[Enteritis cystica profunda]. / L'entérite kystique profonde.

Enteritis cystica profunda is a rare and benign disease defined as the invagination of the intestinal epithelium into the submucosa and more profound layers of intestinal wall leading to the formation of mucin-filled cystic spaces. We reported the case of a 45-year-old female, suffering from a Crohn's disease, with a Koenig's syndrome, diarrhea, abdominal pain and weight loss. The colonoscopy and the abdominopelvic scan showed a terminal ileal stenosis, with parietal calcifications. A surgical ileocecal resection was decided. Gross examination of the ileocecal resection showed a thickening of the ileal wall, with many mucin-filled cysts measuring 1mm to 2cm, with some calcifications. The ileal mucosa was ulcerated, and showed a stenotic sector extending over 3cm. Histological examination showed acute ulcerated ileitis lesions, with chronic ileitis lesions and stenosis, compatible with the known diagnosis of Crohn's disease. There were also many cysts into the ileal wall. They were lined with a regular ileal epithelium. The cysts contained mucus, with some calcifications. Some cysts were ruptured, with extravasation of mucus within the wall. Cystica profunda can be found anywhere along the digestive tract. The physiopathology is not yet well understood, but it seems to be favored by chronic aggression of the intestinal wall. This pathology most often coexists with Crohn's disease. The main differential diagnosis is mucinous adenocarcinoma. Cystica profunda does not require any specific treatment.

Increased Primary Bile Acids with Ileocolonic Resection Impact Ileal Inflammation and Gut Microbiota in Inflammatory Bowel Disease.

BACKGROUND: Most Crohn's disease [CD] patients require surgery. Ileitis recurs after most ileocolectomies and is a critical determinant for outcomes. The impacts of ileocolectomy-induced bile acid [BA] perturbations on intestinal microbiota and inflammation are unknown. We characterized the relationships between ileocolectomy, stool BAs, microbiota and intestinal inflammation in inflammatory bowel disease [IBD]. METHODS: Validated IBD clinical and endoscopic assessments were prospectively collected. Stool primary and secondary BA concentrations were compared based on ileocolectomy and ileitis status. Primary BA thresholds for ileitis were evaluated. Metagenomic sequencing was use to profile microbial composition and function. Relationships between ileocolectomy, BAs and microbiota were assessed. RESULTS: In 166 patients, elevated primary and secondary BAs existed with ileocolectomy. With ileitis, only primary BAs [795 vs 398 nmol/g, p = 0.009] were higher compared to without ileitis. The optimal primary BA threshold [≥228 nmol/g] identified ileitis on multivariable analysis [odds ratio = 2.3, p = 0.04]. Microbial diversity, Faecalibacterium prausnitzii and O-acetylhomoserine aminocarboxypropyltransferase [MetY] were decreased with elevated primary BAs. Amongst ileocolectomy patients, only those with elevated primary BAs had diversity, F. prausnitzii and MetY reductions. Those with both ileocolectomy and intermediate [p = 0.002] or high [≥228 nmol/g, p = 9.1e-11]] primary BA concentrations had reduced F. prausnitzii compared to without ileocolectomy. Those with ileocolectomy and low [<29.2 nmol/g] primary BA concentrations had similar F. prausnitzii to those without ileocolectomy [p = 0.13]. MetY was reduced with ileitis [p = 0.02]. CONCLUSIONS: Elevated primary BAs were associated with ileitis, and reduced microbial diversity, F. prausnitzii abundance and enzymatic abundance of MetY [acetate and l-methionine-producing enzyme expressed by F. prausnitzii], and were the only factors associated with these findings after ileocolectomy.

One appendix too many: a severe IBD-U case in remission after appendectomy.

A 39-year-old female was diagnosed with inflammatory bowel disease unclassified (IBD-U) in 2011, during the post-partum period, after developing chronic bloody diarrhea and marked weight loss. She underwent colonoscopy showing erosive ileitis and pancolitis. Biopsies were compatible with unspecified chronic ileocolitis.

[The somewhat different cause of an obturation in stenosing terminal ileitis : Case report of a 39-year-old male patient with Crohn's disease]. / Die etwas andere Ursache einer Obturation bei stenosierender Ileitis terminalis : Fallbericht eines 39-jährigen Patienten mit M. Crohn.

A possible classical complication of Crohn's disease is the formation of a stenosis, which can occur throughout the course of the disease and can present differently depending on the narrowing of the lumen. This article reports the case of a 39-year-old male patient with a stenosing terminal ileitis, which was ultimately only manifested after obstruction by a foreign body.

Short- and Long-term Outcomes Following Side-to-side Strictureplasty and its Modification Over the Ileocaecal Valve for Extensive Crohn's Ileitis.

BACKGROUND AND AIMS: Postoperative recurrence remains a challenging problem in patients with Crohn's disease [CD]. To avoid development of short bowel syndrome, strictureplasty techniques have therefore been proposed. We evaluated short- and long-term outcomes of atypical strictureplasties in CD patients with extensive bowel involvement. METHODS: Side-to-side isoperistaltic strictureplasty [SSIS] was performed according to the Michelassi technique or modification of this over the ileocaecal valve [mSSIS]. Ninety-day postoperative morbidity was assessed using the comprehensive complication index [CCI]. Clinical recurrence was defined as symptomatic, endoscopically or radiologically confirmed, stricture/inflammatory lesion requiring medical treatment or surgery. Surgical recurrence was defined as the need for any surgical intervention. Endoscopic remission was defined as ≤i1, according to the modified Rutgeerts score. Deep remission was defined as the combination of endoscopic remission and absence of clinical symptoms. Perioperative factors related to clinical recurrence were evaluated. RESULTS: A total of 52 CD patients [SSIS n = 12; mSSIS n = 40] were included. No mortality occurred. Mean CCI was 10.3 [range 0-33.7]. Median follow-up was 5.9 years [range 0.8-9.9]. Clinical recurrence [19 patients] was 29.7% and 39.6% after 3 and 5 years, respectively. Surgical recurrence [seven patients] was 2% and 14.1% after 3 and 5 years, respectively. At the end of the follow-up, 92% of patients kept the original strictureplasty and deep remission was observed in 25.7% of the mSSIS patients. None of the perioperative variables considered showed a significant association with clinical recurrence. CONCLUSIONS: SSIS is safe, effective, and provides durable disease control in patients with extensive CD ileitis.

Epidemiology and Perioperative Mortality of Exploratory Laparotomy in Rural Ghana.

Background: Perioperative mortality rate (POMR) has been identified as an important measure of access to safe surgical and anesthesia care in global surgery. There has been limited study on this measure in rural Ghana. In order to identify areas for future quality improvement efforts, we aimed to assess the epidemiology of exploratory laparotomy and to investigate POMR as a benchmark quality measure. Methods: Surgical records were reviewed at a regional referral hospital in Eastern Region, Ghana to identify cases of exploratory laparotomy from July 2017 through June 2018. Patient demographics, health information, and outcomes data were collected. Logistic regression was used to identify predictors of perioperative mortality. Findings: The study included operations for 286 adult and 60 pediatric patients. Only 60% of patients were covered by National Health Insurance (NHI). The overall POMR was 11.5% (12.6% adults; 6.7% pediatric). Sixty percent of mortalities were referrals from outside hospitals and the mortality rate for referrals was 13.5%. Odds of mortality was 13 times greater with perforated peptic ulcer disease (OR = 13.1, p = 0.025) and 12 times greater with trauma (OR = 11.7, p = 0.042) when compared to the most common operation. Female sex (OR = 0.3, p = 0.016) and NHI (OR = 0.4, p = 0.031) were protective variables. Individuals 60 years and older (OR = 3.3, p = 0.016) had higher mortality. Conclusion: POMR can be an important outcome and quality indicator for rural populations. Interventions aimed at decreasing emergent hernia repair, preventing perforation of peptic ulcer disease, improving rural infrastructure for response to major trauma, and increasing NHI coverage may improve POMR in rural Ghana.

Longer small bowel segments are resected in emergency surgery for ileocaecal Crohn's disease with a higher ileostomy and complication rate.

BACKGROUND: Repeated intestinal resections may have disabling consequences in patients with Crohn's disease even in the absence of short bowel syndrome. Our aim was to evaluate the length of resected small bowel in patients undergoing elective and emergency surgery for ileocolic Crohn's disease. METHODS: A prospective observational study was conducted on patients undergoing surgery for ileocolonic Crohn's disease in a single colorectal centre from May 2010 to April 2018. The following patients were included: (1) patients with first presentation of ileocaecal Crohn's disease undergoing elective surgery; (2) patients with ileocaecal Crohn's disease undergoing emergency surgery; (3) patients with recurrent Crohn's disease of the distal ileum undergoing elective surgery. The primary outcomes were length of resected small bowel and the ileostomy rate. Operating time, complications and readmissions within 30 days were the secondary outcomes. RESULTS: One hundred and sixty-eight patients were included: 87 patients in the elective primary surgery group, 50 patients in the emergency surgery group and 31 in the elective redo surgery group. Eleven patients (22%) in the emergency surgery group had an ileostomy compared to 10 (11.5%) in the elective surgery group (p < 0.0001). In the emergency surgery group the median length of the resected small bowel was 10 cm longer than into the group having elective surgery for primary Crohn's disease. CONCLUSIONS: Patients undergoing emergency surgery for Crohn's disease have a higher rate of stoma formation and 30-day complications. Laparoscopic surgery in the emergency setting has a higher conversion rate and involves resection of longer segments of small bowel.

Ileítis isquémica idiopática, con sangrado digestivo oscuro manifiesto / Idiopathic ischaemic ileitis, with overt obscure digestive bleeding

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Stepwise Training in Laparoscopic Surgery for Complex Ileocolonic Crohn's Disease: Analysis of 127 Training Episodes.

INTRODUCTION: The inflammation encountered in Crohn's disease makes a minimally invasive approach challenging due to a thickened mesentery, fistulas, abscesses, and large phlegmons with high reported rates of conversion and septic complications. Aim of this study was to evaluate the feasibility of a stepwise approach to training in laparoscopic surgery for complex Crohn's disease. METHODS: Every surgical procedure was divided in 4 different training tasks: access and exposure, bowel mobilization, division of the mesentery, anastomosis. Extensive adhesiolysis and division and repair of fistulae were considered as additional tasks when present. The laparoscopic competence assessment tool was used to evaluate the safety and proficiency of the surgical performance. The primary outcome was the rate of training tasks successfully completed by surgical trainees. RESULTS: One hundred and twenty seven training episodes were included and 86 were performed by trainees (67.7%). Fistula division was the less commonly performed training task (25%), while mobilisation and anastomosis were performed by the supervised trainee in 90% and 85% of the cases. Safety and proficiency scores were significantly higher for senior trainees compared to junior trainees. CONCLUSIONS: Laparoscopic surgery for complex Crohn's disease can be safely performed in a supervised setting with acceptable operating time, postoperative length of hospital stay, and 30 day morbidity.

T cell clonal expansions in ileal Crohn's disease are associated with smoking behaviour and postoperative recurrence.

T cell clonal expansions are present in the inflamed mucosa of patients with Crohn's disease (CD) and may be implicated in postoperative recurrence after ileocolonic resection. METHODS: T cell receptor (TCR) analysis was performed in 57 patients included in a prospective multicentre cohort. Endoscopic recurrence was defined by a Rutgeerts score >i0. DNA and mRNA were extracted from biopsies collected from the surgical specimen and endoscopy, and analysed by high throughput sequencing and microarray, respectively. RESULTS: TCR repertoire in the mucosa of patients with CD displayed diverse clonal expansions. Active smokers at time of surgery had a significantly increased proportion of clonal expansions as compared with non-smokers (25.9%vs17.9%, p=0.02). The percentage of high frequency clones in the surgical specimen was significantly higher in patients with recurrence and correlated with postoperative endoscopic recurrence (area under the curve (AUC) 0.69, 95% CI 0.54 to 0.83). All patients with clonality above 26.8% (18/57) had an endoscopic recurrence. These patients with a high clonality were more frequently smokers than patients with a low clonality (61% vs 23%, p=0.005). The persistence of a similar TCR repertoire at postoperative endoscopy was associated with smoking and disease recurrence. Patients with high clonality showed increased expression of genes associated with CD8 T cells and reduced expression of inflammation-related genes. Expanded clones were found predominantly in the CD8 T cell compartment. CONCLUSION: Clonal T cell expansions are implicated in postoperative endoscopic recurrence. CD patients with increased proportion of clonal T cell expansions in the ileal mucosa represent a subgroup associated with smoking and where pathogenesis appears as T cell driven. TRIAL REGISTRATION NUMBER: NCT03458195.

Malabsorción de ácidos biliares en pacientes con diarrea crónica y enfermedad de Crohn / Bile acid malabsorption in patients with chronic diarrhea and Crohn's disease

Introducción y objetivo: la enfermedad de Crohn (EC) es una de las entidades que constituyen la enfermedad inflamatoria intestinal. Sus síntomas principales son la diarrea y el dolor abdominal. El objetivo de nuestro estudio es analizar la utilidad de 75SeHCAT en pacientes con EC que presentan diarrea crónica, con sospecha de malabsorción de ácidos biliares (MAB), y establecer si existe relación con las características de la enfermedad y la resección intestinal. Pacientes y método: se realizó un estudio observacional transversal a 39 pacientes con diagnóstico de EC y diarrea crónica. A todos se les realizó una gammagrafía con 75SeHCAT para el diagnóstico de MAB, tras descartar actividad de la enfermedad. Resultados: se analizó un total de 19 mujeres y 20 hombres con una mediana de edad de 44 años y con una clasificación de Montreal A2 L1 B1 en el mayor porcentaje. El 84,6% de los pacientes presentaban antecedentes de resección intestinal. El porcentaje global de MAB fue del 97,4% (el 100% de los pacientes intervenidos y el 83,3% de los pacientes no intervenidos), y esta fue grave en el 92,1% de los casos. Se estableció tratamiento con quelantes de ácidos biliares, que obtuvo una respuesta del 72,7%. Se analizó la existencia de una posible relación entre las características de la enfermedad y de la cirugía intestinal y la respuesta al tratamiento con quelantes con el grado de MAB que presentaban los pacientes (moderada o grave). El resultado obtenido fue no significativo. Conclusión: la MAB es una causa muy frecuente de diarrea en pacientes con EC en remisión endoscópica o radiológica, estando presente en todos los pacientes analizados con resección intestinal previa. La respuesta al tratamiento con quelantes de ácidos biliares estuvo presente en el 73% de los pacientes

Introduction and aim: Crohn's disease (CD) is a form of inflammatory bowel disease and is mainly characterized by diarrhea and abdominal pain. The aim of our study was to analyze the usefulness of performing a 75SeHCAT scan in CD patients with chronic diarrhea and suspected bile acid malabsorption (BAM). In addition, we aimed to determine whether there was a relationship with the clinical features of the disease and a previous bowel resection. Patients and methods: this was an observational cross-sectional study of 39 patients with a diagnosis of CD and chronic diarrhea. All cases underwent a 75SeHCAT scan for BAM diagnosis, after discarding disease activity. Results: the study cohort included 19 females and 20 males. The median age was 44 years and the majority of patients were A2 L1 B1 according to the Montreal classification; 84.6% of patients had undergone a previous bowel resection. BAM was present in 97.4% of patients (100% and 83.3% of patients with and without previous surgery, respectively), which was severe in 92.1% of cases. Treatment with bile acid sequestrants was initiated and a favorable response was obtained in 72.2% of patients. The relationship between BAM degree (moderate or severe), bowel surgery and the response to bile acid sequestrant treatment was also analyzed but not statistically significant. Conclusion: BAM is a frequent cause of diarrhea in CD patients in endoscopic or radiological remission. This condition was present in all patients with a history of a bowel resection. A response to bile acid sequestrants treatment was observed in 73% of patients

Faecal Calprotectin and Magnetic Resonance Enterography in Ileal Crohn's Disease: Correlations Between Disease Activity and Long-Term Follow-Up.

BACKGROUND AND AIMS: Magnetic resonance enterography [MRE] is the gold standard for assessing ileal inflammation in Crohn's disease [CD]. The aim of the present study was to correlate faecal calprotectin [FC] to MRE via a simple score in an exclusive ileal cohort with long-term follow-up for association with time to surgery or biologic therapy. METHODS: In total, 150 MRE studies with matched FC [±30 days] were identified from the Edinburgh FC Register [2008-12; n = 18138]. Scans were re-read blinded to clinical data, independently, by two expert gastrointestinal radiologists, to generate a simple MRE score [range 0-10] from assessment of the worst intestinal segment plus total disease extent. RESULTS: In total, 119 MRE scans were evaluated from 104 patients with ileal CD [L1 or L3 with panproctocolectomy]. Receiver operating characteristic analysis showed an area under the curve of 0.77 [0.67-0.87, p < 0.0001] for FC and MRE score >1, with an optimal cut-off of 145 µg/g for severe inflammation on MRE with 69.3% [57.6-79.5] sensitivity and 71.4% [53.7-85.4] specificity. Long-term follow-up over a median [interquartile range] of 2086 days [1786-2353] revealed FC ≥ 145 µg/g was associated with reduced biologic-free survival until 3 years following MRE, whereas MRE score [severe vs absent] was associated with reduced surgery- and biologic-free survival throughout follow-up. Backwards stepwise logistic regression revealed that length of ileal disease (odds ratio [OR] 3.8, 1.1-13.2, p = 0.034) and increased bowel wall thickness at MRE [OR 4.2, 1.6-10.7, p < 0.0001] or female sex [OR 5.2, 1.5-18.7, p = 0.011] increased the risk of biologic use or surgery, respectively. CONCLUSIONS: FC correlates well with MRE assessment of ileal CD with MRE parameters associated with long-term biologic- and surgery-free remission.

Previous colonic resection is a risk factor for surgical relapse in Crohn's disease.

BACKGROUND: Despite the improvement of medical therapies, nearly half of patients with Crohn's disease require surgery within 10 years after diagnosis. However, intestinal resection is not curative and recurrence may occur. AIMS: To evaluate post-surgical outcomes for patients with Crohn's disease in a large monocentric cohort, and to identify variables associated with clinical and surgical relapse. METHODS: Patients with Crohn's disease who had surgery for ileal and colonic Crohn's disease between 2004 and 2016 and on at least one-year follow-up following surgery were included. RESULTS: One hundred ninety-three patients were included in the study. Crohn's disease recurrence concerned 53% of patients after a median 56-month (6-158) follow-up and 29% of patients required a second surgical intervention. At logistic regression analysis, active smoking and young age at diagnosis were identified as independent risk factor for post-surgical relapse (p = 0.01), while colonic or ileocolonic resection was recognized as a risk factor for surgical Crohn's disease relapse (p = 0.003). CONCLUSIONS: Post-surgery recurrence is frequent for patients with Crohn's disease. Active smoking and young age at diagnosis are risk factors for Crohn's disease recurrence. As compared with patients undergoing small-bowel surgery, patients with colonic resection are proner to relapse requiring a second surgical intervention.

Ultrasound and computed tomography of sacculitis and appendicitis in a rabbit.

A 9-month-old neutered male rabbit was referred for lethargy, anorexia, and gastrointestinal stasis. Routine hematology, serum biochemistry, and diagnostic imaging were performed. Computed tomography revealed a wall thickening of the sacculus rotundus and appendix, which was further confirmed on abdominal ultrasound. Full thickness biopsies were collected with histopathology diagnosing a chronic multifocal heterophilic granulomatous sacculitis and appendicitis. The patient was treated medically and at 6 weeks follow-up, clinical signs and intestinal changes had completely regressed. Inflammation of the sacculus rotundus and appendix should be considered as a cause of gastrointestinal stasis in rabbits.

Chronic ileitis with transmural migration of ingested foreign body treated by laparoscopy.

BACKGROUND: Ingestion of foreign bodies such as fish bone or chicken bone is relatively common in adults; however, resultant transmural migration is extremely rare. METHODS: We present a case of a 79-year-old woman with chronic low-grade abdominal pain, worsening over the last 4 days. Computed tomography revealed segmental small bowel wall thickening with chronic inflammation suggestive of Crohn's ileitis and oral steroids were commenced; only later, ingestion of a foreign body was suspected. RESULTS AND CONCLUSION: At diagnostic laparoscopy, a linear foreign body resembling a wooden splinter was identified. It had partly migrated through the chronically inflamed bowel wall without causing perforation or abdominal contamination. It was removed laparoscopically without an enterotomy or bowel resection. Microscopy revealed non-viable bone, likely fish or chicken bone. The patient made an uneventful recovery and was discharged 3 days later. Herein we emphasise on the differential diagnosis and presentation of chronically ingested foreign bodies, as well as the feature of chronic ileitis with uncomplicated transmural migration of the ingested foreign body that was treated laparoscopically without an enterotomy.

Case report of a pair of siblings with cryptogenic multifocal ulcerating stenosing enteritis: A rare disease easily to be misdiagnosed as Crohn disease.

RATIONALE: The rare disease cryptogenic multifocal ulcerous stenosing enteritis (CMUSE) is characterized by multiple and recurring small intestinal ulcers with stenosis of unknown causes. In clinic, it is difficult to be differentiated from the inflammatory bowel disease, especially the Crohn disease. PATIENT CONCERNS: Here we report a pair of siblings who suffered from long-time anemia and abdominal pain and misdiagnosed with inflammatory bowel disease (IBD) for many years. DIAGNOSES: They were finally diagnosed with CMUSE with intestinal obstruction. INTERVENTIONS AND OUTCOMES: They both accepted surgical treatment and recovered well. No abdominal symptom appeared in the two-year follow-up. LESSONS: This report underscores that CMUSE patients may have a long course of suffering from anemia and abdominal pain, normal inflammatory markers and normal colon, and sometimes have a family history of CMUSE. Surgery of segmental bowel resection is a good way to solve intractable intestinal obstruction in CMUSE.