Rupp et al. Journal of Medical Case Reports 2014, 8:163 http://www.jmedicalcasereports.com/content/8/1/163

JOURNAL OF MEDICAL

CASE REPORTS

CASE REPORT

Open Access

Protracted primary cytomegalovirus infection presenting as ileoanal pouchitis in a nonimmunosuppressed patient: a case report Christian Rupp1, Esther Herpel2, Paul Schnitzler3, Anna Zawierucha1, Philipp Zwickel1, Lukas Klute1, Martina Kadmon4, Wolfgang Stremmel1 and Annika Gauss1*

Abstract Introduction: Pouchitis often occurs after proctocolectomy and ileal pouch-anal anastomosis for ulcerative colitis. It is usually deemed idiopathic and commonly responds to antibacterial therapy. To date, only a few cases of cytomegalovirus pouchitis have been documented, and only a single report describes pouchitis in a case of assumed primary cytomegalovirus infection. Case presentation: A 26-year-old Caucasian woman underwent proctocolectomy and ileal pouch-anal anastomosis for refractory ulcerative colitis and adenocarcinoma. After 28 months she developed bloody diarrhoea, abdominal pain, fever, nausea and general malaise suggesting severe pouchitis. Antibiotic treatment reduced humoral inflammation, but failed to resolve her fever. A pouchoscopy revealed distinct pouchitis, and cytomegalovirus infection was diagnosed from pouch biopsies by polymerase chain reaction as well as conventional histology and immunohistochemistry. The infection was confirmed in her blood by polymerase chain reaction and pp65 antigen test, and was clearly defined as the ‘primary’ infection by serial serological tests. Intravenous treatment with ganciclovir (10mg/kg body weight/day) led to resolution of symptoms and negative cytomegalovirus deoxyribonucleic acid and pp65 within a few days. When symptoms and laboratory evidence of cytomegalovirus infection recurred a few days after completing 20 days of therapy with ganciclovir and valganciclovir, a second course of ganciclovir treatment was initiated. Conclusions: Cytomegalovirus infection of the ileoanal pouch is an important differential diagnosis of pouchitis even in non-immunosuppressed patients and can be treated with ganciclovir. Keywords: Cytomegalovirus, Pouchitis, Primary infection, Ulcerative colitis

Introduction Idiopathic pouchitis is a common problem after ileal pouch-anal anastomosis (IPAA) for ulcerative colitis (UC). It can usually be managed with antibiotics [1]. More rarely, specific causes of pouchitis can be identified, for example Clostridium difficile or cytomegalovirus (CMV) infection [1-6]. CMV has infected between 40 and 100% of the adult population [7-9]. This facultative pathogen causes clinical illness in a small percentage of the infected, with the greatest risk during the intrauterine period or in immunocompromised patients [10]. * Correspondence: [email protected] 1 Department of Gastroenterology and Hepatology, University Hospital Heidelberg, INF 410, 69120 Heidelberg, Germany Full list of author information is available at the end of the article

CMV can aggravate pre-existing inflammatory bowel disease (IBD), especially in immunosuppressed patients, whereas CMV colitis has only rarely been described in immunocompetent patients without IBD [6,11-13]. There are few reports on CMV infection after restorative proctocolectomy in patients with UC. A single case of a patient with putative ‘primary’ CMV infection of the pouch was published in 1998, and the patient was successfully treated with ganciclovir [4]. In that case report, the definition of primary CMV infection versus reactivation was derived from acute symptoms, pouch biopsies with positive immunohistochemistry and the presence of CMV immunoglobulin (Ig) M and G antibodies in the blood. Since no data on antibody avidity or serological analyses from samples taken prior to the onset of symptoms were

© 2014 Rupp et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

Rupp et al. Journal of Medical Case Reports 2014, 8:163 http://www.jmedicalcasereports.com/content/8/1/163

Page 2 of 6

presented, a reliable differentiation from CMV reactivation is not possible. Two other case reports describe chronic refractory pouchitis due to CMV infection [14]. Very recently, McCurdy et al. published a retrospective case series of seven patients with CMV infection of the ileoanal pouch, of whom five were immunocompromised, most of them being liver transplant patients for primary sclerosing cholangitis. In that paper, it was not stated whether any of the patients suffered from primary CMV infection [6]. The only systematic analysis on the prevalence of CMV in the ileoanal pouch detected CMV genes and proteins in 41.6% of patients with pouchitis after proctocolectomy for UC, but only in 11.2% of otherwise comparable patients with normal pouch findings [5]. Recently, Tribonias et al. raised the question of whether CMV in pouch mucosa might be the ‘real enemy or the innocent bystander’ [3]. The answer may be influenced by case-specific factors and even then difficult to determine conclusively. Here, we report a rare case of primary CMV infection with pouchitis as the prevailing disease manifestation in a patient after proctocolectomy and IPAA for UC.

Case presentation In 2011, a 26-year-old Caucasian woman with concomitant coeliac disease underwent proctocolectomy and IPAA for refractory UC and adenocarcinoma of the colon. She did not undergo chemotherapy. Three months later, her ileostomy was closed. After 1 year, she suffered from acute pouchitis, which was successfully treated with ciprofloxacin and metronidazole. She was admitted to our hospital 2.5 years after her proctocolectomy, with severe diarrhoea, abdominal cramps, discharge of blood, nausea, emesis, generalised malaise and

chills with fever up to 38.5°C. Prior to admission, she was not taking any medication. The physical examination showed tachycardia (104 beats per minute) and tenderness of her lower abdomen. Laboratory data revealed high C-reactive protein (CRP 117mg/L:

Protracted primary cytomegalovirus infection presenting as ileoanal pouchitis in a non-immunosuppressed patient: a case report.

Pouchitis often occurs after proctocolectomy and ileal pouch-anal anastomosis for ulcerative colitis. It is usually deemed idiopathic and commonly res...
504KB Sizes 0 Downloads 3 Views

Recommend Documents


Cytomegalovirus pouchitis in a patient with Crohn's disease.
Colectomy with ileoanal pouch formation is usually contraindicated in patients with Crohn's disease (CD) due to the risk of recurrent disease and pouch failure. We report the case of a patient, initially thought to have ulcerative colitis (UC), who u

Colonic perforation in a patient with systemic lupus erythematosus accompanied by cytomegalovirus infection: A case report.
Cytomegalovirus (CMV) infection of the gastrointestinal tract is an uncommon illness, but can be observed in immunocompromised patients. Systemic lupus erythematosus (SLE) patients are generally at high risk of CMV infection. Here we report a subacut

Cytomegalovirus retinitis in an immunocompetent patient: A case report.
To report a case of cytomegalovirus (CMV) retinitis in an immunocompetent adult patient.

Transient antiphospholipid syndrome associated with primary cytomegalovirus infection: a case report and literature review.
Viral infection is known to induce transient autoimmunity in humans. Acute cytomegalovirus (CMV) infection is implicated in occasional thrombosis formation. We here, for the first time, report a 19-year-old female who had an acute CMV infection, lead

Hypopituitarism Presenting as Adrenal Insufficiency and Hypothyroidism in a Patient with Wilson's Disease: a Case Report.
Wilson's disease typically presents symptoms associated with liver damage or neuropsychiatric disturbances, while endocrinologic abnormalities are rare. We report an unprecedented case of hypopituitarism in a patient with Wilson's disease. A 40-year-

Synchronous cytomegalovirus and Clostridium difficile infection of the pouch: a trigger for chronic pouchitis?
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 w

Extracavitary primary effusion lymphoma presenting as a cutaneous tumor: a case report and literature review.
Primary effusion lymphoma is an unusual form of aggressive B-cell lymphoma universally associated with human herpesvirus 8 (HHV8) that involves mostly human immunodeficiency virus (HIV)-infected patients. Characteristically, it presents as a malignan

Cancer of unknown primary (CUP) presenting as a Spigelian hernia: a case report.
Cancer of unknown primary (CUP) is a well-known entity that can present in a multitude of clinical presentations. Based on the extent and organ of involvement, the presentation will be varied. For instance, patients presenting with axillary lymphaden