Pregnancy luteoma: a rare presentation and expectant management

Introduction

Pregnancy luteoma (PL) is a non-neoplastic tumor-like lesion of the ovary which was first described by Sternberg in 1963 [1]. Human chorionic gonadotropin (HCG) is responsible from the increased and uncontrolled cell proliferation on ovarian stroma and this pathophysiological mechanism may be the underlying reason for PLs [2], [3]. PLs appear as solid ovarian masses and can be detected during routine ultrasonography. The size of these cysts are most frequently between 5 and 10 cm, however, there are some reported PL cases which are extremely large (up to 20 cm) [3]. Some women can suffer from general symptoms like pelvic pain, lumbalgia and constipation. However, the majority of these rare lesions are detected incidentlly during cesarean section without any complaints. [2], [3]. Moreover, luteomas can be hormonally active (especially androgen secretion) and may cause maternal virilization [2].

We present a case of ovarian cysts mimicking a malignant ovarian mass during pregnancy, which was followed-up with expectant management until birth for the definitive diagnosis.

Case presentation

A 38-year-old primigravid woman presented to our clinic at 19 weeks of pregnancy with abdominal pain, abdominal tenderness, tachycardia and tachypnea. Her antenatal records were normal, she was diagnosed with abortus imminens in the 7^th week of pregnancy, and given oral progesterone supplement (micronized progesterone 100 mg/day) for 4 weeks. She was only taking oral vitamin supplements when she presented to our clinic.

On the first examination, a live fetus with a biometry of 20 weeks’ proportion was noted. The right ovary was found to be asymmetrically larger, and had a 51 × 44 × 51 mm heterogen solid mass with peripheral vascularization, Also, there were ascites in the abdominal cavity (Figure 1). She was hospitalized with suspicion of Meigs’ syndrome, ovarian torsion or ovarian malignancy.

Figure 1:

The ovarian mass and the ascites – at the first visit.

(A) Ascites in the abdominal cavity. (B) 51 × 44 × 51 mm heterogen solid mass with peripheral vascularization.

The results of tumor markers were: Ca125:2850 U/mL and alpha-fetoprotein (AFP): 49, 63 ng/mL, respectively. For the further evaluation of the ovarian mass, a lower abdominal magnetic resonans imaging (MRI) without intravenous contrast was performed. In the MRI, a right ovarian mass of 62 × 47 mm, with solid nature was reported. The mass was characterized as hyperintense on the T2 weighted images and hypointense on T1 weighted images (Figure 2). The right ovarian mass was suspected of a high posibility of malignancy.

Figure 2:

Right ovarian mass of 62 × 47 mm on MRI.

During hospitalization, due to worsening clinical condition and persisting tachypnea 2000 mL of ascites was drained and sent for cytological evaluation. The ascites were exudative in nature, and the cytological evaluation revealed benign cells. After the paracentesis the patient’s symptoms dramatically improved.

The patient was informed about the differential diagnosis and she decided to have close follow-up of the ovarian mass until delivery without any surgical intervention. During the follow-up, the fetus showed normal anatomy at the second trimester ultrasound screening, and the growth was proportional with the gestational weeks throughout pregnancy. A normal glucose challenge test was noted at the 24^th week of pregnancy. For the preterm contractions the patient was given two doses of betamethasone for lung maturation at the 30^th gestational week. The contractions relieved spontaneously. The patient showed no signs of hirsutism during the antenatal follow-up.

The mass showed no change in size and nature, and minimal ascites were observed until delivery without any need for additional paracentesis. The patient gave birth to a 3340 g male baby with an APGAR score of 9/10 at 38 weeks’ by elective cesarean section. During exploration a 5–6 cm solid adnexal mass was detected in the right ovary, no other gross pathology was detected in the pelvis. A unilateral salpingo-oophorectomy was performed and a frozen specimen was sent for investigation at surgery. The frozen section was reported as a sex-cord stromal tumor, so no other surgical intervention was made.

The mother and the baby were discharged from the hospital on the 3^rd postoperative day. The final pathology specimen was reported as PL.

Discussion

Ovarian cysts in pregnancy are usually related to pre-existing ovarian conditions, a new ovarian pathology presenting in pregnancy is very rare [3]. The incidence of adnexal mass in pregnancy is reported to be 0.05–3.2% [4]. The histological distribution of malignant ovarian tumors are germ cell tumors (35.5%), borderline ovarian tumors (27.9%), invasive epithelial carcinoma (25%) and sex-cord stromal tumors (11.8%). On the other hand, benign ovarian masses may be seen including benign cystic teratoma (7–37%), serous cystadenoma (5–28%), mucinous cystadenoma (3–24%), endometrioma (0.8–27%), paraovarian cysts (<5%), and leiomyoma (1–2.5%) [3], [4], [5]. Overall, malignant ovarian mass in pregnancy is a very rare condition with a rate of 1 in 1000 pregnancies [3], [6], [7].

Ovarian masses can be diagnosed during routine prenatal ultrasound or incidentlly during cesarean section [3]. Detection of an adnexal mass in pregnancy is important to prevent adverse perinatal and maternal outcomes. It has been reported that, there is an increased risk of spontaneous abortion, preterm contractions/preterm delivery, intrauterine fetal demise and vaginal bleeding in pregnancies if surgical interventions are performed for adnexal masses [4]. Furthermore, early diagnosis of an ovarian malignancy can be useful for prompt treatment.

PL is one of the benign ovarian masses occurring in pregnancy [3]. It is usually diagnosed incidentally at the time of cesarean section and most cases resolve completely in months after delivery [2], [3]. In our case, the patient presented with symptoms of abdominal pain, abdominal tenderness, tachycardia and tachypnea. These symptoms are not usually associated with PLs, they are more common with malignant ovarian masses. There are not many cases of luteomas presenting with these symptoms in the literature. There is a case described by Wang with massive ascites and elevated CA125 levels at the 15^th week of pregnancy after ovulation induction therapy. Due to the suspicion of malignancy, laparatomy was performed but a PL was detected [7]. It is clear that, PLs must be kept in mind in the differential diagnosis of malignant ovarian cysts.

PL always regress after birth. If clinical findings are compatible with a benign mass (or luteoma), unnecessary surgical interventions should be avoided because of the increased risk of miscarriage and preterm delivery [2], [3], [5]. In our presented case, the first examination was concordant with a malign mass. However, the paracentesis had shown benign cells and it was very helpful in the paliative management of the patient. It was not only useful for patient relief but also in the exclusion of malignancy. We evaluated the mass size and fetal growth by serial ultrasonography to avoid possible surgical complications until delivery.

PLs can be hormonally active and cause sudden onset of virilization in the mother and even in the female fetus in some cases [3], [8], [9]. We did not observe any signs of virilization in the presented case. However, clinicians should consider PL diagnosis in case of maternal virilisation occurence.

In conclusion, pregnancy luetomas are benign lesions, however, they can mimic advanced stage malignant ovarian masses in pregnancy. If surgical urgencies are excluded in clinically benign cyts, the patient can safely be followed-up until term with close monitoring.