Clinical Case Summary

Introduction: Gallstone ileus is an uncommon complication of cholelithiasis and a rare cause of intestinal occlusion. While most luminal obstruction occurs in the terminal ileum, only 1-3% of impactions occur proximally, leading to gastric outlet obstruction, a condition known as Bouveret’s syndrome.
Case: A ninety-year-old woman with a past medical history of heart failure and obesity presented to the emergency department with abdominal pain, nausea and vomiting for one week. At admission, she was hypotensive, tachycardic, with a distended abdomen with diffuse pain.
Laboratory tests showed raised inflammatory markers, acute kidney injury and hyperlactatemia. Total bilirubin and liver enzymes were within normal range.
Computed tomography demonstrated a 36mm gallstone in the jejunum with distention of the bowel proximally. Additionally, it showed remarkable distention of stomach with stasis, pneumobilia and a 25mm gallstone in the second portion of the duodenum.
Gastroenterology and General Surgery discussed the case and an urgent laparotomy was planned with a gastroscopy in the same operative time to remove the proximal gallstone.
Intraoperatively, the jejunal gallstone was identified and an enterectomy was performed. Subsequently, a gastroscopy was conducted which showed a gallstone impacted in the second portion of the duodenum. The gallstone was mobilized to the duodenal bulb with a net and it was retrieved with the same device, with no complications.
The initial postoperative course was uneventful.
Conclusions: The authors highlight this case because it is a rare presentation of two different forms of gallstone ileus simultaneously in the same patient. Furthermore, we also highlight the success of the endoscopic approach. Despite the unequivocal surgical indication of the intestinal occlusion, endoscopy has been gaining more recognition for treatment of Bouveret’s syndrome since it is minimally invasive. In this case, it avoided an additional enterotomy.

Introduction

The randomized controlled OPTIC trial reported that optimised mercaptopurine treatment was superior to placebo in achieving clinical, endoscopic and histological outcomes at one year in ulcerative colitis (UC) patients failing 5-aminosalicylates after receiving induction treatment with corticosteroids.

Aims & Methods

We aimed to assess cost-effectiveness and cost-utility of therapeutic drug monitoring (TDM)-guided mercaptopurine treatment in UC. Data were collected during the OPTIC trial to perform a health economic evaluation for mercaptopurine treatment versus placebo in UC patients. The economic evaluation was performed from a health care and societal perspective using a cost-effectiveness and cost-utility analysis with a time horizon of one year. Volumes and costs of in-hospital care, out-of-hospital care, out-of-pocket expenses and productivity loss were assessed. Main outcomes were the costs per patient who achieved combined corticosteroid-free clinical remission and endoscopic improvement at one year and the costs per quality-adjusted life-year (QALY).

Results

In total, 59 patients were randomized to the intervention (n = 29) and control (n = 30) group. Patients on mercaptopurine treatment showed a non-significant gain in QALYs of 0.047 (-0.024 to 0.117) (P=0.184) corrected for baseline health utility. Non-significant differences in costs were €63 (-€1267 to €1434; P=0.927) in favour of placebo from a health care perspective and -€742 (-€3683 to €2016; P=0.644) in favour of mercaptopurine from a societal perspective, including patients’ out-of-pocket expenses and employers’ productivity loss. The probability of optimised mercaptopurine treatment being cost-effective was 0.80 at a willingness to pay per additional QALY of €20,000.

Conclusion

Optimised mercaptopurine treatment in UC patients failing 5-aminosalicylates is a cost-effective strategy from a societal perspective.

References

1. de Boer NKH, Peyrin-Biroulet L, Jharap B, et al. Thiopurines in inflammatory bowel disease: new findings and perspectives. J Crohns Colitis 2018;12:610–20.
2. Löwenberg, M., Volkers, A., van Gennep, S., Mookhoek, A., Montazeri, N., Clasquin, E., Duijvestein, M., van Bodegraven, A., Rietdijk, S., Jansen, J., van Asseldonk, D., van der Zanden, E., Dijkgraaf, M., West, R., de Boer, N., & D'Haens, G. (2023). Mercaptopurine for the Treatment of Ulcerative Colitis: A Randomized Placebo-Controlled Trial. Journal of Crohn's & Colitis, 17(7), 1055–1065.

Disclosure

NA

Introduction

Biliary stenting by endoscopic retrograde cholangiopancreatography (ERCP) is a common palliative endoscopic procedure for unresectable malignant biliary obstruction or for patients unfit for surgery.

Aims & Methods

We conducted a multicentre retrospective cohort study in 3 centres in Portugal, involving patients submitted to first palliative biliary drainage for malignant biliary obstruction by ERCP, between 2015 and 2022. This study aimed to identify predictive factors of 90-day mortality after endobiliary drainage and propose a predictive model. We have analysed data from 217 patients. Statistical analysis was performed using SPSS v.29.

Results

In our cohort, 51,2% (N=111) of patients were female and the mean age was 74 years. 52,5% (N=114) had stage IV disease and in 88% (N=191) the biliary obstruction resulted from primary tumour involvement. The most frequent tumour was primary pancreatic cancer (53,5%, N=116), followed by cholangiocarcinoma (26,7%, N=58). Metallic stents were used in 53,9% (N=117) of the patients, mainly with fully uncovered self-expandable metallic stents (66,7%, N=78). In the multivariate regression model, the independent predictors for 90-day mortality after first ERCP-guided stent placement were primary tumour size <3 cm (p = 0,014; OR 0,394; 95% CI, 0,187-0,830), stage IV malignant disease (p = 0,005; OR 3,776; 95% CI, 1,507-9,465), intrahepatic biliary obstruction (p = 0,004; OR 3,727; 95% CI, 1,518-9,150) and palliative chemotherapy after ERCP biliary drainage (p <0,0001; OR 0,44; 95% CI, 0,015-0,132). Using these variables, we developed a prediction model for the risk of mortality within 90 days after biliary drainage (Hosmer-Lemeshow X² = 2,907, p = 0,893) with R² of 44,1% in our cohort. The AUROC for our prediction model was 0,833 (p <0,0001; 95% CI, 0,774-0,892). By the Youden’s J statistic, the optimal cut-off value for was 0,4, with a sensitivity of 73,9% and specificity of 76,3%. Patients with a score < 0,2 had a low likelihood of death, whereas a score ≥ 0,9 had a high likelihood of death.

Conclusion

The present study was able to identify statistically significant predictive factors of 90-day mortality after ERCP-guided biliary stenting in unresectable malignant biliary obstruction, which may help to stratify the risk of death within 90 days post-stent placement. Since all these predictive factors can be determined before ERCP, this risk stratification could help in the definition of the proper palliative strategy for each patient.

References

Termsinsuk, P., Charatcharoenwitthaya, P., & Pausawasdi, N. (2022). Development and validation of a 90-day mortality prediction model following endobiliary drainage in patients with unresectable malignant biliary obstruction. Frontiers in oncology, 12, 922386. https://doi.org/10.3389/fonc.2022.922386;
Sripongpun, P., Attasaranya, S., Chamroonkul, N., Sookpaisal, T., Khow-Ean, U., Siripun, A., Kongkamol, C., Piratvisuth, T., & Ovartlarnporn, B. (2018). Simple Clinical Score to Predict 24-Week Survival Times in Patients with Inoperable Malignant Distal Biliary Obstruction as a Tool for Selecting Palliative Metallic or Plastic Stents. Journal of gastrointestinal cancer, 49(2), 138–143. https://doi.org/10.1007/s12029-017-9918-9;
Afshar, M., Khanom, K., Ma, Y. T., & Punia, P. (2014). Biliary stenting in advanced malignancy: an analysis of predictive factors for survival. Cancer management and research, 6, 475–479. https://doi.org/10.2147/CMAR.S71111.

Introduction

Differential diagnosis of solid pancreatic tumours (SPT) is a clinical challenge. Development of advanced imaging techniques associated with EUS, such as elastography and contrast enhancement, has been a step forward in this context. EUS-guided detective flow imaging (DFI) is a very recent technology allowing the evaluation of microvascularization and low-velocity blood flow without the need of contrast agents.

Aims & Methods

Aim of our study was to evaluate the findings obtained by EUS-DFI in SPT and compare them with contrast enhanced harmonic EUS (CEH-EUS).
Methods: A prospective, single-centre, observational study in patients who underwent EUS for the evaluation of SPT was designed. Procedures were performed with a linear echoendoscope (Fujifilm 740UT) attached to the ultrasound system Arietta 850. Lesions were characterized according to the vascularization pattern (hypervascular, isovascular and hypovascular) described with both methods. Final diagnosis was based on histology after EUS-guided biopsy. Data area shown as mean ± SD and percentage.

Results

44 patients were included (mean age 67 years, range 24-88, 23 males). Mean tumour size was 26.1 ± 15.7 mm. Fourteen (31.8%) lesions were located in the head of the pancreas, 19 (43.2%) in the body, 9 (20.5%) in the tail and 2 (4.5%) in the uncinate process. Final diagnosis was adenocarcinoma in 24 patients (54.5%), neuroendocrine tumour in 13 (29.5%), inflammatory mass in 3 (6.8%), autoimmune pancreatitis in 1 (2.3%), pancreatic necrosis in 1 (2.3%), accessory spleen in 1 (2.3%) and lipoma in 1 (2.3%). Agreement regarding the vascular pattern of SPT between CEH-EUS and EUS-DFI was reached in 97.7% of the cases. All adenocarcinomas were hypovascular, inflammatory masses and autoimmune pancreatitis isovascular, pancreatic necrosis avascular, and accessory spleen and lipoma hypervascular at both CEH-EUS and EUS-DFI. All 13 neuroendocrine tumours were hypervascular at CEH-EUS, 12 of them (90%) at EUS-DFI.

Conclusion

EUS-DFI allows evaluating the microvascular pattern of SPT with a similar accuracy than CEH-EUS, but without the need of using contrast agents.

Disclosure

Julio Iglesias-Garcia. Advisor Pentax, FujiFilm, Mediglobe

Clinical Case Summary

The authors present a case of a 22-year-old male with no previous medical history, apart from COVID-19 in January 2022, which had a mild course. He was referred to a medical appointment in our department after routine blood tests, from March 2022, that revealed abnormal liver biochemistry elevation (ALT 476U/L, AST 139U/L, GGT 135U/L, normal ALP and total bilirubin), which was normal in the previous year. He reported no symptoms and physical examination was unremarkable. There was no chronic medication, recent introduction of drugs, herbal products or epidemiology for zoonoses.
Abdominal ultrasound and CT scan with IV contrast showed discrete hepatomegaly with homogeneous steatosis, without focal lesions, and splenomegaly.
An extensive work-up excluded hepatotropic agents, metabolic causes, autoimmune aetiologies and haematological or lymphoproliferative disorders. The serum protein electrophoresis and immunofixation, immunoglobulins count, tumour markers and celiac disease serology were also normal. During this period, the patient maintained the same liver elevation pattern with stable values, without any alteration in total bilirubin, albumin, coagulation and hemogram, except for the appearance of discrete thrombocytopenia. We remained asymptomatic, with no signs of hepatic decompensation or haemorrhagic diathesis.
The percutaneous liver biopsy documented septal fibrosis and regenerative nodules, interface hepatitis, focal hepatocellular necrosis and, notably, the presence of numerous multinucleated giant hepatocytes, allowing the diagnosis of post-infantile giant cell hepatitis (PIGCH), in cirrhotic phase.
We present a case of PIGCH, with rapid progression to cirrhosis, whose aetiology remains unknow. Viral aetiologies account for 20% of the cases. Given the temporal association between SARS-CoV-2 infection and liver enzymes’ elevation, we cannot exclude this agent as a possible culprit. To our knowledge, this is the first case reporting this possible trigger.

References

Xiao, Y., Gao, Z. H., & Deschenes, M. (2021). Autoimmune post-infantile giant cell hepatitis: a case report and review of the literature. Canadian liver journal, 4(2), 104–109. https://doi.org/10.3138/canlivj-2020-0024;
Jiao, J., & Zhang, X. (2022). Post-infantile Giant Cell Hepatitis: A Literature Review and Meta-analysis. Journal of clinical and translational pathology, 2(3), 100–107. https://doi.org/10.14218/jctp.2022.00016.

Introduction

The use of a gluten-free diet (GFD) in healthy people has been steadily increasing worldwide based upon the unproven belief that avoiding gluten enhances overall wellness. In contrast, evidence indicates that voluntary GFD has an impact on living costs, psychosocial profile and may cause malnutrition in the long-term^1,2. Since dietary changes may significantly affect gut microbiota composition³, we tested whether a 30-day unnecessary GFD in healthy subjects has an impact on diversity and richness of gut microbiota.

Aims & Methods

116 fecal samples were collected from 30 thoroughly examined healthy subjects (21 females, 9 males; 34±14 years) recruited at St. Anna University Hospital in Ferrara, Italy. All subjects had no demonstrable metabolic, cardiovascular, respiratory, rheumatic and neurological illnesses and were asymptomatic for gastrointestinal symptom (established by Gastrointestinal Symptom Rating Scale, GSRS). Stool samples, collected at day 0, and 3, 7, 30 days from GFD were sequenced via shotgun metagenomic and computationally profiled (bioBAkery 3). Data were analyzed on MetaPhlAn 4.0 and HUMAnN 3.0 for taxonomic and functional profiling, respectively and relative abundance transformed via arcsin-sqrt. Only species present at any time point in at least 5 subjects were considered (600 species). Statistical analysis was performed using a linear mixed model and Friedman’s ANOVA. P values were combined and corrected for multiple hypothesis testing (q value).

Results

Species alpha-diversity did not significantly change during 30 days of GFD (p >0.05). However, during the GFD, 14 species significantly decreased (q < 0.05) in relative abundance. Most of the decreased bacteria belonged to Bifidobacterium genus (e.g. Bifidobacterium longum p=0.000000003; q=0.000001) or those involved in complex carbohydrate metabolism.

Conclusion

In healthy subjects, a 30-day GFD did not significantly affect gut eubiosis. However, consistent changes in the relative abundance of gut microbiota species showed a reduction of Bifidobacterium genus thus potentially affecting intestinal and immunological homeostasis and health status⁴.

References

1. Cardo, A. et al. Nutritional Imbalances in Adult Celiac Patients Following a Gluten-Free Diet. Nutrients 13, 2877 (2021).
2. Dennis, M., Lee, A. R. & McCarthy, T. Nutritional Considerations of the Gluten-Free Diet. Gastroenterology Clinics of North America 48, 53–72 (2019).
3. Caio, G. et al. Effect of Gluten-Free Diet on Gut Microbiota Composition in Patients with Celiac Disease and Non-Celiac Gluten/Wheat Sensitivity. Nutrients 12, 1832 (2020).
4. Tojo, R. et al. Intestinal microbiota in health and disease: Role of bifidobacteria in gut homeostasis. World J Gastroenterol 20, 15163–15176 (2014).

Introduction

Pancreatic cyst prevalence is about 2% to 3% in the general population(1,2). It is important to classify them accurately, given the fact that in premalignant lesions surgery could be curative. On the other hand, unnecessary pancreatic surgery could be associated with significant morbidity. The mucinous cysts are considered the premalignant types, traditionally the carcinoembryonic antigen (CEA) has been described as the analysis of cyst fluid to discern them from the non-mucinous. Recently, the cyst glucose value has been described as a better discriminant, however, there are different cut-off levels founds across the studies(3). The distribution of glucose in the body is almost uniform (4), a higher metabolic rate within a cyst translates into lower intracystic glucose, which correlates with high-grade injury (3). Therefore, we decided to explore the diagnostic performance of the glucometry-to-glucometry cyst ratio.

Aims & Methods

We conducted a prospective cohort from January 2021 to October 2022 at the endoscopy department at the National Institute of Medical Sciences and Nutrition “Salvador Zubiran” (INCMNSZ) in Mexico City. We included patients older than 18 years who underwent endoscopic ultrasound with FNA of a pancreatic cyst and had a fluid analysis of glucose, CEA, and amylase. We performed glucometry on-site on the patient and the cyst fluid with a FreeStyle Optium Neo glucometer. Glucometry to glucometry cyst ratio (glucometry patient/glucometry cyst) was calculated for each patient. Follow-up was performed and a final diagnosis was established by a multidisciplinary team, biopsy or surgical specimen. Finally, we performed a 2x2 table and a ROC curve with Youden index to establish the best cut off for diagnostic performance of glucometry-to-glucometry cyst ratio.

Results

24 patients were included 14 with mucinous cyst diagnosis and 10 with non-mucinous diagnosis, the median age was 58.5 (25-84), and 83% were female. CEA mean was 2331.7± 3359.3 ng/mL for mucinous vs 23.8± 62.6 ng/mL for non-mucinous (p= <0.001). The on-site cyst glucometry was 39.3± 22.1mgdL vs 112± 42.9 for the mucinous and non-mucinous respectively (p= 0.001). The glucometry/cyst glucometry relation was 3.30± 0.84 (mucinous) vs 1.13± 0.69 (non-mucinous), p= 0.001. The AUC was 0.971 (0.915 -1.000), 0.948 (0.853 – 1.00), and 0.964 (0.900 – 1.000) for glucose relation, glucometry, and CEA respectively. The sensitivity, specificity, PPV, NPV, and precision for CEA (192ng/dL) was 70%, 92.8%, 87.5%, 81.2%, and 83.3% while glucometry (<50mg/dL) was 80%, 85.7%, 83.3%, 85.7%, and 83.3%. Notably, the glucometry relation (>2.45) found a 90%, 92.8%, 90%, 92.8%, and 91.6% for the same parameters.

Conclusion

The glucometry-to-glucometry cyst relation is a novel, cheap, accessible, and superior diagnostic approach for the classification of mucinous and non-mucinous pancreatic cysts. Further studies with bigger sample sizes are needed to confirm these findings.

References

1. Laffan TA, Horton KM, Klein AP, et al. Prevalence of unsuspected pancreatic cysts on MDCT. AJR Am J Roentgenol 2008;191:802-7.
2. de Jong K, Nio CY, Hermans JJ, et al. High prevalence of pancreatic cysts detected by screening magnetic resonance imaging examinations. Clin Gastroenterol Hepatol 2010;8:806-11
3. Guzman-Calderon E, Md BM, Casellas JA, Aparicio JR. Intracystic Glucose Levels Appear Useful for Diagnosis of Pancreatic Cystic Lesions: A Systematic Review and Meta-Analysis. Dig Dis Sci 2021
4. Zierler K. Whole body glucose metabolism. Am J Physiol 1999;276:E409-26