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Theca lutein cysts a.k.a. hyperreactio luteinalis are benign cysts arising in the ovary as a result of increased sensitivity to hCG (human Chorionic Gonadotrophin). However 60 % have been noted in uncomplicated singleton pregnancies. They are usually bilateral and commonly associated with trophoblastic disease with sizes reaching upto 10cm. Only 51 cases associated with normal pregnancy have been reported in the literature. Usually they are managed conservatively and regress in the postnatal period 4. To our knowledge we are presenting the first case of a large, progressive theca lutein cyst presenting for the first time in the postpartum period. The cyst was managed laparoscopically.

INTRODUCTION

CASE REPORT

A 33 year old, gravida 2 para 1 patient with a history of polycystic ovaries and scoliosis, booked for antenatal care at 12 weeks gestation. The initial booking scan was at 12 weeks and a routine anomaly scan at 19 weeks were normal. At 40 weeks gestation, a healthy male infant weighing 3.36 Kg was delivered vaginally following a very short uneventful labour as a homebirth, lasting less than an hour in total. APGAR scores were 9 at 1 minute and 10 at 5 minutes.

The patient presented at four weeks postpartum with increasing abdominal distension and pain. A transvaginal and abdominal ultrasound scan reported a large, unilocular, anechoic cyst, measuring 144 mm x 120 mm x111mm, volume 1004ml extending to the subcostal margin, at 36/40 size (Fig.1, Fig.2 , Fig.3, Fig.4) Within 3 days, the cyst had enlarged to 224mm x 221 mm x 132 mm, volume 3421ml, unilocular, aseptate and thin-walled. The presumptive diagnosis was that of a serous cystadenoma, however a borderline tumour could not be ruled out due to the size and volume of the cyst. Tumour markers were all negative and Haemoglobin and CRP levels were normal.

DISCUSSION Ovarian cysts and tumours in the postpartum period are rare, the incidence of ovarian cancer is 1 in 12, 500 -25,000 pregnancies 1. It is thought that relative risk for ovarian carcinoma is lowest at 2 years post childbirth, suggesting a possible protective effect of childbirth and the puerperal period 2. Indeed a similarly low incidence is estimated for ovarian tumours and cysts in pregnancy 3. Hyperreactio luteinalis (multiple theca lutein cysts) which is usually associated with trophoblastic disease of pregnancy can present at any stage of pregnancy and occasional reports in the puerperium also. These cysts can be associated with marked ovarian oedema, haemorrhage, torsion rupture, haemoperitoneum, excessive ovarian androgen production (20 % ) and rarely fatality. There are reports of obstructive symptoms in labour and shoulder dystocia secondary to hyperreactio luteinalis5. It is common to observe regression of these cysts in the puerperium, however occasionally these persist for longer. It is common to expect ovarian volume and androgen concentrations to return to normal by 3 months postpartum4.

Due to increasing size and pain, a decision for urgent surgical exploration was made. Veress needle (Rocketmedical, Washington, England))was used for pneumoperitoneum and a 5mm port was used for laparoscopic entry through Palmer’s (left upper quadrant) point. The patient was found to have a large thick walled cyst arising from the right ovary extending from pelvic brim to subcostal margin. The cyst was excised with monopolar scissor and straw coloured contents aspirated using a 5mm suction. The internal structure was examined with the 5mm to rule out any suspicious areas. A cystectomy was carried out initially, however due to lack of any healthy residual ovarian tissue and moderate amount of bleeding from the ovarian hilum, a right salpingo-oopherectomy was performed. The patient made a good recovery and was discharged home the following day. Histology showed a fibrous tan coloured, partly congested tissue cyst capsule measuring 420 x 130 x 120 mm. Microscopically, a cyst lined with luteinised cells and the cyst wall is composed of fibro-connective tissue with ectatic blood vessels, which are features of a theca-lutein cyst.At the time of writing this report, the patient is 28 weeks into her third spontaneous pregnancy.

The presentation of a theca lutein cyst in this case report was atypical on numerous accounts. This cyst presented only in the postpartum period. For the duration of the pregnancy, there were no clinical complaints of distension, pain or peritonism. There was no ultrasonographic evidence of the cyst during the pregnancy. Theca lutein cysts characteristically present during the pregnancy, and regress in the postpartum period. However in this case, the theca lutein cyst enlarged during the postpartum period and was either non-existent or not detectable for the duration of pregnancy. It is unlikely that the cyst was present at this size during labour as the labour was uneventful, indeed the patient had an uncomplicated home birth. Uncharacteristically, in this case there were normal levels of bHCG in a singleton pregnancy. In hyperreactio luteinallis, theca lutein cysts are usually bilateral however, in this case, there left ovary appeared normal.

There is smaller blood loss with laparoscopic ovarian cystectomy when compared to open ovarian cystectomy 6. The estimated blood loss that the patient in this case report endured was 500ml which is acceptable given the size of the cyst, avoided blood transfusion and was discharged the next day. Laparoscopic surgery alllows for quick discharge and shorter hospital stay whichi si advantageous for women on the post-partum period. The alternative would be a lower transverse laparotomy with aspiration of the cyst contents to debulk followed by its removal. Laparoscopy also allowed careful examination of the exterior of the cyst and also interior of of the cyst and any potential omental/bowel attachments. A possible disadvantage of laparoscopic surgery is in the treatment of borderline tumours. This would have been indicated in the ultrasound scan findings and progressive presentation. This would require a full supra and inferior midline laparotomy which would cause significant morbidity. There is a statistically significant decrease in hospital stay when comparing laparoscopic management of ovarian dermoid cysts in comparison to open management of ovarian dermoid cysts6. The patient presented in this case report was discharged on day one post-operatively. Laparoscopy is associated with smaller costs also, this may be associated with the shorter hospital stay alone. A potential disadvantage of laparoscopic management of ovarian cysts is that operating time is greater, however this is not always the case6,7. Previous studies have found that cyst diameter in those who are treated by laparotomy tends to be larger6. However in this case, a 40cm cyst was successfully treated through laparoscopic surgery. Also, cyst contents are more likely to be spilt due to cyst rupture during laparoscopic management of ovarian cysts which is associated with increased rates of recurrence, as well as possible seeding of previously unrecognised malignancy 7.

CONCLUSIONIn conclusion, a theca lutein cyst should be considered as a possible differential diagnosis for large ovarian cysts presenting in the postpartum period. Laparoscopic management of such cysts of this size should be considered due to well-documented advantages of laparoscopic surgery over laparotomy, but would likely depend on the skill of the surgeon.

1. Nadereh Behtash3,1, Mojgan Karimi Zarchi 2*, Mitra Modares Gilani 3, Fatemeh Ghaemmaghami3, Azamsadat Mousavi3 and Fahimeh Ghotbizadeh3 : Ovarian carcinoma associated with pregnancy: A clinicopathologic analysis of 23 cases and review of the literature BMC Pregnancy and Childbirth 2008, 8:3

2. Qin Liu, Mats Lambe, Inkyung Baik, Sven Cnattingius, Tomas Riman,, Anders Ekbom, Hans-Olov Adami and Chung-Cheng Hsie. A Prospective Study of the Transient Decrease in Ovarian Cancer Risk Following Childbirth. Cancer Epidemiol Biomarkers Prev December 2006 15; 25083. Ueda M, Ueki M: Ovarian tumors associated with pregnancy. Int J Gyn Obstet 1996, 55:59-654. Niamh Phelan, Gerard S. Conway: Management of Ovarian Disease in Pregnancy. Best Practice and Research clinical endocrinology and metabolism 25; 2011:285-9925. FRÉDÉRIQUE PENAULT-LLORCA: Pathology of non-neoplastic ovarian enlargements6. Laberge PY, Levesque S: Short-term morbidity and long-term recurrence rate of ovarian dermoid cysts treated by laparoscopy versus laparotomy. J Obstet Gynaecol Can. 2006 Sep;28(9):789-93.7. Medeiros L, Rosa DD, Bozzetti MC, Fachel JMG, Furness S, Garry R, Rosa MINES, Stein AT: Laparoscopy versus laparotomy for benign ovarian tumour (Review). The Cochrane Library 2009, Issue 1

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