requires a detailed search of causes to avoid another incident.
Jarrett SA, Tito S, Chan M
… +3 more, Jarrett DE, Lo KB, DePalma R
Case Rep Gastroenterol
· 2024 · PMID 38197013
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INTRODUCTION: The purpose of this case series was to review a rare subset of tumors known as gastric lipomas, which are typically found incidentally. The motivation for this study arose from the identification of 2 cases...INTRODUCTION: The purpose of this case series was to review a rare subset of tumors known as gastric lipomas, which are typically found incidentally. The motivation for this study arose from the identification of 2 cases within our institution in a short period. CASE PRESENTATION: The study involved a review of the diagnosis and management of 2 patients presenting with gastric lipomas at our institution after symptoms of gastrointestinal bleeding. With the advent of new radiologic investigations such as computed tomography and magnetic resonance imaging and advances in endoscopy, there are new approaches to identifying and managing these tumors. On further evaluation of the literature, we found that despite the availability of minimally invasive endoscopic techniques such as mucosal resection and submucosal dissection in the setting of large tumors, most patients tend to have to resort to surgical management. CONCLUSION: This case series underscores the rarity of gastric lipomas and their often-incidental discovery. Further investigation into endoscopic approaches for managing these tumors is needed, and additionally, there is a need to explore a potential association between gastric lipomas and malignancy, as chronic inflammation of the overlying mucosa may play a significant role.
Kawanaka H, Tajiri K, Muraishi N
… +3 more, Murayama A, Nukui T, Yasuda I
Case Rep Gastroenterol
· 2024 · PMID 38188593
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INTRODUCTION: Immune checkpoint inhibitors are sometimes associated with immune-related adverse events during or after treatment. Among these, aseptic meningitis is a rare and serious complication. We report the first ca...INTRODUCTION: Immune checkpoint inhibitors are sometimes associated with immune-related adverse events during or after treatment. Among these, aseptic meningitis is a rare and serious complication. We report the first case of atezolizumab-induced aseptic meningitis, which occurred during treatment for advanced hepatocellular carcinoma (HCC). CASE PRESENTATION: A 74-year-old woman diagnosed with advanced HCC and treated with first-line atezolizumab plus bevacizumab developed anorexia, fatigue, and fever, after three treatment cycles. Cerebrospinal fluid examination showed slightly increased cell count and protein level but no infection or malignancy. Contrast enhancement along the cerebral sulcus was evident in contrast-enhanced magnetic resonance imaging, and the patient was diagnosed with aseptic meningitis associated with atezolizumab. Steroid therapy soon improved her clinical symptoms, and the contrast enhancement along the cerebral sulcus disappeared. CONCLUSION: Clinicians should monitor to avoid serious immune-related adverse events, such as aseptic meningitis, in patients during treatment of HCC with immune checkpoint inhibitors and make the diagnosis as soon as possible.
Burute N, Aseyev O, Hasan F
… +5 more, Allard-Ihala E, Shahrour W, Kotb A, Harris W, Rozenberg R
Case Rep Gastroenterol
· 2024 · PMID 38188592
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INTRODUCTION: Primary appendiceal carcinoma is rare and comprises up to 1% of all colorectal malignancies. Its invasion into adjacent organs, such as the bladder and rectum, especially as a presenting characteristic, is...INTRODUCTION: Primary appendiceal carcinoma is rare and comprises up to 1% of all colorectal malignancies. Its invasion into adjacent organs, such as the bladder and rectum, especially as a presenting characteristic, is even less common. CASE PRESENTATION: A 75-year-old asymptomatic male tested positive on a screening fecal immunochemical test (FIT). Colonoscopy showed a rectosigmoid tumor and normal appendiceal orifice. Staging MRI surprisingly showed that the cancer was, in fact, of appendiceal origin, coursed posteriorly to invade the rectosigmoid and form adhesions with the urinary bladder. Staging CT did not show metastatic disease. Low anterior resection, en bloc appendectomy, and right hemicolectomy were performed along with cystectomy and ileal conduit. Hematoxylin and eosin stains showed appendiceal adenocarcinoma invading through the appendiceal wall into the rectal muscularis and submucosa. Features of neuroendocrine carcinoma were not identified on immunohistochemistry. This was a colonic type of adenocarcinoma of the appendix. CONCLUSION: This is a rare case of appendiceal carcinoma invading the rectum and presenting as a positive screening fecal immunochemical test in an asymptomatic individual. We effectively demonstrate the use of preoperative MRI to identify the appendiceal origin of the tumor, as well as to demonstrate the extent of tumor spread, which assisted with operative management and treatment planning.
Case Rep Gastroenterol
· 2023 · PMID 38111805
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INTRODUCTION: Down syndrome is due to trisomy 21 and is characterized by intellectual disability, dysmorphic facial features, congenital malformations, and gastrointestinal abnormalities. There is an increased appreciati...INTRODUCTION: Down syndrome is due to trisomy 21 and is characterized by intellectual disability, dysmorphic facial features, congenital malformations, and gastrointestinal abnormalities. There is an increased appreciation of congenital portosystemic shunts in Down syndrome patients. Congenital portosystemic shunts have been associated with many defects in body systems, including cardiac, metabolic, and neurological. CASE PRESENTATION: Herein, we describe a portosystemic shunt in a Down syndrome patient that resulted in hyperammonemia with altered mental status and choreiform movements. Computed tomography angiography of the abdomen and pelvis identified a connection between the right portal vein and inferior vena cava. An 18 mm Amplatzer PFO closure device was placed within the congenital shunt, significantly improving symptoms. The patient has no sequelae from the related shunt or the device at the 2-year follow-up. We extensively reviewed the literature and identified cases of portosystemic shunts in Down syndrome patients. Shunts can either be extrahepatic or intrahepatic and are classified by vasculature connections. CONCLUSION: From our literature review and case presentation, we identify other conditions in patients, including cardiac and gastrointestinal defects. We then review the available treatment options, whether observation or surgical, depending on the patient's clinical picture.
Case Rep Gastroenterol
· 2023 · PMID 38090636
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INTRODUCTION: Dapsone is known to cause drug-induced liver injury (DILI) but can rarely induce the formation of hepatic granulomas. We describe a patient with jaundice who demonstrated granulomas on liver biopsy in respo...INTRODUCTION: Dapsone is known to cause drug-induced liver injury (DILI) but can rarely induce the formation of hepatic granulomas. We describe a patient with jaundice who demonstrated granulomas on liver biopsy in response to dapsone. Her symptoms were only evident once steroids, used to also treat her pyoderma gangrenosum, had been tapered. CASE PRESENTATION: In this case, a 67-year-old female was hospitalized due to 1 day of jaundice. She had started dapsone and prednisone concurrently 7 weeks ago to treat her pyoderma gangrenosum. Steroids were discontinued 4 days prior to symptoms. Her laboratories were notable for newly elevated alkaline phosphatase (756 U/L), aspartate transaminase (199 U/L), alanine transaminase (273 U/L), and total bilirubin (12.6 mg/dL). Dapsone was held due to suspicion for DILI. A liver biopsy was performed and disclosed non-necrotizing hepatic granulomas. After infectious and autoimmune causes were excluded, dapsone was determined to be the cause of her hepatic granulomas. Her bilirubin and liver enzymes steadily normalized over the next 4 weeks following discontinuation of dapsone. CONCLUSION: Thus, dapsone-related liver injury may present following a steroid taper if dapsone and steroids had been initially started together. Hepatic granulomas, though rare, can be seen when dapsone causes DILI.