[Intraperitoneal cystic lymphangioma and Crohn's disease: an exceptional association]. / Kystique intra-péritonéal et maladie de Crohn: à propos d'une association exceptionnelle.

Cystic lymphangioma is a rare benign malformative tumor of the lymphatic vessels which may occur in various locations. Intra-abdominal cystic lymphangioma is less frequent than cervicoaxillary cystic lymphangioma. Clinical presentation is polymorphic. Diagnosis is based on imaging data but it requires histological confirmation. Surgery is the gold standard treatment. We here report a rare case of acquired intraperitoneal cystic lymphangioma secondary to subtotal colectomy in a female patient with severe evolutive Crohn's disease treated with anti-TNF alpha. The patient presented with irreducible right painful paramedian mass with no impulse on coughing, suggesting the diagnosis of strangulated eventration within a surgical scar from midline laparotomy. She underwent emergency surgery. Surgical exploration showed multi-cystic intraperitoneal mass protrunding through the right paramedian eventration. Anatomo-pathological examination helped to confirm the diagnosis of cystic lymphangioma. Postoperatively, the mass was punctured twice to evacuate the fluid, because of incomplete surgical resection. This is the first reported case of cystic lymphangioma in a patient under anti-TNF alpha. It could be caused by disruption of the immune system and more specifically of the lymphocyte population. This association has not hitherto been established and experimental studies are necessary to accept or refuse this hypothesis.

Haemostatic balance in cirrhosis.

Despite the prolongation of coagulation tests, recent studies reported an increased frequency of thromboembolic events in patients with cirrhosis. The aim of this study was to evaluate the haemostatic balance in cirrhotic patients through assessing the variation of pro- and anticoagulant factors and evaluating the in-vitro thrombin generation in patients with cirrhosis and in healthy patients. Fifty-one cirrhotic patients with or without thromboembolic events and 50 controls matched by age and sex were enrolled. Procoagulant (factors VII, II, V, VIII, and XII) and inhibitor (protein C, protein S and antithrombin) factor activities were determined. Thrombin generation was measured as endogenous thrombin potential (ETP). Haemostatic balance was assessed by means of both procoagulant to inhibitor coagulation factor ratios and ETP with to without protein C activation ratios. There were 24 males and 27 females. The mean age was 57.8 years [16-91 years]. Pro and anticoagulant factors were significantly lower in patients than in controls (P < 0.001) except for factor VIII and protein S. In fact factor VIII level was significantly higher in patients than in controls and protein S levels were not significantly different between patients and controls. Almost all the pro to anticoagulant factor ratios were higher in cirrhotics than in controls, especially the factor VIII to protein C ratios which increased significantly from Child Pugh A to C (P < 0.001), the ratio of ETP with to without protein C activator was higher in patients than in controls, but did not reach a significant level (0.8 vs. 0.52) There was no statistically significant difference between Child classes. When comparing patients with history of thrombosis (n = 7) to those matched by age and sex and without history of thrombosis (n = 14), the ratios were not statistically different between the two groups. Haemostatic changes in cirrhosis tend to rebalance the haemostatic system. This state often results in a hypercoagulable state attested by increased pro- to anticoagulant factor ratios and a normal thrombin generation.

Surgical management of primary crohn's disease. descriptive study about 226 patients.

BACKGROUND: The surgery is required in more than 80% of patients with Crohn's disease (CD). Studies before confirm the specific genetic variation of CD in the Tunisian population compared with the others ethnic groups. AIM: This article aims to study the epidemiological, anatomical and therapeutic principles of surgical forms of CD in a cohort of Tunisian patients. METHODS: We report a retrospective study from January 1998 to September 2010 that studied 226 patients originated only from Tunisia (in North Africa), operated on for MC. We had been interested in epidemiological, anatomical, clinical, therapeutic, topographic progression of the disease, the procedure and the postoperative follow-up. RESULTS: The median age was 33 years. The average time between the onset of the disease and the surgical procedure was 31 months. The diagnosis of CD was established preoperatively in 213 patients (94%). The diagnosis was made intraoperatively because of an acute complication in 5 cases (2.2%) and postoperatively in 8 cases (3.5%). The most common location was the ileocecal junction in 184 cases (81.4%). Achieving the most common was the mixed form (stricture and fistula) in 123 cases (54.4%). Operative mortality was 0.04% (n = 1). Specific morbidity was 8.4% (n = 19). In long term, a surgical recurrence was noted in 17 patients (7.5%). In multivariate analysis the independent risk factors for surgical recurrence were: smooking (p = 0.012, ORs = 3.57) and post-operative medical treatment (p = 0.05, ORs = 2.6). CONCLUSIONS: Achieving stenosing and fistulizing the ileocecal junction is the most frequent surgical form in Crohn's disease. Our series is unique for a lower rate of the postoperative recurrence (7.5%).

Radiation exposure in Crohn's disease patients.

Introudction Crohn's disease (CD) is a lifelong condition. Multiple imaging investigations are often performed during follow-up. This could cause overexposure to radiation. The aim of our study was to determine mean radiation dose in patients  with  at least a 5-year course of CD and to determine possible risk factors associated with exposure to high doses of radiation. Methods We conducted a retrospective study including patients whose CD was diagnosed between 1998 and 2005. Epidemiologic features of patients, characteristics of the disease,  types of imaging investigations that were performed during follow-up and cumulative radiation effective dose were determined. Risk factors associated with exposure to high doses of radiation were then determined. Results One hundred sixty seven patients were included.  There were 92 males (55.1%) and 75 females (44.9%) with mean age at dianosis of 31.4±12.3years. Global radiation dose was 18.8±18.9 mSv. Twenty seven patients (16,2%) were exposed to more than  35 mSv and 4 patients (2.4%) had an exposure  of more than  75 mSv. Use of Infliximab, age at disease onset ≤ 24 years old and number of flares ≥ 8  were independent risk factors of radiation exposure more than 35 mSv with adjusted Odds ratios (OR) : 2.5 [2.1- 5.3]; 1.6 [1.2- 4.7] and 3.2 [2.1- 7.8] respectively. Similarly,  use of Infliximab and number of flares ≥ 8  were independent risk factors of radiation exposure more than 75 mSv with adjusted OR : 4.3 [2.8-9.5] and 7 [3.2-11.2] respectively. Conclusion Radiation risk seems to be increased with severe course of  CD. Both referring physicians and radiologists have the responsibility to minimise radiation exposure. Entero-magnetic resonance imaging (Entero-MRI)  may reduce this risk.

Preventive and curative effect of Pistacia lentiscus oil in experimental colitis.

AIM: to investigate the anti-inflammatory effect of the Pistacia lentiscus oil in experimental colitis model. MATERIALS AND METHODS: Colitis was induced in male rats by instillation of 2,4,6-trinitrobenzenesulfonic acid (TNBS) in all groups. The experimental groups consisted of: 5 rats received Lentisc oil 2months before colitis induction (preventive group), 5 rats received the oil on the day of colitis induction (curative group) and 5 control rats. Lentisc oil was extracted from the ripe fruit of the plant by the cold press method and was analyzed by spectro-chromatography. Lentisc oil has been inserted with a standard diet at the dose of 30mg oil/100g of food/rat. RESULTS: The lentisc oil sample is composed mainly by Oleic acid (47.96%), Palmitic acid (27.94%) and Linoleic acid (20.22%).There was a significant difference between control rats and treated rats with lentisc oil concerned body mass (p=0.009), bleeding index (p=0.005 and p=0.018) and diarrhea (p=0.012). Histological examination revealed a clear difference between the control and preventive groups with disappearance of erosion, decreased of cryptitis, irregular crypts and crypt loss in the preventive group. Curative group showed a significant decrease of ulceration, hyperplasia, cryptitis, irregular crypts and crypt loss compared to the control group. There was an attenuation of inflammation in the preventive group compared to the curative group without statistically significant. CONCLUSION: Lentisc oil administration could provide a protective effect on intestinal inflammation in colitis rats induced by TNBS mainly when it is administered at a young age in preventive mode. This beneficial effect would involve a modification of arachidonic acid metabolism.

Changes of Crohn's disease phenotype over time.

Background - Crohn's disease is a clinically heterogeneous condition. Our aim was to identify the phenotype evolution of Crohn's disease over time according to the Montreal Classification and to precise predictive factors of the need for immunosuppressant treatment or surgery. Methods - We included Crohn's disease patients who were followed up for at least 5 years. We excluded patients who were lost to follow up before five. Patients were classified according to the Montreal classification for phenotype at diagnosis and five years later. The evolution of phenotype over time and the need for surgery, immunosuppressive or immunomodulatory drugs were evaluated. Results - One hundred twenty consecutive patients were recruited: 70 males and 50 females. At diagnosis, 68% of patients belong to A2 as determined by the Montreal classification. Disease was most often localized in the colon. The disease location in Crohn's disease remains relatively stable over time, with 93.4% of patients showing no change in disease location. Crohn's disease phenotype changed during follow up, with an increase in stricturing and penetrating phenotypes from 6% to 11% after 5 years. The only predictive factor of phenotype change was the small bowel involvement (OR=3.7 [1.2-7.6]). During follow-up, 82% of patients have presented a severe disease as attested by the use of immunosuppressive drugs or surgery. The factors associated with the disease severity were: small bowel involvement (L1), the stricturing (B2) and penetrating (B3) phenotypes and perineal lesions (OR=17.3 [8.4-19.7]; 12 [7.6-17.2]; 3[1.7-8.3] and 2.8 [2.2-5.1] respectively), without association with age, sex or smoking habits. Conclusion - Crohn's disease evolves over time: inflammatory diseases progress to more aggressive stricturing and penetrating phenotypes. The ileal location, the stricturing and penetrating forms and perineal lesions were predictive of surgery and immunosuppressant or immunomodulatory treatment.

[Nocturnal gastroesophageal reflux. About a pH metric series]. / Le reflux gastro-oesophagien acide nocturne pathologique. A propos d'une série pHmétrique.

BACKGROUND: Nocturnal gastroesophageal reflux has been shown to be associated with the more severe forms of gastroesophageal reflux disease (GERD), particularly with extraesophageal manifestations as well as complications of mucosal damage. AIM: To determine the frequency of nocturnal gastro esophageal reflux disease on 24-hour esophageal pH monitoring in patients with digestives or extra-digestives symptoms and to evaluate the clinical and pHmetric characteristics of nocturnal reflux in these patients. METHODS: We conducted a retrospective study based on results of 24- hour esophageal pH monitoring during a 11-year period in patients with or without digestive symptoms of gastroesophageal reflux disease. The nocturnal gastroesophgeal reflux was defined. RESULTS: We studied 696 patients (299 men, 397 women; mean age: 34.05 years). Gastroesophageal reflux was found in 350 patients (50%). Nocturnal reflux was observed in 240 patients (34.3%), mostly in association with pathological reflux in the total period (223 cases). Compared to the diurnal period, the nocturnal period was characterized by fewer number of reflux episodes (21.9±27.4 vs 67.4±5.,1 ; p<0.0001), more longer duration of reflux episodes (24.4±37.9 minutes vs 13.9± 17.5 minutes ; p<0.001), and a lower symptomatic correlation (26% vs 45% ; p=0.0005). CONCLUSION: Nocturnal reflux is associated with overall reflux on the 24 hour examination. Nocturnal period is characterized by longer reflux episodes, less number of reflux episodes and less symptomatic correlation.

[Surgical management of entero-vesical fistulas in Crohn's disease]. / Traitement chirurgical des fistules entéro-vésicales secondaires à la maladie de Crohn.

BACKGROUND: Crohn's disease (CD) is a chronic inflammatory disease of the intestine that can cause an attack by contiguity of the urinary tract. Although the shape is common and fistulizing 35% of all patients with CD, entero-urinary fistulas are rare and only seen in 2-8% of patients. aim: To report the frequency of occurrence of this complication among the group of surgical forms of CD. Describe the different pathophysiological mechanisms of occurrence of entero-vesical fistula (EVF) during the CD. methods: We report, retrospectively, seven observations of EVF complicating MC made during the period from 01/01/1998 until 31/12/2010. results: The mean age of patients was 30 years. There were 3 men and 4 women. All patients had clinical signs and radiological EVF. In six patients, CD was ileo-caecal and the ileo-vesical fistula was between the last loop and the bladder. In one patient, the CD was located only in colon, and the fistula was between sigmoid colon and bladder. Level of the bladder, it was a false EVF in five patients and a true EVF in two patients. In these last two, the fistula of 2 mm, was on the top of the bladder. Treatment consisted in all cases by a disconnect between the digestive tract and bladder, resection with restoration of digestive continuity, and if the case of true EVF, a freshening the edges of the fistula with suture of the bladder's wall and drainage. The postoperative course was uneventful in six patients and marked by an outbreak intraperitoneal abcess in one patient who had evolved under medical treatment. After a mean of eleven months, no recurrence surgery was noted. CONCLUSION: Despite advanced treatment in the context of CD, the indication in EVF is a surgical treatment. Surgery helps fight against the consequences of septic urinary tract, but also to launder bowel disease and reduce the risk of recurrence in the short term.

Stem cell transplantation as rescue therapy for refractory Crohn's disease: a sytematic review.

BACKGROUND: Crohn's disease is a chronic relapsing- remitting affection. It has a strong immunologic component which represent the target of standard therapies including immunosppressants and biological therapies. However, many patients remain refracory or intolerant to these therapies. AIM: The aim of this review is to determine the effects of stem cell transplantation in patients with refractory Crohn's disease. METHODS: Systematic review of observational studies, clinical trials and case reports that focused on the effectiveness and safety of stem cell transplantation in patients with refractory Crohn's disease. RESULTS: Hematopoietic stem cell transplantation seems to be efficient in maintaining clinical and endoscopic remission in patients with Crohn's disease refractory or intolerant to current therapies. However, it has been associated to high morbidity and mortality due to chemotherapy. Mesenchymal stem cell transplantation could induce remission in patients with fistulising refractory Crohns disease with no severe side effects. Its impact on luminal Crohns disease is still controversial. CONCLUSION: Stem cell transplantation seems to hold promising in patients with refractory Crohn's disease. However, because of the high morbidity and mortality related to chemotherapy, hematopoietic stem cell transplantation should be used as last resort to control this disease. Effectiveness of mesenchymal stem cell transplantation in luminal Crohn's disease has yet to be proven.

[Dietary intake in Crohn's disease: results of a prospective comparative study]. / Enquête alimentaire au cours de la maladie de Crohn : résultats d'une étude prospective comparative.

BACKGROUND: Diet is thought to have an important role in the etiopathogenesis of Crohn's disease. In the other hand, Crohn's disease is frequently associated with nutritional deficiencies probably as result of disease activity and poor oral intake. AIMS: To investigate the dietary intake in patients with Crohn's disease in comparison with matched population controls and to assess the correlation between the results of the dietary enquete, nutritional status and disease activity. METHODS: We conducted a prospective case control study in patients with Crohn's disease and matched controls. All subjects were evaluated in respect of dietary intake (based on "NUTISTAR" logiciel) and nutritional status. RESULTS: We studied 23 patients and 23 controls. There was no statistical difference between patients and controls according to the proportion of carbohydrates, fat and protein intakes. Energy intakes were significantly lower in patients with Crohn's disease (1991 + 678 kcal/j) compared to controls (2537 + 345 kcal/j) (p=0.007) ; and in active disease (1353 + 308 kcal/j) compared to inactive disease patients (2481 + 415 kcal/j) (p<0.0001). In Crohn's disease patients, correlation study showed that energy intakes were correlated with CDAI (p<0.0001 ; r = - 0.74) and BMI (p=0.03 ; r = 0.45). CONCLUSION: In Crohn's disease, an inadequate dietary intake is correlated with nutritional status and disease activity.

[Epithelial cell in intestinal homeostasis and inflammatory bowel diseases]. / Rôle de la cellule épithéliale dans l'homéostasie intestinale et les maladies inflammatoires chroniques de l'intestin.

Crohn's disease (CD) and ulcerative colitis (UC) are the principal inflammatory bowel diseases (IBD) which physiopathology is currently poorly elucidated. During these diseases, the participation of the epithelial cell in the installation and the perpetuation of the intestinal inflammation is now clearly implicated. In fact, the intestinal epithelium located at the interface between the internal environment and the intestinal luminal, is key to the homeostatic regulation of the intestinal barrier. This barrier can schematically be regarded as being three barriers in one: a physical, chemical and immune barrier. The barrier function of epithelial cell can be altered by various mechanisms as occurs in IBD. The goal of this article is to review the literature on the role of the epithelial cell in intestinal homeostasis and its implication in the IBD.

Diagnosis and management of refractory celiac disease: a systematic review.

BACKGROUND: Refractory celiac disease is defined by persisting malabsorptive symptoms in spite of a strict gluten free diet for at least 6 to 12 months. Alternatives to gluten free diet seem to be still controversial. AIM: To describe the clinical and epidemiologic aspects of refractory celiac disease, and to identify therapeutic options in this condition. METHODS: Systematic review and critical analysis of observational studies, clinical trials and case reports that focused on diagnosis and management of refractory celiac disease. RESULTS: Refractory celiac disease can be classified as type 1 or type 2 according to the phenotype of intraepithelial lymphocytes. Great complications such as enteropathy-associated T-cell lymphoma may occur in a subgroup of these patients mainly in refractory celiac disease type 2. Curative therapies are still lacking. CONCLUSION: Refractory celiac disease remains a diagnosis of exclusion. Its prognosis remains still dismal by the absence yet of curative therapies. However, some new treatments seem to hold promise during few cohort-studies.