Clinical Case Summary

The syndrome of buried lumen-apposing metal stents (LAMS) occurs in 4.7% of cases and is caused by a foreign body reaction characterised by tissue overgrowth embedded in the stent. This complication often requires surgery or "stent-in-stent" endoscopic procedures (1). Stent in a stent safely and effectively recanalises LAMS placed as esophagogastric anastomosis (2), gallbladder drainage (3), and endoscopic ultrasound (EUS)-directed transgastric ERCP (EDGE) (4). We report the first EUS-directed transenteric ERCP (EDEE) case using this approach. In 2023 a patient with Roux-en-Y hepaticojejunostomy (HJ) substenosis and recurrent cholangitis was admitted to our institution. The first attempt to perform an EUS-guided gastrojejunostomy (GJ) as part of EDEE failed; EUS -Hepaticogastrostomy (HGS) was conducted. One month later, a Hot-Axios LAMS (15mm x 15mm) EUS-GJ was placed, and prior 15 mm balloon dilatation, the second part of the EDEE operation was conducted, accessing the surgical HJ with a paediatric colonoscope and placing a Nagi stent (16mm x 20mm) towards the substenosis. The LAMS completely buried itself during the 6-month Nagi removal follow-up, making it impossible to determine its gastric mucosal location. We placed a fully covered self-expandable metal stent (10mm x 8cm) on the HGS for antegrade Nagi stent extraction. Recurrent HJ substenosis and bile duct stones induced cholangitis in 2024. EUS-guided identification and cannulation of the buried LAMS involved passing a 19-G fine needle biopsy needle through the LAMS, advancing a Jagwire deep into the jejunum, and bridging it with a second Hot-Axios LAMS (15mm x 15mm) using a non-cautery enhanced (cold LAMS-in-LAMS) technique.A frontal scope was used to remove bile duct stones before the latest 15 mm balloon was expanded, and a third 15 mm x 10 mm LAMS was placed across the substenotic HJ. EUS-guided management of buried LAMS was safe and effective, with normal liver function tests and clinical outcomes.

References

1) Nayar, M., Leeds, J. S., UK & Ireland LAMS Colloborative, & Oppong, K. (2022). Lumen-apposing metal stents for drainage of pancreatic fluid collections: does timing of removal matter?. Gut, 71(5), 850–853. https://doi.org/10.1136/gutjnl-2021-325812
2) Bazaga Pérez de Rozas, S., Chahal, P., Sánchez-Ocaña, R., García-Alonso, F. J., Carbajo, A. Y., de la Serna-Higuera, C., & Manuel, P. M. (2019). Buried lumen-apposing metal stent (LAMS) in esophagogastric anastomosis: the LAMS-in-LAMS rescue treatment. Endoscopy, 51(6), 600–601. https://doi.org/10.1055/a-0861-99074)
3) Ligresti, D., Cipolletta, F., Amata, M., Cimavilla, M., Traina, M., & Tarantino, I. (2018). Buried lumen-apposing metal stent (LAMS) following endoscopic ultrasound-guided gallbladder drainage: the LAMS-in-LAMS rescue treatment. Endoscopy, 50(8), 822–823. https://doi.org/10.1055/a-0624-2050
4) Honan, B. M., Mulki, R., & Sánchez-Luna, S. A. (2024). Bridging the Buried GATE: LAMS-in-LAMS Rescue Technique for Endoscopic Ultrasound-Directed Transgastric ERCP. ACG case reports journal, 11(9), e01497. https://doi.org/10.14309/crj.0000000000001497

Clinical Case Summary

A 62-year-old woman with hypothyroidism and multiple sclerosis presented to our care to underwent outpatient esophagogastroduodenoscopy (EGD) after one episode of hematemesis, which had occurred 20 days earlier following the use of Naproxen (440 mg/day) for low back pain. Proton pump inhibitor (PPI) therapy had been initiated afterward. EGD revealed a 20 mm ulcer in the gastric body with an adherent clot. Upon gastric distension, oozing bleeding from the ulcer was observed and treated with adrenaline injection and endoscopic clips. Diffuse oozing from the gastric mucosa and multiple suction-related hematomas were also noted. The patient was hospitalized and started on high-dose intravenous PPI therapy and fasting. H. pylori serology was negative. Physical exam showed hepatomegaly and lower limb edema. Lab tests revealed elevated cholestatic enzymes, proteinuria, leukocytosis, hypoalbuminemia, mild anemia, and a monoclonal spike on serum protein electrophoresis. Abdominal ultrasound confirmed hepatomegaly. Autoimmune, viral, and metabolic liver diseases were excluded. Liver magnetic resonance raised suspicion of a storage disorder. Liver biopsy confirmed AL-type amyloid deposits. A follow-up EGD was performed, in absence of further GI bleeding, to obtain gastric biopsies. It showed a 1 cm ulcer with an adherent clot in the fundus. Again, insufflation triggered diffuse oozing from the ulcer and gastric mucosa with suction-related hematomas. Histology confirmed amyloid deposits. Suspecting multiple myeloma, immunofixation identified Bence-Jones proteinuria and hypergammaglobulinemia. Spinal MRI showed vertebral collapse at D12–L1; cardiac MRI excluded cardiac involvement. MM with secondary AL amyloidosis was diagnosed. Treatment with targeted therapy led to symptom resolution.This case highlights the importance of considering rare systemic diseases in atypical GI bleeding, especially when triggered by minimal stimuli such as gastric distension.

References

1. Histological images of liver and stomach with amyloid deposits.
2. Endoscopic images of gastrointestinal bleeding manifestations in patients with gastric amyloidosis

Clinical Case Summary

Radiation proctitis is a common complication of radiotherapy and up to 80% of patients with chronic radiation proctitis experience rectal bleeding. Here we report the case of a 56-year-old man with type 3 von Willebrand disease (VWD) and high-titer VWF inhibitors who developed life-threatening rectorrhagia following prostate cancer radiotherapy, requiring urgent hospitalization and almost daily blood transfusions. Endoscopy revealed rectal angiodysplasia secondary to radiation proctitis as the bleeding source. Initial management included administration of recombinant FVIIa and tranexamic acid, paired with endoscopic hydrogel application and argon plasma coagulation (APC). Despite this, bleeding persisted with formation of rectal ulcers, prompting administration of prothrombin complex concentrate, extended half-life recombinant FVIII, plasma-derived VWF/FVIII concentrate, factor XIII, fibrinogen, octreotide, and bevacizumab. All interventions were ineffective and thus, after four weeks, off-label emicizumab in combination with rFVIIa was initiated, resulting in temporary improvement and subsequent discharge.
Four days later, the patient developed rectorrhagia and hemorrhagic shock. Repeated APCs and tamponade were unsuccessful, prompting a bipolar sigmoidostomy placement. Still, bleeding persisted, warranting an innovative and unconventional approach which included endoscopic application of autologous platelet-rich fibrin (PRF) and regular platelet transfusions despite normal platelet counts. Only after PRF applications were started hemostasis was gradually achieved. The patient continued emicizumab prophylaxis with biweekly outpatient PRF applications. At 10-month follow-up, he remained free of anemia and recurrence of gastrointestinal bleeding.
This case demonstrates successful management of refractory bleeding in inhibitor-complicated type 3 VWD using a combination of emicizumab and local PRF application when conventional approaches failed.

References

Figure 1. Endoscopic view of a large rectal ulceration with active bleeding, adherent clots, and visible blood vessels.
Figure 2. Endoscopic view of a healing rectal ulceration covered with fibrin.

Clinical Case Summary

A 36-year-old man with a history of chronic diarrhea was admitted with isolated oedemato-ascitic syndrome and general deterioration.On examination,he had conjunctival subicterus bilateral lower limb edema, thoracoabdominal collateral venous circulation and ascites.As part of the etiological work-up of the oedemato-ascitic syndrome,Cardiac and renal causes were ruled out.Chest X-ray showed a hilar opacity with tracheal retraction.GeneXpert test on sputum confirmed pulmonary tuberculosis.Paracentesis showed non-infected transudate ascites with a serum-ascites albumin gradient at 11 g/L.Blood tests showed microcytic hypochromic anemia,normal platelet count,and elevated liver tests with AST at 18 times the upper limit of normal (ULN), ALT at 11× ULN, alkaline phosphatase at 3.3× ULN,normal GGT,prothrombin time at 25%,and a direct hyperbilirubinemia of 24 mg/L.Viral serologies were negative.Serum protein electrophoresis showed a beta-gamma bridging pattern with hypergammaglobulinemia.Antinuclear antibodies were positive, while the remaining autoimmune panel and Wilson's disease screening were negative.Abdominal CT angiography showed hepatomegaly with hypertrophy of the caudate lobe,a “flip-flop” pattern,stenosis of the intrahepatic inferior vena cava, and non visualization of the hepatic veins confirming Budd-Chiari syndrome(BDS).The etiological work-up of the chronic diarrhea revealed negative HIV serology,normal TSH,and elevated IgA anti-transglutaminase antibodies.Ileocolonoscopy showed partial villous atrophy in the ileum,supporting the diagnosis of coeliac disease (CD).Given the radiological features suggestive of BDS,a thrombophilia work-up was performed and was negative.An autoimmune etiology related to CD was retained as the most likely cause of BDS.A gluten-free diet resolved the diarrhea.Anticoagulation was started, but persistent portal hypertension and difficulty maintaining therapeutic anticoagulation led to referral for a portosystemic shunt.

References

An actual image of the thoracoabdominal collateral venous circulation of the patient, and his abdominal CT angiography.

Introduction

The incidence of rectal neuroendocrine neoplasms (R-NENs) has been increasing annually. While most R-NENs are solitary, multiple lesions are relatively rare. Previous studies have suggested that patients with ≥8 lesions are more likely to exhibit lymphatic invasion and metastasis, indicating higher risk. However, the clinical and pathological features, prognosis, and endoscopic treatment outcomes of multiple R-NENs remain poorly understood. This study aimed to clarify the clinical characteristics and endoscopic treatment efficacy of multiple R-NENs through a retrospective analysis of cases from our hospital, providing evidence for clinical management.

Aims & Methods

This retrospective study included 28 patients with multiple R-NENs treated endoscopically at the Cancer Hospital of the Chinese Academy of Medical Sciences between January 2010 and January 2024. Patients were divided into two groups based on lesion count: the "several group" (n < 8) and the "numerous group" (n ≥ 8). All patients underwent preoperative imaging to exclude lymph node metastasis and distant metastases. Data on demographics, lesion characteristics, endoscopic procedures, pathological findings, and follow-up outcomes were collected and analyzed.

Results

We compared endoscopic treatment outcomes between patients with several (＜8, n=24) and numerous (≥8, n=4) rectal neuroendocrine tumors. The numerous group had a larger median maximum diameter (9.50 mm vs. 6.00 mm, p=0.337) and included one G2 case (25.0%) versus none in the several group, though differences were not significant. Notably, endoscopic complete resection was achieved in 100% of patients in both groups. All other parameters, including margin positivity, vascular invasion, neural invasion, and R0 resection rate, showed no significant differences. However, the numerous group had a significantly higher recurrence rate (75.0% vs. 0%, p=0.001). These findings highlight lesion count as a critical prognostic indicator for recurrence after endoscopic treatment.

Conclusion

Patients with ≥8 rectal neuroendocrine tumors exhibited more aggressive disease behavior, characterized by higher recurrence rates. These findings underscore the importance of lesion count as a prognostic indicator and suggest that patients with ≥8 lesions may require more intensive follow-up and management. For patients with <8 lesions, endoscopic treatment is effective when lymph node and distant metastases are excluded preoperatively. Future studies with larger datasets are needed to validate these findings and explore optimized treatment strategies.

Disclosure

The authors declare no conflicts of interest in relation to this study.

Introduction

Gut metabolites are suggested to be potential etiological factors of irritable bowel syndrome (IBS). However, the potential mechanisms remain unclear.

Aims & Methods

This study aimed to elucidated the effect and mechanism of gut metabolite homocysteine(Hcy) in IBS development. A stress-induced IBS model was established using neonatal maternal separation (NMS), while a post-inflammatory IBS (PI-IBS) model was constructed by intracolonic administration of trinitrobenzene sulfonic acid (TNBS). A high-methionine diet was applied to elevate homocysteine levels in both blood and feces of mice or rats. Visceral hypersensitivity was assessed by measuring abdominal withdrawal reflex (AWR) and visceral motor responses (VMR). Carmine red was used to determine whole intestinal transit time. The composition of the gut microbiota in rats fecal samples was analyzed by using 16S rRNA sequencing. A series of cell and molecular experiments, including immunostaining, western blot and ELISA was conducted to illustrate the mechanism.

Results

We found that fecal Hcy levels were significantly elevated in both patients with IBS and rats with IBS induced by water avoidance stress (WAS). Moreover, fecal Hcy levels were positively correlated with the severity of IBS symptoms in patients. Next, we investigated the effects of Hcy on the gut-brain axis in NMS rat model and TNBS mice model. Our study showed that elevated Hcy in stool exacerbated visceral hypersensitivity, increased intestinal permeability and disrupted the mucus barrier of IBS rats/mice. Interestingly, folic acid supplementation lowered the Hcy levels and alleviated the effect of Hcy in exacerbating IBS phenotype. Mechanistically, treatment of Hcy inhibited the expression of NLR family pyrin domain containing 6 (NLRP6) and downregulated mucin MUC2 expression in LS174T and intestinal organoids, and impaired the expression of tight junction proteins ZO1 and Occludin in Caco2. Genetic overexpression of NLRP6 and PPARγ agonist Rosiglitazone rescued the inhibitory effect of Hcy on MUC2 expression. In addition, by using 16S rRNA sequencing, we found that Hcy reduced gut bacterial diversity and lowered the abundances of Coprococcus, Roseburia and Acetitomaculum in NMS rats.

Conclusion

Our study showed that gut metabolite Hcy was involved in the pathogenesis of IBS by disrupting mucus barrier and disturbing gut microbiota, indicating Hcy as a therapeutic target and folic acid administration as a potential strategy for IBS management.

Clinical Case Summary

Condyloma acuminatum represents the clinical expression of epidermal infection caused by the human papillomavirus (HPV) one of the most common sexually transmitted diseases. These lesions range from benign papules to potential precursors of anal squamous cell invasive carcinoma.
Conventional treatments include surgical excision, cryotherapy, immunotherapy and CO₂ laser ablation.Endoscopic approaches such as radiofrequency ablation (RFA) and argon plasma coagulation (APC) are emerging as therapeutic alternatives with promising efficacy and safety profiles.
We report the case of a 49-year-old woman with no relevant medical or surgical history presented with rectal bleeding. Colonoscopy revealed a flat, polypoid lesion circumferentially involving approximately 3–4 cm of distal rectum (Fig1). Biopsy confirmed HPV-6–related condyloma. She underwent transanal minimally invasive surgery (TAMIS) in 2023.
One year later, symptoms recurred. Colonoscopy showed a similar lesion now extending over more than 5 cm of the lower rectum (Fig2-3).Given the lesion's recurrence, extensive involvement, and challenging location, endoscopic RFA was selected as the treatment modality. Using a HALO90° catheter (BARRX Medical Inc.) introduced through a flexible colonoscope, high-energy pulses (1000 pulses at 15 Joules per pulse) were applied to the affected mucosa, achieving near-complete ablation. Residual lesions and the anal canal were subsequently treated with APC (AXIAL catheter, 0.8 L/min, 30 W)(Fig4). The procedure was completed on an outpatient basis without immediate complications.
Post-procedurally, the patient reported transient anal pain and mild bleeding. At 3-month follow-up, mucosal healing was adequate (Fig5), with minor residual lesions successfully treated with APC (Fig6).
This case highlights the utility of combined endoscopic RFA and APC in managing extensive, recurrent rectal condyloma refractory to surgery reducing morbidity and preserving anorectal function.

References

1. Mark Schiffman, Susanne Krüger Kjaer, Chapter 2: Natural History of Anogenital Human Papillomavirus Infection and Neoplasia, JNCI Monographs, Volume 2003, Issue 31, June 2003, Pages 14-19, https://doi.org/10.1093/oxfordjournals.jncimonographs.a003476
2. Ramamoorthy S, Liu YT, Luo L, Miyai K, Lu Q, CarethersJM:Detection of multiple human papillomavirus genotypes in analcarcinoma. Infect Agent Cancer, 2010; 5:17.
3. Wiley DJ, Douglas J, Beutner K, et al.:External genital warts:Diagnosis, treatment, and prevention. Clin Infect Dis, 2002; 35(Suppl 2), S210-24.
4. Goldstone, R.N., Hasan, S.R., Drury, S. et al. A trial of radiofrequency ablation for anal intraepithelial neoplasia. Int J Colorectal Dis 32, 357–365 (2017). https://doi.org/10.1007/s00384-016-2679-2
5. Goldstone, R. N., Hasan, S. R., & Goldstone, S. E. (2017). Brief report: radiofrequency ablation therapy for anal intraepithelial neoplasia: results from a single-center prospective pilot study in HIV+ participants. JAIDS Journal of Acquired Immune Deficiency Syndromes, 76(4), e93-e97.
6. Smulian, A. G., Moore, D. M., Robertson, J. C., & Kralovic, S. M. (2014). Phase I Study Demonstrates Safety and Tolerability of Radiofrequency Ablation (RFA) of the Anal Mucosa. HIV Clinical Trials, 15(1), 36–44. https://doi.org/10.1310/hct1501-36.
7. Bulsiewicz WJ, Shaheen NJ. The role of radiofrequency ablation in the management of Barrett’s esophagus. Gastrointest Endosc Clin N Am 2011; 21: 95-109.