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| CASE REPORT |
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| Year : 2021 | Volume
: 13
| Issue : 2 | Page : 56-59 |
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Metastatic ovarian cancer presenting as multiple hepatic cysts and paraneoplastic manifestation
Dauda Ayuba Dayar1, Yusuf Abiola Raheem1, Abubakar Yerima2, Lawan Mohammed3, Abba Bukar Zarami4
1 Department of Internal Medicine, University of Maiduguri Teaching Hospital, Maiduguri, Borno State, Nigeria
2 Department of Medicine, University of Maiduguri Teaching Hospital, Maiduguri, Borno State, Nigeria
3 Department of Radiology, University of Maiduguri Teaching Hospital, Maiduguri, Borno State, Nigeria
4 Department of Human Pathology, Faculty of Basic Clinical Sciences, College of Medical Sciences, University of Maiduguri, Maiduguri, Borno State, Nigeria
| Date of Submission | 11-Oct-2021 |
| Date of Acceptance | 19-Dec-2021 |
| Date of Web Publication | 24-Feb-2022 |
Correspondence Address:
Dr. Yusuf Abiola Raheem
Department of Internal Medicine, University of Maiduguri Teaching Hospital PMB 1414, Maiduguri, Borno State.
Nigeria
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/njgh.njgh_9_21
Background: Metastatic ovarian diseases are often found on the peritoneal surfaces including the liver, bowel, bladder, and uterus. Recent studies showed liver metastases as the most common presentation of stage-IV disease in patients with epithelial ovarian cancer (EOC). Paraneoplastic syndromes in ovarian cancers may be associated with rheumatologic disorders like polymyositis due to autoimmune mechanisms. Case Presentation: We report a case of a 61-year-old postmenopausal woman who presented with a 2-month history of progressive right hypochondrial swelling and pain, with progressive weight loss and weakness of the limbs. Examination revealed tender hepatomegaly with proximal muscle weakness. Abdominopelvic computed tomography (CT) scan revealed enlarged liver with multiple cystic lesions and right adnexal mass. CA125 and creatine kinase were elevated and ultrasound scan (USS)-guided trucut biopsy of both the hepatic and adnexal masses revealed papillary cystadenocarcinoma. Conclusion: We, therefore, concluded that the diagnosis in our patient is metastatic ovarian cancer to the liver with paraneoplastic phenomenon of polymyositis. Keywords: Papillary cystadenocarcinoma, paraneoplastic syndrome, polymyositis
How to cite this article:
Dayar DA, Raheem YA, Yerima A, Mohammed L, Zarami AB. Metastatic ovarian cancer presenting as multiple hepatic cysts and paraneoplastic manifestation. Niger J Gastroenterol Hepatol 2021;13:56-9 |
How to cite this URL:
Dayar DA, Raheem YA, Yerima A, Mohammed L, Zarami AB. Metastatic ovarian cancer presenting as multiple hepatic cysts and paraneoplastic manifestation. Niger J Gastroenterol Hepatol [serial online] 2021 [cited 2022 Jun 30];13:56-9. Available from: https://www.njghonweb.org/text.asp?2021/13/2/56/338256 |
| Introduction | |  |
Malignant lesions of the ovary can be broadly divided into primary and secondary when they arise from the normal structures of the ovary or elsewhere, respectively. The primary lesions include epithelial ovarian carcinoma (EOC, which accounts for almost 70% of all ovarian malignancies), germ-cell tumors, sex-cord stromal tumors, and other rare types. Metastatic ovarian diseases are often found on the peritoneal surfaces including the liver, bowel, bladder, and uterus. Approximately 12%–33% of patients with EOC will be diagnosed with FIGO stage IV, de novo metastatic disease at initial diagnosis.[1] Liver metastases are found in up to 50% of patients dying of EOC. Recent studies showed liver metastases as the most common presentation of stage-IV disease in patients with EOC.[2] However, liver metastasis in the form of massive and multiple hepatic cysts is rare.
Often, cancers can present as paraneoplastic syndromes, which are disorders associated with systemic cancer which are caused by mechanisms other than direct invasion or metastasis.[3] Some paraneoplastic syndromes in ovarian cancers may be associated with rheumatologic disorders due to autoimmune mechanisms. Antibodies to some tissue antigens as well as circulating immune complexes have been identified in the sera of patients with metastatic EOC.[4] For instance, dermatomyositis has been associated with ovarian cancer;[5] however, polymyositis (without dermatologic features) has been identified as a presenting symptom of ovarian cancer only in rare cases.[6],[7],[8] We, therefore, report a rare case of an advanced metastatic ovarian cancer that presented with massive and multiple hepatic cysts associated with the paraneoplastic phenomenon of polymyositis.
| Case presentation | | |
Our case is a 61-year-old woman who presented with a 2-month history of progressive right hypochondrial swelling and progressive weakness of the limbs. She had associated right hypochondrial pain with dragging sensation but there was no history of jaundice, hematemesis, hematochezia, malena, pruritus, or passage of pale bulky stool. However, she had progressive weight loss and anorexia, with no associated dysphagia or odynophagia. She is not a known Hepatitis B or C virus patient and has been vaccinated for hepatitis B 3 years prior to presentation. Also, there was no history of ingestion of poorly stored grains or traditional medications. She does not smoke or take alcohol in any form. She also had a progressive weakness with inability to stand from a sitting position associated with difficulty in raising her arms to tie her scarf, neck pain, and poor neck control. She had no history of vaginal discharge, bleeding or hirsutism. She, however, had a history of pelvic pain since the onset of her symptoms. She used a combined oral contraceptive for 6 months about 40 years ago. She was P2+0 A2 and her last childbirth was 28 years ago. She was divorced 21 years ago and 16years postmenopausal.
On physical examination, she was found to be wasted with pedal edema, but no skin lesions. She had tender hepatomegaly and ascites. Her temporomandibular joint was not tender but had decreased joint movement at the jaw, cervical spine, and shoulder. Her global muscle power was 2/5, weakness at the proximal muscles which were tender associated with diminished reflexes and tone.
An initial assessment of decompensated chronic liver disease with possible malignant transformation and proximal myopathy secondary to polymyositis was made.
Abdominopelvic computed tomography (CT) scan revealed a grossly enlarged liver with a lobulated outline and multiple thin-walled hypodense cystic lesions of varying sizes, with the largest measuring 21.1 cm x 8.5 cm x 15.7 cm in the right lobe of the liver, which is nonenhancing on post IV contrast study. An irregular enhancing thick-walled multiseptated cystic mass was also noted in the right adnexium measuring 12.3 cm x 7.8 cm x 9.6 cm. No evidence of intramural nodule within it or mass effect on the adjacent pelvic structures. The overall result of the CT scan suggested multiple hepatic cysts and complex right adnexal mass [Figure 1]. | Figure 1: (A) Contrast-enhanced coronal reformatted CT image of the abdomen showing multiple cystic lesions in the liver. (B) Contrast-enhanced axial CT image of the pelvis showing multiseptated right adnexium mass
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Liver function test parameters showed albumin 28 g/L (normal 35–50), ALP 92 iu/L (normal 40–129), AST 873 iu/L (normal <12), ALT 441 iu/L (normal <12), and uric acid 642 µmol/L (normal 142– 416). Electrolytes, bilirubin, hematologic indices, and clotting profile were all within normal limits. She was screened and found to be negative for hepatitis B, hepatitis C, and human immunodeficiency virus (HIV) infections. Creatine kinase was 2620 U/L (normal up to 170). Alpha-fetoprotein was also within the normal limit. Elevated levels of the creatine kinase in addition to weakness of her proximal muscles which were tender confirmed the diagnosis of the polymyositis.
She had two ultrasound scan (USS)-guided Percutaneous Aspiration, Injection, and Reaspiration (PAIR) procedures with absolute alcohol, done about 2 weeks apart. Approximately 3.6 L of straw-colored fluid was drained from the largest cyst during the first aspiration while 1.2 L was drained during the second aspiration. The drained fluid was subjected to cytology and microscopy, culture and sensitivity with a negative yield. Serological tests for Echinococcus and Entamoeba were not available. Repeat USS a day after the first aspiration further revealed multiple complex hepatic cysts and multiple hepatic solid masses. This, therefore, made us entertain an alternative diagnosis of hepatocellular carcinoma (HCC) with polymyositis. She was commenced on prednisolone and azathioprine as well as physiotherapy, with gradual tapering of prednisolone. Complementary transvaginal USS revealed multiple solid masses sandwiching the uterus and the bladder with a color score of 3. The uterus and the cervix were however normal for her age. These features suggested an additional impression of malignant adnexal masses.
USS-guided trucut biopsies of both the hepatic and adnexal masses revealed features of papillary cystadenocarcinoma of the ovary with metastasis to the liver on histology [Figure 2]. Ovarian tumor marker (CA125) was found to be elevated to 80 µ/mL (normal <35). However, immunohistochemical staining for molecular typing of the cancers was not done due to nonavailability. A final diagnosis of papillary cystadenocarcinoma of the ovary with liver metastasis and polymyositis was made. Her muscle power improved to 3/5 after about 2 weeks on the medications (prednisolone and azathioprine) and physiotherapy. She was counseled for chemotherapy which she declined and subsequently discharged on request. | Figure 2: Photomicrographs of metastatic papillary cystadenocarcinoma shown on (A) indicated by star. The arrow showed normal liver parenchyma H & E ×100. (B) Ovarian tissue displaying papillary cystadenocarcinoma, as indicated by the symbol papillary pattern. H & E ×200
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| Discussion | | |
We report a rare case of metastatic ovarian cancer presenting initially as multiple cystic and solid masses in the liver with proximal muscle weakness. The solid masses in the liver were initially obscured by the large cysts; however, they became obvious after the aspiration of the cysts. Liver cystic tumors may be solitary or multiple and can vary from benign to malignant which may pose a diagnostic challenge in clinical practice. Primary liver neoplasm presenting as cystic lesions is uncommon, and usually are solitary. These may be seen in giant cavernous hemangioma and rarely in HCC due to cystic degeneration.[9] Hepatic cystic metastasis is the most important diagnosis to be excluded when multiple cystic lesions are observed in the liver. In general, two different pathologic mechanisms can explain the cyst-like appearance of hepatic metastases. First, it may be due to the tumors outgrowing their blood supply, leading to central necrosis and cystic degeneration, which is commonly seen in metastasis from sarcoma, melanoma, and certain subtypes of lung and breast carcinoma.[10] Second, cystic metastases may also be seen with mucinous adenocarcinomas, such as colorectal or ovarian carcinoma.[11] Ovarian metastases usually spread by way of peritoneal seeding rather than hematogenous.[12]
Malignancy-associated myopathies, especially dermatomyositis and polymyositis, have been reported in the past, with only a few reports of ovarian cancer presenting initially as polymyositis.[6],[7],[8] However, none of the reported cases had a similar presentation with our index case.
We, therefore, concluded that the diagnosis in our patient is metastatic ovarian cancer to the liver as confirmed by histology of the pelvic mass and the raised CA125 levels. Therefore it is important to have a high index of suspicion of the possibility of gynecological malignancy with possible metastasis in any patient with a similar presentation like ours.
| Conclusion | | |
Although we missed part of the patient’s management, she declined chemotherapy, we hope that this case report will raise the awareness and index of suspicion of clinicians of such a rare presentation.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Ataseven B, Chiva LM, Harter P, Gonzalez-Martin A, du Bois A FIGO stage IV epithelial ovarian, fallopian tube and peritoneal cancer revisited. Gynecol Oncol 2016;142:597-607.  |
| 2. | Lu M, Li Z, Yang C, Deng K, Tan Q, Song W, et al. Sites of distant metastases and overall survival in ovarian cancer: A study of 1481 patients. Gynecol Oncol 2018;150:460-5. |
| 3. | Darnell RB, Posner JB Paraneoplastic Syndrome. New York: Oxford University Press, Inc.; 2011. p. 52-3. |
| 4. | Poulton TA, Crowther ME, Hay FC, Nineham LJ Immune complexes in ovarian cancer. Lancet 1978;2:72-3. |
| 5. | Stockton D, Doherty VR, Brewster DH Risk of cancer in patients with dermatomyositis or polymyositis, and follow-up implications: A Scottish population-based cohort study. Br J Cancer 2001; 85:41-5. |
| 6. | Ghosh A, Malak TM, Pool AJ Polymyositis and ovarian carcinoma: A case report. Arch Gynecol Obstet 2007;275:195-7. |
| 7. | Iavazzo C, Vorgias G, Papadakis M, Manikis P, Mavromatis I, Akrivos T Polymyositis in a patient with recurring ovarian cancer and history of unrelated breast cancer. Arch Gynecol Obstet 2007;276:81-4. |
| 8. | Kalogiannidis I, Papanikolaou A, Xanthakis I, Makedos A, Prapas N Clear cell ovarian carcinoma following polymyositis diagnosis: A case report and review of the literature. Hippokratia 2008;12: 181-5. |
| 9. | Jangwon L, Namhee L, Hye KY, Yeon JL, Sung JP Cystic degeneration of hepatocellular carcinoma mimicking mucinous cystic neoplasm: Case report. Korean J Gastroenterol 2019;73:303-7. |
| 10. | Lewis KH, Chezmar JL Hepatic metastases. Magn Reson Imaging Clin N Am 1997;5:319-30. |
| 11. | Sugawara Y, Yamamoto J, Yamasaki S, Shimada K, Kosuge T, Sakamoto M Cystic liver metastases from colorectal cancer. J Surg Oncol 2000;74:148-52. |
| 12. | Lundstedt C, Holmin T, Thorvinger B Peritoneal ovarian metastases simulating liver parenchymal metastases. Gastrointest Radiol 1992;17:250-2. |
[Figure 1], [Figure 2]
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