Ovarian hemangioma presenting as pseudo–Meigs’ syndrome with elevated CA125

Kaneta, Yoshibumi; Nishino, Ruriko; Asaoka, Kentaro; Toyoshima, Kiwamu; Ito, Kimihiko; Kitai, Hirokatsu

doi:10.1046/j.1341-8076.2003.00088.x

Cited by 26 publications

(24 citation statements)

References 11 publications

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“…Studies have shown Meigs may also present with a fourth component: an increase in serum CA-125 (25)(26)(27)(28)(29). Although not formally recognized as a Meigs syndrome neoplasm, hemangioma of the ovary has manifested similarly as a solitary ovarian tumor, ascites, and pleural effusion in 2 patients (3,5). The clinical spectrum of Meigs, particularly with the inclusion of CA-125, overlaps with a recurrent presentation or triad in some patients with ovarian hemangioma: a benign ovarian tumor, increased CA-125 (usually in the hundreds), and massive ascites.…”

Section: Discussionmentioning

confidence: 97%

“…Hemangiomas are a component of numerous syndromes, but, to date, ovarian hemangiomas have only been associated with Kasabach-Merritt syndrome (20,21), pseudo-Meigs syndrome (3,5), and an unnamed triad of unilateral or bilateral hemangioma, CA-125 elevation, and massive ascites (2)(3)(4)(5). Kasabach-Merritt (also known as hemangioma with thrombocytopenia) presents as a vascular tumor and consumptive thrombocytopenia or coagulopathy that can lead to fatal internal bleeding (22).…”

Section: Discussionmentioning

confidence: 99%

“…presentations of this seemingly insignificant vascular tumor-for example, several case reports have demonstrated a consistent history of unilateral (2)(3)(4) or bilateral (5) ovarian hemangioma, elevated CA-125, and massive ascites in a subset of patients. Discussion has also been generated by descriptions of ovarian teratomas showing a frankly vascular proliferation (6,7) and whether such cases indicate a possible germ cell origin of ovarian hemangioma.…”

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confidence: 96%

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Ovarian Hemangiomas Do Not Harbor EWSR1 Rearrangements

Schoolmeester

Greipp²,

Keeney³

et al. 2015

International Journal of Gynecological Pathology

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Hemangiomas of the ovary are rare with a majority described as individual reports of unusual clinical presentations or morphologic findings. Both the expected and unexpected pathologic features of these tumors in the ovary are not well detailed. Therefore, we collected the largest series of ovarian hemangiomas to comprehensively define their clinicopathologic associations and examine the significance of hormone receptors in their pathogenesis. In addition, a novel EWSR1-NFATC1 fusion has recently been described in a case of hemangioma of bone. To our knowledge, EWSR1 rearrangement has not been evaluated in hemangiomas of other sites or in a case series. Accordingly, we used fluorescence in situ hybridization to investigate EWSR1 status in a majority of our cases. Clinical presentation was variable and dependent on tumor size. Patient age ranged 48 to 87 yr (median 63 yr). Tumors involved the right (n=6) and left (n=3) ovaries with laterality unknown in 1 case, and size ranged from 0.2 to 5.0 cm (median 1.0 cm). Three of 4 radiologic reports were either equivocal or could not exclude malignancy. Seven cases were of the cavernous type and 3 were mixed cavernous and capillary type. All lesions formed a single discrete, circumscribed mass that displaced the surrounding cortical stroma. The cavernous type showed dilated, thin-walled vessels and vascular thrombi, some of which were associated with dystrophic calcification. In addition to cavernous morphology, the mixed form exhibited features of capillary hemangioma such as lobulated growth of capillary-sized vascular spaces that lacked atypia or multilayering and were linked to a larger feeding vessel. Each tumor expressed CD31, CD34, FLI-1, ERG, but not D240. The hemangioma stromal cells, but not endothelium, expressed estrogen and progesterone receptors in every case. Stromal luteinization was seen in 2 cases. Follow-up ranged 1 to 139 mo and all patients were disease free. All cases were negative for EWSR1 rearrangement; however, 2 cases demonstrated additional intact copies of EWSR1 indicating aneusomy 22 or a structural abnormality of chromosome 22 resulting in apparent duplication of the EWSR1 gene region (at 22q12). Although an uncommon entity, awareness of ovarian hemangioma's unique and diverse clinical presentation as well as its potential to radiologically imitate malignant ovarian neoplasms are important.

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Section: Discussionmentioning

confidence: 97%

Section: Discussionmentioning

confidence: 99%

mentioning

confidence: 96%

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Ovarian Hemangiomas Do Not Harbor EWSR1 Rearrangements

Schoolmeester

Greipp²,

Keeney³

et al. 2015

International Journal of Gynecological Pathology

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“…3,8,9 Some of the complications of ovarian haemangioma can be listed as pseudo-Meigs syndrome, 10 stromal leutinization, 11 stromal hyperplasia and thrombocytopenia. 1,3 Histopathogenesis of ovarian haemangioma is topic of debate with many theories coming up.…”

Section: Discussionmentioning

confidence: 99%

Ovarian Hemangioma: a rare case in a young girl

Patvekar

Kolate

Bhatnagar

et al. 2015

Int J Reprod Contracept Obstet Gynecol

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“…However, elevated serum CA125 has been reported in various cases of pseudo-Meigs' syndrome associated with metastatic ovarian cancer [9], ovarian benign tumors such as struma ovarii [10], hemangioma [11] and germ cell tumor [6], uteral [12] and renal tumor [13]), and collagen vascular disease simulating pseudo-Meigs' syndrome [14]. Thus, elevated CA125 has to be interpreted with caution in managing relevant patients.…”

Section: Discussionmentioning

confidence: 99%

Pseudo-Meigs’Syndrome as a Cause of Exertional Dyspnea: A Case Report

Ehara¹,

Koga

You

et al. 2009

Kurume Med. J.

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INTRODUCTIONIt is not rare to see patients with bilateral pleural effusion in clinical practice, and it is not unusual for such patients to present with exertional dyspnea [1]. Although several cardiopulmonary diseases account for most cases of pleural effusion [2], it occurs in diverse conditions and diseases. Bilateral pleural effusion develops in several systemic conditions such as hypoalbuminemia and collagen vascular diseases, and even in conditions unrelated to the cardiopulmonary system, including ovarian diseases such as Meigs' syndrome [3], a rare condition characterized by ovarian fibroma complicated by ascites and pleural effusion. We describe herein a patient presenting with exertional dyspnea who was diagnosed with pseudoMeigs' syndrome accompanying ovarian cancer. CASE REPORTA 61-year-old woman sought medical attention because of exertional dyspnea and an occasional wheeze that had developed one month previously without a noticeable triggering episode. She never smoked, consumed alcohol or took any medications. She underwent appendectomy at the age of nineteen, and was diagnosed when she was fifty as having myoma uteri, which was in stable condition on periodic check-ups. Her mother and a brother had diabetes, and another brother died of gastric cancer. Vital signs on presentation were as follows; blood pressure, 146/96 mmHg; pulse rate, 94 per minute with a regular rhythm; body temperature, 36.7°C; and respiration rate, 22 per minutes. She was neither anemic nor icteric. Finger clubbing was not noted. Neither lymphadenopathy nor venous dilation was noted on the neck. Thoracic examinations revealed diminished breath sound and dullness on percussion bilaterally. No wheeze was audible on this occasion. Heart sounds were not particular. Abdomen was flat, and there were no tender areas or masses on palpation. Neurological status was normal. Routine laboratory values were within the normal Kurume Medical Journal, 56, 85-87, 2009 Summary: A 61-year-old otherwise healthy woman presented with gradually worsening exertional dyspnea. Routine examinations revealed bilateral pleural effusion with no other notable cardiopulmonary diseases. Systemic examinations showed ascites and a pelvic tumor, which turned out to be right ovarian endometrioid adenocarcinoma. Surgical removal and chemotherapy against the ovarian cancer resulted in disappearance of the ascites and pleural effusion, establishing a diagnosis of pseudo-Meigs' syndrome. It is common for reported cases of pseudoMeigs' syndrome to initially present with dyspnea, therefore it is important to consider this disorder when attempting a differential diagnosis in female patients presenting with dyspnea without other noticeable conditions.

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Ovarian hemangioma presenting as pseudo–Meigs’ syndrome with elevated CA125

Cited by 26 publications

References 11 publications

Ovarian Hemangiomas Do Not Harbor EWSR1 Rearrangements

Ovarian Hemangiomas Do Not Harbor EWSR1 Rearrangements

Ovarian Hemangioma: a rare case in a young girl

Pseudo-Meigs’Syndrome as a Cause of Exertional Dyspnea: A Case Report

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