Idiopathic granulomatous mastitis: A report of three cases

Idiopathic granulomatous mastitis: A report of three cases

European Journal of Radiology Extra 56 (2005) 1–5 Idiopathic granulomatous mastitis: A report of three cases Aysin Pourbagher ∗ , Naime Tokmak, Mir A...

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European Journal of Radiology Extra 56 (2005) 1–5

Idiopathic granulomatous mastitis: A report of three cases Aysin Pourbagher ∗ , Naime Tokmak, Mir Ali Pourbagher, Zafer Koc Department of Radiology, Baskent University, Adana Teaching and Medical Research Center, Yuregir 01250, Adana, Turkey Received 12 May 2005; accepted 20 July 2005

Abstract This article describes the clinical presentation and mammographic, gray-scale and color Doppler sonographic findings for three women with idiopathic granulomatous mastitis. In each case, a complete history was obtained, a thorough physical examination was performed, and routine laboratory tests were done. Two of the three lumps were tender at the time of presentation; the third was not painful. All three patients had diabetes mellitus. Mammography was done in two cases, and both showed asymmetric density in the area of the palpable mass. Ultrasonography showed multiple hypoechoic lesions in two patients and intraductal papillomatous lesions in one patient. In two cases, there was increased vascularization within and around the lesions. Radiologic findings in cases of idiopathic granulomatous mastitis are non-specific; histopathologic analysis is necessary for a definitive diagnosis. © 2005 Elsevier Ireland Ltd. All rights reserved. Keywords: Idiopathic granulomatous mastitis; Mammography; Ultrasonography

1. Introduction Idiopathic granulomatous mastitis is a rare, benign, chronic inflammatory disease of the breast [1]. The clinical and radiological features of this condition can mimic breast carcinoma. Its etiology is unknown [2]. In this article, we report and discuss the clinical and radiologic findings for three patients with idiopathic granulomatous mastitis who also had diabetes mellitus.

2. Case 1 A 33-year-old woman presented with a tender lump in her right breast that had been present for 2 months. She had had three vaginal deliveries, and the most recent had been 5 years prior to presentation. The patient had used oral contraceptives for 2 years but had discontinued these after her second pregnancy. She had been diagnosed with diabetes mellitus 3 years prior to presentation, and had been on oral antidi-

abetic therapy ever since. Physical examination revealed a well-circumscribed, firm but mobile 2 cm × 2 cm mass in the upper outer quadrant of the patient’s right breast. The lesion was painful on palpation. Results of standard laboratory analyses (excluding blood glucose level) were normal. Owing to the patient’s young age, only mediolateral breast radiographs were obtained. Mammography showed asymmetric density in the upper outer quadrant of the right breast. No discrete mass or suspicious microcalcifications were visible (Fig. 1A). Ultrasonography (US) showed multiple, heterogeneous, hypoechoic lesions with irregular contours and posterior acoustic shadowing (Fig. 1B). There was skin thickening over the lesion area and ipsilateral multiple axillary lymphadenopathy was evident as well. Power Doppler US revealed increased vascularization within and around the lesions (Fig. 1C). The radiologic findings suggested mastitis. Histopathological examination of a biopsy specimen identified the condition as idiopathic granulomatous mastitis.

3. Case 2 ∗

Corresponding author. Tel.: +90 322 327 2727; fax: +90 322 327 1270. E-mail addresses: [email protected] (A. Pourbagher), [email protected] (N. Tokmak), [email protected] (M.A. Pourbagher), [email protected] (Z. Koc). 1571-4675/$ – see front matter © 2005 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ejrex.2005.07.013

A 29-year-old woman presented with a very tender and erythematous left breast that had a palpable mass with sinus tracts to the skin. She had first noted the abnormalities 2

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Fig. 1. Mediolateral oblique mammography (A), sonography (B), and Power Doppler sonography of the right breast of a 33-year-old woman: (A) note the irregular density in the upper outer quadrant of the breast; (B) ultrasonography shows an irregular, non-homogeneous, hypoechogenic lesion with posterior acoustic enhancement in the same region; (C) Power Doppler ultrasonography shows increased vascularization within and around the lesions.

months earlier. The patient had two deliveries (the last one, 2 years prior to presentation) and she had a history of oral contraceptive use. She had been diagnosed with diabetes mellitus 2 years prior to presentation. Physical examination revealed a painful, firm, immobile, 4 cm × 2 cm mass in the

upper outer quadrant of the left breast. The overlying skin showed signs of inflammation. No axillary nodes were palpable. The only abnormal finding on standard laboratory testing was elevated blood glucose. Owing to the patient’s age and the extreme tenderness of her breast, mammography was not

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Fig. 2. (A) Sonography of the left breast of a 29-year-old woman shows a solid, lobular, homogeneous, hypoechoic lesion with focal posterior acoustic enhancement; (B) Power Doppler ultrasonography shows increased vascularization within the lesions.

performed. US showed multiple solid hypoechoic lesions measuring 12–32 mm diameter in the retroareolar region, as well as hyperechoic intraductal foci and skin thickening (Fig. 2A). Power Doppler US showed increased vascularization within the lesions (Fig. 2B). The radiologic findings suggested mastitis. Histopathological examination revealed idiopathic granulomatous mastitis.

4. Case 3 A 46-year-old woman presented with a non-tender lump in her left breast and bloody discharge from the nipple. Her most recent delivery had been 20 years prior to presenta-

tion. There was no history of oral contraceptive use. The patient had been diagnosed with diabetes mellitus 4 years earlier. Physical examination revealed a painless, mobile mass encompassing the upper outer quadrant and periareolar region of the left breast. The overlying skin showed no signs of inflammation, and no axillary lymph nodes were palpable. Mammography showed asymmetric density in the area of the palpable mass. The abnormal area had no distinct margins and contained no calcifications (Fig. 3A). US showed ductal ectasia and multiple solid, ovoid, hyperechoic lesions of 11–17 mm diameter inside the ectatic ducts in the retroareolar region (Fig. 3B). These solid lesions suggested intraductal papilloma. Color Doppler US revealed no apparent vascular structures within these lesions.

Fig. 3. Mediolateral oblique mammography (A) and sonography (B) of the left breast of a 46-year-old woman: (A) observe the asymmetric density encompassing the upper quadrant and retroareolar region of the breast; (B) sonography shows ovoid hyperechoic lesions within an ectatic duct.

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Examination of a biopsy revealed idiopathic granulomatous mastitis.

5. Discussion Granulomatous mastitis, also called granulomatous lobular mastitis or granulomatous lobulitis, is an uncommon chronic inflammatory disease of the breast [1]. The etiology of this disorder is unknown, but the clinical and radiological features can mimic breast carcinoma. To avoid unnecessary mastectomies, it is vital that surgeons, pathologists and radiologists be aware of this condition [2]. Granulomatous mastitis was first described as a specific entity in 1972 by Kessler and Wolloch [3]. Since then, there has been extensive documentation of the clinical and pathological findings of idiopathic granulomatous mastitis, but only a few articles have described imaging findings, including mammography and gray-scale and color Doppler US data [1,4–10]. Most patients who develop this form of mastitis are of reproductive age, ranging from 17 to 42 years (mean, 32 years) [4,7]. Our 46-year-old patient was outside this range. Granulomatous mastitis typically appears during pregnancy or lactation [11], or within 6 years of the most recent pregnancy, but it can develop up to 15 years postpartum [1]. One of the patients in our series (the oldest woman) developed lesions 20 years after her last pregnancy. The authors of a review of 49 granulomatous mastitis cases reported that one-third of the affected women had previously used oral contraceptives [10]. In line with this, two of our three cases featured a history of oral contraceptive use. The most common clinical presentation of idiopathic granulomatous mastitis is a unilateral, firm, discrete breast mass, often accompanied by inflammation of the overlying skin [11]. Most cases are unilateral, and regional lymphadenopathy is detected in up to 15% of patients [4,7]. In two of our cases, the affected breast was edematous and the skin overlying the lesion area was thickened. Only one of the three women exhibited ipsilateral axillary lymphadenopathy. As noted, the cause of granulomatous mastitis is still unclear. Autoimmune disease, prior use of oral contraceptives, and reaction to childbirth are some of the proposed etiological factors [1,2,7]. All three of our patients had diabetes mellitus, but no link with this disease has been documented to date. Our observed connection with diabetes may be coincidental, or diabetes mellitus may actually predispose women to granulomatous mastitis. Previous reports have provided no laboratory findings that relate to this possible association. It would be valuable to investigate this potential link in future studies. Han et al. [1], Yilmaz et al. [7], and Memis et al. [8] documented the imaging findings of idiopathic granulomatous mastitis in 9, 12, and 15 women, respectively. In these cases, mammography revealed normal appearance or asymmetric density or a mass. The most common mammography finding was asymmetric density. The authors noted that the abnor-

mality was more obvious when images were compared to those of the contralateral breast. None of the affected breast regions featured distinct margins or mass effect, and none of the lesions caused breast distortion or contained microcalcifications. Some reports have described multiple, small, ill-defined masses within the breast parenchyma [1,7,8,12]. The mammography findings in our three cases were similar to the non-specific ones documented previously. The sonographic appearances of idiopathic granulomatous mastitis are classified in five categories: normal; mass lesion; focal area different from surrounding normal breast tissue; multiple, relatively well-circumscribed, heterogeneous tubular or nodular hypoechoic lesions; or irregular hypoechoic lesions with focal posterior shadowing [1,7,8]. Memis et al. noted nodular echogenic areas at retroareolar and periareolar locations in two of their 15 cases [8]. We observed the same in one of our patients. Our other two patients had similar gray-scale US and color Doppler US findings: multiple solid hypoechoic lesions and increased vascularity of the lesions and surrounding tissue. According to the literature, the typical histologic appearance of granulomatous mastitis is non-caseating granuloma formation within a predominantly lobular distribution of inflammatory infiltrate. The infiltrate is composed of histiocytes, a few polymorphonuclear leukocytes, and foreignbody and Langerhans’-type multinucleate giant cells. Special staining for bacteria, acid-fast bacilli, and fungi is negative [1,7,13]. There is no ideal treatment for idiopathic granulomatous mastitis, but the most common approach is wide local excision with or without steroid therapy [2]. Recurrence, fistula formation, and secondary infection are well-known complications of this condition [11]. In conclusion, granulomatous mastitis is a rare inflammatory disease of the breast that can mimic breast carcinoma. Accurate preoperative diagnosis is often difficult, and mammography and color Doppler US are of minimal value for ruling out differential diagnoses. Gray-scale US provides more specific findings that help identify granulomatous mastitis, but histopathologic analysis is necessary for a definitive diagnosis. It would be interesting to examine the possible relationship between granulomatous mastitis and diabetes mellitus in larger series of patients.

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