Cecal vanishing tumor associated with cytomegalovirus CASE REPORT

World J Gastrointest Oncol 2010 November 15; 2(11): 417-420
ISSN 1948-5204 (online)
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doi:10.4251/wjgo.v2.i11.417
© 2010 Baishideng. All rights reserved.
CASE REPORT
Cecal vanishing tumor associated with cytomegalovirus
infection in an immunocompetent elderly adult
Shinsuke Kawasaki, Satoshi Osawa, Ken Sugimoto, Takahiro Uotani, Masafumi Nishino, Takanori Yamada,
Mitsushige Sugimoto, Takahisa Furuta, Mutsuhiro Ikuma
hospital visit without any further treatment. Inflammatory pseudotumors associated with CMV infection should
be considered as a differential diagnosis of tumorous lesions in the colon, even in immunocompetent adults.
Shinsuke Kawasaki, Satoshi Osawa, Ken Sugimoto, Takahiro
Uotani, Masafumi Nishino, Takanori Yamada, Mutsuhiro
Ikuma, First Department of Medicine, Hamamatsu University
School of Medicine, 1-20-1 Handayama, Higashi-ku, Hamamatsu
431-3192, Japan
Mitsushige Sugimoto, Takahisa Furuta, Center for Clinical
Research, Hamamatsu University School of Medicine, 1-20-1
Handayama, Higashi-ku, Hamamatsu 431-3192, Japan
Author contributions: Kawasaki S and Osawa S wrote the
manuscript; Sugimoto K, Furuta T and Ikuma M contributed to
the paper design and coordination; Uotani T, Nishino M, Yamada
T and Sugimoto M contributed in performing the endoscopic
examination and follow-up; all the authors have read and approved
the final manuscript.
Correspondence to: Satoshi Osawa, MD, PhD, Assistant
Professor, First Department of Medicine, Hamamatsu University
School of Medicine, 1-20-1 Handayama, Higashi-ku, Hamamatsu
431-3192, Japan. sososawa@hama-med.ac.jp
Telephone: +81-53-4352261 Fax: +81-53-4349447
Received: July 1, 2010
Revised: September 14, 2010
Accepted: September 21, 2010
Published online: November 15, 2010
© 2010 Baishideng. All rights reserved.
Key words: Cytomegalovirus colitis; Vanishing tumor;
Elderly adult; Inflammatory pseudotumor; Immunocompetent host
Peer reviewers: Macaulay Onuigbo, MD, MSc FWACP
FASN, Department of Nephrology, Midelfort Clinic, Mayo
Health System, Eau Claire, WI 54702, United States; Vaios
Karanikas, BSc (Hons), PhD, Associate Professor, Department
of Immunology, Faculty of Medicine, School of Health Sciences, University of Thessaly, Mezourlo, Larissa 41110, Greece
Kawasaki S, Osawa S, Sugimoto K, Uotani T, Nishino M, Yamada T, Sugimoto M, Furuta T, Ikuma M. Cecal vanishing tumor associated with cytomegalovirus infection in an immunocompetent
elderly adult. World J Gastrointest Oncol 2010; 2(11): 417-420
Available from: URL: http://www.wjgnet.com/1948-5204/full/v2/
i11/417.htm DOI: http://dx.doi.org/10.4251/wjgo.v2.i11.417
Abstract
Gastrointestinal involvement in cytomegalovirus (CMV)
infection is well documented among immunocompromised patients and is also observed in immunocompetent individuals. The presentation of this infection can
sometimes mimic those of other diseases, thus making
accurate diagnosis difficult. We herein report a rare case
of an immunocompetent elderly adult with gastrointestinal CMV infection that presented as a vanishing tumor
at the cecum. A 76-year old man initially presented with
lower abdominal pain. Colonoscopy revealed a tumorous lesion with irregular ulceration observed at the ileocecal valve. Histological findings of a biopsy specimen
revealed intranuclear inclusions which were positive for
CMV on immunohistochemical staining. However, this
tumorous lesion disappeared within 7 wk from the initial
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INTRODUCTION
Gastrointestinal involvement in cytomegalovirus (CMV)
infection is well documented among immunocompromised patients. However, recent reports reveal its occurrence in immunocompetent individuals as well as with
some instances of life-threatening manifestations, particularly in elderly adults with other comorbidities[1-3]. The
clinical characteristics of this infection can sometimes
mimic those of other diseases, making accurate diagnosis
difficult[4]. Although most prototypical lesions present
with well-defined punched-out ulceration, endoscopic
findings of gastrointestinal CMV infection may pres-
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Kawasaki S et al . Vanishing tumor associated with CMV infection
ent with various appearances such as erosion, ulceration,
mucosal hemorrhage and stenosis[4]. Awareness of these
characteristics and accurate diagnosis by histological examination are essential for antiviral therapy. In contrast,
self-limited CMV infection in the absence of antiviral
therapy is also reported in immunocompetent hosts[1,5].
We herein report a rare case of an immunocompetent
elderly adult with gastrointestinal CMV infection that presented as a vanishing tumor at the ileocecal valve.
A
CASE REPORT
A 76-year old man initially presented with lower abdominal pain persisting over 4 d. Although he had a past history of cerebral infarction and hypertension, his general
condition was good and his performance status was well
maintained. Physical examination revealed tenderness in
the right lower quadrant of the abdomen. Rebound tenderness was not detected. Laboratory investigations indicated increased white blood cell count (11 300 cells/mm3;
normal range 3600-9200 cells/mm3), elevated C-reactive
protein (9.53 mg/dL; normal range 0.70-1.17 mg/dL) and
elevated γ-GTP levels (228 IU/L; normal range 12-73
IU/L). Serum creatinine levels (0.76mg/dl; normal range
0.70-1.17 mg/dL) were normal. Fasting plasma glucose
levels (93 mg/dL; normal range 60-110 mg/dL) and hemoglobin A1c levels (5.2%; normal range 4.3%-5.8%)
were normal. The levels of tumor markers, carcinoembryonic antigen (CEA) and carbohydrate antigen 19-9
(CA19-9), were normal while the level of soluble IL-2
receptor was slightly elevated (538 U/mL; normal range
145-519 U/mL). Other laboratory tests yielded normal
results.
After initial clinical and laboratory evaluation, antibiotic therapy using ceftriaxone (CTRX) and levofloxacin
(LVFX) for a probable diagnosis of acute appendicitis,
diverticulitis or bacterial ileitis was initiated. His clinical
condition improved gradually and the abdominal pain
disappeared within a week. Two weeks later, a followup colonoscopy was performed. A tumorous lesion with
irregular ulceration was observed at the ileocecal valve
and multiple diverticula were also detected in the ascending colon and cecum (Figure 1). Abdominal computed
tomography revealed an enhanced mass lesion of 2 cm
diameter in the cecum (Figure 2). Histological findings
of a biopsy specimen from the tumorous lesion revealed
intranuclear inclusions. Immunohistochemical staining for
CMV revealed scattered positive cells (Figure 3). Following pathological diagnosis, the patient tested negative for
HIV, positive for antinuclear antibody (1:80), negative for
anti-DNA antibody and complement activity (C3 and C4)
was in the normal range. Because his abdominal symptoms disappeared and physical examination revealed no
tenderness in the right lower quadrant of the abdomen,
we performed an endoscopic examination by double balloon enteroscopy at 49 d after the initial hospital visit to
re-evaluate the focal lesion for antiviral therapy and to distinguish it from a possible coexisting neoplasia, especially
a lymphoma. However, the tumorous lesion at the ileoce-
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B
Figure 1 Endoscopic findings of the tumorous lesion at the ileocecal
valve. A: Tumorous lesion with a diameter of 2 cm is observed at the ileocecal
valve and multiple diverticula are detected in the cecum; B: The tumorous lesion
disappeared 30 d after initial examination without any antiviral therapy.
Figure 2 Computed tomography scan revealed an enhanced mass lesion
with a diameter of 2 cm in the cecum (indicated by an arrow).
cal valve had vanished and both immunohistochemical
staining for PCR and CMV from the biopsy sample were
negative. We could not detect any other inflammatory or
neoplastic lesion in the ileum and colon. Twelve months
of follow-up observation later, the patient appears healthy
with no evidence of recurrence of CMV colitis.
DISCUSSION
CMV is a member of the herpes virus family and is a
common pathogen with serology indicating prior exposure in 40%-100% of the general population[6]. Although
human CMV infection can be seen at all stages of life, it
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Kawasaki S et al . Vanishing tumor associated with CMV infection
eral reports reveal that CMV secondarily colonizes bowel
mucosa previously affected by another inflammatory
process such as inflammatory bowel disease[17] and colitis
with other pathogens[18,19]. CMV is also documented to
behave nonpathogenically in some cases[4]. In this case, no
evidence of inflammatory bowel disease or other types of
colitis was detected in the histopathological examination.
In a meta-analysis of CMV colitis outcomes in immunocompetent hosts (44 cases), the rate of spontaneous
remission was 31.8% but > 50% in patients younger than
55 years old. Death occurred in 31.8% patients older than
55 years old. Highest mortality rates were associated with
immune-modulating conditions such as diabetes mellitus,
renal failure and malignancies. Younger (< 55 years) and
otherwise healthy patients were able to recover from CMV
infection without antiviral therapy[1]. According to this meta-analysis, antiviral therapy with ganciclovir or foscarnet
seems to be mandatory for older patients and for patients
with immune-modulating conditions. Clinicopathological
significance of CMV remains to be elucidated and might
differ among individual cases.
In conclusion, we report a rare case of an immunocompetent elderly adult with a gastrointestinal CMV infection that showed a vanishing tumor at the ileocecal valve
of the cecum. Inflammatory pseudotumors associated
with CMV virus infection should be considered as differential diagnosis of tumorous lesion in the colon, even
in immunocompetent hosts. This case certainly represents
a clinical characteristic of CMV colitis in immunocompetent hosts. However, further clinical experience is required
to clarify the need for antiviral therapy in an immunocompetent host.
A
B
Figure 3 Histological findings of endoscopic forceps biopsy specimens.
A:Hematoxylin-Eosin staining ×100; B: Immunohistochemical staining for
cytomegalovirus reveals positive cells with inclusion.
often affects those who are immunosuppressed and can
commonly cause retinitis, pneumonitis or enteritis in such
patients[7]. CMV complications usually occur in advanced
acquired immune deficiency syndrome when CD4 lymphocyte counts drop below 50 cells/mm3 and among
transplant recipients as a reactivation of latent infection.
Most primary CMV infections in immunologically healthy
adults are asymptomatic or associated with a mild mononucleosis-like syndrome[7].
In the colonic manifestation of CMV infection, multiple erosions or ulcers are common and sometimes may
cause perforation in severe cases[8]. However, rare manifestations of CMV infections of the colon include solitary
mucosal ulcers, toxic megacolon[9], pseudomembrane
formation[10,11] and ischemic colitis[12]. Isolated cases of
pseudoneoplastic appearance of CMV colitis associated
with polypoid masses or strictures have been documented
in literature pertaining to HIV-positive patients[13] and
HIV-negative patients[14-16]. Most of these cases involved
surgical resection and/or antiviral therapy. However, this
is the first report, as far as we could find, detailing the disappearance of a CMV-associated tumor in the absence of
antiviral therapy.
In this case, the initial symptoms were similar to acute
appendicitis, diverticulitis or bacterial ileitis, and laboratory
data indicated successful management of inflammation
by initial antibiotic therapy. Therefore, CMV colitis concomitant with pathogenic bacterial infection could be a
possibility when the patient first visited our hospital. Sev-
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REFERENCES
1
Galiatsatos P, Shrier I, Lamoureux E, Szilagyi A. Metaanalysis of outcome of cytomegalovirus colitis in immunocompetent hosts. Dig Dis Sci 2005; 50: 609-616
2 Rafailidis PI, Mourtzoukou EG, Varbobitis IC, Falagas ME.
Severe cytomegalovirus infection in apparently immunocompetent patients: a systematic review. Virol J 2008; 5: 47
3 Ryu KH, Yi SY. Cytomegalovirus ileitis in an immunocompetent elderly adult. World J Gastroenterol 2006; 12: 5084-5086
4 Goodgame RW. Gastrointestinal cytomegalovirus disease.
Ann Intern Med 1993; 119: 924-935
5 Einbinder Y, Wolf DG, Pappo O, Migdal A, Tsvang E, Ackerman Z. The clinical spectrum of cytomegalovirus colitis in
adults. Aliment Pharmacol Ther 2008; 27: 578-587
6 de la Hoz RE, Stephens G, Sherlock C. Diagnosis and treatment approaches of CMV infections in adult patients. J Clin
Virol 2002; 25 Suppl 2: S1-S12
7 Sissons JG, Carmichael AJ. Clinical aspects and management of cytomegalovirus infection. J Infect 2002; 44: 78-83
8 Chetty R, Roskell DE. Cytomegalovirus infection in the gastrointestinal tract. J Clin Pathol 1994; 47: 968-972
Orloff JJ, Saito R, Lasky S, Dave H. Toxic megacolon in cy9
tomegalovirus colitis. Am J Gastroenterol 1989; 84: 794-797
10 Olofinlade O, Chiang C. Cytomegalovirus infection as a
cause of pseudomembrane colitis: a report of four cases. J
Clin Gastroenterol 2001; 32: 82-84
11 Battaglino MP, Rockey DC. Cytomegalovirus colitis presenting with the endoscopic appearance of pseudomembranous colitis. Gastrointest Endosc 1999; 50: 697-700
419
November 15, 2010|Volume 2|Issue 11|
Kawasaki S et al . Vanishing tumor associated with CMV infection
12 Siegal DS, Hamid N, Cunha BA. Cytomegalovirus colitis
mimicking ischemic colitis in an immunocompetent host.
Heart Lung 2005; 34: 291-294
13 Rich JD, Crawford JM, Kazanjian SN, Kazanjian PH. Discrete gastrointestinal mass lesions caused by cytomegalovirus in patients with AIDS: report of three cases and review.
Clin Infect Dis 1992; 15: 609-614
14 Falagas ME, Griffiths J, Prekezes J, Worthington M. Cytomegalovirus colitis mimicking colon carcinoma in an HIVnegative patient with chronic renal failure. Am J Gastroenterol 1996; 91: 168-169
15 Imam SZ, Khan A, Jaso JM, Foringer JR. Quiz page. CMV
colitis presenting as a colonic mass. Am J Kidney Dis 2010;
55: A35-A37
16 Maiorana A, Torricelli P, Giusti F, Bellini N. Pseudoneo-
plastic appearance of cytomegalovirus-associated colitis in
nonimmunocompromised patients: report of 2 cases. Clin
Infect Dis 2003; 37: e68-e71
17 Kandiel A, Lashner B. Cytomegalovirus colitis complicating
inflammatory bowel disease. Am J Gastroenterol 2006; 101:
2857-2865
18 Sugisaki K, Maekawa S, Mori K, Ichii O, Kanda K, Tai M,
Suzuki T, Ochiai H, Ejiri Y, Takahashi M, Hakozaki H. Selflimited colitis during the course of rubella and cytomegalovirus infection in an immunocompetent adult. Intern Med
2004; 43: 404-409
19 Fan X, Scott L, Qiu S, Raju GS, Shabot M. Colonic coinfection of histoplasma and cytomegalovirus mimicking carcinoma in a patient with HIV/AIDS. Gastrointest Endosc 2008;
67: 977-978; discussion 978
S- Editor Wang JL
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