Journal of Crohn's and Colitis

Issue Contents list

2010-06-01

Neurologic manifestations in inflammatory bowel diseases: Current knowledge and novel insights

2009-12-04

Abstract: Background: Crohn's disease (CD) and ulcerative colitis (UC), widely known as inflammatory bowel diseases (IBD), are thought to result from an inappropriate activation of the mucosal immune system driven by intestinal bacterial flora.Methods: Although the extraintestinal manifestations of IBD are well documented, the association of IBD with neurologic and neuromuscular involvement is rare and often controversial, with sporadic and conflicting data on its prevalence and spectrum. In addition, a serious number of the latter manifestations may become life-threatening, playing a very important role in disease morbidity. To define the pattern of neurologic involvement in IBD, the most important manifestations in these patients have been reviewed, exploring also their clinical significance.Results: There is evidence that UC and CD can manifest both in the PNS and CNS. Thrombotic complications are common in IBD patients, but cerebral vascular involvement is rare.Conclusions: Neurologic manifestations in IBD patients are more common than previously estimated and may follow a different pattern of involvement in CD and UC. Small numbers of patients currently preclude a better characterization of the clinical spectrum and a better understanding of pathogenesis.

Active and passive smoking behaviour and cessation plans of patients with Crohn's disease and ulcerative colitis

2009-12-02

Abstract: Background: Smoking is a remarkable risk factor in inflammatory bowel disease (IBD), with negative effects on Crohn's disease (CD) and positive effects on ulcerative colitis (UC). This makes different changes in smoking behaviour after diagnosis between CD and UC likely. Changes in active smoking, cessation plans and passive smoking were studied in IBD patients.Methods: 820 IBD patients were sent a questionnaire on active and passive smoking, and cessation plans. A total of 675 (82%) patients (380 CD and 295 UC) responded.Results: More ever smoking UC patients stopped smoking before diagnosis than CD patients (63% vs 22%; p<0.001), resulting in 30% former smokers at diagnosis in UC and 13% in CD (p<0.001). The smoking cessation rates at and after diagnosis are equal between CD and UC. Half of the CD patients stopped smoking after diagnosis leading to less present smokers in CD than in a control population (26% (95% confidence interval: 21.1%–29.9%) vs 33%). For both CD (22% vs 35%; p=0.044) and UC (24% vs 53%; p=0.024) continuing smokers after diagnosis were less often higher educated than quitters. Cessation plans (89%), passive smoking in childhood and present passive smoking were not different between CD and UC patients.Conclusion: There are no differences in changes in smoking behaviour at and after diagnosis between CD and UC patients, suggesting a lack of knowledge in these patients about the link between their disease and smoking behaviour. However, CD patients seem less refractory to smoking cessation than the general population. Therefore it is worthwhile putting energy in helping CD patients stop smoking.

OCTN1 variant L503F is associated with familial and sporadic inflammatory bowel disease

2009-10-26

Abstract: Purpose: A two-allele haplotype of TC (OCTN1 rs1050152 and OCTN2 -207G→C) is associated with Crohn's disease (CD). The association has been replicated in different populations, but also failed in some studies. The present study is to replicate the association of OCTN1 rs1050152 and examine another variant rs272879 with familial and sporadic inflammatory bowel disease (IBD) in a cohort from central Pennsylvania, USA.Methods: The study samples (n=465) included 212 inflammatory bowel disease patients (CD=115, UC=97), including 103 familial (CD=55, UC=46) and 111 sporadic (CD=60, UC=51) IBD, 139 non-IBD family members from a familial IBD registry, and 114 unrelated healthy controls. A total of 12 OCTN1 variants within exonic sequences were examined. Two nonsynonymous SNPs, rs1050152 (L503F) and rs272879 (L395V) were genotyped by a PCR-based RFLP/cRFLP method and statistically analyzed. These samples with an additional 141 unrelated healthy samples were also genotyped for rs1050152 using the SNPlex™ Genotyping System.Results: The OCTN1 rs1050152 is associated with CD (OR=1.745, 95% CI=1.019–2.990, χ2=4.129, p=0.042) and with IBD (OR=1.68, 95% CI=1.052–2.676, χ2=4.732, p=0.030); while the variant rs272879 is not associated with IBD, CD or ulcerative colitis (UC). The distribution of the rs1050152 variant showed a high level of the T allele in male UC (OR=2.585, 95% CI=1.139–5.869, p=0.023) and IBD (OR=2.039, 95% CI=1.024–4.059, p=0.042) patients, and in female CD patients (OR=2.329, 95% CI=1.038–5.226, ρ value=0.039).Conclusion: The present results replicated the association of the OCTN1 rs1050152 (L503F) variant with CD and IBD overall. A weak gender-specific effect of rs1050152 (L503F) on male UC and female CD was observed.

A colorectal mosaic pattern might be an endoscopic feature of collagenous colitis

2009-10-26

Abstract: Background and aims: The endoscopic aspect of the colorectal mucosa in those patients with collagenous colitis is usually normal, or with non-specific changes. Until now it had never been related to a mucosal pattern of mosaic type. Our aim was to determine the diagnostic accuracy of the presence of mosaic pattern in the colorectal mucosa for collagenous colitis.Methods: Patients who had undergone a colonoscopy with random biopsies performed in the diagnostic evaluation of chronic diarrhea between 2004 and 2008 were studied. We defined patients with chronic diarrhea and mosaic mucosal pattern as “cases”, and patients with chronic diarrhea without mosaic pattern as “controls”. The odds ratio (OR) of finding a collagenous colitis in view of a mosaic pattern in colon was determined; as well as sensitivity and specificity; positive and negative likelihood ratios (LR+, LR−), considering this finding as a diagnostic instrument for collagenous colitis.Results: 252 patients who had undergone colonoscopý with biopsy due to chronic diarrhea were analyzed. In 6 patients, a mosaic pattern was identified in the colorectal mucosa. The histological diagnose of 36 of the 252 patients (14%) was microscopic colitis, 27 of which (11%) had collagenous colitis. The colonoscopy was found normal in 21 of these 27 patients; in 2 patients, congestion or petechiae was found in the rectum; and in 4 patients (15%), all women, a mosaic pattern was found in the rectosigmoid mucosa. The OR of this finding was 19.4 (CI95% 3.9–95.4) for collagenous colitis. It had a sensitivity of 14.8% (CI95% 6.8–20), a specificity of 99.1% (CI95% 98.2–99.7), LR+ of 16.6 (CI95% 3.7–76.4), and LR− of 0.86 (CI95% 0.80–0.95) for a collagenous colitis.Conclusion: The mosaic pattern in the colorectal mucosa of patients studied due to chronic diarrhea could be a distinguishing feature of collagenous colitis.

Does fecal calprotectin predict relapse in patients with Crohn's disease and ulcerative colitis?

2009-11-18

Abstract: Background and aims: An evaluation is made of the utility of fecal calprotectin in predicting relapse in patients with inflammatory bowel disease (IBD). The possible differences in its predictive capacity in Crohn's disease (CD) versus ulcerative colitis (UC), and the different phenotypes, are also examined.Methods: This is a prospective study with 135 patients diagnosed with IBD in clinical remission for at least 3months. The patients submitted a stool sample within 24hours after the baseline visit, for the measurement of fecal calprotectin. All patients were followed-up on for one year.Results: Sixty-six patients had CD and 69 UC. Thirty-nine (30%) suffered from relapse. The fecal calprotectin concentration was higher among the patients with relapse than in those that remained in remission: 444µg/g (95% CI 34–983) versus 112µg/g (95% CI 22–996); p<0.01. Patients with CD and calprotectin>200µg/g relapsed 4 times more often than those with lower marker concentrations. In UC, calprotectin>120µg/g was associated with a 6-fold increase in the probability of disease activity outbreak. The predictive value was similar in UC and CD with colon involvement and inflammatory pattern. In this group, calprotectin>120µg/g predicted relapse risk with a sensitivity of 80% and a specificity of 60%. Relapse predictive capacity was lower in patients with ileal disease.Conclusions: Fecal calprotectin may be a useful marker for predicting relapse in patients with IBD. Its predictive value is greater in UC and CD with colon involvement and inflammatory pattern, compared with ileal CD.

Clinical trial: Preliminary efficacy and safety study of a new Budesonide-MMX® 9mg extended-release tablets in patients with active left-sided ulcerative colitis

2009-10-26

Abstract: Background: Ulcerative colitis (UC) is a chronic inflammatory disease with relapses. Many patients need systemic corticosteroids to induce clinical remission.Aim: Efficacy and safety of Budesonide-MMX® 9mg tablets, a new oral, extended-release formulation, were evaluated in patients suffering from active, left-sided UC with colitis activity index (CAI) <14.Methods: 36 patients were treated once daily for 4weeks with Budesonide-MMX® 9mg tablets or placebo. In an additional 4-week period, all patients received Budesonide-MMX®. CAI, endoscopic index and histology were assessed after 4 and 8weeks. Primary end-point was remission, and/or CAI reduction by 50% after 4weeks. Morning cortisol was assayed after 4 and 8weeks, and a short ACTH-test was performed at week 8.Results: 32 patients were analysed. After 4weeks, 47.1% of the patients in the Budesonide-MMX® 9mg tablets group achieved the primary end-point vs. 33.3% of patients on placebo. In addition, 47.1% of budesonide patients and another 33.3% of placebo recipients improved without remission by 4weeks. The CAI reduction was significant with Budesonide (p<0.0001) tablets and not with placebo (p=0.1). Neither morning cortisol nor pituitary–adrenal axis was more frequently suppressed with Budesonide tablets than with placebo.Conclusions: Budesonide-MMX® 9mg tablets induced a fast and significant clinical improvement of active left-sided UC without suppression of adrenocortical functions and without important toxicity EudraCT number 2004-000896-33.

Anti-colitis and -adhesion effects of daikenchuto via endogenous adrenomedullin enhancement in Crohn's disease mouse model

2009-11-30

Abstract: Background and aims: Adrenomedullin (ADM) is a member of the calcitonin family of regulatory peptides, and is reported to have anti-inflammatory effects in animal models of Crohn's disease (CD). We investigated the therapeutic effects of daikenchuto (DKT), an extracted Japanese herbal medicine, on the regulation of endogenous ADM in the gastrointestinal tract in a CD mouse model.Methods: Colitis was induced in mice by intrarectal instillation of 2,4,6-trinitrobenzenesulfonic acid (TNBS); afterwards, DKT was given orally. Colonic damage was assessed on day 3 by macroscopic and microscopic observation, enzyme immunoassays of proinflammatory cytokines in the colonic mucosa, and serum amyloid A (SAA), a hepatic acute-phase protein. To determine the involvement of ADM, an ADM antagonist was instilled intrarectally before DKT administration. The effect of DKT on ADM production by intestinal epithelial cells was evaluated by enzyme immunoassay and real-time PCR.Results: DKT significantly attenuated mucosal damage and colonic inflammatory adhesions, and inhibited elevations of SAA in plasma and the proinflammatory cytokines TNFα and IFNγ in the colon. Small and large intestinal epithelial cells produced higher levels of ADM after DKT stimulation. A DKT-treated IEC-6 cell line also showed enhanced ADM production at protein and mRNA levels. Abolition of this effect by pretreatment with an ADM antagonist shows that DKT appears to exert its anti-colitis effect via up-regulation of endogenous ADM in the intestinal tract.Conclusion: DKT exerts beneficial effects in a CD mouse model through endogenous release and production of ADM. Endogenous ADM may be a therapeutic target for CD.

Impact of a patient-support program on mesalamine adherence in patients with ulcerative colitis — A prospective study

2009-11-18

Abstract: Background: Patient adherence to medications, particularly mesalamine, is reported to be low in patients with ulcerative colitis. We sought to determine whether a nurse-delivered patient-support program could improve medication adherence in these patients.Methods: Patients prescribed mesalamine for ulcerative colitis prospectively received either a 23week, nurse-delivered, patient support program (PSP) by phone, or standard care (SC). Medication adherence and quality of life were measured before and at 3 and 6months after the program started.Results: Eighty-one patients completed the study; 60 who received standard care, and 21 who received the PSP. Patients were in remission (mean SCAI score 3) at enrollment. Mean % of prescribed mesalamine refilled was 71% and 74% in the SC and PSP groups at 3months (p=0.7), and 73% and 84% at 6months (p=0.4). The proportion of adherent patients at 3months (39% vs 44%, p=0.7) and 6months (50% vs 67%, p=0.3) were similar between the SC and PSP groups. There was no association between use of the PSP and adherence at 3 (OR 1.2, 95% CI 0.4 to 3.8) or 6months (OR 2, 95% CI 0.6 to 7). The change from baseline in SIBDQ scores were similar between SC and PSP groups at 3months (+0.3 vs +0.2, p=0.8), and 6months (+0.6 vs +0.2, p=0.2).Conclusions: This nurse-delivered patient-support program did not significantly improve medication adherence or quality-of-life beyond standard care at short and medium-term time-points. Simply discussing and measuring adherence improved mesalamine adherence in both groups in this study.

Pregnancy and IBD treatment: This challenging interplay from a patients' perspective

R.E. Mountifield, R. Prosser, P. Bampton, K. Muller, J.M. Andrews — 2009-12-07

Abstract: Introduction: Current data suggest that exacerbations of Inflammatory Bowel Disease (IBD) during pregnancy worsen perinatal outcomes. However, patients' perceptions regarding the interaction between pregnancy and IBD management are unexplored.Aims: To (1) obtain pregnancy outcome data from local female IBD patients, and (2) to gain insight into patients' understanding of the interaction between IBD and pregnancy, and how this affects medication-taking behaviour.Methods: Female IBD subjects aged 18–50years were surveyed by questionnaire. This large retrospective study sought patient who reported pregnancy outcomes and examined the relationship between major adverse outcomes, IBD activity and treatment. Subjective data regarding patients' perceptions about IBD management and pregnancy were sought.Results: 219 females were surveyed, 143 completing a questionnaire (68.1%). 342 pregnancies occurred, 298 of which outcome data were available. Overall IBD women reported adverse pregnancy outcome rates comparable to the local population. Major adverse outcomes were more frequent in the subgroup with severe disease during pregnancy (5/14 (35.7%)) than those with inactive disease (14/284 (4.9%)), (OR 6.8 (95% CI 1.7–26.3), p=0.006). Adjusting for disease severity, neither corticosteroid, azathioprine nor 5ASA affected pregnancy outcome. Most female patients (84%) reported (unwarranted) concerns about the effect of IBD medications on pregnancy, free text responses indicating that this was of greater concern than any effect of IBD exacerbation.Conclusions: Unwarranted fear of adverse medication effect on pregnancy is highly prevalent in women with IBD, yet awareness of the harmful effect of IBD exacerbation during pregnancy is poor. This information gap between patients and their gastroenterologists warrants attention.

Conception outcomes and opinions about pregnancy for men with inflammatory bowel disease

2009-11-30

Abstract: Background and aims: Patients with inflammatory bowel disease (IBD) who want to have children are anxious to receive medical treatment. The consensus regarding pregnancy has not been surveyed for male IBD patients. The present study was investigated opinions among male IBD patients about pregnancy, conception and neonatal outcomes for partners.Methods: Subjects comprised 364 of 386 patients enrolled (94.3%). Subjects received a questionnaire regarding their opinions and thoughts about pregnancy. The course of partner's conceptions and presence of neonatal malformations was also surveyed.Results: The rate of live births for partners of male IBD patients was 91.6% (219/239). Most patients with CD (29/33; 88%) had their children after surgery had been performed. The rate of expressing hopes to have a child tended to be higher for patients with UC (93/128; 73%) than for patients with CD (61/97; 63%; p=0.21). Furthermore, the rate of hesitation was significantly higher in CD patients (34/107; 32%) than in UC patients (38/188; 20%; p=0.03).Patients considered that safety of medication (51%) and maintenance of remission (41%) was more important than receiving no treatment for IBD (19%) when planning to conceive. Mesalamine and infliximab were more favorable at conception than sulfasalazine and immunomodulators.Conclusions: This is the first report to survey the thinking of male IBD patients regarding pregnancy. Most male IBD patients considered “maintaining remission” as important at conception. Our study provides important information for IBD patients and for the treating physician when planning to conceive.

Colorectal carcinoma and preceding fistula in Crohn's disease

Anna Sobala, Friedrich Herbst, Gottfried Novacek, Harald Vogelsang — 2009-11-24

Abstract: Background: An increased risk for intestinal carcinoma is known in Crohn's disease, but there are also several reports on patients with perianal fistula and later carcinoma at this location — so to call fistula associated carcinoma.Methods: We retrospectively investigated 591 patients with CD who underwent abdominal surgery during the last 10years (1997–2006) and found seven patients (5 male, 2 female, median age: 53years (range 37–74)) with colorectal cancer (Dukes A–C), four of them (57%) with fistula associated CRC. These seven patients with CRC were matched 1:3 to randomly selected Crohn's patients based on age. The medical records of these 21 patients (11 with perianal fistula (52%)) were evaluated with respect to duration and pattern of intestinal involvement of CD, fistula history, intestinal surgery, perianal surgery, prior immunosuppressive and 5-ASA derivative intake.Results: Colorectal cancer was significantly (p=0.048) associated with longstanding anorectal fistula (median=11years (range 0–28years)) in the CRC group compared to the matched Crohn's patients (median=1year (range 0–6years)). Earlier colonic surgery seemed to protect from later malignancy (p=0.036). No significant symptoms preceded rectal carcinoma, except for new blood drainage from fistula in 2 patients. Two patients underwent ileocolonoscopy within 1year before the diagnosis of malignancy and 2 patients underwent MRI of the pelvic region within 4months.Conclusion: Colorectal carcinoma is frequently associated with the presence of longstanding anorectal fistula.

Prevalence and clinical impact of endoscopic pseudomembranes in patients with inflammatory bowel disease and Clostridium difficile infection

2009-11-24

Abstract: Background and aim: Limited data suggests that pseudomembranes are uncommon in patients with inflammatory bowel disease (IBD) and C. difficile associated disease (CDAD), but the reason for this is unknown.We aimed to evaluate the rate of pseudomembranes in this population, identify predictive factors for pseudomembranes' presence and assess its clinical impact.Methods: This was a sub-study of a retrospective European Crohn's & Colitis Organization (ECCO) multi-center study on the outcome of hospitalized IBD patients with C. difficile. The present study included only patients who underwent lower endoscopy during hospitalization, and compared demographic and clinical parameters in the group of patients with discernable pseudomembranes versus those without.Results: Out of 155 patients in the original cohort, 93 patients underwent lower endoscopy and constituted the study population. Endoscopic pseudomembranes were found in 12 (13%) of these patients. Patients with pseudomembranes presented more commonly with fever (p=0.02) compared to patients without pseudomembranes. No difference between the two groups was found with respect to the use of immunosuppressant drugs, background demographics or disease characteristics. Neither was there a difference between the group with or without pseudomembranes in the frequency of severe adverse clinical outcome or in the duration of hospitalization. On multi-variate analysis the presence of fever remained independently associated with the finding of pseudomembranes (OR 6, 95% CI 1.2–32, p=0.03).Conclusions: This study documents that hospitalized IBD patients with CDAD have low rate of endoscopic pseudomembranes, which is not accounted for by the use of immunosuppressant drugs. IBD patients with CDAD and discernable pseudomembranes more commonly present with fever, but their clinical outcome is similar to patients without pseudomembranes.

Primary cytomegalovirus infectious colitis complicating Crohn's disease successfully treated with oral valganciclovir

2009-12-14

Abstract: Most cases of cytomegalovirus (CMV) colitis that develop in patients with inflammatory bowel disease (IBD) are caused by reactivation of a latent virus. Primary CMV infections are rare in adult patients. Treatment with immunosuppressive agents increases the infection risk in patients with IBD. We present a 26year old lady with primary CMV colitis, superimposed on underlying Crohn's colitis. The diagnosis was confirmed by a viral-like prodrome, a positive CMV IgM titer, presence of low avidity IgG antibodies to CMV, high CMV DNA titers in the plasma, and immunohistological detection of CMV positive cells in her colonic mucosa. The patient responded to initial treatment with intravenous ganciclovir with a fall in plasma levels of CMV DNA, treatment was completed with oral valganciclovir until plasma CMV DNA levels became undetectable.

Fournier's gangrene complicating ulcerative pancolitis

2009-12-21

Abstract: Fournier gangrene is a very rare and a rapidly progressing, polymicrobial necrotizing faciitis or myonecrosis of the perineal, perianal and genital regions, with a high mortality rate. Infection is associated with superficial traum, urological and colorectal diseases and operations. The most commonly found bacteria are Escherichia coli followed by Bacteroides and streptococcal species. Diabetes mellitus, alcoholism, and immunosuppression are perpetuating co-factors.Fournier's gangrene complicating inflammatory bowel disease has been reported in three patients so far, two with Crohn's disease.A 78-year-old man diagnosed with ulcerative pancolitis was referred for fever, and painful perianal and scrotal swelling after perianal surgery for a horseshoe-type perianal abscess. Since bowel disease diagnosis, patient was on mesalazine and achieved long-term remission. Perianal abscess occurred suddenly one week before perianal surgery without any evidence of pre-existing fistula or other abnormalities. Physical examination showed extensive edema and crepitus of perineum and genitalia and patient had symptoms of significant toxicity.The diagnosis of Fournier's gangrene was made and patient underwent emergency surgery with extensive surgical debridement of the scrotal and perianal area and Hartman procedure with a diverting colostomy. In addition, patient started on therapy with mesalazine 3gr, methylprednisolone 16mg, parenteral nutrition and broad spectrum of antibiotics. Two days after the first operation the patient needed a second operation for perianal debridement. On the fourth day, blood cultures showed E. coli. Patient had an uneventful recovery and was discharged after 34days of hospitalization. On follow up, disease review is scheduled and colostomy closure is planned.

The association between Crohn's disease and desmoid tumors: A novel case and review of the literature

Anton W. Bungay, Andrew J. Smith, Eugene Hsieh, Fred G. Saibil — 2010-04-26

Abstract: Desmoids are rare tumors resulting from the proliferation of fibroblasts. They occur in association with familial adenomatous polyposis (FAP), but they may also occur in the post-traumatic peri-partum or post-abdominal surgery setting, and a few present spontaneously. Presenting features of desmoids are protean and largely relate to the anatomical area of involvement.We describe a 50year old male not known to have Crohn's disease and without FAP who presented with multiple desmoids. Investigation of post-operative diarrhea confirmed a diagnosis of Crohn's disease. This is the first report of a male patient, who had never undergone prior abdominal surgery, presenting with Crohn's disease and abdominal desmoid tumors. The reasons why Crohn's disease and desmoids may be associated are explored, focusing particularly on alternations in the fibrogenic cytokine TGF-β now known to be involved in the pathogenesis of both diseases.

Methotrexate-induced pneumonitis in a patient with Crohn's disease

Giovanna Margagnoni, Valeria Papi, Annalisa Aratari, Luca Triolo, Claudio Papi — 2009-12-18

Abstract: Pulmonary toxicity is a well recognised but infrequent adverse event of treatment with methotrexate. The vast majority of cases have occurred in patients with rheumatoid arthritis; here we present the case of a 44-year old woman with ileo-colonic Crohn's disease who developed methotrexate pneumonitis. The patient had a 10year history of Crohn's disease and, in the last 18months, she was treated with oral methotrexate because of steroid-dependency and intolerance to thiopurines. She was admitted to the hospital because of acute dyspnoea, non-productive cough and fever. High-resolution CT scan showed diffuse bilateral areas of ground-glass opacity, and pulmonary function tests disclosed a mild obstructive pattern with a decrease in carbon monoxide diffusing capacity. Blood cultures for pathogenic bacteria or fungi were negative as well as serologic tests against major pneumotropic agents. Methotrexate-induced lung injury was considered: the drug was discontinued and the patient received a steroid course with rapid symptomatic improvement. After 4weeks pulmonary function tests and high-resolution chest CT scan were normal. To our knowledge this is the second reported case of methotrexate-induced pneumonitis occurring in a patient with Crohn's disease. A definite diagnosis has been made not invasively according to clinical, laboratory and radiological criteria and excluding any infectious aetiology of the pulmonary findings.

Necrotizing external otitis on a Crohn's disease patient treated with infliximab

Gabriela Duque, Francisco Portela, Maria Carmo Migueis — 2009-11-18

This letter is about a 27years old female with the diagnosis of Crohn's disease since July 2004, involving the duodenum, ileum and right colon. She was first treated with Mesalamine (1g 3 id for 20d, suspended because of the absence of efficacy), Prednisolone (four episode treatment, tapered), Azathioprine (since July 2004, up to 2.5mg/Kg/d).

Crohn's disease presenting with multiple intestinal "perforation"

Seyfettin Köklü, Erdem Koçak, Serap Erel, Soykan Dinç — 2009-12-02

Crohn's disease is a disorder that is characterized by transmural inflammation and may involve any part of the gastrointestinal tract. Obstructive clinical presentations can be seen during the course of Crohn's disease. Free perforation of the intestine in Crohn's disease is an uncommon occurrence, with incidence of 1% to 3% in Western countries. Multiple intestinal perforations during the course of Crohn's disease have been reported very rarely. Herein, we present a patient, who was diagnosed with Crohn's disease after presenting with multiple intestinal perforations.

An unusual case of anorectal carcinoma in a patient with Crohn's disease

Piero Narilli, Quirino Lai, Donatella Meo — 2010-01-06

Ulcerative colitis (UC) and Crohn's disease (CD) increase the risk of tumoral evolution, with approximately 1% of patients who develop a colorectal cancer during their life. However, whereas there are several studies that analyzed the role of UC in tumoral transformation, available data on CD are still limited. The evaluation of the risk of cancer in CD presents several methodological problems, due to the heterogeneous nature of the disease (“jump lesions”, partial intestinal resections). We report an unusual case of anorectal carcinoma developed in a patient with CD. In February 2008, a 63-year-old Caucasian man with a 40-year history of CD was admitted to our center with a deterioration of anorectal fistulous disease. His surgical history included an appendectomy at the age of 18 and two ileocolic resections for subocclusive disease due to CD at the age of 23 and at the age of 53. The colonoscopy showed a stenosis of the anal canal, extending for about 5cm, which involved the distal rectum: at histological examination, this lesion resulted in a high-grade dysplasia. Magnetic resonance scans confirmed these findings, also reporting an inhomogeneity of the sphincter complex, associated with multiple solutions of continuity in the wall of bowel and numerous fistulous tracts (). The patient underwent surgery: an abdomino-peritoneal amputation according to Miles was performed. A 32cm-segment of left colon was removed, comprehending also a 4.5cm-anal tract, the mesorectal tissue, the perianal coetaneous tissue and various entero-cutaneous fistulas (). Post-operative course was regular and the patient was discharged in good condition in post-operative 12th day. Histological examination showed a lesion with the characteristics of the mucinous adenocarcinoma infiltrating the perianal adipose tissue and the anal cutaneous superficial dermis (pT2, N0, Mx, and G3). Currently, the patient is alive and is receiving chemotherapy. The role of CD in the development of colorectal tumors is unclear: however, several studies have shown a surprising equivalence between CD and UC in the risk of cancer, when compared to similar extension of colonic disease. In CD patients, the risk of tumoral transformation is related to the combined effects of chronic inflammation and individual genetic factors. However, development of carcinoma on anorectal fistulous disease due to CD is an uncommon finding. The symptoms of chronic inflammation may often occult the presence of malignancies: the discovery of high-grade dysplasia requires aggressive surgery, due to the presence of 20% of preoperatively undetected cancers. The role of CD in determining the development of colorectal carcinoma is currently underestimated. However, the development of an anorectal carcinoma due to CD is rare: its evaluation with multidisciplinary strategies is mandatory, obtaining early diagnosis and an immediate surgical approach.

Instruction for Authors

2010-06-01