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LETTER TO EDITOR
Year : 2011  |  Volume : 48  |  Issue : 3  |  Page : 364-366
 

A pseudo case of atypical pseudo-Meigs syndrome


1 Redcliffe Hospital, Queensland, Australia
2 The Prince Charles Hospital, Queensland, Australia

Date of Web Publication14-Sep-2011

Correspondence Address:
D A Thaker
Redcliffe Hospital, Queensland
Australia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-509X.84914

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How to cite this article:
Thaker D A, Stride P J, Dettrick A. A pseudo case of atypical pseudo-Meigs syndrome. Indian J Cancer 2011;48:364-6

How to cite this URL:
Thaker D A, Stride P J, Dettrick A. A pseudo case of atypical pseudo-Meigs syndrome. Indian J Cancer [serial online] 2011 [cited 2019 Aug 24];48:364-6. Available from: http://www.indianjcancer.com/text.asp?2011/48/3/364/84914


Sir,

A 54-year-old lady presented with right-sided pleural effusion. She had been admitted 1 year earlier with similar complaints, when pleural aspiration revealed an exudate, thought to have been due to a resolving infection and requested follow-up radiology was omitted. Pleural fluid biochemistry again revealed an exudate. Blood investigations ruled out autoimmune and other nonmalignant causes of effusion. Tumor marker studies were normal as well. The CT chest and abdomen [Figure 1] and pelvic ultrasound revealed a lesion arising from the left adnexa with a 6 cm focus of central calcification compatible with a dermoid. The pleural effusion was drained and subsequently bilateral oophorectomy was performed. Histopathology identified a right-sided benign serous cystadenoma and a left-sided benign cystic teratoma in which respiratory epithelium, squamous epithelium, skin adnexal structures, bone with marrow, myxoid stroma, and mucinous epithelium were identified [Figure 2]. A draft case report was composed by the excited authors, but diagnostic hubris followed with the recurrence of an even larger right pleural effusion, 2 months after surgery, thus excluding Meigs syndrome according to Meigs original definition. A thoracoscopic-assisted pleural biopsy was performed, and pleural membrane with small white nodules was removed. Microscopy revealed diffuse mesothelial malignancy involving the pleura with skip areas of reactive mesothelium.
Figure 1: Chest and abdominal CT, showing pleural effusion (white arrow) and ovarian teratoma (black arrow)

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Figure 2: Ovarian teratoma H and E, ×40 original magnification showing respiratory epithelium, stroma, and bone

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Immunohistochemistry of the tumor cells was consistent with a malignant mesothelioma [Figure 3] and [Figure 4]. Subsequent repeat enquiries with the patient and her husband revealed that patient's husband had work-based exposure to asbestos and patient had washed his clothes for many years previously. The association of ascites and a pleural effusion with an ovarian tumor was first recorded in the 19 th century. Universal recognition occurred when Meigs described seven cases of pleural effusion, ascites, and benign ovarian fibroma in 1937. [1] In 1954, Meigs limited the syndrome to cases where the removal of the tumor cures the disease. [2] Meigs syndrome is a desirable unifying, though not always correct diagnosis, as it detects an uncommon problem that crosses the boundaries of single organ specialists, and is a curable benign problem. Schmitt et al. suspected this diagnosis in a patient with ascites, hydrothorax, simple serous ovarian inclusion cysts, and an elevated CA 125; however, systemic lupus erythematosus was the subsequent correct diagnosis. [3] The pathophysiology of ascites in Meigs syndrome remains speculative, with proposed mechanisms including production of ascitic fluid by the tumor, lymphatic obstruction, hormonal stimulation, release of inflammatory mediators, or tumor torsion. The pleural effusion is thought to be caused by migration of fluid and protein perhaps by lymphatic channels across the diaphragm, but the mechanism of pleural effusion development is even more obscure in the absence of ascites. The effusion is classically, though not exclusively a transudate. Malignant disease should be excluded, but low titers of CA 125 have little significance, neither implying nor excluding ovarian carcinoma. [4]
Figure 3: Mesothelioma, pleural biopsy, H and E, ×40 showing exuberant papillary growth on the surface and invasion into chest wall fat

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Figure 4: Mesothelioma, pleural biopsy, ×40 original magnification. Immunohistochemistry, calretinin antibody strong reaction also showing invasion

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Fibromas and thecomas are the most common benign solid ovarian tumors, comprising at least 80% of the total. Approximately 1% of these are associated with Meigs syndrome. Majzalin et al. reviewed the first 128 reported cases of Meigs syndrome up to 1964. Majzalin found that 65% of pleural effusions were right-sided as in our case. [5] Ascites and hydrothorax associated with a benign ovarian tumor other than a fibroma is designated pseudo-Meigs syndrome. Atypical Meigs syndrome is characterized by an ovarian tumor and a pleural effusion, but no ascites, as in our case. We initially suspected this to be a previously unreported case of atypical pseudo-Meigs syndrome associated with a teratoma, but subsequent progress confirmed the old adage that an uncommon presentation of a common disease is more likely than a common presentation of a very rare disease. The presence of an asymptomatic ovarian tumor should not retard appropriate investigation of a recurrent pleural effusion.

 
  References Top

1.Meigs J, Cass J. Fibroma of the ovary with ascites and hydrothorax: With a report of seven cases. Am J Obstet Gynaecol 1937;33:249-67.  Back to cited text no. 1
    
2.Meigs J. Fibroma of the ovary with ascites and hydrothorax: Meigs' syndrome. Am J Obstet Gynaecol 1954;67:962-85.  Back to cited text no. 2
    
3.Schmitt R, Weichert W, Schneider W. Pseudo-pseudo Meigs' syndrome. Lancet 2005;366:1672.  Back to cited text no. 3
    
4.Moran-Mendoza A, Alvarado-Luna G, Calderillo-Ruiz G. Elevated CA125 level associated with Meigs' syndrome: Case report and review of the literature. Int J Gynaecol Cancer 2006;16:315-8.  Back to cited text no. 4
    
5.Majzalin C, Stevens F. Meigs syndrome: Case report and review of the literature. J Int Coll Surg 1964;42:625-30.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]

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