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December 2015

Disseminated Cutaneous Cytomegalovirus Infection Following Total Body Electron Beam Irradiation for Mycosis Fungoides

Author Affiliations
  • 1Department of Internal Medicine, University of Texas Southwestern Medical Center, Dallas
  • 2Eastern Pathology and Dermatology Associates and Surgery Center, Beckley, West Virginia
  • 3Department of Dermatology, Duke University Medical Center, Durham, North Carolina
  • 4Department of Pathology, Duke University Medical Center, Durham, North Carolina
  • 5Department of Medicine, Duke University Medical Center, Durham, North Carolina
  • 6Durham VA Medical Center, Durham, North Carolina
JAMA Dermatol. 2015;151(12):1380-1381. doi:10.1001/jamadermatol.2015.2233

Cutaneous cytomegalovirus (CMV) infections are rare and are often associated with poor prognosis from an underlying malignant condition or immunosuppression.1,2 We report a unique case of disseminated cutaneous CMV infection and CMV viremia following total body electron beam (TBEB) irradiation.

Report of a Case

A woman in her 60s presented with pruritic pustules and generalized ulcers after starting TBEB irradiation for mycosis fungoides (MF). Her history was significant for an initial diagnosis of MF, stage IIA (T2N1M0B0), 1 year prior to presentation. After an initial response to psoralen plus UV-A (PUVA) monotherapy, her disease progressed to large-cell transformation after PUVA therapy was ended. She developed further plaque lesions and a new tumor despite restarting treatment with PUVA plus oral bexarotene (150 mg/d). She had recurrent cutaneous herpes simplex virus (HSV) infections and methicillin-resistant Staphylococcus aureus (MRSA) folliculitis. She also had a history of myelodysplastic disease manifesting with thrombocytopenia, anemia, and leukocytosis that did not require active treatment.

Treatment with TBEB irradiation for MF was started while she was taking oral acyclovir and doxycycline. She responded well until 2 weeks into the TBEB regimen (cumulative dose, 15/36 Gy), when she developed lower-extremity weakness, fever, and weight loss and was admitted to the hospital. She had numerous, shallow 4- to 8-mm, punched-out oval ulcerations and pustules scattered diffusely on her head, neck, trunk, and upper and lower extremities (Figure 1). She had larger, irregularly shaped ulcerations with a fibrinous base on her legs.

Figure 1.
Clinical Presentation of Cutaneous Cytomegalovirus Infection
Clinical Presentation of Cutaneous Cytomegalovirus Infection

Shallow, punched-out, oval ulcerations are seen scattered diffusely on the trunk and extremities.

Blood and skin cultures grew MRSA that was sensitive to trimethoprim-sulfamethoxazole, vancomycin, linezolid, and daptomycin. Findings of polymerase chain reaction (PCR) studies of cutaneous ulcerations were negative for varicella zoster virus and HSV. A punch biopsy of a cutaneous ulcer revealed epidermal atrophy, papillary dermal edema, and perivascular and interstitial mixed inflammation. Scattered endothelial cells in small dermal vessels and dermal fibroblasts showed large eosinophilic intranuclear inclusions surrounded by a clear halo (Figure 2). Immunohistochemical stains for CMV highlighted several endothelial cells and fibroblasts (Figure 2, inset). No PCR testing for CMV was performed on skin specimens because the positive immunohistochemical findings were considered confirmatory. Cytomegalovirus DNA was detected by plasma PCR.

Figure 2.
Pathologic Findings From Cutaneous Cytomegalovirus (CMV) Ulcer Specimens
Pathologic Findings From Cutaneous Cytomegalovirus (CMV) Ulcer Specimens

In the primary image, scattered endothelial cells in small dermal vessels demonstrate large eosinophilic intranuclear inclusions surrounded by a clear halo (arrowhead) (hematoxylin-eosin, original magnification ×40). The inset highlights several endothelial cells and fibroblasts (CMV immunohistochemical stain, original magnification ×40).

She was diagnosed with disseminated cutaneous CMV infection with CMV viremia. There was no evidence of other organ involvement or of further progression of her MF or hematologic disease. Treatment with TBEB was discontinued, and she completed 3 weeks of treatment with intravenous ganciclovir and vancomycin followed by resolution of her cutaneous ulcerations. Subsequent plasma CMV PCR findings were negative, and she was discharged home.

Discussion

Systemic CMV infection in immunocompromised patients may present a wide range of cutaneous manifestations, including ulcers (classically perianal), purpuric eruptions, morbilliform eruptions, vesiculobullous eruptions, and necrotic papules.1,2 Isotopic immune responses are not commonly associated with CMV, although 1 report describes a chronic CMV infection developing along herpes zoster scars in an patient with human immunodeficiency virus.3 In our patient, the CMV infection presented in a widely disseminated cutaneous fashion after initiation of TBEB irradiation similar to what is seen in eczema herpeticum—a condition causally linked to HSV4 but not CMV. The TBEB irradiation could possibly have been the inciting factor that reactivated a latent CMV infection and induced a disseminated cutaneous eruption.

It is important to remember that coinfections frequently occur with CMV, and these must be included in the differential diagnoses even in the presence of another infectious diagnosis that can seemingly explain the cutaneous findings. In this case, our patient had a history of recurrent cutaneous HSV and grew MRSA from both skin and blood. Identification of this condition is important: although cutaneous CMV is rare, it can portend a poor prognosis and be a leading sign of systemic infection.5

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Article Information

Corresponding Author: Adela R. Cardones, MD, Duke University Medical Center, Box 3135, Room 3307, Purple Zone, Duke South, 40 Medical Center Dr, Durham, NC 27710 (adela.cardones@duke.edu).

Published Online: August 12, 2015. doi:10.1001/jamadermatol.2015.2233.

Conflict of Interest Disclosures: None reported.

References
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Kaisar  MO, Kirwan  RM, Strutton  GM,  et al.  Cutaneous manifestations of cytomegalovirus disease in renal transplant recipients: a case series. Transpl Infect Dis. 2008;10(3):209-213.
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Katibi  OS, Dlova  NC, Mosam  A.  Cutaneous cytomegalovirus infection on multi dermatomal herpes zoster scars: an isotopic immune response. Clin Exp Dermatol. 2015;40(1):42-44.
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Goldgeier  MH, Cohen  SR, Braverman  IM, Stenn  KS.  An unusual and fatal case of disseminated cutaneous herpes simplex: infection in a patient with cutaneous T cell lymphoma (mycosis fungoides). J Am Acad Dermatol. 1981;4(2):176-180.
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Guo  RF, Gebreab  FH, Tang  EH, Piao  Z, Lee  SS, Perez  ML. Cutaneous ulcer as leading symptom of systemic cytomegalovirus infection. http://www.hindawi.com/journals/criid/2015/723962/. Accessed July 2, 2015.
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