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A Treatment-Refractory, Perianal Tumor Arising in an Otherwise Healthy Older Adult Woman

Educational Objective
Based on this clinical scenario and the accompanying image, understand how to arrive at a correct diagnosis.
1 Credit CME

A woman in her 80s presented with an 8-month history of a growing, painful tumor on her right buttock. The patient felt well otherwise and denied experiencing fevers, chills, sweats, weight loss, or malaise. Her medical history was notable for chronic kidney disease and genital herpes simplex virus (HSV). She reported no history of genital warts, HIV, or inflammatory bowel disease. A biopsy was performed, and results were consistent with a noninfectious, granulomatous ulcer. Based on the clinical appearance and prior pathologic findings, the patient received treatment of oral antibiotics, intralesional steroids, topical steroids, and immunomodulators, without improvement. Findings from colorectal surgical evaluation of the internal anal mucosa were negative for malignant neoplasm.

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C. Herpes vegetans

Histologic results showed epithelial hyperplasia, ulceration, and focal multinucleated keratinocytes (Figure 1B and C). Immunostaining was positive for HSV (Figure 2) but negative for varicella-zoster virus. Microbial cultures were negative for deep fungal infection and acid-fast bacteria but positive for HSV type 2. Given the clinicopathologic findings, herpes vegetans was diagnosed.

Herpes vegetans is a verrucous presentation of HSV in immunocompromised patients, including those with HIV, congenital immunodeficiency disorders, malignant neoplasm, and organ transplants.1 The condition is exceedingly uncommon in immunocompetent patients.2 Coinfections of HSV with other cutaneous infections, such as fungal and human papillomavirus (HPV) infections, have also presented as verrucous plaques, which can complicate the diagnosis.3 Herpes vegetans is frequently reported to be recalcitrant to oral antiviral therapy,4 and although, to our knowledge, precise rates of drug resistance have not been elucidated, antinucleoside resistance has been associated with several characteristics of the disease, including prolonged symptom duration, recurrent outbreaks, and prior failed therapy attempts.5

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Article Information

Corresponding Author: Erika L. Hagstrom, MD, Department of Dermatology, Oregon Health and Science University, 3303 SW Bond Ave, Center for Health & Healing Bldg 1, 16th Floor, Portland, OR 97239 (hagstroe@ohsu.edu).

Published Online: February 12, 2020. doi:10.1001/jamadermatol.2019.4823

Conflict of Interest Disclosures: None reported.

Additional Contributions: We thank the patient for granting permission to publish this information. We also thank Kevin P. White, MD, Department of Dermatology, Oregon Health and Science University, for his assistance with histopathological diagnosis, and Jina Makadia, MD, Division of Infectious Diseases, Oregon Health and Science University, for her assistance with treatment in this case. They were not compensated for their contributions.

References
1.
Rashid  A, Nanjappa  S, Dessureault  S, Messina  JL, Greene  JN.  Herpes vegetans and review of verrucous lesions of the anogenital region.  Infect Dis Clin Pract. 2017;25(6):294-300. doi:10.1097/IPC.0000000000000537Google ScholarCrossref
2.
Bae-Harboe  YS, Khachemoune  A.  Verrucous herpetic infection of the scrotum and the groin in an immuno-competent patient: case report and review of the literature.  Dermatol Online J. 2012;18(7):7.PubMedGoogle Scholar
3.
Toro  JR, Sanchez  S, Turiansky  G, Blauvelt  A.  Topical cidofovir for the treatment of dermatologic conditions: verruca, condyloma, intraepithelial neoplasia, herpes simplex and its potential use in smallpox.  Dermatol Clin. 2003;21(2):301-309. doi:10.1016/S0733-8635(02)00116-XPubMedGoogle ScholarCrossref
4.
Beasley  KL, Cooley  GE, Kao  GF, Lowitt  MH, Burnett  JW, Aurelian  L.  Herpes simplex vegetans: atypical genital herpes infection in a patient with common variable immunodeficiency.  J Am Acad Dermatol. 1997;37(5, pt 2):860-863. doi:10.1016/s0190-9622(97)80012-6PubMedGoogle ScholarCrossref
5.
Reyes  M, Shaik  NS, Graber  JM,  et al; Task Force on Herpes Simplex Virus Resistance.  Acyclovir-resistant genital herpes among persons attending sexually transmitted disease and human immunodeficiency virus clinics.  Arch Intern Med. 2003;163(1):76-80. doi:10.1001/archinte.163.1.76PubMedGoogle ScholarCrossref
6.
Kutlubay  Z, Engin  B, Zara  T, Tüzün  Y.  Anogenital malignancies and premalignancies: facts and controversies.  Clin Dermatol. 2013;31(4):362-373. doi:10.1016/j.clindermatol.2013.01.003PubMedGoogle ScholarCrossref
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de Hullu  JA, van der Zee  AGJ.  Surgery and radiotherapy in vulvar cancer.  Crit Rev Oncol Hematol. 2006;60(1):38-58. doi:10.1016/j.critrevonc.2006.02.008PubMedGoogle ScholarCrossref
8.
Wang  SH, Chi  CC, Wong  YW, Salim  A, Manek  S, Wojnarowska  F.  Genital verrucous carcinoma is associated with lichen sclerosus: a retrospective study and review of the literature.  J Eur Acad Dermatol Venereol. 2010;24(7):815-819. doi:10.1111/j.1468-3083.2009.03531.xPubMedGoogle ScholarCrossref
9.
Gholam  P, Enk  A, Hartschuh  W.  Successful surgical management of giant condyloma acuminatum (Buschke-Löwenstein tumor) in the genitoanal region: a case report and evaluation of current therapies.  Dermatology. 2009;218(1):56-59. doi:10.1159/000165359PubMedGoogle ScholarCrossref
10.
Kreuter  A, Pantelaki  I, Michalowitz  AL,  et al.  CD30-positive primary cutaneous anaplastic large cell lymphoma with coexistent pseudocarcinomatous hyperplasia.  Clin Exp Dermatol. 2018;43(5):585-588. doi:10.1111/ced.13416PubMedGoogle ScholarCrossref
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