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CASE REPORT
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Infiltrating duct cell carcinoma in lactating adenoma: A rare case in pair


1 Department of Pathology, Shri Lal Bahadur Shastri Government Medical College, Himachal Pradesh, Mandi, India
2 Department of Surgery, Shri Lal Bahadur Shastri Government Medical College, Mandi, Himachal Pradesh, India
3 Department of Pathology, Dr R.K.G.M.C, Hamirpur, Himachal Pradesh, India
4 Department of Pharmacology, Dr R.K.G.M.C, Hamirpur, Himachal Pradesh, India

Date of Submission20-Jul-2021
Date of Acceptance01-Sep-2021
Date of Web Publication01-Feb-2022

Correspondence Address:
Kavita Kumari,
Department of Pathology, Dr R.K.G.M.C. Hamirpur, Himachal Pradesh
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jcrt.jcrt_1183_21

 > Abstract 


Lactating adenomas, though considered being benign, sometimes turn out to be malignant. We are hereby presenting two cases of lactating adenomas which turned out to be harboring infiltrating duct cell carcinoma (DCC). Two lactating women with mass in the breast were referred from surgery for fine-needle aspiration cytology and biopsy. On histopathology, it was found that both had infiltrating DCC (either as collision tumor or as a malignant transformation of lactating adenoma). These cases point toward some association between lactating adenoma and DCC, however, small it may be. Therefore, any mass lesion in the breast should be followed up under strict vigilance for an early diagnosis and management of this deadly disease. A large sample size and elaborate study are required to frame its prognostic relevance. Like estrogen receptor and progesterone receptor, prolactin and prolactin receptor expression can be exploited for developing newer receptor-targeted therapy.

Keywords: Breast lump, infiltrating duct cell carcinoma, lactating adenoma



How to cite this URL:
Sharma R, Rattan A, Kumari K, Sharad S. Infiltrating duct cell carcinoma in lactating adenoma: A rare case in pair. J Can Res Ther [Epub ahead of print] [cited 2022 Nov 29]. Available from: https://www.cancerjournal.net/preprintarticle.asp?id=337036




 > Introduction Top


A lactating adenoma, the most common breast lesion encountered during pregnancy and lactation, is a localized focus of lobular hyperplasia, presenting as a distinct breast mass. They are thought to be benign breast lesions that regress spontaneously but are sometimes aggressive in nature, presenting as an ulcerative lesion of the breast and causing diagnostic dilemmas.[1] Lactating adenoma associated with infiltrating duct cell carcinoma (DCC) is also acknowledged in the literature. Pathological changes due to neoplasia might be considered normal by the examining physician, due to which a pregnant female has a 2.5-fold higher risk of being diagnosed with metastatic breast cancer.[2] Breast cancer diagnosed during pregnancy or 12-month postpartum is known as pregnancy-associated breast cancer (PABC).[3] It is reported in 1/3000 pregnancies.[4] In western countries, 7%–14.5% of all breast cancers are PABCs.[5] Whereas institutional studies conducted on Indian women shows the incident rate of 0.7%.[6] Apart from estrogen and progesterone, prolactin and prolactin receptor expression are also responsible for tumor induction.


 > Case Report Top


The present cases report comprises two cases of lactating adenoma associated with infiltrating duct cell carcinoma. A 29-year-old lactating mother attended the surgical outpatient department for gradually increasing swelling in the right breast for 7–8 months. A nontender diffuse lump of irregular consistency was identified measuring approximately 8 cm × 6 cm size. Routine investigations were unremarkable. Radiological study with mammography and ultrasonography (USG) showed an ill-defined hypoechoic space-occupying lesion of approximately 6 cm × 4 cm identified in the upper inner quadrant of the right breast (12–3 o'clock position) with right axillary lymph nodes. The report was given as “BIRADS IV and suspicious lesion.” Fine-needle aspiration cytology (FNAC) showed epithelial cell clusters arranged in an acinar pattern. Individual cells had vesicular nuclei, prominent nucleoli, and bubbly cytoplasm. In addition, a loosely cohesive cluster of atypical cells was also seen. The background was foamy protenaceous. The report was signed out as “cytomorphological features are suspicious for malignancy with associated lactational changes in right breast.” Core-needle biopsy showed histopathological features of DCC. On serial sectioning of mastectomy specimen with an attached axillary tail (17 cm × 14 cm × 9 cm), a 6-cm × 4-cm × 4-cm gray–white area was identified, extending up to the lateral resection margin. It was hard and gritty to cut. Adjacent to the gray–white area, a gray–brown area of 4 cm × 2 cm × 4 cm was also identified. The area was cystic and soft on sectioning. Twenty lymph node/tumor nodule dissected out (4 mm−2 cm size). Microscopic examination (M/E) of the sections from gray–white areas showed features of DCC [Figure 1]a, [Figure 1]b and [Figure 1]d, whereas M/E of the gray–brown area showed proliferating tubules arranged back to back with little surrounding stroma. The cells showed lactational changes [Figure 1]c. The final pathological report was signed out as “infiltrating DCC not otherwise specified with lactating adenoma.” With involved lateral margin and lymphovascular invasion. Metastatic deposits were seen in the 17/20 lymph node/tumor nodule. Bloom–Richardson score was 2 + 3 + 2 = 7, Grade: II: TNM classification – pT3N3M1. On immunohistochemistry (IHC), the tumor was triple negative. We lost the follow-up after 7 months.
Figure 1: (H and E, stain) (a) ×4. Gray–white area adjacent to gray–brown area. The right upper part shows a circumscribed area of closely packed glands (yellow arrow). The left lower sideshow infiltrating duct cell carcinoma (green arrow). (b) ×10 Cords and tubules of tumor cells. (c) . Uniform glands with secretory changes. Vacuolated cytoplasm and eosinophilic secretions in the lumen. (d) ×40. Cells showing nuclear cellular pleomorphism high N/C ratio, hyperchromatic nuclei, and moderate amount of eosinophilic cytoplasm

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The second case was a 34-year lactating female who presented with a palpable mass in the right breast for 3–4 months. A well-defined soft-to-firm lump approximately 5 cm in diameter was palpated. On mammography, a small suspicious-looking hypoechoic lesion was detected. FNAC showed only epithelial cells clusters having finely vacuolated cytoplasm in a lipoprotenacious background. On repeat, USG-guided FNA, apart from abovesaid findings, few loosely cohesive atypical cells with vesicular nuclei, conspicuous nucleoli, and a moderate amount of cytoplasm were also identified. Cytomorphological features were suggestive of lactating adenoma with few atypical cells. Keeping in view the mammography, the possibility of malignancy was also kept and biopsy was advised which showed a component of ductal carcinoma. A mastectomy specimen with an axillary tail (16 cm × 12 cm × 7 cm) was received, which on serial sectioning showed a large brown spongy mass of size 6 cm × 6 cm × 4 cm with a small gray white area in the center measuring 2 cm in diameter. Sections from large spongy mass showed features of lactating adenoma with interspersed malignant cells [Figure 2]a. The small gray–white area showed features of infiltrating ductal carcinoma [Figure 2]b, [Figure 2]c, [Figure 2]d. The report was signed out as infiltrating DCC with lactating adenoma. Bloom–Richardson score was 3 + 2 + 3 = 8, Grade – III, and TNM classification – pT1cN0M0. On IHC, the tumor was hormone negative and human epidermal growth factor receptor 2 (HER2) positive.
Figure 2: (H and E, stain). (a) ×4. Compact aggregate of lobules exhibiting secretory hyperplasia. (yellow arrow). Hyperchromatic malignant cells with disruption of the glandular structure at place (green arrow). (b) ×10. Intact back to back uniformly arranged glands with secretory changes (yellow arrow) and malignant cells in the same focus with haphazard arrangement (green arrow). (c) ×40. Cell showing pleomorphism, high N/C ratio, vesicular nuclear chromatin, prominent nucleoli, and eosinophilic cytoplasm. Brisk Mitosis. (d) ×10 The tumor cells are arranged singly, solid pattern, and tubule form

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 > Discussion Top


Lactating adenoma with infiltrating DCC is an extremely rare condition. To the best of our knowledge, no more than twenty cases have been reported in the literature to date. In our study, two lactating females had a simultaneous occurrence of a lactating adenoma and infiltrating ductal carcinoma. Case one had to infiltrate DCC adjacent to lactating adenoma. Saglam A, Can B. reported a similar case of lactating adenoma along with infiltrating DCC at the periphery.[7] S Geschicker et al. and Khanna et al. also reported lactating adenoma with an associated infiltrating carcinoma.[8] Hertel et al. reported a case of invasive ductal adenocarcinoma developing in the previous excision site of a lactating adenoma. A case of in situ ductal carcinoma developing inside lactating adenoma was reported by Kumar et al.[9] which was similar to our second case in which duct carcinoma (2 cm) was located right at the center of a large lactating adenoma (6 cm × 5 cm × 4 cm). IHC study by Genin et al. concluded that PABC has two times more frequent HER2 overexpression and hormone-negative status.[10] Our second case seems to follow the observation, whereas the triple-negative status of our first case challenges it. In the majority of case reports, infiltration of lactating adenoma by the malignant cell is seen; hence, it is suggested that there is a strong possibility of collision tumor. Our first case appears to harmonize with the suggestion, whereas some researchers supported the theory of transformation from lactating adenoma to adenocarcinoma, as seen in our second case. Given all the above said evidences, it is suggested that benign lactating adenoma per SE may have malignant potential, either by malignant transformation of lactating adenoma itself or preexisting premalignant condition.

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 initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Acknowledgment

The authors express their special thanks to Professor and HOD of the Department of Pathology, Dr Rashmi Kaul Raina, Dr RPGMC, Tanda, Himachal Pradesh.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 > References Top

1.
Phung HT, Nguyen LT, Nguyen HV, Nguyen CV, Nguyen HT. Aggressive lactating adenoma mimicking breast carcinoma: A case report. Int J Surg Case Rep 2020;70:17-9.  Back to cited text no. 1
    
2.
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3.
Wang B, Yang Y, Jiang Z, Zhao J, Mao Y, Liu J, et al. Clinicopathological characteristics, diagnosis, and prognosis of pregnancy-associated breast cancer. Thorac Cancer 2019;10:1060-8.  Back to cited text no. 3
    
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Ravikanth R, Kamalasekar K. Imaging of lactating adenoma: Differential diagnosis of solid mass lesion in a lactating woman. J Med Ultrasound 2019;27:208-10.  Back to cited text no. 4
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Hou N, Ogundiran T, Ojengbede O, Morhason-Bello I, Zheng Y, Fackenthal J, et al. Risk factors for pregnancy-associated breast cancer: A report from the Nigerian breast cancer study. Ann Epidemiol 2013;23:551-7.  Back to cited text no. 5
    
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Gogia A, Deo SV, Shukla NK, Mohanti BK, Raina V. Pregnancy associated breast cancer: An institutional experience. Indian J Cancer 2014;51:167-9.  Back to cited text no. 6
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7.
Saglam A, Can B. Coexistence of lactating adenoma and invasive ductal adenocarcinoma of the breast in a pregnant woman. J Clin Pathol 2005;58:87-9.  Back to cited text no. 7
    
8.
Khanna M, Manjari M, Khanna A. Lactating adenoma with infiltrating ductal carcinoma breast in a pregnant woman. Indian J Cancer 2015;52:585-6.  Back to cited text no. 8
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9.
Kumar H, Narasimha A, Bhaskaran, Divya Rani MN. Concurrent lactating adenoma and infiltrating ductal carcinoma: A case report. J Clin Diagn Res 2015;9:D14-5.  Back to cited text no. 9
    
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Genin AS, Lesieur B, Gligorov J, Antoine M, Selleret L, Rouzier R. Pregnancy-associated breast cancers: Do they differ from other breast cancers in young women? Breast 2012;21:550-5.  Back to cited text no. 10
    


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