Pediatr Gastroenterol Hepatol Nutr.  2015 Mar;18(1):60-65. 10.5223/pghn.2015.18.1.60.

Disseminated Cytomegalovirus Infection and Protein Losing Enteropathy as Presenting Feature of Pediatric Patient with Crohn's Disease

Affiliations
  • 1Department of Pediatric Gastroenterology Hepatology and Nutrition, Karadeniz Technical University, Trabzon, Turkey. muratcak@hotmail.com
  • 2Department of Pathology, Faculty of Medicine, Karadeniz Technical University, Trabzon, Turkey.

Abstract

We report a pediatric patient admitted with abdominal pain, diffuse lower extremity edema and watery diarrhea for two months. Laboratory findings including complete blood count, serum albumin, lipid and immunoglobulin levels were compatible with protein losing enteropathy. Colonoscopic examination revealed diffuse ulcers with smooth raised edge (like "punched out holes") in the colon and terminal ileum. Histopathological examination showed active colitis, ulcerations and inclusion bodies. Immunostaining for cytomegalovirus was positive. Despite supportive management, antiviral therapy, the clinical condition of the patient worsened and developed disseminated cytomegalovirus infection and the patient died. Protein losing enteropathy and disseminated cytomegalovirus infection a presenting of feature in steroid-naive patient with inflammatory bowel disease is very rare. Hypogammaglobulinemia associated with protein losing enteropathy in Crohn's disease may predispose the cytomegalovirus infection in previously healthy children.

Keyword

Crohn disease; Cytomegalovirus infections; Protein-losing enteropathies

MeSH Terms

Abdominal Pain
Agammaglobulinemia
Blood Cell Count
Child
Colitis
Colon
Crohn Disease*
Cytomegalovirus
Cytomegalovirus Infections*
Diarrhea
Edema
Humans
Ileum
Immunoglobulins
Inclusion Bodies
Inflammatory Bowel Diseases
Lower Extremity
Protein-Losing Enteropathies*
Serum Albumin
Ulcer
Immunoglobulins
Serum Albumin

Figure

  • Fig. 1 (A) Colonoscopic finding of the patient on admission. Note the ulcer with smooth raised edge (like "punched out holes"). (B) Aphthous ulcers in the cecum suggesting Crohn's disease.

  • Fig. 2 (A) Cyrptitis and active chronic inflammation in the lamina propria of the colonic mucosa suggesting active colitis (H&E, ×200). (B) Immunostaining for cytomegalovirus (CMV) antigen, brownish appearance in the vascular endothelial cells indicates CMV-positive cells (immunohistochemistry, ×400).

  • Fig. 3 Treatment regimen of the patient. IV, intra-venous.


Reference

1. Crough T, Khanna R. Immunobiology of human cytomegalovirus: from bench to bedside. Clin Microbiol Rev. 2009; 22:76–98.
Article
2. Kim JJ, Simpson N, Klipfel N, Debose R, Barr N, Laine L. Cytomegalovirus infection in patients with active inflammatory bowel disease. Dig Dis Sci. 2010; 55:1059–1065.
Article
3. Lawlor G, Moss AC. Cytomegalovirus in inflammatory bowel disease: pathogen or innocent bystander. Inflamm Bowel Dis. 2010; 16:1620–1627.
Article
4. Umar SB, DiBaise JK. Protein-losing enteropathy: case illustrations and clinical review. Am J Gastroenterol. 2010; 105:43–49.
Article
5. Ho M. The history of cytomegalovirus and its diseases. Med Microbiol Immunol. 2008; 197:65–73.
Article
6. Yoshino T, Nakase H, Ueno S, Uza N, Inoue S, Mikami S, et al. Usefulness of quantitative real-time PCR assay for early detection of cytomegalovirus infection in patients with ulcerative colitis refractory to immunosuppressive therapies. Inflamm Bowel Dis. 2007; 13:1516–1521.
Article
7. Matsuoka K, Iwao Y, Mori T, Sakuraba A, Yajima T, Hisamatsu T, et al. Cytomegalovirus is frequently reactivated and disappears without antiviral agents in ulcerative colitis patients. Am J Gastroenterol. 2007; 102:331–337.
Article
8. Berk T, Gordon SJ, Choi HY, Cooper HS. Cytomegalovirus infection of the colon: a possible role in exacerbations of inflammatory bowel disease. Am J Gastroenterol. 1985; 80:355–360.
9. Pfau P, Kochman ML, Furth EE, Lichtenstein GR. Cytomegalovirus colitis complicating ulcerative colitis in the steroid-naive patient. Am J Gastroenterol. 2001; 96:895–899.
Article
10. Ghidini B, Bellaiche M, Berrebi D, Viala J, Hugot JP, Mougenot JF, et al. Cytomegalovirus colitis in children with inflammatory bowel disease. Gut. 2006; 55:582–583.
Article
11. Inoue K, Wakabayashi N, Fukumoto K, Yamada S, Bito N, Yoshida N, et al. Toxic megacolon associated with cytomegalovirus infection in a patient with steroid-naïve ulcerative colitis. Intern Med. 2012; 51:2739–2743.
Article
12. Ferrante M, Penninckx F, De Hertogh G, Geboes K, D'Hoore A, Noman M, et al. Protein-losing enteropathy in Crohn's disease. Acta Gastroenterol Belg. 2006; 69:384–389.
Article
13. Sferra TJ, Pawel BR, Qualman SJ, Li BU. Ménétrier disease of childhood: role of cytomegalovirus and transforming growth factor alpha. J Pediatr. 1996; 128:213–219.
Article
14. Orvar K, Murray J, Carmen G, Conklin J. Cytomegalovirus infection associated with onset of inflammatory bowel disease. Dig Dis Sci. 1993; 38:2307–2310.
Article
15. van Dorp WT, Jonges E, Bruggeman CA, Daha MR, van Es LA, van Der Woude FJ. Direct induction of MHC class I, but not class II, expression on endothelial cells by cytomegalovirus infection. Transplantation. 1989; 48:469–472.
Article
16. Domènech E, Vega R, Ojanguren I, Hernández A, Garcia-Planella E, Bernal I, et al. Cytomegalovirus infection in ulcerative colitis: a prospective, comparative study on prevalence and diagnostic strategy. Inflamm Bowel Dis. 2008; 14:1373–1379.
Article
17. Turner D, Travis SP, Griffiths AM, Ruemmele FM, Levine A, Benchimol EI, et al. European Crohn's and Colitis Organization. Porto IBD Working Group, European Society of Pediatric Gastroenterology, Hepatology, and Nutrition. Consensus for managing acute severe ulcerative colitis in children: a systematic review and joint statement from ECCO, ESPGHAN, and the Porto IBD Working Group of ESPGHAN. Am J Gastroenterol. 2011; 106:574–588.
Article
Full Text Links
  • PGHN
Actions
Cited
CITED
export Copy
Close
Share
  • Twitter
  • Facebook
Similar articles
Copyright © 2024 by Korean Association of Medical Journal Editors. All rights reserved.     E-mail: koreamed@kamje.or.kr