Clinical Case Summary

A 57-year-old man was admitted to our department because of edema of both lower extremities and diarrhea for more than 1 year. The patient underwent a subtotal gastrectomy more than 10 years ago. On admission, the albumin was 15.2 g/L, and the effect of supplementation was poor; renal function, immunology and other tests showed no abnormality. Enhanced CT scan of the whole abdomen suggested that the proximal small intestine of the gastrointestinal anastomosis connected to the adjacent transverse colon by a localized duct-like structure (fig.1). Gastroscopy revealed an opening on the left side of the gastric-small bowel anastomosis, which was observed as a colon after entering the scope, and a regular colonic pouch and smooth colonic mucosa were visible (fig.2). X-ray of small bowel imaging showed early colonic visualization (fig.3). In summary, gastrocolic fistula was considered. The patient's bilateral lower extremity edema was caused by hypoproteinemia, which turned out to be caused by a gastrocolic fistula that prevented most of the food from passing through the small intestine, affecting protein absorption. The patient therefore underwent surgery. 1-month-postoperative follow-up showed the albumin had increased to 32.3g/L, significant improvement of edema and 2-3 times per day of defecation.

Clinical Case Summary

A 74-year-old man with a history of distal gastrectomy was hospitalized because of left lower extremity edema. Occlusion of left common iliac vein (CIV) and severe stenosis of left external iliac vein (EIV) were found (Video.1). Stents were used to release the edema. In order to investigate the pathogenesis, contrast-enhanced computed tomography was then carried out. Diffuse enlargement and continued enhancement of pancreas were found (Figure.1). Soft-tissue mass with low density grew around inferior mesenteric artery, left common iliac artery (CIA), left internal and external iliac arteries (Figure.2). Left CIV and EIV were compressed by the soft-tissue mass. Elevated serum immunoglobulin G4 (IgG4) was 598mg/dl. Therefore, the diagnosis of autoimmune pancreatitis (AIP) was confirmed. The soft-tissue mass was considered as AIP-associated retroperitoneal fibrosis (RF). The left lower extremity edema was actually attributed to AIP. Glucocorticoid was initially administered at 60mg/day for 2 weeks, and tapered by 5mg every week. Until to 20mg/day, glucocorticoid was tapered by 2.5mg every week. And it was finally maintained at a dose of 5mg/day.
Sausage-like shape is a typical sign of AIP¹. That serum IgG4 is more than 135mg/dl can be used for differential diagnosis from pancreatic cancer². According to the guideline², AIP was definitely confirmed based on diffuse enlargement of pancreas and elevated serum IgG4. Patients sometimes suffer from extra-pancreatic lesions, which are so called other organ involvement², such as RF. In this patient, the space between left CIA and vertebra became much narrow due to AIP-associated RF. Its compression contributed to the occlusion of left CIV that ran through the gap, resulting in the clinical symptoms of left lower extremity venous reflux disorder. This is the first AIP patient who primarily presented with left lower extremity edema. And no patient has been reported to suffer from left CIV occlusion caused by AIP-associated RF.

References

1. Masamune AA-O, Kikuta K, Hamada S, et al. Nationwide epidemiological survey of autoimmune pancreatitis in Japan in 2016. Journal of gastroenterology 2020;55(4):462-470.
2. Okazaki K, Kawa S, Kamisawa T, et al. Amendment of the Japanese consensus guidelines for autoimmune pancreatitis, 2020. J Gastroenterol 2022;57:225-245.

Clinical Case Summary

A 29-year-old man was admitted to the hospital with complaints of rectal tenesmus, a sensation of incomplete evacuation, diarrhea occurring 6-8 times daily, and occasional constipation. Digital rectal examination revealed a hard, immovable mass 5 cm from the anus, with an upper margin that could not be palpated. Laboratory tests showed a C-reactive protein (CRP) level of 4.68 mg/L and an erythrocyte sedimentation rate (ESR) of 28.0 mm/h. The tuberculosis-interferon gamma release assay and fecal occult blood test were positive. Computed tomography (CT) and rectal endoscopic ultrasound (EUS) showed a 6 cm mass in the lower rectum, thickened rectal walls with uneven enhancement, multiple enlarged lymph nodes and a solid perianal nodule measuring 5.0×4.0 mm. Endoscopy revealed irregular thickening of rectal wall, nodular lesions with erythema and erosion from 10 cm proximal to the anal canal, and intestinal stenosis with a fistula. Multiple biopsies from the colonoscopy showed chronic mucosal inflammation with hyperplastic glandular epithelial cells and mucus, but no malignancy was detected.
The patient received sequential treatments of metronidazole, mesalazine, and diagnostic antitubercular drugs, but showed no improvement. Finally, an endoscopic mucosal resection (EMR) was performed. Biopsy findings revealed crypt branching and distortion, reactive epithelial hyperplasia, a few chronic inflammatory cells in the lamina propria, and fibrosis of the submucosa, consistent with mucosal prolapse.
Rectal mucosal prolapse syndrome (RMPS) poses significant diagnostic challenges due to its overlapping clinical, endoscopic, and histopathological features with other rectal disorders, such as Crohn' disease, intestinal tuberculosis, and colorectal carcinoma^1-3. Detailed endoscopic evaluation and deep histopathological analysis is essential. In patients with a long history of abnormal defecation habits, RMPS should be considered in the differential diagnosis of rectal masses.

References

1. Hayasaka J, Hoteya S, Tomizawa K, et al. The Long-term Efficacy of Endoscopic Submucosal Dissection in the Treatment of Symptomatic Mucosal Prolapse Syndrome. Intern Med 2021;60:1005-1009.
2. Tang X, Han C, Sheng L, et al. Rectal mucosal prolapse with an emphasis on endoscopic ultrasound appearance. Dig Liver Dis 2021;53:427-433.
3. Chiang JM, Changchien CR, Chen JR. Solitary rectal ulcer syndrome: an endoscopic and histological presentation and literature review. Int J Colorectal Dis 2006;21:348-56.

Clinical Case Summary

We present a 78 years old male with a seven year history of loose stools and weight loss, besides frequent belching, bloating and flatulence. Previous investigations included colonoscopy, gastroscopy, SeHCAT scan and CT scan which were unremarkable apart from severe diverticulosis. His medical history includes diverticulitis episodes. Faecal elastase was low suggestive of pancreatic exocrine insufficiency. Trial of pancreatic enzyme replacement therapy led to some improvement, but he continued to report intermittent bloating.
Repeat colonoscopy was arranged. During the colonoscopy procedure, the colonoscope surprisingly entered the duodenum on passing around the sigmoid colon, suggesting the presence of a wide fistula between the colon and duodenum. Subsequent barium contrast fluoroscopy confirmed a fistulous tract between the third part of duodenum and the sigmoid-descending junction (figure 1). Upon closer review of his previous CT scans, a similar fistula tract was revealed, though it was never reported before (figure 2).
Benign Duodeno-colic Fistula (DCF) is an unusual complication of different gastrointestinal inflammatory conditions. The most common cause being perforated duodenal ulcer¹ .Other causes include IBD, regional enteritis, tuberculous lymphadenitis and duodenal diverticulum. Most frequently DCF presents with malabsorption symptoms like weight loss and diarrhoea, which may be observed in up to 80%² of cases. Colicky abdominal pain³ and eructation are also observed.
In our case the most likely cause for the DCF was severe sigmoid diverticulosis which is extremely rare. His symptom presentations are likely secondary to bacterial overgrowth.
The gold standard confirmatory test for DCF is barium contrast fluoroscopy⁴. Whilst the curative treatment for symptomatic cases being surgical correction⁵, this was deemed unnecessary as he was well and symptoms were only intermittent. He was treated with oral antibiotics when symptoms flared to good effect.

References

1. HERSHENSON, L. M. and J. B. KIRSNER. Duodenocolic Fistula. Gastroenterology, 19:864, 1951.
2. Higgins, P. M. (1963) Benign Duodenocolic Fistula. Canadian Medical Association Journal. 1963 Oct 26; 89(17): 893–895
3. Melissas J., Schoretsanitis G., Daskalakis M., Tsiftsis D.D.: Sigmoidoduodenal fistula as a rare complication of colonic carcinoma: report of a case. Surg Today 2003; 33: pp. 623-625.
4. Barton D.J., Walsh T.N., Keane T., Duignan J.P.: Malignant duodenocolic fistula. Report of a case and review of the literature. Dis Colon Rectum 1987; 30: pp. 636-637
5. Kamath AS, Iqbal CW, Pham TH, et al. Management and Outcomes of Primary Coloduodenal Fistulas. J Gastrointest Surg 2011;15(10):1706-11.

Clinical Case Summary

Introduction: Heterotopic gastric mucosa (HGM) is not uncommon throughout the gastrointestinal (GI) tract, nevertheless its presence in the lower GI is considered as a rare entity. The purpose of this study is to report a clinical case of HGM located in the rectum, which was presented with intermittent bleeding. The medical history of the patient and the way was treated are presented.
Case presentation: A 40-year-old male patient with an unremarkable medical history presented to the outpatient clinic suffering from anal pain, accompanied with intermittent rectal bleeding. At colonoscopy, a solitary flat elevated lesion with central depression, approximately 2 cm in size, was identified in the lower part of the rectum. Histological examination of the biopsy specimen showed heterotopic gastric mucosa while Giemsa stain was positive for Helicobacter Pylori (HP). First-line treatment for HP eradication including a 14-day course of proton pump inhibitor (PPI), amoxicillin, clarithromycin and metronidazole was administered. One month after therapy was completed, the patient underwent a Urea Breath Test (UBT) and an HP antigen stool examination, which were both negative. Meanwhile, the symptoms had been resolved. Unfortunately, the patient never returned to follow the standard procedure of proper clinical and endoscopic re-evaluation.
Conclusion: HGM can be either congenital, as a result of an error in the differentiation of the endodermal stem cells during organogenesis, or acquired, as a metaplasia during the repair of damaged epithelium. When located in rectum, HGM can be presented with symptoms such as bleeding, abdominal or anal pain. Moreover, complications such as ulcer and even adenocarcinoma have been reported. Although endoscopic or surgical resection is considered as the radical therapy of the HGM in rectum, administration of PPIs, inhibitors of histamine-receptors or HP eradication treatment, in cases HP is present, is effective in short-term symptoms relief.

References

1) Iacopini F, Gotoda T, Elisei W, Rigato P, Montagnese F, Saito Y, Costamagna G, Iacopini G. Heterotopic gastric mucosa in the anus and rectum: first case report of endoscopic submucosal dissection and systematic review. Gastroenterol Rep (Oxf). 2016 Aug;4(3):196-205. doi: 10.1093/gastro/gow006. Epub 2016 Apr 21. PMID: 27103738; PMCID: PMC4976682.
2) Devereaux CE, Devereaux RG. Heterotopic gastric mucosa of the rectum with a review of the literature. J Clin Gastroenterol. 1994 Jul;19(1):41-5. doi: 10.1097/00004836-199407000-00011. PMID: 7930432.
3) Chrysanthos, N. & Anagnostopoulou, Evagelia & Daskalaki, Anna & Patsavela, Stamatia. (2015). Images of the Month. The American journal of gastroenterology. 110. 498. 10.1038/ajg.2014.251.
4) Chen WG, Zhu HT, Yang M, Xu GQ, Chen LH, Chen HT. Large heterotopic gastric mucosa and a concomitant diverticulum in the rectum: Clinical experience and endoscopic management. World J Gastroenterol 2018; 24(30): 3462-3468 [PMID: 30122884 DOI: 10.3748/wjg.v24.i30.3462]

Introduction

Lower abdominal pain in patients with diverticular disease (DD) can be very challenging in clinical practice. In fact, it is matter of debate whether symptomatic uncomplicated diverticular disease (SUDD) could be considered a disease its own or it represents the coexistence of irritable bowel syndrome (IBS) in patients with colonic diverticulosis. Moreover, after an episode of acute diverticulitis, patients may complain of chronic-recurrent lower abdominal pain, only sometimes related to a recurrent diverticulitis.

Aims & Methods

We aimed to assess clinical features of lower abdominal pain, in terms of presence, severity and characteristics of short-lasting and long-lasting, and the possible overlap with IBS in patients with SUDD and previous diverticulitis (PD). We utilized baseline data from the REMAD (Diverticular Disease Registry), a 5-yr prospective, observational, cohort study, involving 47 Italian centres. Patients who fulfilled criteria for diagnosis of SUDD (n=300) and PD (n=212) were asked to fill in questionnaires for characterization of abdominal pain in the past 6 months: i) questionnaire for short-lasting abdominal pain (<24 hours); ii) questionnaire for long-lasting abdominal pain (>24hours); iii) questionnaire for IBS following Rome III Criteria.

Results

One hundred and forty-eight SUDD and 118 PD patients fulfilled all questionnaires. Seventy-five patients out of 266 (28.2%) fulfilled criteria for IBS diagnosis following Rome III Criteria. IBS criteria were reported more frequently in SUDD (37.2%) compared to PD patients (17.1%) (p<0.001). The prevalent IBS subtype was the diarrhoea-one (IBS-D), that was more frequent in SUDD compared to PD (22.3% vs 10.2%, p=0.009), followed by the constipation subtype (IBS-C: 12.2% vs 5.9%, p=0.084) and mixed subtype (IBS-M: 2.7% vs 0.8%, p=0.268). Considering the abdominal pain pattern, several differences were found between SUDD and PD in terms of long-lasting abdominal pain characteristics. The number of episodes was significantly higher in SUDD (6.6±11.9) compared to PD patients (3.4± 6.9) (p <0.001). PD patients reported long-lasting pain more frequently localized in the lower left abdomen (p <0.001), while in SUDD it was more frequently diffuse (p= 0.008) or localized in the lower right quadrant (p=0.016). Features associated with long-lasting abdominal pain were more often reported in patients with PD than SUDD [fever (p<0.001), confinement to bed (p=0.021), medical consultations (p=0.026), antibiotic therapy (p<0.001), and hospitalization (p<0.001)]. Compared to PD, SUDD patients reported a significantly more frequent amelioration of pain with antispasmodics use (43.8% vs 36.1%, p=0.003) and a more frequent onset of long-lasting pain after acute gastrointestinal symptoms (i.e. fever, vomit or diarrhoea) (90.8% vs 72.4% p=0.002). Episodes of short-lasting abdominal pain were significantly more frequently reported in SUDD than in PD patients (90.5% vs 78.8% p = 0.007), although there were no significant differences in terms of severity, localization, and pain’s relief with defecation between the two groups of patients.

Conclusion

This study shows that less than a third of patients with diverticular disease reported an overlap with IBS. Moreover, SUDD patients have a different pattern of lower abdominal pain compared to PD (i.e. length of pain, number of episodes, localization, amelioration with antispasmodics, associated features). Recognizing these differences could help clinician’s through a correct differential diagnosis addressing the diagnostic approach and therapeutic management.

Introduction

Treatment of complex fistula and residual anastomotic insufficiencies remain a challenge even in an interdisciplinary setting. Endoscopic standard techniques for closure like clips, OTSC or SEMS are limited to special indications. Endoscopic suturing may offer an approach to close complex fistula orifices.

Aims & Methods

Endoscopic suturing was used for interventional closure of complex insufficiencies in the lower GI-tract. 5 consecutive patients were treated using standard Endosuturing (SutuArt, Olympus) or modified suture strategy using an additional working channel (ACEES). In three patients residual insufficiencies in the rectum were visible after complex vacuum therapy. In one patient a recto-vaginal fistula was clinical apparent. In one patient an iatrogenic perforation was occurred within dilation therapy for anastomotic stenosis. Due to size or location of the defects a standard closure technique was either not applicable or failed in a prior attempt.

Results

Technical success, defined as macroscopic defect closure, was achievable in 5 of 5 patients. Mean time for intervention was 57(±14)min. For 3 patients bimanual work using additional working channel enhanced endoscopic suturing (ACEES) was performed. In endoscopic surveillance permanent closure was achieved in 4 of 5 patients.

Conclusion

Endoscopic suturing is a newly available technique. Beyond its basic indication for defect closure after ESD this procedure, also in combination with bimanual suturing (ACEES), may offer an excellent opportunity for treatment of complex fistula.

Disclosure

nothing to disclose