Clin J Gastroenterol DOI 10.1007/s12328-014-0458-6
CASE REPORT
Synchronous cytomegalovirus and Clostridium difficile infection of the pouch: a trigger for chronic pouchitis? Ioannis Papaconstantinou • Evanthia Zampeli • Dionysios Dellaportas • Charalambos Giannopoulos • Maria Sotiropoulou • George Polymeneas Giorgos Bamias • Spyros Michopoulos
•
Received: 29 October 2013 / Accepted: 22 January 2014 Ó Springer Japan 2014
Abstract Pouchitis occurs in up to one half of patients after restorative proctocolectomy with ileal pouch-anal anastomosis (IPAA). Cytomegalovirus (CMV) and Clostridium difficile are among the commonest secondary identifiable etiologies. A 17-year-old male with ulcerative colitis underwent IPAA due to refractory disease. Nine months later he experienced bloody diarrhea and fever. Laboratory testing and endoscopy confirmed pouch inflammation. Testing for C. difficile toxins A and B was
positive. Histology revealed affluent inclusion bodies and immunohistochemistry detected reactivity against CMV protein. Treatment with metronidazole and vancomycin offered partial improvement, whereas the addition of gancyclovir led to a successful recovery. One month after completion of treatment symptoms recurred. Repeat testing precluded an identifiable infectious cause and the diagnosis of idiopathic chronic pouchitis was established. The patient is currently on maintenance treatment with the probiotic compound VSL#3.
I. Papaconstantinou D. Dellaportas G. Polymeneas 2nd Department of Surgery, Aretaieion Hospital, University of Athens, 76 Vas. Sofias Avenue, 115 28 Athens, Greece e-mail: [email protected]
Keywords Ulcerative colitis Ileal-pouch anal anastomosis Pouchitis CMV Clostridium difficile
D. Dellaportas e-mail: [email protected]
Introduction
G. Polymeneas e-mail: [email protected]
Restorative proctocolectomy with ileal pouch-anal anastomosis (IPAA) is the surgical treatment of choice for the majority of patients with medically refractory ulcerative colitis (UC), UC-associated dysplasia, or familial adenomatous polyposis [1]. IPAA is related to a number of complications and mostly with the development of pouch inflammation which occurs almost exclusively in patients with UC. Pouchitis has a cumulative prevalence of up to 50 % [2]. Clinical presentation is variable and may occasionally result in diagnostic challenges. Pathogenesis of pouchitis is not clearly defined and most cases are classified as idiopathic. However, approximately 20–30 % of patients who present with pouchitis may have secondary identifiable etiologies which offer the opportunity of definitive and curative treatment [3]. Among the commonest causes are cytomegalovirus (CMV) and Clostridium difficile infection. Such secondary causes of pouchitis are usually suspected when patients do not
E. Zampeli (&) C. Giannopoulos M. Sotiropoulou S. Michopoulos Department of Gastroenterology, ‘Alexandra’ General Hospital, 80 Vas. Sofias Avenue, 115 28 Athens, Greece e-mail: [email protected] C. Giannopoulos e-mail: [email protected] M. Sotiropoulou e-mail: [email protected] S. Michopoulos e-mail: [email protected] G. Bamias Academic Department of Gastroenterology, First Department of Propaedeutic Internal Medicine, Laikon General Hospital, Ethnikon and Kapodistriakon University, 17 Aghiou Thoma, 115 27 Athens, Greece e-mail: [email protected]
123
Clin J Gastroenterol Fig. 1 Endoscopy showed patchy erythema, edema, granularity, friability and mucus exudate of the pouch mucosa
respond to the antibiotic regimens that constitute the standard of care for pouchitis. We present a case of synchronous CMV and C. difficile infection of the pouch in a 17-year-old male who subsequently developed chronic pouchitis.
Case report A 17-year-old male with refractory UC underwent IPAA in 2012. Nine months later, he presented with abdominal pain, bloody diarrhea and fever. There was no history of antibiotics or other drug administration before symptom initiation. Laboratory markers of inflammation were elevated (C-reactive protein and erythrocyte sedimentation rate). Endoscopy revealed pouchitis of moderate severity (Fig. 1). He was initially started on ciprofloxacin (500 mg b.i.d.) and mesalazine suppositories (500 mg b.i.d.) with only partial improvement. A fecal sample was tested positive for C. difficile toxin A and B. Additionally, on histological examination of endoscopically obtained biopsies from the pouch the presence of multiple inclusion bodies was noted. The presence of CMV was confirmed by positive immunostaining in immunohistochemistry (Fig. 2). Serology was positive for past CMV exposure [IgG(?), IgM(-)]. Of note, CMV infection had repeatedly been excluded before surgery. In addition, no CMV infection was detected in the resected colectomy specimen. Ciprofloxacin was discontinued and metronidazole commenced (500 mg t.i.d.). Due to persistence of symptoms and a new positive stool specimen for C. difficile toxin 3 days later, oral vancomycin was added at a dose of 125 mg q.i.d. As there was still only a partial response five days later, the patient was admitted and started on gancyclovir (10 mg/kg/day). The combination of vancomycin/ gancyclovir was maintained for 10 days. The patient gradually recovered and was discharged from the hospital after completion of 21 days on gancyclovir. He remained well until a month later when bloody diarrhea and urgency
123
Fig. 2 a H&E stain disclosed intense inflammatory infiltration. b Immunohistochemistry revealed CMV intranuclear inclusion bodies in multiple cells
ensued. Fecal specimens and histology were negative for C. difficile and CMV, respectively. A diagnosis of chronic idiopathic pouchitis was established based on the European Crohn’s and Colitis Organization guidelines [4]. He responded well to a course of metronidazole and is currently on maintenance treatment with VSL#3.
Clin J Gastroenterol
Discussion Pouchitis is defined as a nonspecific inflammation of the ileal pouch reservoir with a cumulative prevalence of up to 50 % in patients with UC undergoing IPAA [2]. According to certain reports, the risk of pouchitis is higher during the first two years following loop-ileostomy closure [5]. Extensive UC, extraintestinal manifestations (i.e., primary sclerosing cholangitis), being a non-smoker, perinuclear antineutrophil cytoplasmic antibody-positive serology and non-steroid anti-inflammatory drug (NSAID) use are possible risk factors for pouchitis (4). Ideally, the diagnosis of pouchitis is based on the triad of compatible symptoms (diarrhea, urgency, incontinence, nocturnal seepage, abdominal cramps, pelvic discomfort), endoscopic findings (erythema, mucous exudates, friability, bleeding, ulcers), and mandatory histological features (inflammatory infiltrate and crypt abscesses) [6]. The differential diagnosis encompasses cuffitis, Crohn’s disease, backwash ileitis, celiac sprue and ‘irritable pouch syndrome’. Pouchitis can be clinically classified as acute (duration of symptoms \4 weeks) or chronic ([4 weeks) [7]; on the basis of response to treatment with antibiotics it is also divided into antibiotic-responsive, antibiotic-dependent, and antibiotic-refractory pouchitis. In the vast majority of cases the etiology of pouchitis is not clearly defined and is considered idiopathic. However, when a specific cause is identified, secondary pouchitis is diagnosed; this occurs in 20–30 % of patients with chronic, antibiotic-refractory pouchitis. The most common etiological factors are infectious causes (C. difficile, CMV, and Candida), NSAIDs, ischemia, radiation injury, etc. [8]. Patients with secondary pouchitis may benefit from eradication of the triggering factors. In our patient, stool samples were twice positive for C. difficile toxins. C. difficile may colonize the pouch without causing inflammation; hence, eradication of the bacterium may have little impact on the natural history of pouchitis. Indeed, in patients with IPAA, the epithelium of the pouch undergoes morphological changes facilitating microbiota colonization. There are no current guidelines for the optimal management of C. difficile pouchitis due to lack of prospective trials. Standard therapy consists of metronidazole and/or vancomycin. Previous reports suggested that metronidazole may not suffice in cases of pouchitis since C. difficile infection may develop while patients are taking metronidazole. Therefore, in patients with C. difficile-associated pouchitis, rifaximin, tinidazole or vancomycin are preferred as first-line treatment. Our case was further complicated by the presence of active CMV colitis. CMV infection is a well-recognized cause of secondary pouchitis in IPAA patients [9]. CMVassociated pouchitis resembles idiopathic pouchitis,
although fever may be more common. In particular, there are no pathognomonic endoscopic features of CMV colitis [10]. Definite diagnosis of CMV pouchitis relies on the detection of the viral proteins by immunohistochemistry and/or detection of CMV-specific DNA in colonic biopsies by polymerase chain reaction (PCR). The most common practice is to base the diagnosis on histology and immunohistochemistry [11]. Histopathological characteristics include mononuclear cells with viral inclusion bodies, which are 2–4-fold larger than the surrounding normal cells. Eosinophilic intranuclear inclusions are often seen in these cytomegalic cells with a diameter of 8–10 mm. They are placed eccentrically and surrounded by a clear halo thus producing the characteristic ‘owl’s eye’ appearance. Immunostaining for CMV antigen using monoclonal antibodies is crucial for the diagnosis of CMV pouchitis, whilst serology is of limited value. Histological sections with multiple inclusion bodies have the highest probability for showing immunoreactivity against CMV proteins by immunohistochemistry. Detection of CMV DNA in the tissue is often determined as a complementary diagnostic tool although it has not been properly validated. The utility of serum assays (pp65 antigenemia or PCR for CMV DNA) for the diagnosis of CMV colitis are compromised by low negative predictive value. Treatment with gancyclovir is the recommended first-line treatment. To our knowledge, this is the first case of synchronous CMV and C. difficile infection of the pouch. As pouchitis developed 9 months after IPAA, the pre-existence of CMV in our patient is highly unlikely. Interestingly, he was not exposed to antibiotics or immunosuppressants over a long period of time. Nevertheless, it is not infrequent for C. difficile infection to develop in inflammatory bowel disease patients without prior antibiotic exposure. CMV is less often encountered in immunocompetent individuals. Due to skepticism regarding the actual role of CMV we did not initially offer antiviral treatment to the patient. However, the patient’s clinical condition with the presence of fever and his incomplete response after a week of treatment with metronidazole and vancomycin led to the addition of gancyclovir. Given that the patient was offered treatment for both agents we cannot rule out the possibility that one of the two was simply an innocent bystander rather than accountable for the patient’s symptoms [12]. Whilst the pathogenesis of pouchitis remains largely unknown, the role of luminal microflora is considered central [13]. The development of idiopathic chronic pouchitis a month after completion of combination treatment for CMV and C. difficile raises the possibility that the infectious agents triggered chronic pouchitis in the present case. Considering that follow-up fecal tests and histology for C. difficile and CMV were negative, treatment of the
123
Clin J Gastroenterol
acute episode was effective. However, chronic perpetuating inflammation of the pouch may have been induced. In conclusion, pouchitis is usually considered idiopathic. In one-third of cases specific causes are identified and should therefore always be sought before commencement of standard treatment. The exact role of infectious agents has not been elucidated, although as in the case we present, one can assume that they may trigger chronic inflammation.
4.
5.
6.
7.
Consent 8.
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal. Conflict of interest peting interests.
The authors declare that they have no com-
9.
10.
11.
References 1. Coffey JC, Winter DC, Neary P, Murphy A, Redmond HP, Kirwan WO. Quality of life after ileal pouch-anal anastomosis: an evaluation of diet and other factors using the Cleveland Global Quality of Life instrument. Dis Colon Rectum. 2002;45:30–8. 2. Stocchi L, Pemberton JH. Pouch and pouchitis. Gastroenterol Clin North Am. 2001;30:223–41. 3. Munoz-Juarez M, Pemberton JH, Sandborn WJ, Tremaine WJ, Dozois RR. Misdiagnosis of specific cytomegalovirus infection of
123
12.
13.
the ileoanal pouch as refractory idiopathic chronic pouchitis: report of two cases. Dis Colon Rectum. 1999;42:117–20. Van Assche G, Dignass A, Bokemeyer B, Danese S, Gionchetti P, Moser G, et al. Second European evidence-based consensus on the diagnosis and management of ulcerative colitis part 3: special situations. J Crohns Colitis. 2013;7(1):1–33. Fazio VW, Ziv Y, Church JM, Oakley JR, Lavery IC, Milsom JW, et al. Ileal pouch-anal anastomoses complications and function in 1005 patients. Ann Surg. 1995;222:120–7. Coffey JC, Rowan F, Burke J, Dochery NG, Kirwan WO, O’Connell PR. Pathogenesis of and unifying hypothesis for idiopathic pouchitis. Am J Gastroenterol. 2009;104:1013–23. Sandborn WJ. Pouchitis following ileal pouch-anal anastomosis: definition, pathogenesis, and treatment. Gastroenterology. 1994;107:1856–60. Shen B, Remzi FH, Lavery IC, Lashner BA, Fazio VW. A proposed classification of ileal pouch disorders and associated complications after restorative proctocolectomy. Clin Gastroenterol Hepatol. 2008;6:145–158 (quiz 124). Moonka D, Furth EE, MacDermott RP, Lichtenstein GR. Pouchitis associated with primary cytomegalovirus infection. Am J Gastroenterol. 1998;93:264–6. Navaneethan U, Shen B. Secondary pouchitis: those with identifiable etiopathogenetic or triggering factors. Am J Gastroenterol. 2010;105(1):51–64. McCurdy JD, Loftus EV Jr, Tremaine WJ, Smyrk TC, Bruining DH, Pardi DS, Raffals LE, Kisiel JB, Coelho-Prabhu N, Kane SV, Faubion WA, Papadakis KA. Cytomegalovirus infection of the ileoanal pouch: clinical characteristics and outcomes. Inflamm Bowel Dis. 2013;19(11):2394–9. Tribonias G, Karmiris K, Giannikaki E, Konstantinidis K, Voudoukis E, Theodoropoulou A, et al. Detection of CMV in pouch mucosa in a patient with acute pouchitis: the real enemy or an innocent bystander? J Crohns Colitis. 2012;6:728–9. Komanduri S, Gillevet PM, Sikaroodi M, Mutlu E, Keshavarzian A. Dysbiosis in pouchitis: evidence of unique microfloral patterns in pouch inflammation. Clin Gastroenterol Hepatol. 2007;5(3):352–60.
CASE REPORT
Synchronous cytomegalovirus and Clostridium difficile infection of the pouch: a trigger for chronic pouchitis? Ioannis Papaconstantinou • Evanthia Zampeli • Dionysios Dellaportas • Charalambos Giannopoulos • Maria Sotiropoulou • George Polymeneas Giorgos Bamias • Spyros Michopoulos
•
Received: 29 October 2013 / Accepted: 22 January 2014 Ó Springer Japan 2014
Abstract Pouchitis occurs in up to one half of patients after restorative proctocolectomy with ileal pouch-anal anastomosis (IPAA). Cytomegalovirus (CMV) and Clostridium difficile are among the commonest secondary identifiable etiologies. A 17-year-old male with ulcerative colitis underwent IPAA due to refractory disease. Nine months later he experienced bloody diarrhea and fever. Laboratory testing and endoscopy confirmed pouch inflammation. Testing for C. difficile toxins A and B was
positive. Histology revealed affluent inclusion bodies and immunohistochemistry detected reactivity against CMV protein. Treatment with metronidazole and vancomycin offered partial improvement, whereas the addition of gancyclovir led to a successful recovery. One month after completion of treatment symptoms recurred. Repeat testing precluded an identifiable infectious cause and the diagnosis of idiopathic chronic pouchitis was established. The patient is currently on maintenance treatment with the probiotic compound VSL#3.
I. Papaconstantinou D. Dellaportas G. Polymeneas 2nd Department of Surgery, Aretaieion Hospital, University of Athens, 76 Vas. Sofias Avenue, 115 28 Athens, Greece e-mail: [email protected]
Keywords Ulcerative colitis Ileal-pouch anal anastomosis Pouchitis CMV Clostridium difficile
D. Dellaportas e-mail: [email protected]
Introduction
G. Polymeneas e-mail: [email protected]
Restorative proctocolectomy with ileal pouch-anal anastomosis (IPAA) is the surgical treatment of choice for the majority of patients with medically refractory ulcerative colitis (UC), UC-associated dysplasia, or familial adenomatous polyposis [1]. IPAA is related to a number of complications and mostly with the development of pouch inflammation which occurs almost exclusively in patients with UC. Pouchitis has a cumulative prevalence of up to 50 % [2]. Clinical presentation is variable and may occasionally result in diagnostic challenges. Pathogenesis of pouchitis is not clearly defined and most cases are classified as idiopathic. However, approximately 20–30 % of patients who present with pouchitis may have secondary identifiable etiologies which offer the opportunity of definitive and curative treatment [3]. Among the commonest causes are cytomegalovirus (CMV) and Clostridium difficile infection. Such secondary causes of pouchitis are usually suspected when patients do not
E. Zampeli (&) C. Giannopoulos M. Sotiropoulou S. Michopoulos Department of Gastroenterology, ‘Alexandra’ General Hospital, 80 Vas. Sofias Avenue, 115 28 Athens, Greece e-mail: [email protected] C. Giannopoulos e-mail: [email protected] M. Sotiropoulou e-mail: [email protected] S. Michopoulos e-mail: [email protected] G. Bamias Academic Department of Gastroenterology, First Department of Propaedeutic Internal Medicine, Laikon General Hospital, Ethnikon and Kapodistriakon University, 17 Aghiou Thoma, 115 27 Athens, Greece e-mail: [email protected]
123
Clin J Gastroenterol Fig. 1 Endoscopy showed patchy erythema, edema, granularity, friability and mucus exudate of the pouch mucosa
respond to the antibiotic regimens that constitute the standard of care for pouchitis. We present a case of synchronous CMV and C. difficile infection of the pouch in a 17-year-old male who subsequently developed chronic pouchitis.
Case report A 17-year-old male with refractory UC underwent IPAA in 2012. Nine months later, he presented with abdominal pain, bloody diarrhea and fever. There was no history of antibiotics or other drug administration before symptom initiation. Laboratory markers of inflammation were elevated (C-reactive protein and erythrocyte sedimentation rate). Endoscopy revealed pouchitis of moderate severity (Fig. 1). He was initially started on ciprofloxacin (500 mg b.i.d.) and mesalazine suppositories (500 mg b.i.d.) with only partial improvement. A fecal sample was tested positive for C. difficile toxin A and B. Additionally, on histological examination of endoscopically obtained biopsies from the pouch the presence of multiple inclusion bodies was noted. The presence of CMV was confirmed by positive immunostaining in immunohistochemistry (Fig. 2). Serology was positive for past CMV exposure [IgG(?), IgM(-)]. Of note, CMV infection had repeatedly been excluded before surgery. In addition, no CMV infection was detected in the resected colectomy specimen. Ciprofloxacin was discontinued and metronidazole commenced (500 mg t.i.d.). Due to persistence of symptoms and a new positive stool specimen for C. difficile toxin 3 days later, oral vancomycin was added at a dose of 125 mg q.i.d. As there was still only a partial response five days later, the patient was admitted and started on gancyclovir (10 mg/kg/day). The combination of vancomycin/ gancyclovir was maintained for 10 days. The patient gradually recovered and was discharged from the hospital after completion of 21 days on gancyclovir. He remained well until a month later when bloody diarrhea and urgency
123
Fig. 2 a H&E stain disclosed intense inflammatory infiltration. b Immunohistochemistry revealed CMV intranuclear inclusion bodies in multiple cells
ensued. Fecal specimens and histology were negative for C. difficile and CMV, respectively. A diagnosis of chronic idiopathic pouchitis was established based on the European Crohn’s and Colitis Organization guidelines [4]. He responded well to a course of metronidazole and is currently on maintenance treatment with VSL#3.
Clin J Gastroenterol
Discussion Pouchitis is defined as a nonspecific inflammation of the ileal pouch reservoir with a cumulative prevalence of up to 50 % in patients with UC undergoing IPAA [2]. According to certain reports, the risk of pouchitis is higher during the first two years following loop-ileostomy closure [5]. Extensive UC, extraintestinal manifestations (i.e., primary sclerosing cholangitis), being a non-smoker, perinuclear antineutrophil cytoplasmic antibody-positive serology and non-steroid anti-inflammatory drug (NSAID) use are possible risk factors for pouchitis (4). Ideally, the diagnosis of pouchitis is based on the triad of compatible symptoms (diarrhea, urgency, incontinence, nocturnal seepage, abdominal cramps, pelvic discomfort), endoscopic findings (erythema, mucous exudates, friability, bleeding, ulcers), and mandatory histological features (inflammatory infiltrate and crypt abscesses) [6]. The differential diagnosis encompasses cuffitis, Crohn’s disease, backwash ileitis, celiac sprue and ‘irritable pouch syndrome’. Pouchitis can be clinically classified as acute (duration of symptoms \4 weeks) or chronic ([4 weeks) [7]; on the basis of response to treatment with antibiotics it is also divided into antibiotic-responsive, antibiotic-dependent, and antibiotic-refractory pouchitis. In the vast majority of cases the etiology of pouchitis is not clearly defined and is considered idiopathic. However, when a specific cause is identified, secondary pouchitis is diagnosed; this occurs in 20–30 % of patients with chronic, antibiotic-refractory pouchitis. The most common etiological factors are infectious causes (C. difficile, CMV, and Candida), NSAIDs, ischemia, radiation injury, etc. [8]. Patients with secondary pouchitis may benefit from eradication of the triggering factors. In our patient, stool samples were twice positive for C. difficile toxins. C. difficile may colonize the pouch without causing inflammation; hence, eradication of the bacterium may have little impact on the natural history of pouchitis. Indeed, in patients with IPAA, the epithelium of the pouch undergoes morphological changes facilitating microbiota colonization. There are no current guidelines for the optimal management of C. difficile pouchitis due to lack of prospective trials. Standard therapy consists of metronidazole and/or vancomycin. Previous reports suggested that metronidazole may not suffice in cases of pouchitis since C. difficile infection may develop while patients are taking metronidazole. Therefore, in patients with C. difficile-associated pouchitis, rifaximin, tinidazole or vancomycin are preferred as first-line treatment. Our case was further complicated by the presence of active CMV colitis. CMV infection is a well-recognized cause of secondary pouchitis in IPAA patients [9]. CMVassociated pouchitis resembles idiopathic pouchitis,
although fever may be more common. In particular, there are no pathognomonic endoscopic features of CMV colitis [10]. Definite diagnosis of CMV pouchitis relies on the detection of the viral proteins by immunohistochemistry and/or detection of CMV-specific DNA in colonic biopsies by polymerase chain reaction (PCR). The most common practice is to base the diagnosis on histology and immunohistochemistry [11]. Histopathological characteristics include mononuclear cells with viral inclusion bodies, which are 2–4-fold larger than the surrounding normal cells. Eosinophilic intranuclear inclusions are often seen in these cytomegalic cells with a diameter of 8–10 mm. They are placed eccentrically and surrounded by a clear halo thus producing the characteristic ‘owl’s eye’ appearance. Immunostaining for CMV antigen using monoclonal antibodies is crucial for the diagnosis of CMV pouchitis, whilst serology is of limited value. Histological sections with multiple inclusion bodies have the highest probability for showing immunoreactivity against CMV proteins by immunohistochemistry. Detection of CMV DNA in the tissue is often determined as a complementary diagnostic tool although it has not been properly validated. The utility of serum assays (pp65 antigenemia or PCR for CMV DNA) for the diagnosis of CMV colitis are compromised by low negative predictive value. Treatment with gancyclovir is the recommended first-line treatment. To our knowledge, this is the first case of synchronous CMV and C. difficile infection of the pouch. As pouchitis developed 9 months after IPAA, the pre-existence of CMV in our patient is highly unlikely. Interestingly, he was not exposed to antibiotics or immunosuppressants over a long period of time. Nevertheless, it is not infrequent for C. difficile infection to develop in inflammatory bowel disease patients without prior antibiotic exposure. CMV is less often encountered in immunocompetent individuals. Due to skepticism regarding the actual role of CMV we did not initially offer antiviral treatment to the patient. However, the patient’s clinical condition with the presence of fever and his incomplete response after a week of treatment with metronidazole and vancomycin led to the addition of gancyclovir. Given that the patient was offered treatment for both agents we cannot rule out the possibility that one of the two was simply an innocent bystander rather than accountable for the patient’s symptoms [12]. Whilst the pathogenesis of pouchitis remains largely unknown, the role of luminal microflora is considered central [13]. The development of idiopathic chronic pouchitis a month after completion of combination treatment for CMV and C. difficile raises the possibility that the infectious agents triggered chronic pouchitis in the present case. Considering that follow-up fecal tests and histology for C. difficile and CMV were negative, treatment of the
123
Clin J Gastroenterol
acute episode was effective. However, chronic perpetuating inflammation of the pouch may have been induced. In conclusion, pouchitis is usually considered idiopathic. In one-third of cases specific causes are identified and should therefore always be sought before commencement of standard treatment. The exact role of infectious agents has not been elucidated, although as in the case we present, one can assume that they may trigger chronic inflammation.
4.
5.
6.
7.
Consent 8.
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal. Conflict of interest peting interests.
The authors declare that they have no com-
9.
10.
11.
References 1. Coffey JC, Winter DC, Neary P, Murphy A, Redmond HP, Kirwan WO. Quality of life after ileal pouch-anal anastomosis: an evaluation of diet and other factors using the Cleveland Global Quality of Life instrument. Dis Colon Rectum. 2002;45:30–8. 2. Stocchi L, Pemberton JH. Pouch and pouchitis. Gastroenterol Clin North Am. 2001;30:223–41. 3. Munoz-Juarez M, Pemberton JH, Sandborn WJ, Tremaine WJ, Dozois RR. Misdiagnosis of specific cytomegalovirus infection of
123
12.
13.
the ileoanal pouch as refractory idiopathic chronic pouchitis: report of two cases. Dis Colon Rectum. 1999;42:117–20. Van Assche G, Dignass A, Bokemeyer B, Danese S, Gionchetti P, Moser G, et al. Second European evidence-based consensus on the diagnosis and management of ulcerative colitis part 3: special situations. J Crohns Colitis. 2013;7(1):1–33. Fazio VW, Ziv Y, Church JM, Oakley JR, Lavery IC, Milsom JW, et al. Ileal pouch-anal anastomoses complications and function in 1005 patients. Ann Surg. 1995;222:120–7. Coffey JC, Rowan F, Burke J, Dochery NG, Kirwan WO, O’Connell PR. Pathogenesis of and unifying hypothesis for idiopathic pouchitis. Am J Gastroenterol. 2009;104:1013–23. Sandborn WJ. Pouchitis following ileal pouch-anal anastomosis: definition, pathogenesis, and treatment. Gastroenterology. 1994;107:1856–60. Shen B, Remzi FH, Lavery IC, Lashner BA, Fazio VW. A proposed classification of ileal pouch disorders and associated complications after restorative proctocolectomy. Clin Gastroenterol Hepatol. 2008;6:145–158 (quiz 124). Moonka D, Furth EE, MacDermott RP, Lichtenstein GR. Pouchitis associated with primary cytomegalovirus infection. Am J Gastroenterol. 1998;93:264–6. Navaneethan U, Shen B. Secondary pouchitis: those with identifiable etiopathogenetic or triggering factors. Am J Gastroenterol. 2010;105(1):51–64. McCurdy JD, Loftus EV Jr, Tremaine WJ, Smyrk TC, Bruining DH, Pardi DS, Raffals LE, Kisiel JB, Coelho-Prabhu N, Kane SV, Faubion WA, Papadakis KA. Cytomegalovirus infection of the ileoanal pouch: clinical characteristics and outcomes. Inflamm Bowel Dis. 2013;19(11):2394–9. Tribonias G, Karmiris K, Giannikaki E, Konstantinidis K, Voudoukis E, Theodoropoulou A, et al. Detection of CMV in pouch mucosa in a patient with acute pouchitis: the real enemy or an innocent bystander? J Crohns Colitis. 2012;6:728–9. Komanduri S, Gillevet PM, Sikaroodi M, Mutlu E, Keshavarzian A. Dysbiosis in pouchitis: evidence of unique microfloral patterns in pouch inflammation. Clin Gastroenterol Hepatol. 2007;5(3):352–60.
