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Year : 2018  |  Volume : 11  |  Issue : 4  |  Page : 348-351  

A rare case of xanthogranulomatous mastitis with intraductal papilloma

Department of Pathology, Hospital and Research Center, Dr. D. Y. Patil Medical College, Dr. D. Y. Patil Vidyapeeth, Pune, Maharashtra, India

Date of Web Publication2-Aug-2018

Correspondence Address:
Sunita Arvind Bamanikar
5 and 6, Siddharth Estates, North Avenue, Kalyani Nagar, Pune - 411 006, Maharashtra
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/mjdrdypu.MJDRDYPU_214_17

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Xanthogranulomatous inflammation is rare and has been described most commonly in kidney and gallbladder. It is extremely rare in breast. We report here a case of a 42-year-old female who came with firm painless lump in the left breast. Mammography suggested suspicious neoplastic retroareolar lesion. Fine-needle aspiration (FNA) smears suggested a benign cystic lesion and excision of the cyst was advised. Histopathological examination revealed breast tissue infiltrated with polymorphonuclear leukocytes, lymphoplasmacytic inflammatory cells, few giant cells, and sheets of foamy histiocytes (xanthoma cells) with a focus of intraductal papilloma. A diagnosis of xanthogranulomatous mastitis with intraductal papilloma was made. Chronic inflammatory lesions are known to produce firm lesions which may mimic neoplastic lesion. Definitive diagnosis could not be achieved in FNA smears due to the absence of adequate number of xanthoma cells. Final diagnosis was made on histopathology examination. Xanthogranulomatous mastitis is an extremely rare entity. Clinical presentation is quite variable. Imaging modalities and FNA are inadequate for definitive diagnosis.

Keywords: Breast, inflammation, intraductal papilloma, xanthoma cells

How to cite this article:
Bamanikar SA, Chandanwale SS, Pathak P, Gambhir A, Sheth J. A rare case of xanthogranulomatous mastitis with intraductal papilloma. Med J DY Patil Vidyapeeth 2018;11:348-51

How to cite this URL:
Bamanikar SA, Chandanwale SS, Pathak P, Gambhir A, Sheth J. A rare case of xanthogranulomatous mastitis with intraductal papilloma. Med J DY Patil Vidyapeeth [serial online] 2018 [cited 2020 Oct 25];11:348-51. Available from: https://www.mjdrdypv.org/text.asp?2018/11/4/348/238172

  Introduction Top

Xanthogranulomatous inflammation is a benign pathological condition associated with rare form of chronic inflammation.[1] It has been seen in many organs but most commonly seen in kidney and gallbladder. It is an uncommon finding in the breast.[2] Although etiology of xanthogranulomatous mastitis remains unknown, it has been suggested that obstruction, hemorrhage, inflammation, and hypoxia in the ducts of the breast can be the cause of xanthogranulomatous inflammation. Very few reports have been published in literature on xanthogranulomatous mastitis.

  Case Report Top

A 42-year-old female presented to our institute surgical outpatient department with abnormal mass in left breast of 1-month duration. The mass was initially small and then increased to present size. She had no comorbidities. Initial investigation of the mass with ultrasound revealed irregular hypoechoic area suggestive of an inflammatory lesion. On mammography, approximately 3 cm irregularly shaped, high density in the retroareolar region was seen suspicious of malignancy, and the patient was advised fine-needle aspiration (FNA) cytology. On examination, the lump was tender, slightly firm and measured 4 cm × 3 cm.

FNA cytology smears showed few cyst macrophages and pigment-laden macrophages against a background of numerous foreign body giant cells, few lymphocytes, and spindeloid cells [Figure 1]. Cytological diagnosis of benign cystic lesion was given, and excision of the lump was advised. Specimen received for pathology diagnosis was multiple irregular fragmented bits of yellow-brown tissue, aggregate measuring approximately 4 cc.
Figure 1: Fine-needle aspiration smears show cyst macrophages, pigment-laden macrophages, and lymphocytes (Leishmann, ×400)

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Microscopic examination revealed diffuse nodular aggregates of foamy histiocytes-xanthoma cells intermingled with chronic inflammatory cells and multinucleated giant cells [Figure 2] and [Figure 3]. Also seen was a focus of intraductal papilloma with apocrine metaplasia with areas of fibrosis and fat necrosis [Figure 4]. No epithelioid cells were seen. Ziehl–Neelsen's stain revealed no acid-fast bacilli. Histopathological diagnosis of xanthogranulomatous mastitis with intraductal papilloma was made. The recovery of the patient postsurgery was uneventful.
Figure 2: Photomicrograph showing dense areas of chronic inflammation, fibrosis, and numerous lipid-laden macrophages (xanthoma cells) and multinucleate giant cell (H and E, ×100)

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Figure 3: Multinucleated giant cell (thick arrow) and (thin arrow) xanthoma cells (H and E, ×400)

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Figure 4: Xanthogranulomatous inflammation with a focus of intraductal papilloma with apocrine metaplasia (H and E, ×100)

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  Discussion Top

Although xanthogranulomatous inflammation is an entity affecting several organs, most commonly the kidney and gallbladder,[3],[4] previous studies have reported that xanthogranulomatous mastitis is very uncommon. Shin et al. in 2005 described the first case of mammary presentation of adult-type “juvenile” xanthogranuloma.[5] Hwang et al. reported bilateral xanthogranuloma in breast in 2007.[6] Koo and Jung reviewed 16 cases of xanthogranulomatous mastitis which was associated with multinucleated giant cell reaction in six cases and fat necrosis and duct ectasia with foamy histiocyte aggregate in five cases each. Xanthogranulomatous inflammation is characterized microscopically by the presence of multinucleated giant cells, lipid-laden macrophages (xanthoma cells), and cholesterol crystals. It also shows accumulation of cells including lymphocytes, plasma cells, and some polymorphonuclear leukocytes.[7] The most typical findings in xanthogranulomatous inflammation are interlacing of foamy macrophages and activated plasma cells.[8]

The etiology of xanthogranulomatous inflammation is unknown. The proposed causes of xanthogranulomatous inflammation include obstruction, hemorrhage, inflammation, and local hypoxia. Defective lipid transport, immunological disturbance, and infection by low-virulence organisms are some mechanisms that lead to xanthogranulomatous inflammation.[7] Koo and Jung suggested that the cause of xanthogranulomatous mastitis can be obstruction and rupture of ectatic duct with foamy histiocyte aggregates. Immunohistochemistry shows these histiocytes positive for CD68 and negative for cytokeratin.[2]

Xanthogranulomatous inflammation may or may not be associated with hyperlipidemia.[9] Xanthogranulomatous mastitis occurs in the age group of 26–70 years with mean age being 44 years. The size of the lesion ranges from 0.5 to 4.8 cm, and the left breast is affected more often than the right breast. Gross specimens often reveal a poorly defined nodular mass with an indistinct normal structure. Bourm et al. suggested that xanthogranulomatous processes result in replacement of the organ tissue involved by xanthoma cells; fibrin and collagen deposition leads to tissue destruction.[8] The inflammatory process can invade the tissue and simulate aggressive process with abscess formation.[10] The presence of diffuse inflammation in breast is associated with mass formation mimicking advanced tumors.[10] Chronic inflammatory lesions are known to produce firm lesions which may mimic neoplastic lesion. In our case, definitive diagnosis could not be achieved in FNA smears due to the absence of adequate number of xanthoma cells. Final diagnosis was made on histopathology examination.

Recently, it has been expressed that infectious processes sustained by Gram-positive and Gram-negative bacteria can affect the soft tissue and appear as xanthogranuloma.[11]

Xanthogranulomatous inflammation in breast can occur in cancer regression due to neoadjuvant chemotherapy,[12] as mammary involvement in Erdheim–Chester disease [13] and malakoplakia.[14] Cancer regression in neoadjuvant chemotherapy is characterized by histiocytes, elastosis, inflammation, and fibrosis which is like findings in xanthogranulomatous mastitis and can be differentiated by chemotherapy history. Microscopically, mammary Erdheim–Chester disease shows extensive infiltrates of foamy histiocytes in a background of dense fibrous stroma with a marked loss of mammary ducts and lobules in breast tissue. However, Erdheim–Chester disease is a multiorgan disease, which involves the skeletal system, eyes, lungs, pituitary gland, kidneys, retroperitoneum, liver, spleen, central nervous system, skin, and heart.[15] Breast involvement is very rare. Malakoplakia or pseudoxanthomatous inflammation is characterized by collection of foamy histiocytes and should be differentiated from xanthogranulomatous inflammation. Malakoplakia shows characteristic Michaelis-Gutmann bodies which stain positive for Von Kossa calcium and Prussian blue.[16]

Diagnosis of xanthogranulomatous mastitis on ultrasonographic examination and mammography is not possible. Imaging features are nonspecific, and in most cases, they are misdiagnosed as a neoplastic condition preoperatively. Histopathological examination gives a confirmatory diagnosis.

  Conclusion Top

Xanthogranulomatous mastitis is an extremely rare entity. Clinical presentation is quite variable but recognizing the features of xanthogranulomatous inflammation may have a significant impact on the management of the patient. The differential diagnosis should be considered. Imaging modalities and FNA are inadequate for the diagnosis. Histopathological examination is essential for definitive diagnosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Hussain T, Elahi B, Long E, Mahapatra T, McManus PL, Kneeshaw PJ, et al. Xanthogranulomatous inflammation involving latissimus dorsi donor site and implant breast reconstruction: Case report and literature review. World J Surg Oncol 2012;10:166.  Back to cited text no. 1
Koo JS, Jung W. Xanthogranulomatous mastitis: Clinicopathology and pathological implications. Pathol Int 2009;59:234-40.  Back to cited text no. 2
Benbow EW. Xanthogranulomatous cholecystitis. Br J Surg 1990;77:255-6.  Back to cited text no. 3
Franco V, Aragona F, Genova G, Florena AM, Stella M, Campesi G, et al. Xanthogranulomatous cholecystitis. Histopathological study and classification. Pathol Res Pract 1990;186:383-90.  Back to cited text no. 4
Shin SJ, Scamman W, Gopalan A, Rosen PP. Mammary presentation of adult-type “juvenile” xanthogranuloma. Am J Surg Pathol 2005;29:827-31.  Back to cited text no. 5
Hwang SH, Son EJ, Oh KK, Kim EK, Jung J, Jung WH, et al. Bilateral xanthogranuloma of the breast: Radiologic findings and pathologic correlation. J Ultrasound Med 2007;26:535-7.  Back to cited text no. 6
Antonakopoulos GN, Chapple CR, Newman J, Crocker J, Tudway DC, O'Brien JM, et al. Xanthogranulomatous pyelonephritis. A reappraisal and immunohistochemical study. Arch Pathol Lab Med 1988;112:275-81.  Back to cited text no. 7
Bourm KS, Menias CO, Ali K, Alhalabi K, Elsayes KM. Spectrum of xanthogranulomatous processes in the abdomen and pelvis: A Pictorial review of infectious, inflammatory, and proliferative responses. AJR Am J Roentgenol 2017;208:475-84.  Back to cited text no. 8
de Oliveira I Junior, Viana CR, Sabino SM, Kerr LM, Vieira RA. Xanthogranulomatous mastitis mimicking locally advanced breast cancer. Breast J 2017;23:227-9.  Back to cited text no. 9
Wong KC, Tsui WM, Chang SJ. Xanthogranulomatous inflammation of terminal ileum: Report of a case with small bowel involvement. Hong Kong Med J 2015;21:69-72.  Back to cited text no. 10
Cozzutto C, Carbone A. The xanthogranulomatous process. Xanthogranulomatous inflammation. Pathol Res Pract 1988;183:395-402.  Back to cited text no. 11
Aktepe F, Kapucuoǧlu N, Pak I. The effects of chemotherapy on breast cancer tissue in locally advanced breast cancer. Histopathology 1996;29:63-7.  Back to cited text no. 12
Barnes PJ, Foyle A, Haché KA, Langley RG, Burrell S, Juskevicius R, et al. Erdheim–Chester disease of the breast: A case report and review of the literature. Breast J 2005;11:462-7.  Back to cited text no. 13
Faucher JN, Galian P, Bourdon AJ, Vermelin G, Jourdanne P. Malacoplakia of the breast. Ann Pathol 1993;13:135-6.  Back to cited text no. 14
Kenn W, Eck M, Allolio B, Jakob F, Illg A, Marx A, et al. Erdheim–Chester disease: Evidence for a disease entity different from langerhans cell histiocytosis? Three cases with detailed radiological and immunohistochemical analysis. Hum Pathol 2000;31:734-9.  Back to cited text no. 15
Rauschkolb EN, Sandler CM, Patel S, Childs TL. Computed tomography of renal inflammatory disease. J Comput Assist Tomogr 1982;6:502-6.  Back to cited text no. 16


  [Figure 1], [Figure 2], [Figure 3], [Figure 4]


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