Recurrent syringomatous adenoma of the nipple following a misdiagnosis: A case report

Syringomatous adenoma of the nipple: A case series and systematic review

“…SANs are rare breast lesions. Although it is known to not metastasize, it can recur if not excised to negative margins 3 . To our knowledge, about little over 40 cases of SAN have been reported in the literature.…”

“…Imaging modalities such as mammography and ultrasound are not as specific and needle biopsies rarely assist in diagnosis, with only 7.1% (2/28) of cases seen during literature review demonstrated typical characteristics of SAN preoperatively. Among cases reviewed, most needle biopsy results were uncertain or misdiagnosed as low‐grade adenosquamous carcinoma or ductal hyperplasia 3,4,9 . Due to this difficulty, patients are usually referred for excisional biopsy or surgical excision to achieve a diagnosis.…”

“…The series demonstrate typical pathological finding of characteristic comma shape, which stems from keratinous cysts along duct-like structures composed of multilayered epithelial cells in a fibrotic stroma invading smooth muscle bundle. 3 Immunohistochemistry such as p63 may be used to demonstrate intact myoepithelium. 9 While there are multiple causes of nipple shape changes, SAN should be considered in the differential diagnoses when initial diagnostic workup reveals common pathological features.…”

“…Syringomatous adenoma of the nipple (SAN) was first described in 1983 by Rosen 1 . It is widely believed that these tumors arise from sweat glands, specifically within the nipple‐areolar complex 1–3 . Some of the presentations include mass associated with pain, itching, ulceration, and nipple discharge or inversion 4 .…”

“…Imaging tools such as mammogram and ultrasound are not specific to distinguish SAN from other breast etiologies. Histologically, they are known to be glandular structures surrounded by myoepithelial layer which infiltrates the smooth muscle stroma containing keratinous cysts, providing a characteristic comma appearance in a dense stromal background 1,3 . Achieving the definitive diagnosis of this pathology in the preoperative setting is still unclear to date; thus, most patients undergo surgical excision.…”