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Phyllodes Tumors of the Breast: A Rare Lesion with Still-Evolving Prognostic Features and Clinical Management

• Breast Imaging (Azadeh Elmi, Section Editor)
• Published: 09 November 2022
• Volume 10 , pages 162–170, ( 2022 )

Cite this article

• Tara A. Retson   ORCID: orcid.org/0000-0002-0009-7733 1 ,
• Soudabeh Fazeli   ORCID: orcid.org/0000-0002-9435-3004 1 ,
• Vivian Lim 1 &
• Haydee Ojeda-Fournier   ORCID: orcid.org/0000-0003-3773-0050 1  

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Purpose of Review

Phyllodes tumor (PT) of the breast is a rare type of fibroepithelial lesion classically presenting in 40–50-year olds. PTs can present a diagnostic challenge with histologic, imaging, and genetic features significantly overlapping other breast lesions, particularly fibroadenomas (FAs).

Recent Findings

PTs are subclassified as benign, borderline, or malignant based on histologic features, with a 10–30% malignancy rate, of which 25–30% will metastasize. Lesion characteristics and histology from image-guided biopsy are used to help distinguish between benign or malignant PTs and other fibroepithelial lesions. Surgical excision is often performed due to PT’s potential for recurrence, rapid growth, and metastasis. However, the state of management is evolving, with conflicting reports presented in the literature regarding the roles of local excision, mastectomy, and radiation treatment.

Here we present the histologic, imaging, genetic characteristics, and clinical management options of PTs, highlighting features to help the radiologist distinguish between PTs and FAs, such as the presence of a leaf-like stromal pattern on histology, rapid growth, and unique imaging characteristics.

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Contemporary Management of Phyllodes Tumors

Surgical treatment of borderline and malignant phyllodes tumors: the effect of the extent of resection and tumor characteristics on patient outcome.

Phyllodes Tumors of the Breast (the Egyptian Experience)

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Dr. Retson is sponsored under NIH training Grant T32EB00597, unrelated to this work.

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Tara A. Retson, Soudabeh Fazeli, Vivian Lim & Haydee Ojeda-Fournier

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Retson, T.A., Fazeli, S., Lim, V. et al. Phyllodes Tumors of the Breast: A Rare Lesion with Still-Evolving Prognostic Features and Clinical Management. Curr Radiol Rep 10 , 162–170 (2022). https://doi.org/10.1007/s40134-022-00403-y

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DOI : https://doi.org/10.1007/s40134-022-00403-y

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Introduction, case presentation, acknowledgements, conflict of interest statement.

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Optimizing aesthetic results in Asian women with giant phyllodes tumors over 10 cm: the periareolar mastopexy approach

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Kun-Han Chen, Yu-Chen Hsu, Optimizing aesthetic results in Asian women with giant phyllodes tumors over 10 cm: the periareolar mastopexy approach, Journal of Surgical Case Reports , Volume 2024, Issue 5, May 2024, rjae342, https://doi.org/10.1093/jscr/rjae342

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Giant phyllodes tumors, typically exceeding 10 cm in size, are neoplastic lesions with malignant potential. Surgical excision in small-breasted Asian women presents unique challenges where expected poor aesthetic outcomes may delay timely medical intervention. The periareolar mastopexy technique offers a comprehensive solution, enabling complete tumor removal alongside mastopexy to achieve optimal breast contouring. This approach consistently delivers favorable aesthetic outcomes, enhancing symmetry and contour. Additionally, the periareolar approach minimizes visible scarring, thereby enhancing patient satisfaction with the cosmetic outcome. Herein, we present a case report of Asian women with giant phyllodes tumors exceeding 10 cm, successfully managed using the periareolar mastopexy technique, emphasizing the importance of optimizing aesthetic outcomes in these challenging cases.

Giant phyllodes tumors, a rare type of fibroepithelial breast neoplasms, are characterized by their significant size, often exceeding 10 cm in diameter. These tumors present a considerable challenge in clinical management due to their potential for rapid growth and tendency to recur locally. Phyllodes tumors have distinct histological features, including stromal overgrowth and leaf-like projections, covering a range from benign to malignant behavior. However, the primary concern with giant phyllodes tumors is their potential for malignancy [ 1 ].

Surgical excision is the standard treatment, aiming to completely remove the tumor while minimizing the risk of recurrence. However, determining the best surgical approach, especially in small-breasted Asian women, poses unique challenges. Concerns about anticipated poor aesthetic outcomes may lead to delays in seeking medical care. These challenges go beyond oncological aspects and also include cosmetic outcomes. This case report provides a comprehensive overview of managing giant phyllodes tumors, emphasizing the complexities of diagnosis and treatment, with a focus on optimizing both oncological and aesthetic outcomes.

A 50-year-old female presented to the outpatient Breast Clinic with a complaint of a painless lump in her right breast persisting for four years, progressively increasing in size. She was a non-smoker with no significant family history of breast disease. The patient sought medical attention 10 months prior to this visit when her tumor was ~10 cm in size as noted on mammography. A core needle biopsy revealed a benign breast lesion. However, she was lost to follow-up due to concerns about potential unfavorable cosmetic outcomes. Subsequently, the tumor progressed to a size comparable to that of a melon, exhibiting a lobulated contour, prompting her decision to pursue treatment.

The clinical picture and mammography before (A, B) and after (C, D) the operation.

Examination revealed a firm, lobulated mass, measuring ~12 × 14 cm, predominantly occupying the breast, with the overlying skin remaining unaffected ( Fig. 1A ). No signs of skin ulceration, nipple inversion, discharge, or enlarged axillary lymph nodes were observed. Mammography indicated a well-defined, high-density lobular tumor measuring 12 cm, lacking any of calcifications ( Fig. 1B ). Breast ultrasound revealed a well-defined homogenous hypoechoic mass lesion larger than 10 cm ( Fig. 2A ). CT scan confirmed a heterogeneous soft tissue lesion within the breast, with no evident involvement of the chest wall, axillary lymph nodes, or distant metastasis ( Fig. 2B ).

Ultrasound (A) and computed tomography (B) image of the tumor.

Periareolar mastopexy approach for giant phylloedes tumor.

Wide local tumor excision by periareolar mastopexy approach was performed to resect the tumor and restore breast symmetry. Pathology report showed a 14-cm borderline phyllodes tumor. Cosmetic outcome was evaluated by the breast and plastic surgeon showed favorable aesthetic results as the treated breast was slightly different than the untreated. Minimal postoperative scarring was observed over the new areolar complex ( Fig. 1C ). Serial breast image follow-up, including mammography and breast echo, showed no evidence of tumor local recurrence for 3 years ( Fig. 1D ).

A patient facing a breast tumor over 10 cm often experiences significant physical discomfort and emotional distress. The size of the tumor implies that it may have been growing for a long time, possibly reaching an advanced stage. It is common for these patients to fear negative treatment outcomes, which can lead to delays in seeking timely care. In such situations, having a surgeon who can offer a positive cosmetic outcome becomes essential in encouraging patients to accept and pursue treatment actively. The periareolar mastopexy approach proves to be a valuable surgical strategy in this context, offering wide surgical exposure. It enables the excision of benign giant breast tumors while concurrently restoring breast shape, resulting in favorable aesthetic outcomes and minimal postoperative scarring [ 2–4 ].

The procedure begins by marking a pair of concentric circumareolar skin incisions ( Fig. 3A ). The inner circle’s diameter is adjusted to match the areolar diameter and ensure symmetry with the opposite breast. Meanwhile, the outer circle’s diameter is determined using four standard reference points: upper, lower, inner, and outer. These points are connected to form a circular pattern. The distances from the upper point to the sternal notch, the lower point to the inframammary fold, the inner point to the sternal notch, and the outer point to the anterior axillary line all corresponded to their respective distances from the nipple in the unaffected contralateral breast.

Initially, a preplanned full-thickness incision is made along approximately one-third to half of the circumference of the outer periareolar circle, targeting the area near the apex of the tumor. This approach ensures easy access to the phyllodes tumor with a clear and optimal view, facilitating its resection with an adequate safety margin ( Fig. 3B ). Following the wide local excision of the tumor, deepithelialization of the entire skin between the inner and outer periareolar incisions was performed, ensuring that no dermoglandular tissue was discarded ( Fig. 3C ). The expanded excessive skin envelope could be mobilized to achieve restoration of breast symmetry. The breast parenchyma was carefully repositioned during the process of approximating the new nipple. For skin closure, a round block suture technique is employed at the outer skin margin to reduce its diameter to that of the normal areola, resulting in only a periareolar scar ( Fig. 3D ).

Surgical excision remains the primary treatment for giant phyllodes tumors, but it is vital to prioritize aesthetic outcomes, which significantly impact body image and quality of life. The periareolar mastopexy approach is particularly recommended when there is no skin or nipple-areolar complex ulceration due to the tumor. This technique allows extensive surgical exposure for excising benign giant breast tumors while restoring breast shape, leading to favorable aesthetic results with minimal periareolar scarring [ 5 ]. The adoption of the periareolar mastopexy approach offers an enhanced treatment option for patients concerned about poor expected surgical outcomes, especially those facing tumors exceeding 10 cm in size. This approach ensures comprehensive care and improved outcomes tailored to individual needs.

We are grateful to the patient for allowing this report.

None declared.

Fernández-Ferreira R , Arroyave-Ramírez A , Motola-Kuba D , et al.  Giant benign mammary phyllodes tumor: report of a case and review of the literature . Case Reports in Oncology 2021 ; 14 : 123 – 33 .

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Hifny MA , Mohamed MA , Abdelhameid M . Application of periareolar mastopexy technique for giant phyllodes tumor resection in an adolescent female with breast asymmetry: a case report and literature review . J Egypt Natl Canc Inst 2020 ; 32 : 1 – 3 .

El-Zoghby A , Wakim H , Ahmed D , et al.  A comparative study assessing surgical outcome of excision of Giant or multiple benign breast lesions using circumareolar incision versus round block technique . Med J Cairo Univ 2021 ; 89 : 461 – 71 .

Cui H , Liu J , Guo J , et al.  A unique four-point approach for removal of giant breast fibroadenoma with marked asymmetry: a modified round block technique . J Invest Surg 2020 ; 33 : 709 – 14 .

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Phyllodes Tumors

Duke breast surgical oncologists are experts in diagnosing and treating phyllodes tumors, a rare type of tumor that grows in the breast. As nationally recognized leaders in phyllodes tumor research and treatment, we perform a comprehensive evaluation to accurately diagnose your phyllodes tumor and genetic testing to determine your risk for future malignancies. We work as a team to perform complicated chest wall (thoracic) surgery when phyllodes tumors return or spread to other parts of the body.

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What Are Phyllodes Tumors?

Unlike most forms of breast cancer, which usually starts in the ducts or glands, phyllodes tumors are rare tumors that start in the connective tissue (called the stroma) of the breast. They comprise less than 1% of all breast cancers.

Types of Phyllodes Tumors

They are classified into three subtypes: benign, borderline, and malignant, based on findings seen under a microscope. Some of these subtypes can overlap. Our specialists are experts in correctly classifying your phyllodes tumor. This ensures you receive the best treatment recommendation.

Benign Phyllodes Tumor Most phyllodes tumors are classified as benign (non-cancerous). The firm masses grow slowly in the breast tissue and typically occur in women in their 40s. They are the least likely phyllodes tumor type to return, but if they do, most continue to be benign.

Benign Phyllodes vs Fibroadenoma Phyllodes tumors may be misdiagnosed as fibroadenomas, which are also firm, benign breast lumps. Fibroadenomas commonly occur in younger women, typically in their teenage years or early 20s. While fibroadenoma and benign phyllodes tumors look similar on an imaging test and even a core needle biopsy, a correct diagnosis can be made after the mass is removed and tested.

Borderline Phyllodes Tumors Phyllodes tumors are classified as borderline if they have characteristics associated with benign and malignant phyllodes tumors.

Malignant Phyllodes Tumors Up to 15% of phyllodes tumors are considered malignant and may become metastatic. These tumors grow rapidly and are more likely to return or spread after treatment. Malignant and metastatic phyllodes tumors require more aggressive treatment and should be managed by a team of experts who specialize in phyllodes tumor treatment.

The Duke Cancer Center is a state-of-the-art patient care facility that provides almost all outpatient services in one convenient location.

Diagnosing Phyllodes Tumors

Imaging Tests Most phyllodes tumors can be seen on ultrasound and mammogram . Breast MRI provides more detailed information on large tumors.

Ultrasound-Guided Core Needle Biopsy A hollow needle punctures the skin and removes a small amount of tissue for examination under a microscope to aid in diagnosis. A definitive diagnosis may not be made until the mass is surgically removed and examined.

Genetic Testing and Counseling Because a rare genetic condition can increase your risk of a malignant phyllodes tumor, you may be referred to our genetic counselors for genetic counseling and testing. This helps us determine your risk of future cancers.

Benign Phyllodes Tumor Treatment

Excisional Biopsy Small benign phyllodes tumors may be removed through a minimally invasive surgical procedure called an excisional biopsy. The tumor and a rim or margin of tissue surrounding the tumor are removed to ensure that only healthy tissue remains.

Lumpectomy or Partial Mastectomy The size of the tumor determines if a lumpectomy is needed. A lumpectomy or partial mastectomy removes a slightly larger portion of the breast than an excisional biopsy and takes a larger margin of healthy tissue around the breast. A lumpectomy aims to preserve the breast shape and contour and create an aesthetically pleasing result.

Follow-Up About 10% of benign phyllodes tumors will return in the breast, usually within the first two years of surgery. The timing of follow-up monitoring, including breast exams and ultrasound imaging, can vary from every six months to every few years, depending on the features of the phyllodes tumor.

Borderline and Malignant Phyllodes Tumor Treatment

Your doctor will consider several factors when determining the right treatment plan for borderline and malignant phyllodes tumors. These factors include:

• How the tumor was removed during the initial operation
• How much breast tissue was preserved or how much is available around the tumor
• Microscopic findings and other lab results

Treatment options include additional surgery, such as a lumpectomy if an excisional biopsy was initially performed, mastectomy, and/or radiation therapy after surgery. Duke’s extensive experience treating phyllodes tumors has helped to improve outcomes for people with borderline and malignant phyllodes tumors.

Locally Recurrent and Metastatic Phyllodes Treatment

Malignant phyllodes tumors may return to the same, or very near the same location (this is called locally recurrent). They may also become metastatic, meaning they return elsewhere in the body. These complex tumors require more extensive treatment, including complete mastectomy, chest wall resection (removes the tumor, chest wall, and muscle if the tumor has spread to the chest), breast reconstruction, and/or radiation therapy.

Complex Chest Wall Surgery for Chest Wall Recurrence Phyllodes tumors that return after mastectomy and have spread deep into the chest wall require a coordinated team of surgeons who routinely remove these rare rumors to prevent a future recurrence. Duke is one of the few cancer centers in the country with a team of breast and thoracic surgeons who are expertly equipped and regularly perform complex surgery to remove the tumor, chest wall, and muscle to ensure a wide, tumor-free margin. The highly skilled team also includes specialized plastic surgeons who reconstruct the area immediately following the procedure.

Why Choose Duke

One-Day Coordinated Consultation with Phyllodes Tumor Specialists If the tumor returns after mastectomy, more extensive surgery may be needed. Your consultation will include a complete range of diagnostic tests. You will meet with members of Duke’s team of phyllodes tumor experts and with a surgical oncologist, medical oncologist, radiation oncologist, and a plastic surgeon when needed. If you have not already undergone genetic testing, you will complete these tests and meet with our genetics counselor, if needed. We gather all the information we require to make an informed decision about your treatment plan that we discuss with you on the day of your appointment.

Nationally Recognized Experts in Phyllodes Tumors Our phyllodes tumor experts are helping to standardize phyllodes tumor treatment and reporting so that centers across the country will use the same information to diagnose and treat all phyllodes tumors.

Leading Research on Phyllodes Tumor Treatment We are also leading research to determine the best approach to phyllodes tumor treatments. Our tumor registry collects phyllodes tumor tissue and uses it to better understand the tumor and test new therapies.

Multi-Specialty Approach to Treatment Decision Making Our team of experts includes specialists in breast surgical oncology and thoracic surgical oncology who routinely perform large chest wall reconstruction surgery for people with malignant phyllode tumors. People come to us for care from around the U.S. because of our unparalleled expertise in complex chest wall surgery and reconstruction. We offer options to people who have been told elsewhere that chest wall reconstruction surgery is not an option.

Our Support Services Follow You Through Your Journey We offer the full range of services to help minimize the side effects of treatment and give you the support you need to cope with the emotional and psychological effects of treatment. Our services include meetings with registered dieticians, family therapists, self-image services, survivorship support, social workers, and palliative care providers.

Where you receive your cancer care is important. Duke University Hospital is proud of our team and the exceptional care they provide. They are why our cancer program is nationally ranked, and the highest-ranked program in North Carolina, according to U.S. News & World Report for 2023–2024.

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Malignant phyllodes tumor of the breast: treatment and prognosis

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• 1 Department of Surgical Oncology, Centre of Oncology, Maria Skłodowska-Curie Memorial Institute, Kraków Branch, Kraków, Poland.
• PMID: 25227987
• DOI: 10.1111/tbj.12333

Surgery remains the mainstay of the treatment in patients with malignant phyllodes tumor of the breast (MPTB); however, the extent of surgery (breast conserving surgery [BCS] versus mastectomy) and the role of adjuvant radiotherapy have been controversial. We report a single institution's experience with MPTB. We discuss controversial therapeutic aspects of this rare tumor. Seventy patients with MPTB treated primarily with surgery were evaluated. The mean age was 50 years (21-76), and the mean size of the tumor was 6 cm. Thirty-four (48.6%) patients were treated with total mastectomy, and 36 (51.4%) were treated with BCS (lumpectomy or wide local excision). Microscopic surgical margins were free of tumor in all cases. In 64 (91.4%) patients, margins were ≥1 cm. Remaining 6 (8.6%) patients treated with BCS margins were <1 cm and subsequently radiotherapy was performed. Among 70 patients, 58 (82.9%) had no evidence of disease (NED) after 5 years. The extent of surgery was not significantly related to the 5-year NED survival rates (82.4% in patients who underwent mastectomy and 83.3% in patients who underwent BCS only or BCS with adjuvant irradiation). The 5-year NED survival rates in BCS (tumor-free margin ≥1 cm) and BCS with irradiation (tumor-free margin <1 cm) groups were identical (83.3%). Our data support the potential use of BCS in patients with MPTB. Mastectomy is indicated only if tumor-free margins cannot be obtained by BCS. Adjuvant radiotherapy may be considered if tumor-free margins are <1 cm.

Keywords: breast sarcoma; malignant neoplasm; phyllodes tumor; prognosis; treatment.

© 2014 Wiley Periodicals, Inc.

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Citation, DOI, disclosures and article data

At the time the article was created Frank Gaillard had no recorded disclosures.

At the time the article was last revised Mohammad Taghi Niknejad had no financial relationships to ineligible companies to disclose.

• Cystosarcoma phyllodes
• Cystosarcoma phylloides
• Phyllodes tumours
• Phyllodes tumors

Phyllodes tumor , also known as cystosarcoma phyllodes , is a rare fibroepithelial tumor of the breast which has some resemblance to a fibroadenoma . It is typically a large, fast growing mass that forms from the periductal stroma of the breast 13 .

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Epidemiology, clinical presentation, radiographic features, treatment and prognosis, history and etymology, differential diagnosis.

• Related articles
• Cases and figures

Phyllodes tumors account for less than 0.3-1% of all breast neoplasms 13 . It is predominantly a tumor of adult women, with very few examples reported in adolescents. The occurrence is most common between the ages of 40 and 60, before menopause (peak incidence ~45 years). This is about 15 years older than the typical age of patients with fibroadenoma.

Patients typically present with a painless, rapid growing breast mass for which imaging is requested. Average sizes can vary from 3 to 5 cm at presentation 13 .

Its original term cystosarcoma phyllodes was coined in view of its leaflike growth pattern 13 . A phyllodes tumor may be considered benign, borderline, or malignant depending on histologic features including stromal cellularity, infiltration at the tumor edge, and mitotic activity. At histologic analysis, the tumor can resemble a giant fibroadenoma  with both epithelial and stromal components being seen.

Fine needle aspiration is inaccurate, and even core biopsy has moderate sensitivity due to tumor heterogeneity causing inadequate sampling ref .

The tumors can be quite large at presentation. Imaging alone is not adequate to differentiate phyllodes tumor from fibroadenoma 13 . Phyllodes tumor are frequently classified as BI-RADS 4 tumor 14 .

Mammography

Typically seen as non-specific large rounded oval or lobulated, generally well-circumscribed, lesions with smooth margins. A radiolucent halo may be present. Calcification (typically coarse and plaque-like) may be seen in a very small proportion 13 . 

General sonographic features are non-specific and can mimic that of a fibroadenoma 7 .

On ultrasound, an inhomogeneous, solid-appearing mass is the most common manifestation. A solid mass containing single or multiple, round or cleft like cystic spaces and demonstrating posterior acoustic enhancement strongly suggests the diagnosis of phyllodes tumor. Vascularization is usually present in the solid components 13 .

In practice, most lesions are indistinguishable from fibroadenomas on both mammography and ultrasound. This is why interval enlargement of a "fibroadenoma" is seen as an indication for a needle biopsy. Large lesions (i.e. >4 cm) may qualify for excision out of hand because needle biopsy may not be representative of the pathology in the whole lesion.

As with mammography, they are typically seen as oval, round, or lobulated masses with circumscribed margins. Signal characteristics can vary with histological grade 11 but in general, are:

• T1: usually of low signal 8
• T2: can be variable ranging from homogenous low 8 to high 4-5  signal
• T1 C+ (Gd): the solid components enhance after contrast administration
• dynamic contrast: the kinetic curve pattern can be gradual slow or have rapid enhancement

An inhomogeneous signal may rarely result in the context of accompanying hemorrhage or cystic spaces 9 . Some suggest the inhomogeneous signal as indicative of benignity 10 .

It is a locally invasive tumor. Treatment is usually with surgical excision. Large tumors may even require a full mastectomy. Both benign and malignant phyllodes tumors have a tendency to recur if not widely excised. Malignant degeneration is seen in 5-25% 4  ( malignant phyllodes tumor ).

After wide local excision, there is relatively frequent local recurrence (up to 25%) and up to 10% can metastasize. The mode of metastases in such cases is by hematogenous route. 

The name is derived from the Greek word: "phullon"   meaning "leaf" .  Phyllodes tumors were first described in 1838 by Johannes Muller as "cystosarcoma phyllodes" .

For ultrasound and MRI appearances consider 6,8 :

• fibroadenoma of the breast : calcification is more common
• primary sarcoma of the breast
• periductal stromal tumor  of the breast : the main distinction being the lack of leaf-like processes 
• 1. Ralph Weissleder. Primer of Diagnostic Imaging. (2007) ISBN: 9780323040686 - Google Books
• 2. Buchberger W, Strasser K, Heim K, Müller E, Schröcksnadel H. Phylloides Tumor: Findings on Mammography, Sonography, and Aspiration Cytology in 10 Cases. AJR Am J Roentgenol. 1991;157(4):715-9. doi:10.2214/ajr.157.4.1654022 - Pubmed
• 3. Yabuuchi H, Soeda H, Matsuo Y et al. Phyllodes Tumor of the Breast: Correlation Between MR Findings and Histologic Grade. Radiology. 2006;241(3):702-9. doi:10.1148/radiol.2413051470 - Pubmed
• 4. Lifshitz O, Whitman G, Sahin A, Yang W. Radiologic-Pathologic Conferences of the University of Texas M.D. Anderson Cancer Center. Phyllodes Tumor of the Breast. AJR Am J Roentgenol. 2003;180(2):332. doi:10.2214/ajr.180.2.1800332 - Pubmed
• 5. Farria D, Gorczyca D, Barsky S, Sinha S, Bassett L. Benign Phyllodes Tumor of the Breast: MR Imaging Features. AJR Am J Roentgenol. 1996;167(1):187-9. doi:10.2214/ajr.167.1.8659370 - Pubmed
• 6. Feder J, de Paredes E, Hogge J, Wilken J. Unusual Breast Lesions: Radiologic-Pathologic Correlation. Radiographics. 1999;19 Spec No(suppl_1):S11-26; quiz S260. doi:10.1148/radiographics.19.suppl_1.g99oc07s11 - Pubmed
• 7. Chao T, Lo Y, Chen S, Chen M. Sonographic Features of Phyllodes Tumors of the Breast. Ultrasound Obstet Gynecol. 2002;20(1):64-71. doi:10.1046/j.1469-0705.2002.00736.x - Pubmed
• 8. Wurdinger S, Herzog A, Fischer D et al. Differentiation of Phyllodes Breast Tumors from Fibroadenomas on MRI. AJR Am J Roentgenol. 2005;185(5):1317-21. doi:10.2214/AJR.04.1620 - Pubmed
• 9. Balaji R & Ramachandran K. Magnetic Resonance Imaging of a Benign Phyllodes Tumor of the Breast. Breast Care (Basel). 2009;4(3):189-91. doi:10.1159/000220604 - Pubmed
• 10. Kinoshita T, Fukutomi T, Kubochi K. Magnetic Resonance Imaging of Benign Phyllodes Tumors of the Breast. Breast J. 2004;10(3):232-6. doi:10.1111/j.1075-122X.2004.21316.x - Pubmed
• 11. Yabuuchi H, Soeda H, Matsuo Y et al. Phyllodes Tumor of the Breast: Correlation Between MR Findings and Histologic Grade. Radiology. 2006;241(3):702-9. doi:10.1148/radiol.2413051470 - Pubmed
• 12. Tan H, Zhang S, Liu H et al. Imaging Findings in Phyllodes Tumors of the Breast. Eur J Radiol. 2012;81(1):e62-9. doi:10.1016/j.ejrad.2011.01.085 - Pubmed
• 13. Plaza M, Swintelski C, Yaziji H, Torres-Salichs M, Esserman L. Phyllodes Tumor: Review of Key Imaging Characteristics. Breast Dis. 2015;35(2):79-86. doi:10.3233/BD-150399 - Pubmed
• 14. Duman L, Gezer N, Balcı P et al. Differentiation Between Phyllodes Tumors and Fibroadenomas Based on Mammographic Sonographic and MRI Features. Breast Care (Basel). 2016;11(2):123-7. doi:10.1159/000444377 - Pubmed

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Giant Borderline Phyllodes Tumor Fungating Through the Skin as Fleshy Polypoid Outgrowths

Mitsuaki yoshida.

1 Department of Pathology and Laboratory Medicine, Kanazawa Medical University, Uchinada, JPN

Akihiro Shioya

Emi morioka.

2 Department of Breast and Endocrine Surgery, Breast Center, Kanazawa Medical University Hospital, Uchinada, JPN

Masafumi Inokuchi

Sohsuke yamada.

We present the case of a 52-year-old female with a giant phyllodes tumor (GPT), which was fungating through the skin that showed fleshy polypoid outgrowths. Histological analysis revealed stromal atypia, mitotic activity, and stromal overgrowth; however, the tumor border was well-defined, and malignant heterologous elements were not observed. Therefore, as some but not all malignant histological characteristics were present, we diagnosed the patient with borderline GPT. In cases of phyllodes tumor (PT) with the unique gross findings of fungation through the skin as fleshy polypoid outgrowths, caution is required for the subsequent course because even if the PT is graded as benign histologically, a malignant process can occur. Pathologists should note that the sampling of the collection site and the ambiguity of the histological grading of PT may affect the final diagnosis of GPT. It is also important to perform surgery with adequate preservation of the resected margins to control recurrence for patients with GPT.

Introduction

Phyllodes tumors (PTs) account for 0.3%-1% of all primary breast tumors and 2.5% of all fibroepithelial neoplasms [ 1 ]. Within this group, giant phyllodes tumors (GPTs) are even rarer, comprising 20% of PTs that grow to ≥10 cm [ 2 ]. According to the World Health Organization’s (WHO) classification, PTs are “tumors that fungate through the skin as fleshy polypoid outgrowths are invariably malignant.” However, without malignant heterologous elements, regardless of macroscopic findings, malignant PTs are diagnosed histologically if all the following features are present: marked stromal nuclear pleomorphism, stromal overgrowth, increased mitoses, increased stromal cellularity, and an infiltrative border. When some but not all adverse histological characteristics are observed, a diagnosis of borderline PT is made.

In this case, we encountered a GPT that was fungating through the skin that showed fleshy polypoid outgrowths. Pathological findings led us to ultimately settle on the diagnosis of “borderline.” However, differences occurred between such unique macroscopic and microscopic findings in the grading of malignancy. Herein, we discuss the pathological diagnostic issues of GPT.

Case presentation

The patient was a 52-year-old female with no remarkable medical history who had developed a tumor in her right breast and underwent fine-needle aspiration cytology at another clinic seven years previously. Cytology results indicated that the mass was benign, and a fibroadenoma was presumed. She had not visited a medical provider since. However, after noticing a rapidly growing tumor on her right breast six months earlier, she visited the hospital with the chief complaint of the tumor breaking through the skin and exposing its components on the skin’s surface along with general malaise and anorexia. Clinical examination revealed a giant tumor in the right breast, which showed fleshy polypoid outgrowths fungating through the skin. Blood pressure was 123/104 mmHg, pulse was 127/minute, and temperature was 37.2°C. Blood tests showed a white blood cell count of 151,00/μL with a neutrophil percentage of 87.3% and a C-reactive protein level of 9.13 mg/dL. The clinical findings were considered inflammation associated with partial necrosis of the tumor site. The blood tumor markers were within reference values: carcinoembryonic antigen (CEA), 2.7 ng/mL; cancer antigen 15-3 (CA15-3), 7.3 U/mL; breast cancer antigen 225 (BCA-225), <20.0 U/mL; and National Cancer Center-stomach-439 (NCC-ST-439), <1.0 U/mL. A fluorodeoxyglucose F 18 (FDG)-positron emission tomography/computed tomography scan showed heterogeneous FDG accumulation inside the right breast mass, and the maximum standardized uptake value was remarkably high (12.89) in the area, showing strong accumulation (Figure  1 ). No metastases to other organs were observed.

A giant mass was found in the right breast. Heterogeneous FDG accumulation was observed in the mass, and the maximum standardized uptake value was remarkably high at 12.89 in the area of strong accumulation

FDG: fluorodeoxyglucose F 18

A tumor biopsy revealed fibroepithelial lesions with a leaf-like growth pattern. Stromal cell density was heterogeneous and varied from low- to high-density areas with atypia. Based on clinical and biopsy findings, malignant PT was suspected. A total right mastectomy was performed. The weight of the right breast specimen was very high (2,905 g). The mass had held a space from the upper to the lower outer quadrants of the right breast and measured 26 × 21 × 13 cm.

Macroscopically, most of the tumor had fungated through the skin as fleshy polypoid outgrowths. Its central portion of the tumor showed hemorrhagic and necrotic changes (Figure  2 ). The cut surface after formalin fixation revealed a grayish-white, fibrous, and elastic appearance with edematous changes. The areas with hemorrhagic changes were a mixture of hematomas showing a reddish-brown color and soft areas showing a grayish-white color (Figure  3 ).

The mass was located in the upper outer and lower outer quadrant areas of the right breast and was 26 × 21 × 13 cm in size. Macroscopically, most of the tumor fungated through the skin as fleshy polypoid outgrowths. The central portion of the tumor showed hemorrhagic and necrotic changes

Most of the tumor had a grayish-white, fibrous, elastic appearance with edematous changes. The areas with hemorrhagic changes were a mixture of hematoma areas showing a reddish-brown color and soft areas showing a grayish-white color

Histologically, the tumor exhibited an exaggerated intracanalicular growth pattern in the fibrous portion with leaf-like projections extending into the variably dilated and elongated lumina. Stromal cellularity and atypical cells were scarce (Figure  4a , ​ ,4b). 4b ). No leaf-like structures were observed in the soft areas. The cell density in the stroma was elevated indicating stromal overgrowth (Figure ​ (Figure4c). 4c ). Cells showing severe atypia with high nuclear/cytoplasmic (N/C) ratios, distinct nucleoli, and atypical mitosis were also observed (Figure  4d ). Mitotic activity was observed as 46 mitoses per 10 high-power fields. No heterologous malignant elements were observed. The border between tumor and background mammary tissue was mostly PT component with mild atypia and well-defined. A few borders were in contact with components with severe atypia; however, their border appeared well-defined rather than permeative.

(a) In the fibrous parts, the tumor exhibited an exaggerated intracanalicular growth pattern with leaf-like projections extending into variably dilated elongated lumina (magnification, ×20; scale bar, 500 μm). (b) Stromal cellularity and atypical cells were scarce (magnification, ×400; scale bar, 20 μm). (c) No leaf-like structures were observed in the soft areas. Cell density in the stroma was elevated indicating stromal overgrowth. The border between the tumor and non-tumor areas was well-defined (magnification, ×20; scale bar, 500 μm). (d) Cells showing severe atypia with a high N/C ratio, distinct nucleoli, and atypical mitosis were observed (magnification, ×400; scale bar, 20 μm)

N/C: nuclear/cytoplasmic

When applied to the histological features of the WHO classification of PT grading, stromal cellularity, stromal atypia, mitotic activity, and stromal overgrowth fulfilled the criteria suggestive of malignancy. However, the tumor border was well-defined, and malignant heterologous elements were not observed; therefore, the diagnosis of borderline GPT was made. After three years post mastectomy, the patient had not experienced recurrence.

We presented a case of GPT that fungated through the skin as fleshy polypoid outgrowths. Despite the WHO’s established classification of the clinical features of PT, no references in the literature have yet supported this finding [ 1 ]. Furthermore, skin ulcerations and fleshy polypoid outgrowths are not included in the histopathological malignancy grading. In several previous case reports, GPT cases with fungation through the skin as fleshy polypoid outgrowths have been graded benign regardless of their macroscopic findings and have not recurred [ 3 - 5 ]. Thus, we did not consider the skin findings in the histological classification of our GPT case, and there had been no recurrence after grading as borderline. However, there is one case of a GPT with fungating proliferation with histological grading of benign in which malignant pleural effusion has been reported, in which the patient died during the postmastectomy follow-up period [ 6 ]. Because we cannot exclude the possibility that GPT that fungated through the skin as fleshy polypoid outgrowths may have a malignant course, we suggest that careful follow-up is necessary for such GPT.

Some reports have shown larger PT size to be significantly correlated with recurrence [ 7 , 8 ], while a study by Yom et al. showed smaller size to be correlated with recurrence [ 9 ]. In a systematic review and meta-analysis by Lu et al., tumor size was not a significant risk factor for local recurrence [ 10 ]. Therefore, it remains unclear whether GPT is a risk for local recurrence compared to PT of less than 10 cm. The risk factors for the local recurrence of PT are more important for histological grade and margin status at excision than for size, and the risk factor for distant metastasis is malignant histological grading [ 1 , 2 , 10 - 12 ]. While histological grading is thus an important indicator for predicting local recurrence and distant metastasis, it must be taken into account that for tumors of large size such as GPT, pathological diagnosis can be affected by the site of sampling. These tumors are notoriously heterogeneous, and the adequate sampling of at least one block per centimeter of maximum tumor dimension with additional sampling of grossly heterogeneous areas is recommended [ 13 , 14 ]. In this case, 50 sections were sampled, with four of the five malignant histological features from the WHO classification being suggestive of malignancy. However, as the tumor border was well-defined, our PT grading remained “borderline” as per the WHO classification. However, it is difficult to prove that there is absolutely no permeative tumor border anywhere in GPT owing to the impracticality of preparing and microscopically searching entire tumors in cases of GPT.

In a survey from Tan et al., nearly half (49%) of pathologists did not require all the histological parameters for PT grading to be on the malignant end of the spectrum (as recommended by the WHO) before diagnosing malignant PT [ 13 ]. Although the guidelines related to PT diagnosis may appear straightforward, their application can be fraught with ambiguity [ 12 ]. Furthermore, how the subdivisions for each microscopic parameter interact to constitute the final grade is subjective [ 12 , 13 ]. In this case, we judged the tumor border to be well-defined as both areas with mild and severe atypia showed a clear border with the background tissue due to expansive growth. However, if another pathologist had reviewed this case, it may have been interpreted as a permeative tumor border condition in a severe atypical area or graded as malignant without assessing the finding of a tumor border as was done in the survey by Tan et al. [ 13 ] The histological grading of GPT should be done with an understanding of these histological diagnostic issues. Moreover, we consider it important to perform the surgery with well-preserved resection margins because local recurrence can be controlled regardless of the ambiguity of the histological grading of the GPT.

Conclusions

This study presents a case of GPT that fungated through the skin as fleshy polypoid outgrowths. This unique clinical and macroscopic feature of PT required caution during the patient’s subsequent course. In addition, pathologists must understand that in the case of GPT, the sampling of the collection site and the ambiguity of the histological grading of the PT can affect the final diagnosis. It is also important to perform surgery with adequate preservation of the resection margins to control recurrence in GPT cases.

Acknowledgments

We would like to thank Editage (www.editage.com) for the English language editing.

Funding Statement

This study was supported by the seventh grant from the Kisshokai Foundation for promoted research and a grant for promoted research from Kanazawa Medical University (S2023-A1). The funding sources had no such involvement in the study design; in the collection, analysis, and interpretation of data; in the writing of the report; and in the decision to submit the article for publication.

The authors have declared that no competing interests exist.

Author Contributions

Concept and design:   Akihiro Shioya, Mitsuaki Yoshida, Sohsuke Yamada, Masafumi Inokuchi

Acquisition, analysis, or interpretation of data:   Akihiro Shioya, Mitsuaki Yoshida, Masafumi Inokuchi, Emi Morioka

Drafting of the manuscript:   Akihiro Shioya, Mitsuaki Yoshida, Sohsuke Yamada

Critical review of the manuscript for important intellectual content:   Akihiro Shioya, Mitsuaki Yoshida, Sohsuke Yamada, Masafumi Inokuchi, Emi Morioka

Supervision:   Akihiro Shioya, Sohsuke Yamada

Human Ethics

Consent was obtained or waived by all participants in this study