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A Case of Malignant Brenner Tumor of the Ovary Incidentally Found in a Patient with Deep Vein Thrombosis

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Abstract

We present the case of a 73-year-old woman who was incidentally found to have a malignant Brenner tumor (MBT) of the ovary during an evaluation for deep vein thrombosis (DVT). The patient presented with swelling in her left leg, non-healing ulcers, weakness, and numbness in her lower limbs. Imaging studies revealed a large multiloculated cystic mass with areas of calcification in the left adnexa extending to the upper abdomen toward the gallbladder fossa. The patient underwent exploratory laparotomy with removal of the ovarian cyst, later diagnosed as a focal MBT in a background of borderline Brenner tumor. Brenner tumors of the ovary are a rare subtype of ovarian neoplasm that accounts for less than 2% of all ovarian tumors. MBTs are even rarer, comprising less than 5% of all Brenner tumors. To our knowledge, this is the first reported case of an MBT incidentally found in a patient with DVT.
Review began 02/28/2023
Review ended 04/02/2023
Published 04/05/2023
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Ortiz et al. This is an open access article
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A Case of Malignant Brenner Tumor of the Ovary
Incidentally Found in a Patient with Deep Vein
Thrombosis
Wendolin J. Ortiz , Michael W. Morris , Mario Cervantes
1. General Surgery, Universidad Autónoma de Baja California, Mexicali, MEX 2. General Surgery, HCA (Hospital
Corporation of America) Houston Healthcare Kingwood, Houston, USA 3. General Surgery, United Memorial Medical
Center, Houston, USA 4. Pathology, HCA (Hospital Corporation of America) Houston Healthcare West, Houston, USA 5.
Pathology, HCA (Hospital Corporation of America) Houston Healthcare Pearland, Houston, USA 6. Pathology, United
Memorial Medical Center, Houston, USA
Corresponding author: Wendolin J. Ortiz, wendolinjortiz@gmail.com
Abstract
We present the case of a 73-year-old woman who was incidentally found to have a malignant Brenner tumor
(MBT) of the ovary during an evaluation for deep vein thrombosis (DVT). The patient presented with
swelling in her left leg, non-healing ulcers, weakness, and numbness in her lower limbs. Imaging studies
revealed a large multiloculated cystic mass with areas of calcification in the left adnexa extending to the
upper abdomen toward the gallbladder fossa. The patient underwent exploratory laparotomy with removal of
the ovarian cyst, later diagnosed as a focal MBT in a background of borderline Brenner tumor. Brenner
tumors of the ovary are a rare subtype of ovarian neoplasm that accounts for less than 2% of all ovarian
tumors. MBTs are even rarer, comprising less than 5% of all Brenner tumors. To our knowledge, this is the
first reported case of an MBT incidentally found in a patient with DVT.
Categories: Obstetrics/Gynecology, Pathology, General Surgery
Keywords: transitional cell carcinoma, ct scan, deep vein thrombosis, brenner tumor, malignant brenner tumor
Introduction
Malignant Brenner tumors (MBT) are an uncommon and aggressive type of ovarian neoplasms that make up
less than 5% of all Brenner tumors and less than 1% of all ovarian malignancies. Brenner tumors are solid,
usually unilateral ovarian tumors made up of abundant stroma, which contain nests of transitional-type
epithelium similar to that of the urinary tract. These nests may occasionally be cystic and lined with
columnar mucus-secreting cells. Brenner tumors are categorized into benign, borderline or proliferative,
and malignant subtypes, with MBT being particularly rare and characterized by stromal invasion under the
microscope.
MBT usually affects women during the perimenopausal and postmenopausal periods, and symptoms such as
abdominal distention, pelvic discomfort, or postmenopausal bleeding may be present. Surgical excision and
pathological evaluation are necessary to diagnose MBT because it lacks specific imaging features. Treatment
consists of surgical resection, and the prognosis of MBT is generally favorable [1-4].
Case Presentation
A 73-year-old African American woman with a medical history of hypertension, hyperlipidemia, type 2
diabetes, and four cerebrovascular accidents presented to the hospital with a recent diagnosis of left lower
extremity deep vein thrombosis (DVT). The patient reported experiencing swelling in her left leg for the past
few months, along with non-healing ulcers in both legs and weakness and numbness in her lower limbs.
On physical examination, the patient had 2+ pitting edema and small ulcers in her lower extremities.
Dorsalis pedis pulses and posterior tibial pulses were bilaterally absent. An abdominal examination revealed
present bowel sounds and a soft, non-tender, and distended abdomen. Venous and arterial Doppler of
bilateral lower extremities confirmed the diagnosis of DVT. The patient was already on anticoagulation, and
a venogram and thrombectomy were planned due to the edema and pain.
A week later, the patient was brought to the hospital due to sudden onset tachycardia. She had a pulse rate of
140 s and was admitted for cardiovascular event evaluation. The patient's organs and systems were
reviewed, and she was negative for chills or fever, vision disturbances, cough, and shortness of breath.
However, she did have bilateral leg edema, left leg pain, urinary frequency, and incontinence. A computed
tomography (CT) scan of the chest was obtained to ensure there was no pulmonary embolism. Although
there was no evidence of acute pulmonary embolism, an 8.8 cm cystic area lesion was visualized in the
anterior upper abdomen. A CT scan of the abdomen and pelvis revealed left mild hydronephrosis and a large
multiloculated cystic mass with areas of calcification within and multiple septations extending from the left
1 2, 3 4, 5, 6
Open Access Case
Report DOI: 10.7759/cureus.37176
How to cite this article
Ortiz W J, Morris M W, Cervantes M (April 05, 2023) A Case of Malignant Brenner Tumor of the Ovary Incidentally Found in a Patient with Deep
Vein Thrombosis. Cureus 15(4): e37176. DOI 10.7759/cureus.37176
adnexa to the upper abdomen toward the gallbladder fossa, measuring 12 cm anterior-posterior, 21 cm
transverse, and at least 20 cm in height (Figure 1).
FIGURE 1: CT of the abdomen and pelvis without contrast
Large multiloculated cystic mass with areas of calcification within and multiple septations extending from the left
adnexa to the upper abdomen shown on the transverse (A) and sagittal (B) planes
CT: computed tomography
Four days later, the patient underwent bilateral femoral, common femoral, external, and common iliac vein
thrombectomy angioplasty. During the angioplasty procedure of the inferior vena cava, a 12 x 40 mm
balloon was used, and it was observed that the pre-treatment stenosis was 50%, which improved to 40%
post-treatment. However, there was some indication of external compression caused by the pelvic mass.
During the next two days, the patient presented with pain in the right upper quadrant on deep palpation and
a decreased appetite.
General surgery was consulted; however, the exploratory laparotomy was postponed due to the patient's
medication regimen for deep vascular problems, including enoxaparin, clopidogrel, and aspirin. The patient
returned the following month for her scheduled exploratory laparotomy with the removal of the large ovarian
cyst. On examination, the abdomen was soft, with positive bowel sounds and no palpable megaly, but a large
mass was observed in the mid epigastric area that was slightly tender on movements and radiated to the left
upper and lower quadrants.
The patient was taken to the operating room. During the surgery, an upper midline incision was made, the
abdomen was emptied and a very large mass was evident. It became obvious that it was attached to the left
ovary and that it was probably a left ovarian mass. It was then excised, dividing the ovarian tissue with the
mass very close to the uterus. The mass was then totally removed, inspected, and opened on the back table.
It was found to be very large, about 10 x 8 inches in size (Figure 2A). The patient tolerated the procedure well
and was sent to the recovery room in good, stable condition.
2023 Ortiz et al. Cureus 15(4): e37176. DOI 10.7759/cureus.37176 2 of 5
FIGURE 2: Gross appearance of the Brenner tumor
A) The Brenner tumor measured approxim ately 10 x 8 inches in size, B) Upon gross examination, the tumor was
found to be a large cystic mass, measuring 21 x 13 x 9.5 cm, with loculated yellowish secretion/debris visible on
the cut section.
The gross examination revealed a large cystic tumor measuring 21 x 13 x 9.5 cm. On a cut section, it was
loculated with yellowish secretion/debris. The capsule/wall thickness ranged from 0.5 to 3.5 cm, and the wall
was partly fibrotic with scattered calcification (Figure 2B).
On histology, focal microinvasion adjacent to a predominantly non-invasive papillary transitional
carcinoma-like area was found, along with squamous epidermal inclusion cysts, glandular cysts with partial
mucinous differentiation, and classic Brenner's tumor with fibrosis. Some of the cysts were partly ruptured
with sub-epithelial chronic inflammation, histiocytosis, and fibrosis. There was also scattered dystrophic
calcification (Figures 3-5). The final diagnosis was a focal MBT in a background of borderline Brenner tumor.
FIGURE 3: Histological image of the Brenner tumor and transitional cell
carcinoma with invasion
Brenner tumor (left) and transitional cell carcinoma with invasion (right) (HE, 20x)
HE: Hematoxylin-eosin
2023 Ortiz et al. Cureus 15(4): e37176. DOI 10.7759/cureus.37176 3 of 5
FIGURE 4: Histological features of the Brenner tumor with transitional
cell islands and squamous differentiation
A) Typical Brenner tumor with transitional cell islands and fibrous stroma (H&E, 100x); B) Nuclear grooves
characteristic of Brenner tumors (H&E, 400x); C) Cystic area within the Brenner tum or with a mixture of
squamous, columnar, and goblet cells (H&E, 20x); D) Squamous differentiation (below), a mixture of columnar
with goblet cells (above) (H&E, 100x)
H&E: Hematoxylin-eosin
FIGURE 5: Histological evidence of tumor invasion into surrounding
tissue
Hematoxylin-eosin (HE) stain of invasion of the tumor cells into the surrounding tissue at 40x magnification (A)
and at 200x (B)
The patient was discharged in stable condition.
Discussion
MBT is an uncommon type of ovarian malignancy and is usually detected incidentally during imaging for
other conditions. The tumor is characterized by the presence of malignant and benign/borderline Brenner
tumor components, with clear stromal invasion by the malignant epithelial components. The absence of
hemorrhage or necrosis, distinct nuclear grooves with urothelial marker expression, and elevated CA125 in
2023 Ortiz et al. Cureus 15(4): e37176. DOI 10.7759/cureus.37176 4 of 5
some patients are among the diagnostic features of MBT. However, distinguishing MBT from transitional cell
carcinoma (TCC), another aggressive high-grade neoplasm with similar histologic features, can be
challenging. The histological features of TCC include a high mitotic rate, severe nuclear atypia, and
extensive necrosis. Immunohistochemical markers, such as CK7, p16, and p63, can aid in the differentiation
of MBT and TCC [2,3].
There are other types of ovarian tumors, such as serous, mucinous, endometrioid, and clear cell carcinomas,
as well as benign Brenner tumors, teratomas, and metastases from urinary tract tumors, which should be
considered in the differential diagnosis of MBT. Metastasis from other organs, such as the urinary bladder or
colorectum, should also be ruled out [4].
Patients with MBT typically experience symptoms similar to those of other epithelial ovarian carcinomas,
such as abdominal pain, distension, and urinary urgency. Imaging studies can assist in the preoperative
differential diagnosis of malignant and benign Brenner tumors, and MBTs usually contain both solid and
cystic components. Most MBTs are unilateral, high-grade tumors with a median size of 10 cm, and most
patients present with localized disease [5,6].
Surgery is the standard of care for all epithelial ovarian tumors, including MBT. Although the prognosis for
MBT is generally good, there is currently no standardized chemotherapy regimen for the treatment of MBT
due to the limited number of reported cases [1,2].
Conclusions
Thorough diagnostic evaluation is crucial in patients who present with symptoms related to DVT, as
emphasized in this case report. The incidental finding of a large ovarian cystic mass during the review of the
patient's cardiovascular symptoms ultimately led to the diagnosis of MBT, a rare subtype of ovarian cancer.
The successful removal of the tumor through surgery underscores the importance of early detection and
prompt treatment.
Additional Information
Disclosures
Human subjects: Consent was obtained or waived by all participants in this study. Conflicts of interest: In
compliance with the ICMJE uniform disclosure form, all authors declare the following: Payment/services
info: All authors have declared that no financial support was received from any organization for the
submitted work. Financial relationships: All authors have declared that they have no financial
relationships at present or within the previous three years with any organizations that might have an
interest in the submitted work. Other relationships: All authors have declared that there are no other
relationships or activities that could appear to have influenced the submitted work.
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3. Bouhani M, Slimane M, Sghaier S, Bouida A, Chargui R, Rahal K: Malignant Brenner tumor of the ovary: one
single institute experience and a review of the literature. Anatol J Family Med. 2020, 3:71-5.
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intestinal obstruction and perforation. BMJ Case Rep. 2020, 13:e235394. 10.1136/bcr-2020-235394
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carcinoma. Med Sci. 2020, 24:3550-4.
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Ovarian cancer are a heterogenous group of tumors, involving a variety of histological tissue ranging from epithelial tissue, connective tissues, specialized hormone secreting cells to germinal and embryonal cells. Brenner Tumor' (BT) is a rare epithelial ovarian tumor accounting for about 1-2% of all ovarian neoplasms. Malignant Brenner tumor (MBT) is much rarer. Although asymptomatic, they can presents as abdominal discomfort, abdominal pain or abnormal uterine bleeding. On gross appearance, it CASE REPORT ARTICLE CASE REPORT resembles fibroma of ovary with cut section appearing gritty and yellowish grey. On histological examination, solid and cystic nests of urothelium-like cells surrounded by abundant dense, fibrous stroma are seen. It closely resembles to transitional cell carcinoma of ovary, latter having worse prognosis and needs to be differentiated from Brenner tumor. Currently, most accepted treatment is primary surgical resection with or without lymph node dissection followed by platinum based chemotherapy. We herein review a case of MBT in a postmenopausal woman with abnormal uterine bleeding with emphasis on clinical features, investigations and primary treatment and discuss the current state of the literature and standards of practice regarding this malignancy.
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INTRODUCTION[|]Malignant Brenner tumors (MBT) of the ovary are rare diseases, representing 1% of all ovarian cancers and 3-5% of Brenner tumors. They carry a poor prognosis. They generally affect women during the perimenopausal and postmenopausal periods. The standard treatment is surgery; however, the indication of adjuvant chemotherapy remains controversial. The present study aims to report our experience in the treatment of MBT of the ovary, to better characterize this disease.[¤]METHODS[|]In this study, a retrospective case series involving four patients diagnosed with MBT of the ovary and treated between 2006 and 2014.[¤]RESULTS[|]Four cases of MBT of the ovary were diagnosed over a seven-year period. The mean age of our patients was 59.3+-11.1 years. Three patients were in the menopause period. The tumor was staged as IC in one case, IIC in one case, and IIIC in two cases of the International Federation of Gynecology and Obstetrics classification. All patients underwent surgery, followed by adjuvant chemotherapy. Three patients underwent a loco-regional recurrence that occurred respectively, after nine months in one patient and 11 months in two patients. The treatment was based on chemotherapy combined with surgery in one case. Two patients presented distant metastasis. The treatment consisted of chemotherapy and surgery. The median follows up period was 49.0 (14.0-64.0) months.[¤]DISCUSSION AND CONCLUSION[|]The treatment approach of MBT of the ovary is not well established since its scarcity and poor prognosis. Thus, more case series and meta-analysis should be conducted.[¤]
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Malignant Brenner tumor (MBT) is a rare ovarian tumor that, given the infrequency of the disease, has not been well documented in the literature. Diagnosing MBT both radiographically and histologically remains a challenge. We report two cases of ovarian MBT, detailing the clinical presentation, radiographic characteristics, and histologic findings with supplementary imaging. Our cases demonstrate the pathologic challenge of histologically diagnosing MBT versus other Brenner tumors and transitional cell carcinoma (TCC) of the ovary.
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A rare case of malignant Brenner tumour of ovary manifesting with intestinal perforation due to colonic infiltration is elaborated in the present report. Brenner’s tumour accounts for 1%–2% of all ovarian neoplasms and malignant Brenner tumour is even rarer and only about 5% of Brenner tumours are malignant. A 62-year-old woman came to surgical emergency with 1-month history of abdominal pain, vomiting and constipation with a palpable mass in right iliac fossa. Abdominal radiograph was suggestive of colonic obstruction. Contrast-enhanced CT of the abdomen revealed cystic right ovarian mass of 10.2×8.8 cm ² with pneumoperitoneum. Exploratory laparotomy was done, which revealed mass arising from right ovary involving terminal ileum, cecum and ascending colon. Possibility of ovarian malignancy was kept. Patient underwent debulking surgery along with ileostomy and descending colon mucous fistula was created. Histology was compatible with malignant Brenner tumour of the ovary.
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Objective: Malignant Brenner Tumor (MBT) is a tumor with an extremely low incidence that morphologically resembles to urothelium. Given the paucity of evidence on the epidemiology and prognosis of MBT, the aim of this retrospective population-based study was to elucidate the demographic and clinico-pathological characteristics of patients with ovarian MBT. Methods: A cohort of patients diagnosed between 1988 and 2012 was drawn from the National Cancer Institute Surveillance and Epidemiology End Results database. For surgically treated patients, Observed and Disease Specific Survival were calculated following generation of Kaplan-Meier curves. Comparisons were made using the log-rank test. Results: A total of 207 patients were identified. Median patient age was 65years and the majority presented with unilateral, high grade tumors with a median size of 10cm. Stage I, II, III and IV disease was noted for 55.4%, 14.4%, 18%, and 12.2% of patients respectively. Only 5.1% had positive lymph nodes for metastatic disease. Five-year disease-specific survival (DSS) of patients with tumors confined to the ovary was 94.5% compared to 51.3% for those with extra-ovarian spread (p<0.001). Lymphadenectomy was not associated with an improved DSS (p=0.2). Conclusions: MBTs are typically unilateral high grade tumors localized to the ovary. Regional lymphatic spread is uncommon and lymphadenectomy does not confer any improvement on survival. Patients with tumors confined to the ovary have an excellent prognosis while extra-ovarian spread is associated with a poor outcome.
Robbins Basic Pathology
  • V Kumar
  • S L Robbins
  • Kumar V
Malignant Brenner tumor: two case reports . Case Rep Womens Health
  • L King
  • R P Gogoi
  • C Hummel
  • A Smith
King L, Gogoi RP, Hummel C, Smith A: Malignant Brenner tumor: two case reports. Case Rep Womens Health. 2018, 20:e00082. 10.1016/j.crwh.2018.e00082