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A case of eruptive syringoma in a vitiligo patient
Journal of the Saudi Society of Dermatology & Dermatologic Surgery (2011) 15, 71–72
King Saud University
Journal of the Saudi Society of Dermatology & Dermatologic Surgery www.ksu.edu.sa www.jssdds.org www.sciencedirect.com
CASE REPORT
A case of eruptive syringoma in a vitiligo patient Omar M. Alakloby *, Nadya Al-Faraidy University of Dammam, King Fahd Hospital of the University, Al-Khobar, Saudi Arabia Received 1 March 2011; accepted 1 April 2011 Available online 2 June 2011
KEYWORDS Crohn’s disease; Metronidazole; Saudi Arabia
Abstract Eruptive syringoma is a rare adnexal tumor characterized by a flesh-colored papules that occurs in crops mainly on the anterior surfaces of the body. The lesions are benign, and treatment options are generally unsatisfactory. The case of a 21-year-old women with a 1-year history of eruptive syringoma and vitiligo of 2 years duration is presented. ª 2011 King Saud University. Production and hosting by Elsevier B.V. All rights reserved.
1. Introduction Syringoma presents as flesh to brown colored papules mainly around the eyes and lips and occasionally on other sites of the body. Eruptive syringoma is a rare clinical presentation of this benign tumor of the sweat glands. It was recently suggested to be hyperplastic inflammatory response and has been reported in association with diabetes mellitus and some neoplastic conditions. * Corresponding author. Address: University of Dammam, College of Medicine, King Fahd Hospital of the University, P.O. Box 40130, Al-Khobar 31952, Saudi Arabia. Tel.: +966 3 8580793; mobile: +966 504814962; fax: +966 3 8595658. E-mail address: [email protected] (O.M. Alakloby). 2210-836X ª 2011 King Saud University. Production and hosting by Elsevier B.V. All rights reserved. Peer review under responsibility of King Saud University. doi:10.1016/j.jssdds.2011.04.003
Production and hosting by Elsevier
We present a case of eruptive syringoma in a vitiligo patient. No relationship between these two skin disorders has been reported so far. 2. Case report Twenty-one year old woman presented to Dermatology Clinic on August 2006 with a history of flesh colored, asymptomatic papules of 1 year duration that initially appeared over the infraorbital region gradually involving the entire face, neck and with larger brown lesions scattered on the chest and forearms. She is a known case of vitiligo since 2 years. Her younger sister has recently been experiencing similar flesh colored lesions around the eyes. On examination, the face and lateral aspects of the neck showed flesh colored asymptomatic papules (Fig. 1). Chest and anterior forearms showed scattered brown papules, with a few plaques of 3–4 mm size (Fig. 2). Depigmented macules of vitiligo were distributed over the elbows, knees and ankles, as well as on a few scars on the arms, but patient did not allow photographs to be taken for vitiligo. Histopathology of the biopsy taken from the chest lesion showed dilated cystic spaces lined by two layers of cuboidal cells, with comma like tails giving the characteristic tadpole
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O.M. Alakloby, N. Al-Faraidy
Figure 2 Papules and plaques of syringoma on the chest wall (breast area). Figure 1 Flesh colored papules on the face mainly on the infraorbital region.
Figure 3 Hematoxilin-eosin stained section with different magnifications showing collections of cystic ducts and some epithelial cords with comma-like tails.
appearance, with epithelial strands of similar cells in a dense fibrous stroma (Fig. 3a–c). 3. Discussion Eruptive syringoma is a rare clinical variant with only few reports in world literature first described by Jacquet and Darier (1887). Recently, some authors claimed that it was a hyperplastic response of the eccrine duct to an inflammatory reaction rather than a true adnexal tumor and proposed the term ‘‘syringomatous dermatitis’’ (Guitart et al., 2003; Garrido-Ruiz et al., 2008). Friedman and Butler proposed a classification according to clinical features and associations. This consisted of four principal clinical variants of syringoma: a localized form, a familial form, a form associated with Down syndrome, and a generalized form that encompasses both multiple and eruptive syringoma (Friedman and Butler, 1987). Lesions are more common in women than men, in Japanese people, and patients with Down syndrome (Patrizi et al., 1998; Butterworth et al., 1964). Patients with diabetes mellitus may present with a histological variant known as clear-cell syringoma (Ambrojo et al., 1989). In one reported case, it was associated with carcinoid tumor (Berbis et al., 1989). Eruptive syringoma may mimic multiple trichoepithelioma, flat warts, xanthomas, eruptive vellus hair cysts, papular dermatoses, pseudoxanthoma elasticum, hydracystoma, milia and sebaceous hyperplasia (Kumaran and Kanwar, 2005). To the best of our knowledge, a link between vitiligo and eruptive syringoma has not been reported in the literature. Considering the family history of similar eruption, and auto-
immune disease, we presented this case to propose a possible immunogenetic factors influencing the coexistence of both syringoma and vitiligo in the same patient and syringoma in another family member.
References Ambrojo, P., Requena Caballero, L., Aguilar Martinez, L., et al., 1989. Clear-cell syringoma: microscopy study. Dermatologica 178, 164–166. Berbis, P., Fabre, J.F., Jancovic, E., et al., 1989. Late-onset syringomas of the upper extremities associated with carcinoid tumor. Arch. Dermatol. Venereol. 125, 848–849. Butterworth, T. et al., 1964. Syringomas and mongolism. Arch. Dermatol. 90, 483. Friedman, S.J., Butler, D.F., 1987. Syringoma presenting as milia. J. Am. Acad. Dermatol. 16, 310. Garrido-Ruiz, M.C., Enguita, A.B., Navas, R., et al., 2008. Eruptive syringoma developed over a waxing skin area. Am. J. Dermatopathol. 30 (4), 377–380. Guitart, J., Rosenbaum, M.M., Requena, L., 2003. ‘‘Eruptive syringoma’’: a misnomer for a reactive eccrine glandular ductal proliferation. J. Cut. Pathol. 30, 202–205. Jacquet, L., Darier, J., 1887. Hiydrade´nomes e´ruptifs, e´pithe´liomes ade´noides des glandes sudoripares ou ade´nomes sudoripares. Ann. Dermatol. Syph. 8, 317–323. Kumaran, M.S., Kanwar, A., 2005. Multiple syringomas of the forehead and scalp: an unusual presentation. Indian J. Dermatol. 50 (3), 171–172. Patrizi, A., Neri, I., Marzaduri, S., et al., 1998. Syringoma: a review of twenty-nine cases. Acta Derm. Venereol. (Stockh) 78, 460– 462.
King Saud University
Journal of the Saudi Society of Dermatology & Dermatologic Surgery www.ksu.edu.sa www.jssdds.org www.sciencedirect.com
CASE REPORT
A case of eruptive syringoma in a vitiligo patient Omar M. Alakloby *, Nadya Al-Faraidy University of Dammam, King Fahd Hospital of the University, Al-Khobar, Saudi Arabia Received 1 March 2011; accepted 1 April 2011 Available online 2 June 2011
KEYWORDS Crohn’s disease; Metronidazole; Saudi Arabia
Abstract Eruptive syringoma is a rare adnexal tumor characterized by a flesh-colored papules that occurs in crops mainly on the anterior surfaces of the body. The lesions are benign, and treatment options are generally unsatisfactory. The case of a 21-year-old women with a 1-year history of eruptive syringoma and vitiligo of 2 years duration is presented. ª 2011 King Saud University. Production and hosting by Elsevier B.V. All rights reserved.
1. Introduction Syringoma presents as flesh to brown colored papules mainly around the eyes and lips and occasionally on other sites of the body. Eruptive syringoma is a rare clinical presentation of this benign tumor of the sweat glands. It was recently suggested to be hyperplastic inflammatory response and has been reported in association with diabetes mellitus and some neoplastic conditions. * Corresponding author. Address: University of Dammam, College of Medicine, King Fahd Hospital of the University, P.O. Box 40130, Al-Khobar 31952, Saudi Arabia. Tel.: +966 3 8580793; mobile: +966 504814962; fax: +966 3 8595658. E-mail address: [email protected] (O.M. Alakloby). 2210-836X ª 2011 King Saud University. Production and hosting by Elsevier B.V. All rights reserved. Peer review under responsibility of King Saud University. doi:10.1016/j.jssdds.2011.04.003
Production and hosting by Elsevier
We present a case of eruptive syringoma in a vitiligo patient. No relationship between these two skin disorders has been reported so far. 2. Case report Twenty-one year old woman presented to Dermatology Clinic on August 2006 with a history of flesh colored, asymptomatic papules of 1 year duration that initially appeared over the infraorbital region gradually involving the entire face, neck and with larger brown lesions scattered on the chest and forearms. She is a known case of vitiligo since 2 years. Her younger sister has recently been experiencing similar flesh colored lesions around the eyes. On examination, the face and lateral aspects of the neck showed flesh colored asymptomatic papules (Fig. 1). Chest and anterior forearms showed scattered brown papules, with a few plaques of 3–4 mm size (Fig. 2). Depigmented macules of vitiligo were distributed over the elbows, knees and ankles, as well as on a few scars on the arms, but patient did not allow photographs to be taken for vitiligo. Histopathology of the biopsy taken from the chest lesion showed dilated cystic spaces lined by two layers of cuboidal cells, with comma like tails giving the characteristic tadpole
72
O.M. Alakloby, N. Al-Faraidy
Figure 2 Papules and plaques of syringoma on the chest wall (breast area). Figure 1 Flesh colored papules on the face mainly on the infraorbital region.
Figure 3 Hematoxilin-eosin stained section with different magnifications showing collections of cystic ducts and some epithelial cords with comma-like tails.
appearance, with epithelial strands of similar cells in a dense fibrous stroma (Fig. 3a–c). 3. Discussion Eruptive syringoma is a rare clinical variant with only few reports in world literature first described by Jacquet and Darier (1887). Recently, some authors claimed that it was a hyperplastic response of the eccrine duct to an inflammatory reaction rather than a true adnexal tumor and proposed the term ‘‘syringomatous dermatitis’’ (Guitart et al., 2003; Garrido-Ruiz et al., 2008). Friedman and Butler proposed a classification according to clinical features and associations. This consisted of four principal clinical variants of syringoma: a localized form, a familial form, a form associated with Down syndrome, and a generalized form that encompasses both multiple and eruptive syringoma (Friedman and Butler, 1987). Lesions are more common in women than men, in Japanese people, and patients with Down syndrome (Patrizi et al., 1998; Butterworth et al., 1964). Patients with diabetes mellitus may present with a histological variant known as clear-cell syringoma (Ambrojo et al., 1989). In one reported case, it was associated with carcinoid tumor (Berbis et al., 1989). Eruptive syringoma may mimic multiple trichoepithelioma, flat warts, xanthomas, eruptive vellus hair cysts, papular dermatoses, pseudoxanthoma elasticum, hydracystoma, milia and sebaceous hyperplasia (Kumaran and Kanwar, 2005). To the best of our knowledge, a link between vitiligo and eruptive syringoma has not been reported in the literature. Considering the family history of similar eruption, and auto-
immune disease, we presented this case to propose a possible immunogenetic factors influencing the coexistence of both syringoma and vitiligo in the same patient and syringoma in another family member.
References Ambrojo, P., Requena Caballero, L., Aguilar Martinez, L., et al., 1989. Clear-cell syringoma: microscopy study. Dermatologica 178, 164–166. Berbis, P., Fabre, J.F., Jancovic, E., et al., 1989. Late-onset syringomas of the upper extremities associated with carcinoid tumor. Arch. Dermatol. Venereol. 125, 848–849. Butterworth, T. et al., 1964. Syringomas and mongolism. Arch. Dermatol. 90, 483. Friedman, S.J., Butler, D.F., 1987. Syringoma presenting as milia. J. Am. Acad. Dermatol. 16, 310. Garrido-Ruiz, M.C., Enguita, A.B., Navas, R., et al., 2008. Eruptive syringoma developed over a waxing skin area. Am. J. Dermatopathol. 30 (4), 377–380. Guitart, J., Rosenbaum, M.M., Requena, L., 2003. ‘‘Eruptive syringoma’’: a misnomer for a reactive eccrine glandular ductal proliferation. J. Cut. Pathol. 30, 202–205. Jacquet, L., Darier, J., 1887. Hiydrade´nomes e´ruptifs, e´pithe´liomes ade´noides des glandes sudoripares ou ade´nomes sudoripares. Ann. Dermatol. Syph. 8, 317–323. Kumaran, M.S., Kanwar, A., 2005. Multiple syringomas of the forehead and scalp: an unusual presentation. Indian J. Dermatol. 50 (3), 171–172. Patrizi, A., Neri, I., Marzaduri, S., et al., 1998. Syringoma: a review of twenty-nine cases. Acta Derm. Venereol. (Stockh) 78, 460– 462.