Infiltrating syringomatous adenoma of the nipple is a rare, benign lesion with distinct clinical and histological features. Origin from ducts of dermal sweat glands has been postulated. Important differential diagnosis include nipple adenoma, tubular carcinoma and adenosquamous carcinoma. Appropriate local management includes accurate diagnosis and complete excision to avoid local recurrences. This report describes the condition in a 39-year-old parous lady

Idress, Romana

Kayani, Naila

Khan, Shaista Masood

Riaz, Nazia

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eCommons@AKUSection of General Surgery Department of SurgeryJuly 2008Infiltrating syringomatous adenoma of nippleNazia RiazAga Khan UniversityShaista Masood KhanAga Khan UniversityRomana IdreesAga Khan UniversityNaila KayaniAga Khan UniversityFollow this and additional works at: http://ecommons.aku.edu/pakistan_fhs_mc_surg_genPart of the Surgery CommonsRecommended CitationRiaz, N., Khan, S., Idrees, R., Kayani, N. (2008). Infiltrating syringomatous adenoma of nipple. Journal of the College of Physicians andSurgeons Pakistan, 18(7), 438-9.Available at: http://ecommons.aku.edu/pakistan_fhs_mc_surg_gen/11438 Journal of the College of Physicians and Surgeons Pakistan  2008, Vol. 18 (7): 438-439INTRODUCTIONSyringomatous adenoma of the nipple is a rare, distinct,benign though locally infiltrative clinical entity. Only31 cases have been reported to-date.1 Histologically, it issimilar to syringoma, a benign neoplasm originating inthe ducts of the dermal sweat glands located in the lips,axilla, buttocks or minor salivary glands.2 Importantaspects of management involve its recognition anddifferentiation from adenosquamous and tubularcarcinoma with associated risk of local recurrence whenincompletely excised.3,4 We report the case of infiltratingsyringomatous adenoma of the nipple with review ofrelevant literature.CASE REPORTA 39-year-old lady presented with 3 months history ofpainless lump over left nipple. Although not a regularbreast self-examiner, she noticed a recent painlessincrease in size of left nipple without any associatednipple discharge or ulceration. She did not notice anylumps in either breast. There was no significant historyof breast diseases in the past. No risk factors wereidentified for breast cancer except late age at firstdelivery.She initially presented elsewhere where an incisionalbiopsy of the left nipple was done and thehistopathology revealed neoplastic lesion of dermaladnexal origin. On examination, upon refferal toAga Khan University Hospital, Karachi, the left nipplewas enlarged and exuberant, but epithelialized withan indistinct retroareolar palpable mass. Right breastwas normal on examination and there was no regionallymphadenopathy. Mammogram did not reveal anyparenchymal abnormality and a bilateral breastultrasound was normal. The histopathology was alsoreviewed at The Aga Khan University Hospital. Itshowed ductules, tubules and strands of small uniformcells infiltrating into the dermis. Some ducts showed acharacteristic comma-shape. Nuclear pleomorphism,hyperchromasia and lack of mitosis were noted. Thefindings were consistent with syringomatous adenomaof the nipple. In addition to routine haematoxylin and eosin stainedslides, immunohistochemical markers for demonstrationof myoepithelial layer-anti smooth muscle antigen andCD10 were performed. The presence of double layerprovided additional evidence in favour of syringomatousadenoma and excluded other differential of tubularcarcinoma and low-grade adenosquamous carcinoma. She underwent wide excision of the nipple areolacomplex via an elliptical incision. Histopathologicalanalysis revealed widely scattered epithelioid strands inthe dermis with eccrine differentiation and papillaryhyperplastic changes in the epidermis (Figure 1a, b).The findings were consistent with syringomatousadenoma. The lesion was 0.2 cm from the painted deepmargin. She did not develop any recurrence at 6 monthsfollow-up. In view of the unusual nature of this lesion, thepatient was advised long-term follow-up.ABSTRACTInfiltrating syringomatous adenoma of the nipple is a rare, benign lesion with distinct clinical and histological features.Origin from ducts of dermal sweat glands has been postulated. Important differential diagnosis include nipple adenoma,tubular carcinoma and adenosquamous carcinoma. Appropriate local management includes accurate diagnosis andcomplete excision to avoid local recurrences. This report describes the condition in a 39-year-old parous lady.Key words: Infiltrating syringomatous adenoma.   Nipple adenoma.   Benign nipple neoplasm.Department of  Surgery1/Pathology2, The Aga Khan UniversityHospital, Karachi.Correspondence: Dr. Nazia Riaz, 65-A, GF-III,Seaview Apartments, Phase-V Extension, D.H.A, Karachi.E-mail: nazia.riaz@aku.eduReceived  December 11, 2007; accepted  May 14, 2008.Infiltrating Syringomatous Adenoma of Nipple  Nazia Riaz1, Shaista Masood Khan1, Romana Idrees 2 and Naila Kayani2CASE REPORTFigure 1 (a and b):  Histopathology of syringomatous adenoma of the nipplewith increasing magnification showing islands of squamous epitheliuma bDISCUSSIONBreast neoplasms with squamous differentiation arerelatively rare and tumours with specific syringomatousfeatures are sparsely reported in the literature. It wasdescribed as a tumour like proliferation of smallsquamous islands in which no connection could befound between the squamous structures and thesurrounding breast parenchyma.5 The authors interpretedthe lesions as a result of squamous metaplasia arisingfrom pre-existing ductal structures. Clinically andpathologically similar lesions to infiltrating syringomatousadenoma but located away from the nipple in the breastparenchyma were also described by Suster et al.6Historically termed as “adenoma”, the term “syringo-matous adenoma of the nipple” was first coined byRosen in 1983.7 In a clinical and pathological study of11 cases in 1989, Jones et al. introduced the term“infiltrating” to emphasize the locally infiltrative nature ofthis benign but peculiar lesion.4Patients with syringomatous adenoma of the nipplehave been reported to range in age from 11-76 yearswith an average age of 40 years.4,7 Though, there is afemale preponderance, it has been reported in menpresenting with nipple ulceration.8The clinical presentation of infiltrating syringomatousadenoma of the nipple is variable. The commonpresentation is that of a solitary, painful nipple masswhich may cause nipple inversion, discharge oritching.4,7,8 Except one, all reported lesions areunilateral and none have been associated with regionallymphadenopathy. There is no specific radiologicalfeatures characteristic to this unusual condition.Histological and microscopic examination reveals aninfiltrative pattern of epithelial islands that are angulatedor comma-shaped as well as tubular or solid inconfiguration. The glandular lumina are small orobliterated. Squamous metaplasia is usually present invariable proportion of epithelial islands, which have aninconspicuous or absent outer myoepithelial layer. Theepithelial elements often invade into the smooth muscleof the nipple, mimicking invasive carcinoma.7,9It is important to differentiate syringomatous adenomafrom tubular carcinoma and low-grade adenosquamouscarcinoma. The glandular structures of tubularcarcinoma are mostly angulated with open lumina andare composed of single cell population as opposedto syringomatous adenoma, which has a variableamount of squamous metaplasia and has characteristic“comma” or “tad pole” shapes.10 Ductal carcinoma in situis often associated with tubular carcinoma. Low-gradeadenosquamous carcinoma may be impossible todifferentiate from syringomatous adenoma, especially ifinvolving the nipple areola complex.Surgical management has varied from excisional biopsyto radical mastectomy.3 Recurrence has been reportedfrom 25-55% in patients treated initially with some formof local excision to subcutaneous mastectomy.4 Localrecurrence has been attributed to be related toincomplete excision. It is evident from the literature thataxillary dissection is not indicated as no nodal metastasishas been found in patients who had axillary dissection.Distant metastasis is unknown with syringomatousadenoma on follow-up of as long as 24 years.4Based upon this case report and review of the literature,infiltrating syringomatous adenoma is an unusual,benign though locally infiltrative clinical entity. It shouldbe included in the differential diagnosis of the patientswho present with lump in the nipple areola complex.Though non-metastasizing, it may recur locally ifincompletely excised. Hence, complete excision withclear margins seems to be an appropriate therapy andextensive surgical procedures should be avoided.Owing to the rarity of this disease, long-term follow-up iswarranted.REFERENCES1. Oliva VL, Little JV, Carlson GW. Syringomatous adenoma of thenipple -- treatment by central mound resection and oncoplasticreconstruction. Breast J 2008; 14:102-5.2. Carter E, Dyess DL. Infiltrating syringomatous adenoma of thenipple: a case report and 20-year retrospective review. Breast J2004; 10: 443-7.3. Ku J, Bennett RD, Chong KD, Bennett IC. Syringomatousadenoma of the nipple. Breast 2004; 13: 412-5.4. Jones MW, Norris HJ, Synder RC. Infiltrating syringomatousadenoma of the nipple. A clinical and pathological study of 11cases. Am J Surg Pathol 1989; 13:197-201.5. Moran CA, Suster S, Carter D. Benign mixed tumours(pleomorphic adenomas) of the breast. Am J Surg Pathol 1990; 14:913-21.6. Suster S, Moran CA, Hurt MA. Syringomatous squamoustumours of the breast. Cancer 1991; 67:2350-5.7. Rosen PP. Syringomatous adenoma of the nipple. Am J Surg Pathol1983; 7:739-45.8. Coulthard A, Liston J, Young JR. Case report: infiltratingsyringomatous adenoma of the breast: appearances onmammography and ultrasonography. Clin Radiol 1993; 47: 62-4.9. Dahlstorm JE, Tait N, Cranney BG, Jain S. Fine needleaspiration cytology and core biopsy histology in infiltratingsyringomatous adenoma of the breast: a case report. Acta Cytol1999; 43:303-7.10. Schnitt SJ, Connolly JL. Pathology of benign breast disorders.In: Harris JR, Lippman ME, Morrow M, Osborne CK, (edi).Diseases of the breast. 3rd ed. Philadelphia: LippincoH Williams andWilkins; 2004.p.86.Journal of the College of Physicians and Surgeons Pakistan  2008, Vol. 18 (7): 438-439 439Infiltrating syringomatous adenoma of nipplel l l l lOl l l l l

Infiltrating syringomatous adenoma of nipple

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Infiltrating syringomatous adenoma of nipple

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