Diabetic Mastopathy in Long Standing Type I Diabetic Mellitus; A Case Report

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1 CASE REPORT J Surg Ultrasound 2017;4:25-29 JSU Journal of Surgical Ultrasound Diabetic Mastopathy in Long Standing Type I Diabetic Mellitus; A Case Report Ra Mi Kim, Hunkyoung Lee 1, Heeboong Park 2 Kangnam Womens Hospital, Suwon, 1 Department of Pathology, Ewha Medical Laboratory, Seoul, 2 Park Surgical Clinic, Suwon, Korea Received February 26, 2017 Revised March 27, 2017 Accepted March 27, 2017 Correspondence to: Heeboong Park Park Surgical Clinic, Hyowon Road 256-7, Suwon 16471, Korea Tel: Fax: dr@pabc.net Diabetic mastopathy is occasionally confused with breast carcinoma radiologically. Diabetic mastopathy is an unusual fibrous disease of the breast that is associated with type 1 diabetes mellitus of long duration. This study was conducted to evaluate the imaging features of diabetic mastopathy and the role of sono-guided biopsy for definite diagnosis with the goal of avoiding unnecessary surgical procedures for its treatment. Keywords: Diabetes mellitus, Mastopathy, Periductal lymphocytic infiltration, Keloid stroma INTRODUCTION Diabetic mastopathy, an uncommon form for lymphocytic mastitis and stromal fibrosis, typically occurs in premenopausal female population suffering from type 1 diabetes insulin-dependent for long duration.(1,2) The prevalence of diabetic mastopathy has been found to be < 1% of benign breast diseases, but the prevalence can range from 0.6% to 13% in type 1 diabetes.(2) The presentation of diabetic mastopathy is clinically as a large, painless, hard breast mass that is usually clinically indistinguishable from breast cancer. It is not the type of change that is typically associated with an increased risk of breast cancer. Radiologically, diabetic mastopathy is difficult to diagnose because it dose not have typical characteristics.(1) Therefore, the aim of this case report is to evaluate the imaging characteristics of diabetic mastopathy and the necessary role of tissue obtain procedure for diagnosis, such as image guided biposy or vaccum - assisted breast biopsy (VABB). We report a case of diabetic mastopathy in a middle aged premenopausal woman with long-standing type I diabetic mellitus. CASE REPORT A 40-year-old woman, with a painless, irregular, firm mass in her right breast, was presented to the surgical department of breast. The patient was affected from diabetes type I insulin dependent and has been treated with insulin injection for 10 years; she was asymptomatic, apparently well-being, and her vital parameters were normal. Journal of Surgical Ultrasound is an Open Access Journal. All articles are distributed under the terms of the Creative Commons Attribution Non-Commercial License ( which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. Copyright c 2017 by The Korean Surgical Ultrasound Society ISSN

J Surg Ultrasound Vol. 4, No. 1, 2017 Fig. 1. Mammography showed a moderately dense, benign macrocalcification at bilateral breast.

Ultrasound demonstrated an ill-defined upper central region of hypoechogenicity and marked intense posterior shadowing. This lesion shows no vascularity.

There was no skin dimpling, peaud'orange edema, nor discharge from nipples. There was no clinical evidence of axillary or supraclavicular lymphadenopathy.

There was no discrete nodule, suspicious microcalcification, Fig. 3. Vaguely nodular arrangement of epithelioid fibroblasts within sclerotic stroma. nor axillary lymphadenopathy (Fig. 1).

2 J Surg Ultrasound Vol. 4, No. 1, 2017 Fig. 1. Mammography showed a moderately dense, benign macrocalcification at bilateral breast. There was no discrete nodule, suspicious microcalcification, nor axillary lymphadenopathy. Fig. 2. Ultrasound demonstrated an ill-defined upper central region of hypoechogenicity and marked intense posterior shadowing. This lesion shows no vascularity. Physical examination of the breasts revealed a five centimeter soft to hard mass in the upper central region of the right breast. There was no skin dimpling, peaud'orange edema, nor discharge from nipples. There was no clinical evidence of axillary or supraclavicular lymphadenopathy. Mammography and breast sonography were performed. Mammography showed a moderately dense, benign macrocalcification at bilateral breast. There was no discrete nodule, suspicious microcalcification, Fig. 3. Vaguely nodular arrangement of epithelioid fibroblasts within sclerotic stroma. nor axillary lymphadenopathy (Fig. 1). Ultrasound demonstrated an ill-defined upper central region of hypoechogenicity and marked intense posterior shadowing. It was difficult to measure the definite size, because of the strong posterior shadowing. Approximately, the size of the mass was cm. It also showed irregular shape, spiculated margin and no calcification. It was indistinguishable from that of breast carcinoma (Fig. 2). However, there was no vascularity unlike malignancy. We performed ultrasound guided core biopsy for diagnosis, and the lesion fibrocystic change was 26

Ra Mi Kim, et al.: Periductal Lymphocytic Infiltration in Breast identified by histology.

attain much more tissue. After VABB (Bexcore R, Medicalpark, Korea), microscopic examination revealed breast tissue with no evidence of malignancy again.

In addition, there was a dense chronic periductal and perilobular inflammatory cell infiltration (Fig. 4), associated with lymphoid follicle formation (Fig.

After 3 months later, we checked ultrasonography again. The size was slightly increased, however, no change of shape and other features.

6 months later, we checked again with physical examination and ultrasonography. There was no significant interval change and other abnormal lesions (Fig. 6).

3 Ra Mi Kim, et al.: Periductal Lymphocytic Infiltration in Breast identified by histology. On the basis of inconclusive result and with consideration to the clinical features suspicious for neoplastic disease, we decided to perform a VABB to attain much more tissue. After VABB (Bexcore R, Medicalpark, Korea), microscopic examination revealed breast tissue with no evidence of malignancy again. There was marked fibrosis and keloidlike hyalinization of the interlobular stroma (Fig. 3). In addition, there was a dense chronic periductal and perilobular inflammatory cell infiltration (Fig. 4), associated with lymphoid follicle formation (Fig. 5). These features were consisted with diabetic mastopathy, and with a diagnosis of diabetic mastopathy, further surgical wide excision was not necessary. After 3 months later, we checked ultrasonography again. The size was slightly increased, however, no change of shape and other features. We recommended her to continue regular breast surveillance every 6 months, clinical evaluations, and imaging studies. 6 months later, we checked again with physical examination and ultrasonography. There was no significant interval change and other abnormal lesions (Fig. 6). Fig. 4. Histological assessment of the VABB is consistent with lymphocytic mastitis. Periductal lymphocytic infiltration around ducts and lobules can be seen. Fig. 6. There was no significant change in ultrasonography after six months. Fig. 5. Mononuclear inflammatory cells aggregate around duct and into the lobule and also around vascular spaces. 27

4 J Surg Ultrasound Vol. 4, No. 1, 2017 DISCUSSION An association between diabetes mellitus and fibrous breast benign disease was reported initially in 1984 by Soler and Khardori, who described 12 patients with longstanding type 1 diabetes mellitus, multiple diabetic complications, and palpable breast masses.(2) Clinically this change is indistinguishable through physical or radiographic findings from breast malignancy.(1-3) The pathogenesis is not completely understood. Three dominant mechanisms are known: 1. Exogenous insulin may result in an inflammatory or immunologic reaction. 2. Hyperglycemia results in abnormal extracellular matrix accumulation. 3. Auto-immunity link with connective tissue diseases.(4) The physical examination is that of hard, irregular, easily movable, discrete, painless breast lumps. It can be solitary or multiple, and unilateral or bilateral. Some patients can have nonpalpable lesion.(5) Mammography generally does not demonstrate a distinct mass, spiculation or microcalcification. Most mammograms are reported as dense breast parenchyme, dense glandular tissue, asymmetric densities and parenchymal deformity.(1,6) The ultrasound findings varied from case to case, however, the most typical ultrasound findings observed for diabetic mastopathy were an irregularly shaped, hypo-echoic mass with posterior shadowing due to the fibrotic nature of the masses. The majority of diabetic mastopathy lesions corresponded to a diagnosis of BI-RADS category 4.(1,3,7) Some studies demonstrated absence of Doppler signal on color flow.(4) Most cancers show increased vascularity, while the absence of it is more frequently associated with benign conditions. Magnetic resonance imaging (MRI) may reveal nonspecific diffuse stromal enhancement without a focal enhancing mass.(4) There is usually increased focal uptake of 18-flourodeoxyglucose on PET-CT scans, but this is also a nonspecific finding.(8) Despite the thorough clinical and radiologic assessment, it is still difficult to differentiate diabetic mastopathy from breast carcinoma. The definitive diagnosis depends on histologic assessment. Fine needle aspiration is not recommended because the firm resistance experienced during the back and forth motions of the needle is stronger than that of benign and malignant breast conditions, making cytological diagnosis difficult. Moreover, there is insufficient cellular yield for diagnostic purposes in 50-70% of cases.(9,10) Ultrasound guided Core biopsy or VABB is the preferred method to diagnose the condition.(6) Histopathologic finding that allowed us to make the diagnosis of diabetic mastopathy was characterized by lymphocytic lobulitis with glandular atrophy, perivascular infiltrate mainly represented by small lymphocytes mixed with some plasma cells and diffuse fibrosis.(11,12) Physicians should be aware of the association of long-standing diabetes mellitus with the development of benign fibro-inflammatory breast lesions when managing these in premenopausal women. We outline the constellation of findings on clinical examination, medical history, imaging studies, and histo-pathologic examination that are required to make the diagnosis of diabetic mastopathy. Although these breast masses may be recurrent, they are not premalignant. In the appropriate setting, the diagnosis can be made by core biopsy or VABB, avoiding unnecessary surgeries in patients with multiple, bilateral, or recurrent lesions. Diabetic mastopathy is considered the complication of long-standing diabetes mellitus. It can confused with malignacy clinically and radiologically. We should avoid unnecessary surgical excision through exact diagnosis. 28

5 Ra Mi Kim, et al.: Periductal Lymphocytic Infiltration in Breast REFERENCES 1. Kim J, Kim EK, Kim MJ, Moon H, Yoon JH. Diabetic mastopathy: imaging features and the role of image-guided biopsy in its diagnosis. Ultrasonography 2016;35: Soler NG, Khardori R. Fibrous disease of the breast, thyroiditis, and cheiroarthropathy in type I diabetes mellitus. Lancet 1984;1: Chan HHL, Lam TPW, Nicholls J, Leong LLY. Lymphocytic mastitis and rheumatoid arthritis. J HK Coll Radiol 2002;5: Meerkotter DA, Rubin G. Diabetic mastopathy: a clinical and radiological challange. SA J Radiol 2010;14: Logan WW, Hoffman NY. Diabetic fibrous breast disease. Radiology 1989;172: Thorncroft K, Forsyth L, Desmond S, Audisio RA. The diagnosis and management of diabetic mastopathy. Breast J 2007;13: Camuto PM, Zetrenne E, Ponn T. Diabetic mastopathy: a report of 5 cases and a review of the literature. Arch Surg 2000;135: Gurion R, Groshar D, Schindel A, Shpilberg O, Raanani P. 18F-fluorodeoxyglucose-Avid mammary mass in a patient with insulin-dependent diabetes mellitus and Hodgkin's lymphoma: relapse or pitfall? Isr Med Assoc J 2006;8: Rollins SD. Fine-needle aspiration cytology of diabetic fibrous mastopathy. Diagn Cytopathol 1993;9: Al-Daheen GAF, Al-Aswad FD, Verma MK. Diabetic mastopathy: a case report. Int J Diabetes Metab 2009; 17: Ghirarduzzi A, Sivelli R, De Simone B, Ambrosini AS, Arcuri MF, Sianesi M. Diabetic mastopathy: case report. Ann Ital Chir 2012;83: Han JH, Kim HJ, Jung WH, Oh KK. Histopathologic findings of mastopathy in diabetes mellitus. The Korean Journal of Pathology 1999;33: Korean J Pathol 1999;33:

Diabetic mastopathy: spectrum of radiological findings.

Diabetic mastopathy: spectrum of radiological findings. Poster No.: C-1519 Congress: ECR 2014 Type: Educational Exhibit Authors: V. Navarro-Aguilar, G. Montoliu, R. M. Viguer Benavent, M. Á. Sánchez Fuster,