Taglietti et al. Journal of Medical Case Reports 2010, 4:334 http://www.jmedicalcasereports.com/content/4/1/334 evidence of the onset of hemolysis in patients with nega- tive Coombs test during CMV infection [5]. In our spe- cific case, the presence of cold agglutinins may be a possible explanation for the onset of hemolysis. We performed a review of the literature by PubMed for relevant articles regarding hemolytic anemia during acute CMV infection in apparently immunocompetent individuals, published between 1980 and 2008. Only 12 cases have been reported (Table 1). Rafailidis et al. [7] performed a systematic review that included 290 immu- nocompetent patients with severe clinical manifestations of CMV infection, of whom only five were found to have hemolytic anemia. Among the 12 cases reported in the literature, the Coombs test was positive in four, negative in three, and was not specified for the remaining five patients (Table 1). Bonnet et al. [8] described 115 patients with acute CMV infection. Twenty-three patients (20%) had hemo- lytic anemia; however, the authors did not distinguish whether the hemolysis was secondary to hypersplenia or directly connected to the CMV infection; thus this study was not reported in Table 1. Regarding therapeutic management, two patients were treated with steroids and anti-CMV therapy (one of whom also had blood transfusions); two received only steroid therapy; three patients were not given any speci- fic treatment; and for the remaining five patients, the treatment was not specified. Interestingly, the prognosis was favorable in all cases, including those patients who did not receive steroids and/or antiviral therapy. One of those cases had a clinical history similar to that of our patient [5], with a hemoglobin level that reached 5.1 g/ dL, and the patient experienced a full and spontaneous recovery without additional medications. As is evident in the literature, no conclusive state- ments regarding specific treatment of hemolytic anemia during acute CMV infection in immunocompetent patients can be made. In our opinion, although steroid and specific antiviral therapy was given in our patient, the policy of “wait and see” in the presence of hemolytic anemia without severe manifestations during CMV infection in an immuno- competent patient could be justified. Conclusions Clinicians should consider CMV infection in the differ- ential diagnosis of hemolytic anemia in immunocompe- tent adults. The true incidence of this complication may be underestimated, because CMV serology may not be routinely obtained in patients with hemolysis. Possible therapeutic options include antiviral therapy and ster- oids, although the best treatment strategy is still controversial. Page 3 of 3 Randomized controlled trials are needed for conclu- sive answers regarding the specific treatment of hemoly- tic anemia due to CMV infection. Consent Written informed consent was obtained from the patient for the 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. Acknowledgements This work was supported by Ricerca Corrente IRCCS. Authors’ contributions FT followed up the patient during the hospitalization, analyzed data from the literature, and wrote the article. ST and PN analyzed data from the literature. CMD was the major contributor in writing the manuscript. AC and EG followed up the patient after the discharge from the hospital. NP reviewed the manuscript. All authors read and approved the final manuscript. Competing interests The authors declare that they have no competing interests. Received: 7 December 2009 Accepted: 21 October 2010 Published: 21 October 2010 References 1. 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Salloum E, Lundberg WB: Hemolytic anemia with positive direct antiglobulin test secondary to spontaneous cytomegalovirus infection in healthy adults. Acta Haematol 1994, 92(1):39-41. doi:10.1186/1752-1947-4-334 Cite this article as: Taglietti et al.: Hemolytic anemia due to acute cytomegalovirus infection in an immunocompetent adult: a case report and review of the literature. Journal of Medical Case Reports 2010 4:334.