Clinical Case Summary

Primary biliary cholangitis (PBC) is a rare disease (19-42 per million) characterized by a T-lymphocyte-mediated attack on small intralobular bile ducts. We are presented with a case of a 46 year old female who came in for generalized pruritis, generalized jaundice and elevated liver enzymes. The patient was completely worked up, and all the most common causes of jaundice turned out negative. Liver biopsy was done and was signed out as a case of PBC which progressed to a decompensated state of liver cirrhosis complicated by portal hypertension. Most of the treatment options were exhausted at this time and the medical team was left with the option to do liver transplantation. During the work-up for the said transplant, there was an incidental finding of a breast cancer which was an absolute contraindication to transplantation. Fortunately, the malignancy was at an early stage (Stage 1) and was managed surgically. A repeat PET CT scan was done for surveillance which revealed no distant metastases. The procedure was tolerated well by the patient and was eligible to be placed back on the transplant list. The patient was in dire need of transplant and having been initially removed from the transplant list pushed her at the back of the list. Fortunately a relative selflessly offered to be a living donor and the patient was for liver transplant. Unfortunately, the patient succumbed to COVID-19 and expired even before the medical team could do the transplant. This case highlights the management of PBC with end-stage liver disease has numerous complications but are manageable. Individualization of patient management is essential when faced with rare and complicated cases which lead to the conclusion that absolute contrainidications are not always absolute as demonstrated in this case that the patient initially thought that she can never undergo liver transplant due to her malignancy but was given another chance at life when she got treated and was declared cancer free.

References

1. The American Association for the Study of Liver Diseases practice guidelines. Hepatology 2000; 31:1005.
2. The risk of surgery in patients with liver disease; https://doi.org/10.1002/hep.510290639
3. Lee J, Belanger A, Doucette JT, Stanca C, Friedman S, Bach N. Transplantation trends in primary biliary cirrhosis. Clin Gastroenterol Hepatol. 2007;5:1313-1315
4. Beuers U, Gershwin ME, Gish RG, Invernizzi P, Jones DEJ, Lindor K, et al. Changing nomenclature for PBC: From ’cirrhosis’ to ’cholangitis’. Clin Res Hepatol Gastroenterol. 2015;39:e57-e59.
5. Lindor Et al. Primary Biliary Cholangitis: 2018 Practice Guidance from the American Association for the Study of Liver Diseases. 2018. 400.
6. Lammers WJ, van Buuren HR, Hirschfield GM, Janssen HL, Invernizzi P, Mason AL, et al. Levels of alkaline phosphatase and bilirubin are surrogate end points of outcomes of patients with primary biliary cirrhosis: an international follow-up study. Gastroenterology. 2014;147:1338-1349 e5; quiz e15.
7. Dickson ER, Grambsch PM, Fleming TR, Fisher LD, Langworthy A. Prognosis in primary biliary cirrhosis: model for decisionmaking. Hepatology. 1989;10:1-7.
8. Martin P, DiMartini A, Feng S, Brown R Jr, Fallon M. Evaluation for liver transplantation in adults: 2013 prac- tice guideline by the American Association for the Study of Liver Diseases and the American Society of Transplantation. Hepatology. 2014;59:1144-1165.

Introduction

The quantitative fecal immunochemical test (FIT) is widely used for colorectal cancer (CRC) screening. While the association between FIT concentration and the risk of advanced colorectal neoplasia (ACN) detection is well established, the predictive performance of current-round FIT concentration alone has limitations. Identifying additional predictive factors could enhance screening. For individuals with a prior colonoscopy, colonoscopy findings and quality metrics may serve as such factors.1 For those who did not undergo colonoscopy due to a negative FIT in the previous round, alternative predictors should be explored. We hypothesized that prior-round FIT concentration could serve as a potential predictor.

Aims & Methods

We aimed to investigate whether prior-round negative FIT concentration could predict CRC and ACN detection by conducting a cross-sectional analysis of data from the FIT arm of the SCREESCO, a nationwide randomized controlled trial on CRC screening in Sweden.1,2 A sample of the Swedish population aged 60 years was randomly assigned to one of three arms: the once-colonoscopy arm, FIT arm, or control arm. Individuals in the FIT arm were invited to undergo two rounds of quantitative FIT screening in the first and third years. A highly sensitive method was used to assess FIT positivity, with a cut-off concentration of ≥10 μg Hb/g feces. We estimated the detection rates of CRC and ACN in second-round colonoscopy based on first-round FIT concentration. Binary regression models were used to assess whether first-round FIT concentration was associated with CRC and ACN detection in the second round. The models included participant characteristics such as sex, body mass index, and smoking status, and second-round FIT concentration, in addition to first-round FIT concentration.

Results

A total of 1,991 individuals were included, with a median FIT concentration of 2.2 μg Hb/g feces (interquartile range: 0.4–4.6) in the first round and 21.8 (13.4–50.8) in the second round. CRC and ACN were detected during second-round colonoscopy in 23 (1.2%) and 267 (13.4%) individuals, respectively. Individuals with first-round FIT concentration of 0–1.9, 2.0–5.9, and 6.0–9.9 μg Hb/g feces had second-round CRC detection rates of 0.8%, 0.9%, and 2.6%, respectively, and ACN detection rates of 9.7%, 12.5%, and 15.9%, respectively. A higher first-round FIT concentration was associated with an increased risk of CRC (risk ratio per unit increase 1.27; 95% confidence interval 1.10–1.47) and ACN (1.08; 1.04–1.12) detection in the second round, after adjusting for the second-round FIT concentration and participant characteristics. For a fixed second-round FIT concentration, the absolute risk of second-round ACN detection increased with higher first-round FIT concentration. For instance, in a nonsmoking or ex-smoking male with a second-round FIT concentration of 20 µg Hb/g feces, the risk increased from 10.0% at < 0.2 µg Hb/g feces in the first round to 14.9% at 5 µg Hb/g feces and 19.6% at 9.9 µg Hb/g feces.

Conclusion

First-round negative FIT concentration is predictive of CRC and ACN detection in the second screening round. Even an increase in low levels of the FIT concentration is associated with future detection of these neoplasms. Incorporating first-round FIT concentration alongside second-round FIT concentration may enhance risk stratification in CRC screening, leading to more efficient use of colonoscopy resources.

References

1. Sekiguchi M, Westerberg M, Löwbeer C, Forsberg A. Gastrointest Endosc. 2025 Feb 4. doi: 10.1016/j.gie.2025.01.037. Epub ahead of print.
2. Forsberg A, Westerberg M, et al. Lancet Gastroenterol Hepatol. 20227: 513-21. doi: 10.1016/S2468-1253(21)00473-8.

Clinical Case Summary

Background: Alcoholic liver disease (ALD) is a major health concern, ranging from hepatic steatosis and alcoholic hepatitis (AH) to cirrhosis, and can progress to acute liver failure (ALF). Extrahepatic manifestations of AH include gastrointestinal bleeding, peripheral neuropathy, and alcoholic cardiomyopathy. However, the association between alcoholic hepatitis and acute inflammatory demyelinating polyneuropathy (AIDP) is rare and scarcely reported.
Case Presentation: A 35-year-old chronic alcoholic female with hypothyroidism presented with jaundice (bilirubin 13.3 mg/dL) and hepatomegaly. Two weeks later, she developed ascending lower limb weakness, absent reflexes, and reduced muscle power (right leg 3+/5, left leg 2/5). Nerve conduction studies were suggestive of demyelinating pathology. Lab workup revealed elevated AST (134 IU/L), ALT (31 IU/L), INR (1.9), and thiamine deficiency (23.7 μg/L). Other potential differentials, including viral hepatitis and autoimmune hepatitis, were ruled out based on negative serology for HBsAg, HCV, CMV, and autoimmune markers (ANA, anti-LKM antibodies). Despite thiamine supplementation and IV immunoglobulin (IVIG) therapy (0.4 g/kg/day for 5 days), the patient showed limited motor improvement. Progressive liver failure and sepsis led to death.
Conclusion: The pathophysiology of AIDP in the context of AH remains unclear but may involve immune dysregulation due to alcohol’s toxic effects on the nervous system. Early detection of AIDP in patients with AH is crucial, as these patients are at risk of respiratory failure and permanent neurological deficits. Although IVIG therapy may offer some improvement, the prognosis remains poor in cases complicated by severe liver failure. This case highlights the importance of recognizing and managing complications like AIDP in patients with AH.

Introduction

Early detection of gastric neoplasms remains challenging, even with enhanced endoscopic imaging. Photodynamic endoscopic imaging (PEI) with 5-aminolevulinic acid (5-ALA) is a novel diagnostic approach that exploits tumor-selective fluorescence. After oral administration, 5-ALA is converted via the heme biosynthesis pathway to protoporphyrin IX (PpIX), which accumulates in neoplastic cells and emits red fluorescence under violet-blue excitation. We developed specialized endoscope prototypes (first-generation prototype using a 410 nm laser, second-generation prototype with an enhanced 410 nm laser, and third-generation prototype employing a high-intensity LED light source) to visualize PpIX fluorescence in the stomach. These successive devices improved image brightness and enabled more objective tumor detection.

Aims & Methods

In this exploratory study at Tottori University Hospital, 54 patients with 60 early-stage gastric lesions (gastric adenomas or early gastric carcinomas) were examined. Patients received oral 5-ALA (20 mg/kg) three hours before endoscopy. PEI was performed using the prototype systems (excitation at 410 nm and detection of ~635 nm red fluorescence). Lesions were identified by their red fluorescent contrast against surrounding mucosa. After the procedure, patients were protected from strong light for 24 hours to prevent photosensitivity. Key outcomes were the fluorescence detection rate of known tumors, discovery of any additional lesions, and safety (adverse events).

Results

The overall fluorescence detection rate was 85.0% (51/60 lesions). PEI newly identified 11 synchronous lesions in 8 patients (14.8%). Among lesions with fluorescence, histological types included tubular adenocarcinoma and adenoma (tub/sig/adenoma: 49/0/13). All signet ring cell carcinomas (5/5) were fluorescence-negative. Among 12 post-H. pylori eradication gastric cancers, 11 (91.6%) exhibited detectable fluorescence. Adverse events included transient elevations in liver enzymes (9 cases), mild hypotension (1 case), and low-grade fever (1 case), all resolving spontaneously. No phototoxic reactions were observed under light-shielding protocols.

Conclusion

Oral 5-ALA-mediated photodynamic endoscopic imaging is a feasible and effective technique for objective visualization of early-stage gastric tumors. This method provides bright tumor-specific fluorescence in gastric adenomas and differentiated early cancers, enhancing lesion contrast and aiding detection. Although diffuse-type (signet ring cell) carcinoma may evade fluorescence due to limited PpIX accumulation, the overall ability of 5-ALA-PEI to highlight tumors could significantly augment current endoscopic screening practices. This emerging molecular imaging approach offers endoscopists a valuable tool for identifying gastric neoplasms, and ongoing device refinements may further improve sensitivity for subtle lesions.

Disclosure

Hiroki Kurumi and Yuichiro Ikebuchi declare no conflict of interest. Hajime Isomoto received research funding from FUJIFILM Corporation.

Clinical Case Summary

Background: Percutaneous endoscopic gastrostomy (PEG) tubes are commonly used for long-term enteral nutrition in critically or chronically ill patients [1]. Buried bumper syndrome (BBS), a rare but serious complication, occurs when the internal bumper migrates along the stoma tract, leading to tissue ischemia, infection, and potential tube dysfunction [2,3]. BBS typically manifests around 18 months post-placement, with acute presentations exceptionally rare [4]. This case highlights an unusual early onset of BBS, emphasizing the importance of vigilant post-procedural care [5].
Case report: An 83-year-old male with multiple comorbidities, including progressive oropharyngeal dysphagia following cervical spine surgery, underwent PEG tube placement for enteral nutrition. Postoperatively, mild oozing was managed by tightening the bumper. Twelve days later, erythema and pressure necrosis were noted due to excessive tightness, prompting loosening. Three weeks post-placement, localized cellulitis developed and was treated with antibiotics. Two months postoperatively, the PEG tube became dysfunctional, and endoscopic evaluation revealed a partially buried bumper. A minimally invasive surgical approach, assisted endoscopically, was performed to extract the bumper and replace the tube with a balloon G-tube. The revised PEG tube functioned effectively, with no recurrence of complications.
Conclusion: This case underscores the importance of early recognition and management of BBS, even in the acute postoperative period [4,7]. Minimally invasive techniques, combined with proper bumper positioning and patient education, are crucial for preventing and managing this complication [3,6]. Vigilant monitoring of PEG sites and timely intervention can significantly reduce morbidity and improve outcomes in patients with BBS [2,7].

References

1. Blumenstein I, Shastri YM, Stein J. Gastroenteric tube feeding: Techniques, problems and solutions. World J Gastroenterol. 2014;20(26):8505–24.
2. Cyrany J, Rejchrt S, Kopacova M, Bures J. Buried bumper syndrome: A complication of percutaneous endoscopic gastrostomy. World J Gastroenterol. 2016;22(2):618–27.
3. Pérez R, De La Serna C. Buried bumper syndrome: A critical analysis of endoscopic release techniques. World J Gastrointest Endosc. 2019;11(9):611–7.
4. Pinho J, Libânio D, Pimentel-Nunes P, Dinis-Ribeiro M. The challenging acute buried bumper syndrome: A case report. GE Port J Gastroenterol. 2018;25(3):151–4.
5. Satiya J, Marcus A. The buried bumper syndrome: A catastrophic complication of percutaneous endoscopic gastrostomy. Cureus. 2019;11(3):e4275.
6. Maly O, Bichard P. Endoscopic management of buried bumper syndrome using a guidewire and external traction: a novel technique. Acta Gastro-Enterol Belg. 2023;86(1):13–6.
7. Venu RP, Brown RD, Pastika BJ, Erikson LW. The buried bumper syndrome: a simple management approach in two patients. Gastrointest Endosc. 2002;56(4):582–4.

Disclosure

Dear Editors,
I, Evelyn Inga, hereby declare that I have no financial, professional, or personal conflicts of interest.
I affirm that I have no affiliations, financial interests, personal relationships, or commitments that could influence my impartiality in reviewing, conducting, or overseeing our study. Additionally, I confirm that I will uphold the highest ethical standards in compliance with institutional and federal regulations.
If any potential conflict arises, I will promptly disclose it to the United European Gastroenterology Association. Please let me know if any further documentation is required.
Sincerely,
Evelyn Inga, MD

Clinical Case Summary

A 59-year-old man was admitted with painless jaundice. Laboratory data revealed elevated liver enzymes and hyperbilirubinemia (ALT 95UI/L, AST 54UI/L, ALP 550UI/L, total bilirubin 6mg/dL and direct bilirubin 4.6mg/dL). CT confirmed an unresectable 4cm perihilar infiltrative lesion with extensive portal vein invasion and cavernous transformation. An US-guided biopsy of the lesion followed by percutaneous transhepatic cholangiography were performed, allowing palliative biliary drainage with non-covered metal stents. Nevertheless, biopsy sample histopathology yielded no intersected neoplastic tissue.
Given the strong suspicion of malignancy and the need for histopathology to guide systemic treatment, endoscopic retrograde cholangiopancreatography (ERCP) with cholangioscopy-guided biopsies was proposed. The procedure started by conventional biliary cannulation. Cholangiography revealed the metallic stents in situ crossing the hilum. Biliary endoscopic sphincterotomy was performed and a 3.2mm cholangioscope (EyeMAX™) was successfully introduced. The common bile duct and the hepatic ducts were carefully explored advancing the cholangioscope through the stents into the intrahepatic ducts. Detailed inspection of the biliary mucosa identified areas of pseudopolypoid appearance covering the stent body in the hilar region with marked friability and aberrant vascularization. Cholangioscopy-guided biopsies were laboriously performed through the mesh of the metal stents using a 1mm biopsy forceps (EyeMAX™ biopsy forceps). No displacement or damage to the stents were observed. Histology confirmed a moderately differentiated cholangiocarcinoma, and palliative chemotherapy could be started.
Biliary non-covered metal stents should only be used after a definite histologic diagnosis of cancer is stablished. Despite the potential technical limitations of this unreported approach, targeted biopsies through the mesh of metallic stents seem effective without compromising stent integrity.

References

Cholangioscopy images.
Fluoroscopy images.
Histopathology images.

Clinical Case Summary

Introduction: Aortoesophageal fistula (AEF) is a rare but fatal cause of upper gastrointestinal bleeding. The disease progression is often rapidly if not promptly recognized.
The most common etiologies include thoracic aortic aneurysm (TAA) and prior aortic surgery. Early diagnosis remain challenge due to nonspecific symptoms. This case series highlights endoscopic features of AEF and the critical role of endoscopists in early detection.
Case present: We report three elderly patients (aged 76–82) who presented to the emergency department with massive hematemesis. One had a history of type B aortic dissection post thoracic endovascular aortic repair (TEVAR) ; the others had no known thoracic disease. None reported mid-thoracic pain. Laboratory data showed hemoglobin 5.1 to 6.2 g/dL without coagulopathy. Esophagogastroscopy (EGD) was performed within 1 hour in two patients, and at 9 hours in the third. EGD showed consistent findings in three cases: a subepithelial bulging mass with central blood clot adherent to the left quadrant (6–9 o’clock) of the mid-esophagus. CT confirmed AEF secondary to ruptured TAA in two patients. Unfortunately, one patient collapsed suddenly in the ED; the others underwent emergent surgery but died within 1 and 21 days postoperatively.(esophagectomy +/- TEVAR).
Discussion: Chiari’s triad (mid-thoracic pain, sentinel bleeding, fatal hematemesis) is classically associated with AEF but was absent in half patients. In our report, EGD can be a most timely and specific modality to early diagnosis of AEF, especially in patients without clear prior aortic disease. The typical endoscopic feature : bulging submucosal mass with central blood adherent clot may serve as a diagnostic clue. And the “hot-zone” of the AEF in EGD was located at left quadrant (six to nine o’clock) of mid esophagus .
Conclusion: Early recognition of typical characteristic and “hot zone” of AEF on EGD may improve timely diagnosis of AEF and guide life-saving interventions.

References

1. Bergqvist D. Arterioenteric fistula. Review of a vascular emergency. Acta Chir Scand 1987;153:81–6.
2. Hollander JE, Quick G. Aortoesophageal fistula: a comprehensive review of the liter- ature. Am J Med 1991;91:279–87.
3. Mortensen A, Hansen NF, Schiodt OM. Fatal aortoesophageal fistula caused by but- ton battery ingestion in a 1-year-old child. Am J Emerg Med 2010;28(984):e5-6.
4. Takeno S, Ishii H, Nanashima A, Nakamura K. Aortoesophageal fistula: review of trends in the last decade. Surg Today. 2020 Dec;50(12):1551-1559.