Ovarian torsion reveals a large hemorrhagic cystadenofibroma in an elderly woman: Diagnostic challenges and imaging characteristics.

I declare the following description “Written informed consent was obtained from the patient for the publication of patient data and associated images.”

Cystadenofibroma is a rare, benign ovarian neoplasm that typically remains asymptomatic. It often resembles a malignant tumor, manifesting as a fluid-filled ovarian mass with tissue encompassing the entire wall. MRI assists in identifying the fibrous component that differentiates cystadenofibroma. The surgical intervention for cystadenofibromas is typically curative, with a positive prognosis. It is essential to evaluate multiple aspects, including tumor dimensions, symptomatology, and the potential for acute complications such as torsion. Although this tumor is benign, it can resemble malignant lesions on imaging, frequently necessitating verification through biopsy or surgical intervention. The prognosis is often positive, with a minimal risk of recurrence following treatment.

Benign ovarian tumors display considerable variability; cystadenoma is frequently encountered, while cystadenofibroma is rarely observed. These tumors comprise both epithelial and stromal components, and the ratios of these parts determine whether the tumor is solid, semi-solid, or fluid Fig. 4 , Fig. 5 , Fig. 6 . Fig. 4 Transvaginal pelvic USG with color Doppler shows a complex cystic mass (thin arrow) with septae and solid components (thick arrow) and no definite intralesional vascularity [ 16 ]. Fig 4: Fig. 5 Intraoperative specimen of the ovarian tumor and the uterus with normal opposite-side adnexa [ 17 ]. Fig 5: Fig. 6 Postoperative specimen of the ovarian tumor—ovarian cystadenofibroma [ 17 ]. Fig 6: Transvaginal pelvic USG with color Doppler shows a complex cystic mass (thin arrow) with septae and solid components (thick arrow) and no definite intralesional vascularity [ 16 ]. Intraoperative specimen of the ovarian tumor and the uterus with normal opposite-side adnexa [ 17 ]. Postoperative specimen of the ovarian tumor—ovarian cystadenofibroma [ 17 ]. A frequently referenced study by Outwater et al. reveals that cystadenofibromas comprise approximately 1%-2% of benign epithelial ovarian tumors. Consequently, while infrequent, these tumors may be identified with standard imaging assessments or surgical interventions conducted for ovarian masses [ 1 ]. A retrospective study published in the Archives of Gynecology and Obstetrics (2010) examined 5000 cases of benign ovarian tumors detected via histology, revealing that cystadenofibromas constituted approximately 1.7% of the cases. These statistics validate the infrequent occurrence of cystadenofibromas, categorizing them among the rarest benign ovarian tumors. Cystadenofibromas predominantly occur in middle-aged to older women, with peak prevalence between the ages of 40 and 60; however, they may also manifest in younger or older women [ 2 ]. The categorization of these tumors is determined by the specific types of epithelial cells they comprise, including serous, mucinous, endometrioid, clear cell, and mixed cell types. Serous cystadenofibromas are the predominant subtype of adenofibromas, with other variants including endometrioid, mucinous, and clear cell types [ 18 ]. The epithelial component, its growth rate, and its interaction with other components fundamentally dictate the classification of these tumors as benign, borderline, or malignant. Nonetheless, most recorded ovarian cystadenofibromas are benign [ 3 , 4 ]. Clinically, the symptoms of various tumors may vary significantly. They may not produce any discernible symptoms, or they may present as overall pain and a sensation of heaviness. However, the majority of cystadenofibromas are asymptomatic and are accidentally identified during pelvic ultrasonography; the hemorrhagic variant may present as acute pelvic pain or even an acute abdomen in the event of rupture or torsion. In the most adverse scenario, as exemplified in the instance we will discuss, these tumors may induce vaginal bleeding and feminization if they synthesize estrogen, a phenomenon attributed by multiple researchers to heightened stromal activity [ 5 , 6 ]. Tumor indicators are incapable of differentiating between benign and malignant tumors. Moreover, laparoscopy is of limited utility in this context, as these tumors exhibit similarities to malignant tumors upon macroscopic examination. Verification of the benign nature of these tumors may only be accomplished through surgical excision and subsequent microscopic examination [ 19 ]. An ultrasonography examination typically identifies a tumor comprising both tissue and cystic elements. This mass may present as a single chamber (unilocular) or possess many chambers (multilocular). The existence of a solid element within the mass wall frequently results in a cancer diagnosis. Pelvic MRI facilitates enhanced characterization accuracy. Outwater et al. discovered that the solid component frequently has a hypointense look on T2-weighted images, mimicking muscle, indicating its fibrous structure [ 5 ]. Takeuchi et al. have shown that the existence of microscopic cystic locules within the solid component is a characteristic feature of cystadenofibroma. The locules exhibit a dark, sponge-like appearance on T2-weighted images [ 1 ]. Cystadenofibroma lacks any distinguishing features. Cystadenocarcinofibroma (CACF) is a malignant variant of cystadenofibroma (CAF). Nonetheless, CACF is an exceedingly rare illness, and there is a paucity of recorded MRI findings for this malignancy in the current literature. Dae Chul Jung et al. conducted a study and noted that all masses in the 11 instances of CAF and 2 cases of CACF were multilocular, with the exception of 1 unilocular case. Forty-three percent of cases displayed a CAF morphology, defined by a multilocular mass with a solid component that appeared hypointense on T2-weighted imaging [ 20 ]. These tumors contained cystic cavities. Likewise, 43% of patients exhibited multilocular masses characterized by thickening of the outer wall or diffuse dissemination, appearing hypointense on T2 without a discernible solid component. The 2 CACFs had a significant increase in the solid component, which demonstrated an intermediate signal intensity on T2-weighted images. The signal from the solid component was generally greater than that of the CAFs [ 7 ]. Cho et al. (2002) examined the efficacy of computed tomography (CT) and magnetic resonance imaging (MRI) in diagnosing cystadenofibromas. The authors delineated the attributes of these tumors using contemporary imaging techniques, emphasizing their frequently heterogeneous presentation, featuring both cystic and fibrous regions, which may obfuscate identification. MRI, especially T2 weighting, is effective in revealing the fibrous stroma characteristic of cystadenofibromas, hence aiding in their separation from malignant tumors [ 8 ]. Furthermore, there exist ovarian masses with a peripheral tissue component that appears hypointense on T2-weighted imaging, such as endometriotic cysts resulting from hemosiderin or a fibrous capsule [ 7 ]. Moreover, fibroma, fibrothecoma, and Brenner tumors are benign neoplasms distinguished by a mostly solid component, exhibiting low signal intensity on T2-weighted imaging [ 9 , 10 ]. It is worth noting that metastases with substantial fibrous tissue, particularly in the gastrointestinal tract, frequently display areas of low signal intensity on T2-weighted imaging and demonstrate considerable amplification [ 11 ]. The differential diagnosis of an ovarian cystadenofibroma in both its classic and hemorrhagic forms necessitates differentiation from many benign and malignant ovarian conditions, owing to their occasionally analogous radiological appearances. Potential diagnoses include simple ovarian cysts, malignant epithelial tumors (e.g., cystadenocarcinomas), endometriotic cysts, ovarian fibroids, and borderline tumors [ 1 ]. Radiologically, a classic cystadenofibroma typically manifests as a mixed mass comprising cystic and solid components, which may suggest an ovarian fibroma or fibroadenoma. Nonetheless, its robust appearance and the presence of fine septations frequently distinguish it from straightforward cysts. In addition, the absence of malignant features helps to exclude it from more serious diagnoses, such as cystadenocarcinomas or borderline tumors. Ultrasound often reveals a mixed-echo mass with hypoechoic and hyperechoic areas, while CT or MRI confirms the benign nature of the fibrous stroma with the low intensity of the T2-weighted signal [ 8 ]. Otherwise, the hemorrhagic form of cystadenofibroma complicates the differential diagnosis due to the presence of blood in the cystic cavity. This may simulate an endometriotic cyst or a simple hemorrhagic cyst. Endometriotic cysts, for instance, exhibit dense contents on ultrasound or MRI and may exhibit internal echoes resembling those observed in hemorrhagic cystadenofibroma. However, MRI imaging can differentiate between these 2 entities by visualizing iron deposits in endometriotic cysts, characteristic of the presence of old blood. Furthermore, simple hemorrhagic cysts are typically transient and possess thinner walls [ 12 ], whereas hemorrhagic cystadenofibromas persist and exhibit a more intricate appearance with solid fibrous components. The treatment of cystadenofibroma of the ovary is mainly surgical, particularly when the tumor becomes symptomatic or large or when the diagnosis remains uncertain after imaging examinations. The objective of surgery is to remove the tumor while preserving ovarian function to the greatest extent feasible, particularly in women of childbearing age. Small benign tumors are frequently treated with partial ovarian cystectomy, which involves removing only the mass while preserving the ovary [ 13 ]. However, in the case of larger or more complex lesions, it may be considered a unilateral oophorectomy. In emergency cases, such as ovarian torsion, a rapid surgical intervention is necessary in order to avoid serious complications, such as ovarian necrosis. Torsion is caused when the ovary twists around its vascular pedicle, leading to an interruption in blood flow. In these situations, surgical management involves the detorsion of the ovary to restore vascularity [ 1 ]. If the ovary is still viable after detorsion, then resection of the tumor may be performed, preserving the ovary. If the torsion has led to ovarian necrosis, an oophorectomy may be necessary. Surgery can be performed either through laparoscopy or laparotomy, depending on the patient's condition and the surgeon's experience. Laparoscopy, a minimally invasive technique, is frequently favored to expedite recovery and mitigate postoperative complications [ 14 ]. Our patient underwent emergency surgery with laparoscopy, where the surgeon proceeded to untwist the ovarian pedicle with a left ovrectomy. Aydin et al. (2016) presented a rare and similar case of torsion of a giant cystadenofibroma in a postmenopausal woman, detailing the emergency surgical management. Their study illustrates the urgency and severity of complications associated with large cystadenofibromas, particularly ovarian torsion. It also emphasizes the positive postoperative recovery after rapid and appropriate surgical intervention, which was also the case for our patient, who progressed well after a short stay in intensive care [ 15 ].

A 61-year-old multiparous woman, amenorrheic for 10 years, arrived with abdominal edema, a sensation of heaviness in the lower abdomen, and acute discomfort in the left lower quadrant, persisting for the past week. She was referred to the gynecological department. There was no previous documentation of her experiencing weight loss or diminished appetite, nor was there a familial history of neoplastic tumors. The examination indicated stable vital signs and an abdominal mass with varying textures from soft to firm, resulting in soreness and pain. Palpation of the left side indicated the existence of free fluid. The speculum examination revealed no abnormalities, however the vaginal examination suggested that the uterus was of normal size but had ascended into the abdomen. The patient was relocated to the radiology department for an urgent ultrasound evaluation. The ultrasound indicated the existence of cystic and solid components in the right adnexa, accompanied by a strong septum, suggesting a complex cyst. Furthermore, free fluid was observed in the abdomen. Regrettably, no photographs are accessible for reference. The CA-125 level was recorded at 45 mIU/L, whereas the levels of CA-19.9 and CEA were determined to be within the normal range. An MRI scan was performed considering the possible existence of a malignant ovarian tumor. The imaging revealed a substantial tumor on the left side of the uterus, exhibiting spurs on the ovarian tissue. The mass had 2 cystic components that were hyperintense on T2-weighted imaging and contained fluid with an intermediate T2 signal ( Fig. 1 ). This fluid also demonstrated the presence of hemorrhagic stigmata ( Fig. 2 ). The tissue segment exhibited diminished signal intensity on both T2 and T1-weighted pictures. The findings comprised cystic locules with a sponge-like morphology that failed to demonstrate enhancement after gadolinium injection. Additionally, the ovarian pedicle on the same side exhibited an irregular and coiled morphology, indicative of torsion, popularly referred to as the spiral sign ( Fig. 3 ). Fig. 1 Axial and coronal sections of a T2-weighted pelvic MRI showing a large, multilocular cystic mass with a solid component (arrows) in the right ovary. The solid component (black arrowheads) shows low signal intensity and it contains multiple, small cystic locules (white arrowheads). and others of low signal tissue. Fig 1 Fig. 2 Axial section of a T1-weighted pelvic MRI showing a right latero uterine pelvic mass in heterogeneous hypo signal with peripheral hemorrhagic areas in spontaneous T1 hypersignal (white arrowheads). Fig 2 Fig. 3 Axial section of a T1-sequence pelvic MRI with fat saturation and GADO injection showing the whirl sign at the level of the right ovarian pedicle (blue arrowheads). Fig 3 Axial and coronal sections of a T2-weighted pelvic MRI showing a large, multilocular cystic mass with a solid component (arrows) in the right ovary. The solid component (black arrowheads) shows low signal intensity and it contains multiple, small cystic locules (white arrowheads). and others of low signal tissue. Axial section of a T1-weighted pelvic MRI showing a right latero uterine pelvic mass in heterogeneous hypo signal with peripheral hemorrhagic areas in spontaneous T1 hypersignal (white arrowheads). Axial section of a T1-sequence pelvic MRI with fat saturation and GADO injection showing the whirl sign at the level of the right ovarian pedicle (blue arrowheads).

Cystadenofibroma of the ovary is an uncommon benign tumor of the ovaries, classified within the category of epithelial and stromal tumors. It frequently mimics a malignant tumor on imaging, appearing as a cyst with a tissue component within its walls. These tumors can be classified as simple or complicated and may occasionally lead to problems, such as intracystic bleeding. Numerous studies indicate that imaging techniques, particularly ultrasound and MRI, are essential for the discovery and characterization of these tumors. Ultrasound generally demonstrates a mixed ovarian mass comprising cystic and solid regions, with intracystic hemorrhage potentially appearing as heterogeneous internal echoes. However, the detection of the fibrous component via MRI facilitates differentiation from other malignant ovarian tumors. Distinguishing an ovarian cystadenofibroma from other benign or malignant ovarian tumors presents a significant daily challenge for radiologists, particularly in cases involving hemorrhagic tumors. Nevertheless, a conclusive diagnosis depends on histological analysis, which can distinguish this tumor from other benign and malignant ovarian tumors.