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CASE REPORT Table of Contents   
Year : 2009  |  Volume : 52  |  Issue : 1  |  Page : 94-96
Bilateral benign non functional struma ovarii with Pseudo-Meigs' syndrome


1 Department of Pathology, Command Hospital (EC) Alipore, Kolkata 700 027, India
2 Department of Obstetrics and Gynecology, Command Hospital (EC) Alipore, Kolkata 700 027, India
3 Department of Endocrinology, Command Hospital (EC) Alipore, Kolkata 700 027, India

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   Abstract 

Bilateral presentation of benign Struma ovarii is rare and has not been reported frequently in published literature. A 70-year-old postmenopausal female presented with progressive ascites, bilateral pleural effusion and elevated CA-125 levels. The contrast-enhanced computed tomography (CECT) of the abdomen and pelvis revealed a heterogenous mass in the left adnexa. These findings were suspicious for an ovarian malignancy. After surgery the diagnosis of non functional, bilateral benign Struma ovarii was made. Struma ovarii is a specialized ovarian teratoma composed predominantly of mature thyroid tissue. It is associated with pleural effusion and ascites (Pseudo-Meigs' syndrome) in 5% of cases. The combination of struma ovarii and elevated CA-125 levels has been reported infrequently. This is a rare case of bilateral benign struma ovarii associated with Pseudo-Meigs' syndrome and elevated CA-125 levels. Surgical excision of the ovarian masses induced immediate resolution of the ascites and pleural effusion and a reduction of the serum CA-125 level.

Keywords: Bilateral struma ovarii, CA 125, Pseudo-Meigs′ syndrome

How to cite this article:
Rana V, Srinivas V, Bandyopadhyay S, Ghosh S K, Singh Y. Bilateral benign non functional struma ovarii with Pseudo-Meigs' syndrome. Indian J Pathol Microbiol 2009;52:94-6

How to cite this URL:
Rana V, Srinivas V, Bandyopadhyay S, Ghosh S K, Singh Y. Bilateral benign non functional struma ovarii with Pseudo-Meigs' syndrome. Indian J Pathol Microbiol [serial online] 2009 [cited 2020 Jan 19];52:94-6. Available from: http://www.ijpmonline.org/text.asp?2009/52/1/94/44978



   Introduction Top


Struma ovarii is a monodermal teratoma in which thyroid tissue predominates and is defined by the presence of thyroid tissue in more than 50% of the tumor. [1] Struma ovarii comprises 1-4% of benign ovarian teratoma. [2] Bilateral struma ovarii is very rare and has not been reported frequently. Struma ovarii usually presents as a pelvic mass and rarely presents as pseudo-Meigs' syndrome. [3] It may be mistaken as a malignant neoplasm when it is associated with elevated CA-125 levels. [4] We report a rare case of bilateral benign struma ovarii, accompanied by ascites, pleural effusion and an elevated CA-125 level.


   Case Report Top


A 70-year-old postmenopausal female presented in April 2007 with progressive abdominal distention of 6 months duration and breathlessness of 1 month duration. There was no history of fever, significant weight loss, jaundice, or bleeding per vaginum. There were no symptoms of hyperthyroidism. On general physical examination, there was no pallor, icterus, lymphadenopathy, pedal edema, or features suggestive of chronic liver disease. A systemic examination revealed ascites and bilateral pleural effusion. Investigations revealed a normal hemogram and serum biochemistry. A chest x-ray confirmed bilateral pleural effusion. A USG of the abdomen revealed ascites, omental thickening and a large pelvic mass with heterogenous echogenecity. The uterus and ovaries could not be identified separately from the pelvic mass. A CECT of the abdomen and pelvis confirmed a heterogenous mass in the left adnexa with the dimensions of 9.4 x 9.1 x 6.8 cm. No peritoneal implant or abdominal lymphadenopathy was seen. There was bilateral pleural effusion with no lung or pleural nodules. A diagnosis of a malignant ovarian neoplasm with ascites and bilateral pleural effusion was made. Repeated ascitic and pleural fluid analysis did not reveal any malignant cells. The serum CA-125 level was 284 U/ml (reference value <35U/ml). Her free-T3 and free-T4 values were within normal limits. In May 2007, the patient underwent a planned laparotomy during which a total abdominal hysterectomy with bilateral salphingoopherectomy and partial omentectomy was done.

Pathology

Gross examination of the left ovarian mass revealed a 7.5 x 5.5 x 4.0 cm mass with a multi-nodular external surface. Cut surface was multicystic with cysts varying from 1-4 cm in diameter filled with a brown jelly-like gelatinous material. The lining of the cysts was smooth with no solid or papillary areas. The right ovary was partially cystic and measured 2.0 x 1.5 x 1.0 cm. Cut surface revealed small cysts filled with a brown gelatinous material. On microscopic examination, the left ovarian mass revealed normal ovarian architecture only in one focus with replacement of the rest of the ovarian parenchyma by benign colloid filled thyroid follicles [Figure 1] and [Figure 2]. No cytological feature of malignancy was seen. No neural tissue, cartilage, or adnexal tissue was seen. Sections from the right ovary showed compressed normal ovarian architecture with replacement of the rest of the parencmhyma by benign colloid filled thyroid follicles. No cartilage, skin with adnexae, or neural tissue was seen. Both ovaries showed thyroid tissue in more than 50% of the respective architecture. The uterus and cervix were histologically normal. Sections from the omentum revealed no pathology. A diagnosis of bilateral benign struma ovarii was made.

In the post-operative period, there was gradual regression of ascites and pleural effusion. Repeat CA-125 levels 7 days after the surgery were 86U/ml and were <35U/ml after 1 month. Presently, the patient is on follow-up with no recurrence of her symptoms.


   Discussion Top


Struma ovarii was first described in 1899 by Boettlin. [5] Its pathogenesis remains controversial. Today, it is considered that struma ovarii is composed of mature thyroid tissue growing within ovarian teratomas. Although approximately 15% of ovarian teratomas contain a small, non-significant focus of thyroid tissue, only 1-3% are characterized by the presence of functional thyroid tissue or thyroid tissue occupying most of the mass. [2] Struma ovarii is defined by the presence of thyroid tissue in more than 50% of the tumor. [1]

The incidence of struma ovarii varies in different studies. Struma ovarii forms 1% of all solid ovarian tumors and 1-4% of benign ovarian teratomas. [2] The contralateral ovary may contain another teratoma, but the struma is rarely bilateral. Bilaterality is reported to be present in 6% of cases with the left ovary being more frequently involved. [6] In spite of exhaustive search of the existing literature, we were not able to find any documented case of bilateral benign non functional struma ovarii. This is a case with uncommon bilateral presentation of benign struma ovarii.

Struma ovarii usually presents after age of 40 and the peak age of incidence is in the fifth decade. The tumor always occurs as a pelvic mass, which may be palpable on physical examination, depending on the size and location. Most cases are incidentally found during clinical and imaging examination or laparotomy. Struma ovarii is usually non functional and only 8% of patients present with symptoms and signs of hyperthyroidism, as a result of autonomous activation of the thyroid tissue. [7] Our patient was clinically and biochemically euthyroid.

Ascites may be present in up to one-third of the cases of struma ovarii. [3] However, the association of ascites and pleural effusion with this tumor is uncommon. Pleural effusion and ascites is known to be associated with an ovarian fibroma/thecoma, a condition originally described by Meigs in 1937. When the same clinical features exist but involve other ovarian or gynecologic tumors, it is referred to as pseudo-Meigs'syndrome. Struma ovarii rarely presents as pseudo-Meigs' syndrome in about 5% of the cases. [4] Pseudo-Meigs' syndrome also occurs with benign cysts of the ovary, leiomyomas of the uterus and teratomas.

An ovarian mass and an elevated serum CA 125 level in a postmenopausal female generally suggest a malignancy. CA125, the glycoprotein defined by the antibody OC 125, is the most important clinical marker for the diagnosis, treatment and follow-up of epithelial ovarian cancer. [8] In a series by O'Connell, et al ., the predictive value of a CA-125 level greater than 35 U/mL was 60% for ovarian cancer and 84% for any type of malignancy. [9] Infrequently, benign struma ovarii is associated with elevated CA-125 levels. [4] If struma ovarii presents as pseudo-Meigs' syndrome and is associated with elevated CA-125 levels, it can be mistaken for a malignant process. [4] Our patient presented with progressive ascites, bilateral pleural effusion, was found to have elevated CA-125 levels and was clinically treated as malignant until proven benign histologically.

Extensive grossing is required to rule out any other component before labeling it as monodermal teratoma. Extensive serial grossing of both ovarian masses in our patient did not reveal any neural, cartilage, or skin with adnexal tissue or any other evidence of a teratoma. There were no features of malignancy.

Malignant transformation of struma ovarii is rare (5-10%) and may be follicular, papillary, or mixed in pattern and in rare cases can include elements of cystadenocarcinoma, Brenner tumor, carcinoid, or melanoma. [10] Most cases of malignant struma ovarii have been diagnosed on the basis of histologic criteria alone, with only about 20 cases presenting clinically appreciable metastatic disease. Metastatic spread, following the pattern of ovarian cancer, occurs in about 5% of malignant cases. [10] In our patient, there was no evidence of malignancy or detectable metastatic deposits. Surgical excision of the ovarian masses induced immediate resolution of the ascites and bilateral pleural effusion and normalization of the serum CA-125 level in our patient. The patient is on follow-up with no recurrence of her symptoms.

This is an uncommon case of bilateral presentation of benign non functional Struma ovarii with ascites, bilateral pleural effusion and elevated CA-125 levels. Surgical removal of the ovarian masses lead to rapid resolution of symptoms and a decrease in CA-125 levels. This case highlights that a pelvic neoplasm in a female presenting with hydrothorax, ascites and elevated CA-125 levels might be benign and that this condition rapidly resolves with surgical removal.

 
   References Top

1.Zaloudek C. Tumors of ovary. In: Fletcher Christopher DM editor. Diagnostic histopathology of tumors. 2 nd ed. Churchill Livingstone; 2000. p. 614.  Back to cited text no. 1    
2.Ayhan A, Yanik F, Tuncer R, Tuncer ZS, Ruacan S. Struma ovarii. Int J Gynaecol Obstet 1993;42:143-6.  Back to cited text no. 2    
3.Amr SS, Hassan AA. Struma ovarii with pseudo-Meigs' syndrome: Report of a case and review of the literature. Eur J Obstet Gynecol Reprod Biol 1994;55:205-8.  Back to cited text no. 3    
4.Loizzi V, Cormio G, Resta L, Fattizzi N, Vicino M, Selvaggi L. Pseudo-Meigs syndrome and elevated CA125 associated with struma ovarii. Gynecol Oncol 2005;97:282-4.  Back to cited text no. 4    
5.Boettlin R. Struma ovarii. Virchow's Arch Pathol Anat 1989;115:493-5.  Back to cited text no. 5    
6.Scully RE. Recent progress in ovarian cancer. Hum Pathol 1970;1:73-98.  Back to cited text no. 6    
7.Zakhem A, Aftimos G, Kreidy R, Salem P. Malignant struma ovarii: Report of two cases and selected review of the literature. J Surg Oncol 1990;32:61-5.  Back to cited text no. 7    
8.Bast RC, Feeney M, Lazarus H, Nadler LM, Colvin RB, Knapp RC. Reactivity of a monoclonal antibody with a human ovarian carcinoma. J Clin Invest 1981;68:1331-7.   Back to cited text no. 8    
9.O'Connell GJ, Ryan E, Murphy KJ, Prefontaine M. Predictive value of carbohydrate antigen-125 for ovarian carcinoma in patients presenting with pelvic masses. Obstet Gynecol 1987;70:930-2.  Back to cited text no. 9    
10.Dardik RB, Dardik M, Westra W, Montz FJ. Malignant struma ovarii: Two case reports and a review of the literature. Gynecol Oncol 1999;73:447-51.  Back to cited text no. 10    

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Correspondence Address:
Vandana Rana
Department of Pathology, Command Hospital (EC), Alipore, Kolkata - 700027
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.44978

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