Xanthogranuloma on the penis of an adult: An unusual localization

Abstract

To the Editor: A 44-year-old man was referred to our department with a 1-month history of lesions on his glans penis. A clinical examination revealed two well defined, orange-yellow to skin-colored, dome-shaped, smooth, 5- to 7-mm asymptomatic papules (Fig 1). General laboratory screening, including lipid levels, renal and liver function tests, and a complete blood count, were within normal limits. A biopsy specimen from one of the papules showed a dermal infiltrate composed of macrophages, neutrophils, lymphocytes, and fibroblasts, along with foam cells and Touton giant cells (Fig 2). A diagnosis of xanthogranuloma (XG) was made. An ophthalmology examination revealed no abnormalities. Two months after the biopsy, the papules disappeared spontaneously and no obvious relapse has been noted.Fig 2Infiltrate composed of macrophages, neutrophils, and lymphocytes, along with foam cells and Touton giant cells. (Hematoxylin–eosin stain; original magnification: ×100.)View Large Image Figure ViewerDownload Hi-res image Download (PPT)XG is an unusual form of non–Langerhans cell histiocytosis that typically affects children and less frequently adults, with the peak incidence being in the late twenties to early thirties, with an equal sex distribution.1Rodriguez J. Ackerman A.B. Xanthogranuloma in adults.Arch Dermatol. 1976; 112: 43-44Crossref PubMed Scopus (104) Google Scholar, 2Whitmore S.E. Multiple xanthogranulomas in an adult: case report and literature review.Br J Dermatol. 1992; 127: 177-181Crossref PubMed Scopus (15) Google Scholar Clinically, it presents as well defined, firm, yellow-red, dome-shaped, asymptomatic papules or nodules, most frequently located on the head and trunk. Penile involvement is exceptionally rare, and there have been only three cases of XG affecting the penis in the literature.3Quintana de la Rosa J.L. Antón Badiola I. Ortiz Rey J. Juvenile xanthogranuloma of the penis.Actas Urol Esp. 2008; 32 ([in Spanish]): 659-661Crossref PubMed Google Scholar, 4Hautmann R.E. Bachor R. Juvenile xanthogranuloma of the penis.J Urol. 1993; 150: 456-457PubMed Google Scholar, 5Laguna Urraca G. Concha López A. Tudela Patón M.P. Verruciform xanthoma of the penis.Actas Urol Esp. 1990; 14 ([in Spanish]): 210-213PubMed Google Scholar Histologically, it is characterized by a dense histiocytic infiltrate within the papillary dermis occasionally extending into the reticular dermis and subcutaneous tissue. The cellular infiltrate includes spindle cells, giant cells (typically of the Touton type), lymphocytes, eosinophils, and neutrophils. Immunohistochemical studies are positive for vimentin, CD68, and factor XIIIa, and negative for S100, CD34, and CD1a.To our knowledge, our case represents the fourth report of XG occurring in the penis of an adult. Xanthogranuloma should be considered in the differential diagnosis of solitary lesions involving the penis. To the Editor: A 44-year-old man was referred to our department with a 1-month history of lesions on his glans penis. A clinical examination revealed two well defined, orange-yellow to skin-colored, dome-shaped, smooth, 5- to 7-mm asymptomatic papules (Fig 1). General laboratory screening, including lipid levels, renal and liver function tests, and a complete blood count, were within normal limits. A biopsy specimen from one of the papules showed a dermal infiltrate composed of macrophages, neutrophils, lymphocytes, and fibroblasts, along with foam cells and Touton giant cells (Fig 2). A diagnosis of xanthogranuloma (XG) was made. An ophthalmology examination revealed no abnormalities. Two months after the biopsy, the papules disappeared spontaneously and no obvious relapse has been noted. XG is an unusual form of non–Langerhans cell histiocytosis that typically affects children and less frequently adults, with the peak incidence being in the late twenties to early thirties, with an equal sex distribution.1Rodriguez J. Ackerman A.B. Xanthogranuloma in adults.Arch Dermatol. 1976; 112: 43-44Crossref PubMed Scopus (104) Google Scholar, 2Whitmore S.E. Multiple xanthogranulomas in an adult: case report and literature review.Br J Dermatol. 1992; 127: 177-181Crossref PubMed Scopus (15) Google Scholar Clinically, it presents as well defined, firm, yellow-red, dome-shaped, asymptomatic papules or nodules, most frequently located on the head and trunk. Penile involvement is exceptionally rare, and there have been only three cases of XG affecting the penis in the literature.3Quintana de la Rosa J.L. Antón Badiola I. Ortiz Rey J. Juvenile xanthogranuloma of the penis.Actas Urol Esp. 2008; 32 ([in Spanish]): 659-661Crossref PubMed Google Scholar, 4Hautmann R.E. Bachor R. Juvenile xanthogranuloma of the penis.J Urol. 1993; 150: 456-457PubMed Google Scholar, 5Laguna Urraca G. Concha López A. Tudela Patón M.P. Verruciform xanthoma of the penis.Actas Urol Esp. 1990; 14 ([in Spanish]): 210-213PubMed Google Scholar Histologically, it is characterized by a dense histiocytic infiltrate within the papillary dermis occasionally extending into the reticular dermis and subcutaneous tissue. The cellular infiltrate includes spindle cells, giant cells (typically of the Touton type), lymphocytes, eosinophils, and neutrophils. Immunohistochemical studies are positive for vimentin, CD68, and factor XIIIa, and negative for S100, CD34, and CD1a. To our knowledge, our case represents the fourth report of XG occurring in the penis of an adult. Xanthogranuloma should be considered in the differential diagnosis of solitary lesions involving the penis.