Introduction

Endoscopic submucosal dissection (ESD) for superficial esophageal squamous cell carcinoma (SESCC) involving the entire circumference carries the extremely high risk of stricture and also raises concerns about curative resection. Therefore, the current Japanese guidelines weakly recommend ESD only for circumferential SESCCs diagnosed as cEP/LPM and sized ≤ 50 mm, while surgery or chemoradiotherapy (CRT) is recommended for circumferential SESCCs other than the above. ¹ However, in clinical practice, ESD is implemented on a case-by-case basis through the discussion in a multi-disciplinary cancer board at each institution because the outcomes of each treatment for circumferential SESCCs remain unclear.²

Aims & Methods

This multicenter prospective observational study aimed to reveal the outcomes of each treatment for circumferential SESCCs. Patients with cT1a-EP/LPM or cT1a-MM/T1b-SM1 N0M0 SESCCs involving the entire circumference were enrolled in this study. At this stage, short-term outcomes, such as the proportion of curative resection after ESD, complete response (CR) after CRT/radiotherapy (RT), and adverse events including esophageal stricture, were evaluated. Curative resection in ESD was defined as pT1a without lymphovascular invasion (LVI). Esophageal stricture was defined as inability to pass the ordinary-sized endoscope thorough the treated site, and refractory stricture was defined as stricture requiring ≥ 6 endoscopic balloon dilation procedures.

Results

A total of 126 patients with circumferential SESCCs were enrolled from 16 Japanese tertiary institutions between May 2021 and November 2024. ESD was performed for 90 (71.4%) patients including 6 undergoing stepwise ESD, surgery for 18 (14.3%), CRT for 16 (12.7%), and RT alone for 2 (1.6%). Median longitudinal length of tumor was 55 (range: 30–140) mm in the ESD group, 80 (50–150) mm in the surgery group, and 70 (30–100) mm in the CRT/RT group. Tumor depth was diagnosed as cEP/LPM in 65.4% (55/84) of the ESD group, 38.9% (7/18) of the surgery group, and 50.0% (9/18) of the CRT/RT group, and the others were diagnosed as cMM/SM1. As stricture prevention method after ESD, local injection or filling of triamcinolone (TA) alone was used in 9 patients (10.7%), and both local injection of TA and oral steroid of prednisolone were used in 75 (89.3%). Among the 63 patients who did not receive additional treatment after ESD, the proportion of stricture and refractory stricture were 46.0% (29/63) and 27.0% (17/63), respectively. Curative resection after ESD was achieved in 58 patients (69.0%), including 46 (83.6%) with cEP/LPM and 12 (41.4%) with cMM/SM1. Of 26 patients with non-curative resection after ESD, 20 (76.9%) received additional treatment of either surgery, CRT, or RT, while the remaining 6 opted for follow-up. Of 18 patients in the surgery group, 7 with cEP/LPM and 4 with cMM/SM1 were diagnosed as pT1aN0 without LVI, while the remaining 7 with cMM/SM1 were diagnosed as pT1b, 3 of whom had lymph node metastasis. Anastomotic stricture was observed in 11.1% (2/18) of patients treated with surgery. Of 18 patients in the CRT/RT group, CR was confirmed in all patients without forming stricture.

Conclusion

Considering the high curative resection rate, ESD could be indicated for circumferential SESCCs diagnosed as cEP/LPM. In contrast, the careful consideration may be required in indicating ESD for circumferential SESCCs diagnosed as cMM/SM1 due to its low curative resection rate. Long-term outcome needs to be confirmed for further treatment comparisons.

References

1. Ishihara R, Arima M, Iizuka T, et al. Endoscopic submucosal dissection/endoscopic mucosal resection guidelines for esophageal cancer. Dig Endosc. 2020; 32: 452–93.
2. Kadota T, Ishihara R, Hatta W, et al. Multi-institutional questionnaire on treatment strategies for superficial entire circumferential esophageal squamous cell carcinoma. DEN Open. 2023; 3: e206.

Introduction

Primary biliary cholangitis (PBC) is often associated with extrahepatic autoimmune diseases (EAIDs) such as Sjögren’s syndrome, autoimmune thyroiditis, and connective tissue disorders. While this association suggests a shared immunological predisposition, its effect on disease prognosis remains unclear.

Aims & Methods

This retrospective study included 69 patients diagnosed with PBC, excluding those with overlap syndromes involving autoimmune hepatitis. Patients were divided into two groups: isolated PBC (n = 35) and PBC associated with EAIDs (n = 34). The two groups were compared based on clinical, immunological, elastometric characteristics, disease progression, and prognostic scores (Paris I, Paris II, Rotterdam, Barcelona, Rochester, GLOBE, UK-PBC). Statistical analysis was conducted using appropriate tests, with a significance threshold set at p < 0.05.

Results

The average age at diagnosis was 55.6 years. The disease was initially suspected based on functional symptoms in 39% of cases (fatigue, pruritus, arthralgia), biological cholestasis in 33.3%, and decompensated cirrhosis in 20.3%. The most common associated autoimmune diseases were Sjögren’s syndrome (17.4%), thyroid disorders (13%), and systemic lupus erythematosus (10.1%). All patients received treatment with ursodeoxycholic acid at 13-15 mg/kg/day. No significant differences were found between the two groups regarding functional symptoms, biological markers (bilirubin, albumin, prothrombin time), autoantibodies (AMA, Anti-GP210, anti-SP100, anti-PML), non-invasive fibrosis scores (Fib4, Fibroindex, Fibroscan, S-index), or hepatic complications (HDH, cirrhosis, hepatic encephalopathy, liver-related death). Prognostic scores were also comparable between both groups: Paris I (p = 0.468), Paris II (p = 0.519), GLOBE score (p = 0.6), UK-PBC (p = 0.49), Rotterdam (p = 0.897), Barcelona (p = 0.181), Rochester (p = 0.727).

Conclusion

The presence of extrahepatic autoimmune diseases in PBC does not appear to affect the clinical, biological, or prognostic profiles of patients. Despite their frequency, these comorbidities do not seem to influence the progression of PBC.

Disclosure

None.

Clinical Case Summary

A 58 year-old woman was admitted for an elective rectosigmoidectomy after two previous uncomplicated diverticulitis episodes. On the third postoperative day, her clinical status deteriorated, and an abdominal CT scan revealed an anastomotic leak, leading to a new surgery and confection of a protective ileostomy. Her condition improved progressively, and she was discharged 10 days after. However, the patient came back to the emergency one week later with abdominal pain, fatigue, and refractory nausea and vomiting, in addition to acute renal failure with severe hyponatremia and hyperkalemia. Because her daily ileostomy’s output reached about 2 liters/day, she was started on high dose opioids and loperamide to decrease the stoma’s output, as well as replacement fluid therapy, improving quickly within 24 to 48 hours. She was discharged in 5 days.
Despite the adequate control of the ileostomy’s output, her second readmission occurred 12 days later, showing the same clinical and laboratorial patterns. Considering she had a typical profile for adrenal insufficiency, fludrocortisone was associated with the antimotility agents, showing a significant improvement within hours. Nonetheless, she discontinued fludrocortisone on her own, and a third readmission occurred in 10. One more time, clinical and laboratorial characteristics were the same, with rapid improvement in the short term. An extensive investigation was conducted, revealing an aldosterone level more than 10 times higher than upper limit of normal, with normal renin levels and an adrenocorticotropic hormone in the lower limit of normal. These paradoxical findings were reinforced with a low urine and high ileostomy sodium dosages, and a diagnosis of a type 3 pseudohypoaldosteronism was made. Definitive treatment with intestinal transit’s reconstruction was performed, with complete resolution.

References

1. Carl C, Dinkelbach L, Mohr J, Perez R, Vera Lopez T, Fricke-Otto S, Niehues T. Case report: Life threatening hyponatremia in infants with urinary tract infections: two cases of type III pseudohypoaldosteronism and review of the literature. Front Pediatr. 2024 Jan 5;11:1233205. doi: 10.3389/fped.2023.1233205. PMID: 38250595; PMCID: PMC10797885.
2. Amir Babiker, Haya Aldabas, Shahad Alanazi, Bashayer Alahmadi, Waleed Tamimi, Mohsen Al Atawi, Fahad Aljuraibah, Angham Almutair. Pseudohypoaldosteronism: A challenging diagnosis with management pitfalls - Case series. Journal of Clinical and Translational Endocrinology: Case Reports, Volume 32, 2024, 100172. ISSN 2214-6245
3. Stojanović, V., Spasojević, S., Radovanović, T., & Doronjski, A. (2017). Pseudohypoaldosteronism: report of three cases. Journal of Endocrinology, Metabolism and Diabetes of South Africa, 22(2), 17–20. https://doi.org/10.1080/16089677.2017.1335532
4. Nakasone, Ruka et al. Three cases of pseudohypoaldosteronism following ileostomy in preterm infants. Pediatrics & Neonatology, Volume 62, Issue 1, 119 - 121

Introduction

Eosinophilic oesophagitis [EoE] is a chronic, antigen-driven, immune-mediated oesophageal disease characterised by eosinophil-predominant inflammation. It typically presents with dysphagia, food bolus impaction, and, in children, feeding difficulties and failure to thrive [1]. Histologically, it is defined by ≥15 eosinophils per high-power field on oesophageal biopsy in the absence of other causes of eosinophilia [2]. The incidence and prevalence of EoE have increased markedly over recent decades, particularly in Western countries, likely due to increased recognition, changing environmental exposures, and altered microbiota [3,4]. Though EoE is increasingly reported worldwide, data specific to Aotearoa New Zealand, and especially regional patterns by ethnicity, remain limited.

Aims & Methods

This study aimed to describe the incidence, demographic distribution, and clinical characteristics of EoE in the Canterbury region over an 11-year period.
We conducted a retrospective analysis of electronic medical records from 2013 to 2023, identifying EoE cases using diagnostic codes and histological confirmation. Demographic data, clinical presentations, and laboratory findings were collected. Crude incidence rates were calculated using average population data for the Canterbury region, stratified by ethnicity (European vs. non-European). Clinical features including food bolus obstruction, oesophageal strictures, and peripheral eosinophilia were assessed. Incidence rates are reported per 100,000 population with 95% confidence intervals (CI).

Results

A total of 219 cases of EoE were identified between 2013 and 2023. The average annual incidence was 3.23 per 100,000 population (95% CI: 1.82–4.65), based on a regional population of 618,409. The mean number of cases per year was 20 (IQR 16–22). Among European individuals (average population 395,690), the mean annual incidence was 4.42 per 100,000 (95% CI: 2.35–6.49), with 17.5 average cases per year (IQR 14–21). Among non-European individuals (average population 222,627), the mean incidence was 1.08 per 100,000 (95% CI: -0.29–2.44), with 2.4 average cases per year (IQR 1–4).
Of the 219 cases, 70 patients (31.9%) presented with oesophageal food bolus impaction. The annual number of such presentations ranged from 3 to 12 per year (median 6, IQR 4–9). Twenty patients (9.1%) had documented stricturing disease on endoscopy, and 60 patients (27.4%) exhibited peripheral eosinophilia at diagnosis.

Conclusion

In our cohort, eosinophilic oesophagitis presents commonly with oesophageal food bolus impaction. There was a higher incidence of EoE observed in individuals of European descent. Just under one-third of patients in our cohort presented with oesophageal food bolus impaction and about 9% had stricturing disease. The frequent presentation with food bolus obstruction and the presence of complications such as strictures highlight the importance of timely recognition and management. Peripheral eosinophilia was found commonly in our cohort but the prevalence of this finding is not adequate to be used as a diagnostic test in EoE, although more studies are needed.

References

1. Dellon ES, Liacouras CA, Molina-Infante J, et al. Updated International Consensus Diagnostic Criteria for Eosinophilic Esophagitis: Proceedings of the AGREE Conference. Gastroenterology. 2018;155(4):1022-1033.e10.
2. Lucendo AJ, Molina-Infante J, Arias Á, et al. Guidelines on eosinophilic esophagitis: evidence-based statements and recommendations for diagnosis and management in children and adults. United European Gastroenterol J. 2017;5(3):335-358.
3. Hruz P, Straumann A, Bussmann C, et al. Escalating incidence of eosinophilic esophagitis: A 20-year prospective, population-based study in Olten County, Switzerland. J Allergy Clin Immunol. 2011;128(6):1349-1350.e5.
4. Arias Á, Lucendo AJ. Epidemiology and risk factors for eosinophilic esophagitis: Lessons for clinicians. Expert Rev Gastroenterol Hepatol. 2020;14(2):103–115.

Disclosure

None

Introduction

Lynch syndrome (LS), the most common hereditary colorectal cancer (CRC) syndrome, is associated with an increased risk of small bowel malignancies. Given the feasibility of ileal intubation during colonoscopy, this study assesses the prevalence of ileal cancer in a large LS cohort and evaluates ileal intubation as a screening tool.

Aims & Methods

An analysis was performed on Lynch syndrome (LS) patients with ileal cancer enrolled in the prospectively maintained database of the German Consortium for Familial Intestinal Cancer (GCFIC). Demographic characteristics and cancer history were extracted. High-risk pathogenic germline variants (PGVs) were defined as MLH1, MSH2, and EPCAM, while MSH6 and PMS2 were considered low risk.
Additionally, data from 236 LS patients who underwent colonoscopy at a tertiary care center between 2018 and 2023 were retrospectively evaluated. A total of 596 complete colonoscopies were reviewed.

Results

Eight ileal cancers (0.28%) were identified, exclusively in high-risk PGV carriers (MLH1: 62.5%, MSH2: 37.5%), predominantly male (87.5%), with a median age of 56 years. Most cancers (75%) were symptom-detected; only one case was identified through screening. In the tertiary care cohort, ileal intubation was achieved in 96.6% of colonoscopies, leading to the identification of five ileal neoplasms (four adenomas, one carcinoma), all in patients with a history of right hemicolectomy or colectomy. The prevalence of ileal neoplasia in patients without an ileocecal valve was statistically significant at 7.7% (χ²(1, N = 236) = 11.8, p < .001).

Table 1: Cohort characteristics of the German Consortium for Familial Intestinal Cancer

Conclusion

Ileal intubation is a valuable extension of standard colonoscopy, especially for male high-risk PGV carriers with prior CRC and ileocecal valve removal. The strong link between ileal neoplasia and loss of the ileocecal valve suggests a role for colonic reflux, inflammation, and microbiome changes in ileal carcinogenesis.

Disclosure

Robert Hüneburg was funded by Deutsche Krebshilfe (DECADE), and Wilhelm-Sander-Stiftung.
Jacob Nattermann was funded by Wilhelm-Sander-Stiftung.

Introduction

Malignant biliary obstruction (MBO) caused by non-hepatobiliary-pancreatic (non-HBP) malignancies is a rare and underexplored indication for endoscopic retrograde cholangiopancreatography (ERCP). These cases often present a clinical dilemma: while ERCP offers a minimally invasive option, its utility remains uncertain in patients with poor prognosis and advanced disease.

Aims & Methods

We aimed to evaluate the effectiveness of ERCP-guided biliary drainage in this context and identify baseline predictors of clinical failure to help refine patient selection.
We conducted a retrospective analysis of all patients who underwent ERCP for malignant biliary obstruction of non-HBP origin between March 2018 and February 2025. Patients lacking essential clinical or follow-up data were excluded. Technical success was defined as adequate stent placement; clinical success as a ≥50% reduction in bilirubin or resolution of cholangitis within 2 weeks. The predictive role of bilirubin level, stricture location, serum albumin, and ECOG performance status was assessed. A ROC analysis evaluated the discriminative ability of pre-drainage bilirubin.

Results

Forty-eight patients were included (mean age: 55.6 years; 56% women). The most common primary cancers were colorectal (31%, n=15), gastric (18%, n=9), breast (14%, n=7), lung (10%, n=5), and ovarian (8%, n=4). Hilar strictures were observed in 54% (n=26) and mid-to-distal strictures in 46% (n=22). All procedures were performed using plastic stents. In most cases involving hilar strictures, long stents (e.g., 7 Fr / 12–15 cm) were used, and a single biliary sector—usually the most dilated or invaded—was preferentially drained. Technical success was achieved in 85.4% of cases. Clinical success occurred in 54.1% of patients, while early mortality (≤30 days) reached 10.4%, exclusively among individuals with ECOG status 4 and serum albumin <2.8 g/dL. Hilar strictures and total bilirubin levels >200 mg/L were significantly associated with lower clinical success rates (38% vs 65%, p=0.03 and 33% vs 62%, p=0.04, respectively). ROC analysis showed an AUC of 0.72 for bilirubin in predicting failure. Technical failures (14.6%) were mainly due to anatomical distortion or papillary inaccessibility.

Conclusion

Endoscopic biliary drainage in this population is technically achievable but offers limited clinical benefit, especially in cases with hilar involvement, severe hyperbilirubinemia, or poor overall condition. These findings emphasize the importance of using simple, baseline clinical indicators to guide treatment decisions, avoid unnecessary interventions, and adapt care strategies to the realities of resource-limited settings.

Disclosure

I have no conflict of interest.

Introduction

Accurate prediction of lymph node metastasis (LNM) in submucosal invasive (T1) colorectal cancer (CRC) is essential for determining the need for additional surgery following endoscopic resection. Conventional histopathological assessments, however, are limited by interobserver variability. This study aimed to develop and externally validate a stacking-based artificial intelligence (AI) model integrating histopathological whole slide images (WSIs) and clinical parameters to predict the risk of LNM in T1 CRC.

Aims & Methods

This retrospective study included 1,056 patients with histologically confirmed T1 CRC treated at Showa Medical University Northern Yokohama Hospital. Of these, 593 cases were used for model training and 100 for internal testing. External validation was conducted on 25 cases from two independent institutions.
The proposed stacking ensemble model incorporated the Multiple Instance Self-Training (MIST) score derived from WSIs, patient sex, and tumor location (colon vs. rectum). Base learners included logistic regression, XGBoost, and random forest, with a meta-learner synthesizing their outputs to generate the final LNM prediction. Model performance was evaluated using the area under the receiver operating characteristic curve (AUC) in both internal and external cohorts.

Results

LNM was observed in 69 (11.6%) training cases, 15 (15.0%) test cases, and 2 (8.0%) external validation cases. The model achieved an AUC of 0.6965 in the internal test cohort and 0.7500 in the external validation cohort, demonstrating stable and generalizable predictive performance.
A representative case involved a 64-year-old man with a 16-mm type 0-Is lesion in the sigmoid colon resected via endoscopic mucosal resection (EMR). Histopathology showed well-differentiated tubular adenocarcinoma (tub1 > tub2), submucosal invasion depth of 3,300 µm (T1b), tumor budding grade BD1, Ly0, V1a, and negative resection margins (HM0, VM0). Despite guideline-based recommendations prompting additional surgical resection, no LNM was found (N0, 0/16), and the patient remained recurrence-free five years postoperatively—consistent with the AI model’s prediction.

Conclusion

The stacking-based AI model exhibited robust and generalizable performance in predicting LNM in T1 CRC, both internally and across external institutions. This approach offers a complementary tool to conventional pathology and may enhance individualized decision-making regarding the necessity of additional surgical intervention.