A Case of Syringomatous Adenoma of the Nipple

Abstract

Syringomatous adenoma of the nipple (SAN) is an extremely rare disease originating in the adnexal gland; it was first reported by Rosen in 1983 [Am J Surg Pathol 1983;7:739–745]. Since then, 34 cases have been reported worldwide. We present the case of a 51-year-old man with SAN, in whom local excision of the tumor was performed. Histologically, the tumor consisted of tubules, ductules and epithelial cell strands, and most of the proliferating ducts presented with a characteristic teardrop or comma-shaped appearance.

Rare benign tumours of the nipple

“…The references of the papers were reviewed and all relevant articles were also examined (total 43). Different entities that were described included: neurofibroma, 5-7 leiomyoma, [8][9][10][11][12][13][14][15][16][17][18] milium, 19,20 florid papillomatosis, 9,[21][22][23][24][25][26][27] syringomatous adenoma, [28][29][30][31][32] nevoid hyperkeratosis, [33][34][35][36][37][38][39][40][41] fibroma, 42,43 pseudolymphoma and [44][45][46][47] hemangioma. 9 Table 1 summarizes the papers for each entity.…”

“…Bulman 8 Doctor et al 9 Nascimento et al 10 Tsujioka et al 11 Pujol et al 12 Velasco et al 13 Kaufman et al 14 Christenson et al 15 Chaudhary et al 16 Ku et al 17 Pavlidis et al 18 Milia Mishra et al 19 Berk et al 20 Florid Papillomatosis Nichols et al 21 Smith et al 22 Doctor et al 9 Perzin et al 23 Miyahara et al 24 Montemarano et al 25 Sugai et al 26 Kijima et al 27 Syringomatous adenoma of the nipple Rosen 28 Coulthard et al 29 Toyoshima et al 30 Wadhwa et al 31 Ichinokawa et al 32 Nevoid hyperkeratosis Levy-Frankel 33 Mold et al 34 Vestey et al 35 Perez-Izquierdo et al 36 D'Souza et al 37 Okan et al 38 Xifra et al 39 Kubota et al 40 Krishnan et al 41 Fibroma of the nipple Iancu et al 42 Parlakgumus et al 43 Pseudolymphoma of the nipple Bafverstedt 44 Ackerman 45 Schmid et al 46 Smyczek-Gargya et al 47 Haemangioma of the nipple Doctor et al 9 reported in the literature until now. [8][9]…”

“…28 According to our review of the literature there have been 35 cases reported worldwide until now. 32 Histologically, the lesion is difficult to distinguish from well-differentiated breast carcinomas, like tubular carcinoma, 30 as it has features between a benign adenoma and a carcinoma. However, presentation and imaging can suggest the more likely diagnosis.…”

“…This is why the term 'infiltrating syringomatous adenoma' has been proposed 29 and it has been called microcystic adenexal carcinoma, sclerosing carcinoma of the sweat ducts and low-grade adenosquamous carcinoma in the literature. 32 The lesion presents as a nipple nodule which may be tender and painful and cause nipple discharge or inversion. Syringomatous adenoma presents usually unilaterally, with only one case of bilateral presentation reported in the literature.…”

Syringomatous adenoma of the nipple: a case report

“…Syringomatous adenoma of the nipple (SAN) is a very rare benign tumor that was first reported by Rosen in 1983 [ 1 ]. SAN shows locally infiltrating growth but does not metastasize [ 2 ]. Nonetheless, in some cases, this tumor may be misdiagnosed as a malignancy owing to its infiltrating growth pattern [ 3 ].…”