Ductal carcinoma in situ and invasive carcinoma originating in a Fibroadenoma with concurrent benign phyllodes tumor: A case report

Abstract

Introduction and importance

This case report presents a rare occurrence of multiple bilateral breast fibroadenomas, one evolving into ductal carcinoma in situ (DCIS) and invasive carcinoma, occurring simultaneously with a benign phyllodes tumor in the same breast. The importance of this case lies in emphasizing the crucial need for surveillance in patients with a long history of fibroadenomas and the necessity to investigate any rapid change in the size of fibroadenoma.

Case presentation

A 35-year-old multiparous female with a 17 year history of bilateral multiple breast lumps presented with recent onset of right breast pain and yellowish nipple discharge. Two lumps in her right breast had demonstrated an increase in size. Examination revealed a significant mass in the retroareolar region of the right breast and another at the 2 o'clock position. Histopathological examination of the biopsy specimens revealed fibroadenoma and benign phyllodes tumor. The patient underwent a bilateral breast lumpectomy. Further histopathological examination revealed ductal carcinoma in situ and invasive carcinoma within a complex fibroadenoma in the right breast and benign phyllodes tumor. Sentinel lymph node biopsy was negative. She had adjuvant radiations and trastuzumab. Regular follow-ups show no recurrence.

Clinical discussion

Fibroadenomas are usually benign but rarely undergo malignant change. Quick response to size changes and early detection greatly enhance patient results.

Conclusion

Ductal carcinoma in situ and invasive breast cancer, a rare malignancy found within a fibroadenoma, necessitates histopathological specimens and immunohistochemical results for accurate diagnosis. Survival rates are significantly enhanced through a multidisciplinary approach.

Highlights

• •We present a case of a 35-year-old woman with a 17-year history of bilateral breast lumps developed DCIS and invasive carcinoma within a fibroadenoma
• •Radiographic images showed lumps of benign nature
• •Lumpectomy was performed and its biopsy revealed Ductal carcinoma in situ and invasive carcinoma
• •Surveillance is crucial in patients with long-standing fibroadenomas and investigation of any rapid size changes.

1. Introduction

Fibroadenoma, a common benign fibroepithelial breast tumor, typically affects women aged 14–35. It's a painless, mobile, firm lump due to overgrowth in a breast lobule. While it often resolves on its own, may require removal. Uncommonly, conditions such as ductal carcinoma in situ, atypical ductal hyperplasia, lobular carcinoma in situ, atypical lobular hyperplasia, and invasive carcinomas can be found within a fibroadenoma [1].

DCIS originating in a fibroadenoma is a seldom encountered case diagnosed incidentally on histopathological examination of the excised fibroadenoma. Its incidence is 0.002–0.125 % [2]. There have been roughly 100 reported cases globally of carcinoma developing within a fibroadenoma. This includes various types such as intra-ductal carcinoma, lobular carcinoma in situ, and invasive carcinoma [3].

The management of the invasive cancer arising incidentally in a fibroadenoma is done according to the guidelines of invasive breast cancer which depends on type and stage of cancer.

Phyllodes tumors are rare breast neoplasms, accounting for 0.3 % to 1 % of all breast tumors. They typically occur in women aged 40–50 and grow rapidly in a unilateral pattern. The physical manifestation of the tumor is a firm, bosselated swelling, often with nipple discharge [4].

We present a case study of a patient who, over a span of 17 years, had multiple bilateral breast fibroadenomas, one of which evolved into ductal carcinoma in situ (DCIS) and invasive carcinoma occurring simultaneously with a benign phyllodes tumor in the same breast but different quadrant. This case emphasizes the crucial need for surveillance in patients who have a long history of fibroadenomasand to investigate any rapid change in size of fibroadenoma. This case report has been reported in line with the SCARE Criteria [5].

2. Case report

A 35-year-old female patient, gravida 4 para 3, with a 17-year history of bilateral multiple breast lumps, presented in the Outpatient Department (OPD) with complaints of right breast pain and yellowish nipple discharge persisting for four days.

The patient had been living with bilateral multiple breast lumps for 17 years. These lumps had remained stable throughout this period, presenting as circumscribed and benign-looking. They had previously been diagnosed as fibroadenomas. However, over the past month, she noticed an increase in the size of two lumps in her right breast. Accompanying this change was the onset of pain in her right breast, described as gradual, pricking, and non-cyclical, with no identified aggravating or relieving factors.

The nipple discharge was unilateral, spontaneous, slightly thick, and yellowish in color.

The patient is a mother to three living children. Her eldest child, born when she was 25 years old, was breastfed for three months. She has undergone one cesarean section.

In terms of family history, there are no reported cases of breast cancer. However, her sister had been diagnosed with stomach cancer.

During the physical examination, a large, firm, and irregular bosselated mass measuring approximately 7 × 8 cm was identified in the retroareolar region of the right breast. The mass was mobile, non-tender, and adherent to the nipple-areola complex. Additionally, another lump of about 2 × 2 cm was palpable at the 2 o'clock position in the right breast. This lump was also firm, mobile, and non-tender. The nipple was retracted with an active yellowish discharge.

On the left side, a large lump of about 3 × 4 cm was palpable at the 6 o'clock position in the left breast. It was mobile, firm, and non-tender. There was no nipple retraction or discharge from the left breast. Axillary and cervical lymph nodes were not palpable.

The patient underwent a radiological investigation, which included mammography and ultrasound.

On the right side, there was a large (33 × 61.8 × 66.3 mm), well-circumscribed, multi-lobulated solid lesion exhibiting peripheral flow on color Doppler, along with a few calcific foci in the retroareolar region. Additionally, another distinct, lobulated, hypoechoic solid mass lesion of about 20.4 × 26 × 22 mm with calcific foci, causing posterior shadowing and showing no flow on color Doppler, was observed at the 2–3 o'clock position. Multiple similar-looking subcentimetric lumps were also noted in the right breast. A few sub-centimetric lymph nodes were visible. All lumps were categorized as BIRADS-III. [Fig. 1, Fig. 2, Fig. 4, Fig. 5] a well-circumscribed, heterogeneous lesion, exhibiting a wider-than-taller orientation with dimensions of 28.6 × 50.9 × 47.9 mm. This lesion, located at the 6 o'clock position, was characterized by internal cystic areas. In addition, several sub-centimetric axillary lymph nodes were detected. It was categorized as BIRADS-II. [Fig. 3, Fig. 6, Fig. 7].

Fig. 1.

Ultrasound of right breast showing large multilobulated solid cum cystic lesion in retroareolar region that was finally diagnosed as benign phyllodestumor.

Fig. 2.

Ultrasound of right breast showing solid well defined lobulated hypoechoic lesion at 2–3 o clock that was diagnosed as ductal carcinoma in situ arising within a fibroadenoma.

Fig. 4.

Right Breast mammography: mediolateral oblique view.

Fig. 5.

Right breast mammography: craniocaudal view.

Fig. 3.

Ultrasound of left breast showing large well defined wider than taller heterogeneous lesion at 6 o clock position that was diagnosed as fibroadenoma.

Fig. 6.

Left breast mammography: mediolateral oblique view.

Fig. 7.

Left Breast mammography: craniocaudal view.

An ultrasound-guided trucut biopsy was performed on two lesions in the right breast and one in the left breast. The biopsy results revealed a fibroepithelial lesion in the right retroareolar lump, with a differential diagnosis of benign phyllodes versus fibroadenoma. The lump at the 2 o'clock position on the right side and the lump at the 6 o'clock position on the left side were both diagnosed as fibroadenomas.

Given these findings, the patient was scheduled for a bilateral breast lumpectomy. During the operation, the lump in the right retroareolar region was found to be densely adherent to the overlying skin and areola. Consequently, the nipple and areola were excised en bloc with the lump. The lump at the 2 o'clock position was also excised, ensuring a rim of normal and healthy tissue was included. On the left side, the lump at the 6 o'clock position was excised with the capsule intact, again ensuring a rim of surrounding normal tissue was included.

The histopathological and immunohistochemical examination of the breast lumps revealed the following:

• Right breast

Lump at 2 o'clock: The lump was diagnosed as invasive carcinoma of no special type, arising in the background of high-grade Ductal Carcinoma In Situ (DCIS). The histological grade was II out of III. The invasive tumor size was 2 mm, and the pathological stage was PT1a, pNx. All margins were negative for DCIS and invasive carcinoma. The size of the complex fibroadenoma was 4.0 × 2.0 × 4.4 cm Fig. 8, Fig. 9, Fig. 10, Fig. 11, Fig. 12.

Fig. 8.

Low power view (4×) showing focus of invasive carcinoma and background Ductal carcinoma in situ.

Fig. 9.

Focus of tumor, background DCIS and hyalinized stroma of fibroadenoma.

Fig. 10.

High power view (20×) of invasive carcinoma showing pleomorphic nuclei with vesicular chromatin.

Fig. 11.

Foci of DCIS within fibroadenoma.

Fig. 12.

Immunohistochemistry for p63 highlighting myoepithelial cells around DCIS and lack of staining in invasive carcinoma.

Lump in the retroareolar region: The lump was diagnosed as a benign phyllodes tumor with clear margins.

• Left breast

Lump at 6 o'clock: The lump was diagnosed as a fibroadenoma with no malignancy detected.

• The immunohistochemical examination of the invasive cancer found within the right breast fibroadenoma revealed the following:

• Estrogen Receptor/Progesterone Receptor (ER/PR): Negative.

• Human Epidermal Growth Factor Receptor 2 (HER 2 neu): 3+ (Positive).

• Ki67 index: 10 %.

The case was discussed in a Multidisciplinary Team Meeting (MDM), where it was decided that the patient would undergo Sentinel Lymph Node Biopsy (SLNB) and receive adjuvant radiotherapy and trastuzumab. Mastectomy was offered to the patient, but she refused.

We performed the sentinel lymph node biopsy in the right axilla without the nipple areolar complex. The dye was injected on the axillary side of the scar from the previous central excision. Three lymph nodes took the dye and were subsequently excised. All were negative for metastatic disease.

Following her referral to the oncology department, the patient has successfully completed radiation therapy and is currently undergoing treatment with Intravenous Trastuzumab. Regular follow-ups are being conducted, and to date, there has been no recurrence of the disease.

3. Discussion

We report a unique case of ductal carcinoma in situ and invasive carcinoma, originating within a fibroadenoma with concurrent benign phyllodes tumor. As per our knowledge, this is the first instance of such a case being documented in medical literature.

Fibroadenoma, the most prevalent benign fibroepithelial tumor, is an encapsulated growth resulting from the hyperplasia of a single breast lobule. It predominantly occurs in women between the ages of 14 and 35 with variable sizes, although it occurs in older women occasionally. It presents as a painless swelling with a rubbery consistency that is smooth, firm, mobile, and non-tender. Like all the solid lumps of the breast, triple assessment should be completed in all patients. A fibroadenoma may shrink over time and does not require excision unless associated with suspicious cytology or when it becomes very large [1].

Phyllodes tumors represent a rare variety of fibroepithelial neoplasms in the breast, accounting for only 0.3 % to 1 % of all breast tumors. These tumors typically present in women between the ages of 40 and 50. The growth pattern is usually unilateral and rapid. The physical manifestation of the tumor is a firm, bosselated lump, often accompanied by necrosis and redness of the skin overlaying the tumor. A bloody discharge from the nipple can occur, which is a result of spontaneous infarction within the tumor. Treatment often involves a wide local excision, ensuring a healthy margin greater than 1 cm, which is frequently curative. This approach aids in preventing local recurrence and achieving optimal patient outcomes [4].

Our patient, diagnosed with multiple bilateral fibroadenomas at 18 years of age, was informed of their benign nature and advised to monitor for changes. After 17 years of stability, she noticed enlargement in two right breast lumps with nipple retraction and discharge. Both lumps were firm, mobile and non-tender.

Fibroadenoma on ultrasound is presented as a well-defined, round or ovoid mass with uniform hypoechogenicity. In 10 % of cases, calcification may be observed [5].

Signs of potential malignancy include irregular form, pronounced hypo-echogenicity, shadowing, an echogenic halo, and tissue distortion [6].

Fibroadenomas on mammography appear as a distinct oval mass, either hypo- or isodense to breast tissue, or as a macrolobulated or partially obscured mass [5].

Phyllodes tumour sonographic features are non-specific and can mimic that of a fibroadenoma. A mass that appears solid and inhomogenous is most frequently observed.

Phyllodes on mammography presents as large, rounded, or lobulated with smooth edges. A clear halo and occasional coarse calcification may be present [7].

In our case there was no sign of potential malignancy on radiographic imaging. The radiographic findings were consistent with benign fibroadenoma and phyllodes tumor.

FNAC and ultrasound guided trucut biopsy of the breast lumps should be done for accurate diagnosis.

Patients with fibroadenoma had a 2.17-fold higher risk of invasive breast cancer, which increased to 3.10-fold in those with complex fibroadenomas [6]. Hubbard, et al., identified criteria for fibroadenoma excision: age over 35, an immobile or ill-defined mass, size exceeding 2.5 cm, or an inconclusive biopsy. These factors indicated a higher risk for non-fibroadenoma lesions within fibroadenomas [8].

In our particular case, a Tru-cut biopsy was conducted which identified the presence of a fibroadenoma. Given the patient's age of 35 years and the observed increase in the size of the fibroadenoma, these factors collectively contributed to the decision to proceed with its excision.

Core needle biopsy can potentially miss a Ductal Carcinoma in Situ (DCIS) within a fibroadenoma. This could occur if the needle samples nearby normal tissue instead of the tumor. In fact, malignant changes within a fibroadenoma are typically discovered incidentally following its excision.

For benign fibroadenoma, surgical treatment involves mass excision. A tumourectomy or lumpectomy can suffice if the surgical margin is cancer-free or only contains ductal or lobular carcinoma in situ within the fibroadenoma. If the resection margin is involved or close, a wider excision may be required. Management strategies depend on the stage and extent of metastasis (local or distant) [9].

In our case, the patient underwent lumpectomy procedure; and the histopathology report concluded it be an invasive cancer with DCIS arising within a fibroadenoma. The margins were free of cancer. The tumor was staged as pT1a and in multidisciplinary team meeting it was decided that sentinel lymph node biopsy will be advised to investigate the possibility of distant metastatic disease.

The patient has undergone adjuvant radiotherapy and is currently receiving intravenous trastuzumab. The advantage of using adjuvant radiotherapy and intravenous trastuzumab for HER2neu positive cases lies in their potential to reduce tumor recurrence.

4. Conclusion

Ductal carcinoma in situ and invasive breast cancer within a breast fibroadenoma is a rare, often misdiagnosed malignancy. Diagnosis accuracy improves with histopathological specimens and immunohistochemical results. Breast-conserving surgery with adjuvant radiotherapy is an effective treatment. Familiarity with clinicopathological features and treatment methods can enhance survival rates and cure rates. A multidisciplinary approach is recommended for patient care.

Ethical approval

Single-case reports are exempted from ethical approval in our institution.

Funding

This research did not receive any grant from funding agencies.

CRediT authorship contribution statement

Afaq Ahmad: The surgical procedure, conceptualization, methodology, data curation, writing original draft. Erum Najeeb: The surgical procedure, Supervision, Editing. Muhammad Burhan ul Haq: Supervision, Editing. Humaira Bashir: Editing, writing review. Muhammad Uzair: writing review, data curation. Nadira Mamoon: Histopathology slides, writing review.

Guarantor

Afaq Ahmad.

Registration of research studies

Not applicable.

Consent

Written informed consent was obtained from the patient for publication of this case report andaccompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Declaration of competing interest

The authors declare no conflict of interest.