Kougioumtsidou Anna, Kougioumtsidou Niki, Athanasopoulou Maria, Pados George, Tarlatzis Vasileios

1st Department of Obstetrics and Gynecology, Aristotelian University of Thessaloniki, Papageorgiou hospital, Thessaloniki, Greece

Correspondence: Kougioumtsidou Anna, 1st Department of Obstetrics and Gynecology, Aristotelian University of Thessaloniki, Papageorgiou hospital, GR-54623, Thessaloniki, Greece. E-mail: This email address is being protected from spambots. You need JavaScript enabled to view it.

 

 


 

 

Abstract

Ovarian goiter is a rare tumor of the ovary that is characterized by a presence of thyroid tissue in the ovary and occupies more than 50% of the total mass of the tumor. It is accompanied by ascites in the 1/3 of cases. Τhe combination of struma ovarii, ascites and increased levels of CA125 is extremely rare. We report a case of a 77 years old woman with ascites, a big mass on the left ovary and increased levels of CA125, which are signs and symptoms of malignant tumor of the ovary.The patient underwent surgical removal of tumor with rapid remission of the ascites and decrease of the levels of CA125. Struma ovarii can imitate a malignant tumor of the ovary, especially when it is accompanied by ascites and increased levels of CA125.

Key words: ovarian goiter, struma ovarii, ascites, thyroid tissue, CA125

 

 

Ovarian goiter is a rare type of tumor of the ovaries, that constitutes 0.3-1.2% of all the ovarian luteomas and 2.7% of the dermoid tumors of the ovary1. It is characterized by a presence of thyroid tissue in the ovary that occupies more than 50% of its total mass. Struma ovarii is usually benign, but in rare occasions (5%) we notice a malignant change2. Preoperative diagnosis is difficult, since there are no notable symptoms and points of the disease. Patients usually come in with asymptomatic ovarian mass, widespread abdominal pain, distension of the abdominal walls due to ascites and abnormal ovarian bleeding. While ascites appears in 1/3 of the cases3, there is rarely an additional increase of the CA125. Few cases of strumaovarii have been reported to be accompanied by a pseudo-Meigs syndrome. Thyrotoxicosis appears in 5% of the cases4.

A 77 years old woman is presented with ascites, ovarian mass, increased levels of CA125, without symptoms of hyperthyroidism. This case was treated at first as a malignant tumor of the ovary and was submitted to exploratory laparotomy surgery.

 

Case report

The case of a 77 years old woman is reported, in menopause (since 20 years) with a maternity medical history of 8 natural childbirths and 3 spontaneous abortions. From the personal history, the following are reported: appendectomy, coronary disease, heart failure under treatment and mild degree of obstructive pulmonary disease. The woman came in the outpatient clinic of the First Department of Obstetrics and Gynecology of the Aristotle University of Thessaloniki at Papageorgiou hospital, due to a two month's widespread abdominal pain and meteorism. During the physical examination it was detected that there was distension of the abdominal walls due to a palpable mass on the lower abdomen, on the anatomical position of the left adnexal region. The woman was admitted to our clinic for further examination. An abdominal ultrasound and a computed tomography of the upper and lower abdomen and the retroperitoneal space were conducted, where moderate degree of ascites and cystic mass to the space of distribution of the left ovary, with dimensions 7 x 5 x 4cm, were detected.

The uterus was of normal size while the right adnexum was not depicted. In the rest of the intra-abdominal organs (liver, spleen, kidneys and adrenal glands) no pathological findings were detected. During the examination, a computed tomography of the chest was done, which showed absence of pleural effusion and absence of swollen mediastrinal and portal lymph nodes. In the lab test, an increased rate of CA125 (264.0 U/ml) was observed, while CEA (1.0 U/ml), CA15-3 (15.6 U/ml), CA19-9 (19.25 U/ml), and AFP (4.83 U/ml) were within normal limits. The rest of the test had no pathological findings.

The patient underwent exploratory laparotomy. Approximately 500ml of a tinged yellow ascetic fluid was found and was sent for a cytology examination. A complete hysterectomy on both andexasides, a removal of a multiple space-occupying tumor on the left ovary (Figure 1) and a partial omentectomy were conducted. No traces of intraperitoneal spread of the disease were noticed.

The histopathological examination of the tumor presented struma ovarii (thyroid tissue of follicles of various diameter filled with colloid) (Image 2), without omental cakes and without malignant cells at the cytology examination of the ascetic fluid. The uterus, the right ovary, the right salpinx and the left salpinx were examined with no pathological findings.

The postoperative course was smooth and the check of the functioning of the thyroid gland two days later was normal. The patient was discharged the seventh postoperative day. The recheck two months later showed remission and improvement of the clinical presentation without evidences of ascites and the CA125 levels were within normal limits.

  

 

image11 low        image12 low

 

 Figure 1. Macroscopic view of the struma ovarii                                   Figure 2. Typical microscopic presentation  of struma ovarii

 

Discussion

Struma ovarii is characterized by a presence of thyroid tissue that occupies more than 50% of the tumor's total mass and it was first described in 18995. The tumor is usually benign, but in rare occasions (5%) we notice a malignant change. Despite the fact that the tumor mainly consists of thyroid tissue, thyrotoxicosis appears only in 5% of the cases. The typical age of appearance is the 5th and 6th decade. It rarely appears before adolescence5. The clinical presentation of struma ovarii is similar to the other cancers of the ovary, with no special characteristics. Abdominal pain, palpable abdominal mass, ascites, abnormal vaginal bleeding are the most common symptoms. In rare occasions it could be accompanied by pseudo-Meigs syndrome and clinical hyperthyroidism3. Differential diagnosis must be done from all the ovarian tumors (benign and malignant), ectopic pregnancy, hydrosalpinx, tubo-ovarian abscess. The treatment is surgical and varies according to the age of the patient and the wish to preserve the fertility.

The increase of CA125 combined with ascites can usually be found in cases of malignant epithelial tumors of the ovary and more rarely in cases of endometrial, colorectal, breast and lung cancer. Moreover, the increase of its levels might accompany situations such as menstruation, pregnancy, endometriosis and ovarian fibroma6. The precise mechanism of the increase of the levels of CA125 in the case of struma ovarii is unknown. The probable cause is the inflammation of the peritoneum, the stimulation of the mesothelial cells and, as a result, the increased production of CA125 from its surface7. Concerning ascites, there are various speculations, such as the obstruction of the peritoneal lymphatic vessels by the overian mass, the inflammatory response of the peritoneal and the increased permeability of the membranes at the surface of the tumor8. An ovarian mass combined with increased levels of CA125 and ascites to a woman in menopause usually points to malignant mass. During the review of the literature, eight reports of cases with the stated findings were discovered3. All cases were treated at first as malignant tumors (Table 1).

In our case, a patient with ovarian mass, ascites and increased levels of CA125 is depicted. She was treated surgically with removal of the tumor and total hysterectomy, with total remission of the ascites and a decrease of the CA125 to normal levels right after the operation that still remains two months later. In conclusion, even though few similar cases are reported in literature, ovarian goiter should be included to the differential diagnosis of adnexal masses that coexist with ascites and increased levels of CA125.

 

table-struma

 

 

Conflict of interest

All authors declare no conflict of interest

 

 

 

References

1.Roth LM, Talerman A. The enigma of struma ovarii. Pathology 2007;39:139-46. PubMed

2. Bal A, Mohan H, Singh SB, Seghal A. Malignant transformation in mature cystic teratoma of the ovary: report of five cases and review of the literature. Arch Gynecol Obstet 2007;275:179-82. PubMed

3. Mui MP, Tam KF, Tam FK, Ngan HY. Coexistence of struma ovarii with marked ascites and elevated CA-125 levels: case report and literature review. Arch Gynecol Obstet 2009;279:753-7. PubMed

4. Yoo SC, Chang KH, Lyu MO, Chang SJ, Ryu HS, Kim HS. Clinical characteristics of struma ovarii. J Gynecol Oncol 2008;19:135-8. PubMed

5. Yassa L, Sadow P, Marqusee E. Malignant struma ovarii. Nat Clin Pract Endocrinol Metab 2008;4:469-72. PubMed

6. Leung YC, Hammond IG. Limitations of CA 125 in preoperative evaluation of a pelvic mass: struma ovarii and ascites. Aust N Z J Obstet Gynecol 1993;33:216-7. PubMed

7. Lin JY, Angel C, Sickel JZ. Meigs syndrome with elevated serum CA125. Obstet Gynecol 1992;80:563-5. PubMed

8. Abad A, Cazorla E, Ruiz F et al. Meigs syndrome with elevated CA125: case report and review of the literature. Eur J Obstet Gynecol Reprod Biol 1999;82:97-9. PubMed

9. Jotkowitz MW, Gee DC. Unique case of massive ascites, extreme elevation of serum CA125 tumor marker. Aust N Z J Obstet Gynecol 1993;33:453-4. PubMed

10. Mancuso A, Triolo O, Leonardi I, De Vivo A. Struma ovarii: a rare benign pathology which may erroneously suggest malignancy. Acta Obstet Gynecol Scand 2001;80:1075-6. PubMed

11. Loizzi V, Capuccini F, Berma ML. An unusual presentation of struma ovarii mimicking a malignant process. Obstet Gynecol 2002;100:1111-2. PubMed

12. Bokhari A, Rosenfeld GS, Cracchiolo B, Heller DS. Cystic struma ovarii presenting with ascites and an elevated CA125 levels. J Reprod Med 2003;48:52-6. PubMed

13. Rim SY, Kim SM, Choi HS. Struma ovarii showing clinical characteristics of ovarian malignancy. Int J Gynecol Cancer 2005;15:1156-9. PubMed

14. Guida M, Mandato VD, Spiezio Sardo A. Coexistence of Graves' disease and benign Struma ovarian in a patient with marked ascites and elevated CA125 levels. J Endocrinol Invest 2005;28:827-30. PubMed