Correspondence Address:
Kaushik Saha
Tata Main Hospitals, Jamshedpur - 831 001, Jharkhand
India

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Dear Editor,

Pregnancy luteoma is a nonneoplastic lesion characterized by solid proliferation of luteinized ovarian theca lutein cells resulting in tumor-like ovarian enlargement. It is seen in association with pregnancy that usually regresses spontaneously with puerperium.[1] Pathogenesis is by stimulation of theca lutein cells by excessive human chorionic gonadotrophin (HCG) due to pregnancy. It may be unilateral or bilateral, usually asymptomatic, discovered incidentally at C-section or postpartum tubal ligation. However, it is rarely symptomatic. We report a case of pregnancy luteoma associated with missed abortion in a 21-year female who presented in the emergency department with acute abdomen and was provisionally diagnosed as ruptured tubal/fimbrial ectopic on ultrasonography (USG). To the best of our knowledge, in English literature, this is the first reported case of ruptured pregnancy luteoma associated with missed abortion masquerading ruptured tubal ectopic pregnancy. A brief review of relevant literature is also conducted.

A 21-year-old female presented to the emergency department with sudden onset pain abdomen. On physical examination, there was mild tachypnea, tachycardia with maintained mean blood pressure, and mild pallor. The patient was amenorrheic since the last two months, and a urine pregnancy test done in the emergency department was positive. Complete blood count revealed mild anemia with hemoglobin value of 10.5 gm/dl, mild neutrophilic leukocytosis, and raised erythrocyte sedimentation rate. Serum beta HCG level was raised (448.3 IU/ml). USG revealed a right adnexal mass, empty uterine cavity, and free fluid in the pouch of Douglus. The left fallopian tube and ovary were sonographically normal. A provisional diagnosis of ruptured tubal/fimbrial ectopic was made. Urgent laparotomy was done. Intraoperatively right fallopian tube appeared unremarkable; however, the right ovary was mildly enlarged with a ruptured cyst [Figure 2]. Partial oophorectomy was done, and the sample was sent for histopathological evaluation. Blood mixed peritoneal fluid of approx. 200 ml was irrigated. The macroscopical evaluation showed an irregular specimen, with no identifiable anatomical structure, tan white to yellow, solid with part cystic and hemorrhagic center, measuring 2.0 cm × 2.1 cm × 1.3 cm [Figure 1]. Microscopical examination of the H and E-stained section revealed solid sheets of large polygonal cells with abundant eosinophilic cytoplasm, central round nuclei, and variably prominent nucleoli. The central area of hemorrhage was seen. No nuclear atypia, mitosis, or pleomorphism was seen. Stroma was minimal with the presence of a delicate thin-walled vessel [Figure 1]. The patient had an uneventful postoperative stay in the hospital. She is doing well with two years of post-surgery follow-up.{Figure 1}{Figure 2}

Pregnancy luteoma (PL) was first described by Sternberg in 1963.[2] It is a rare nonneoplastic ovarian condition, possibly hyperplastic, with post-partum regression that develops in the later weeks of gestation.

PL is usually asymptomatic, except for the production of androgen in occasional cases resulting in hirsutism or virilization. Burandt et al.[3] reported androgenic manifestation in four out of 20 cases. More than 50% of female infants of virilized mothers are virilized; however, male fetuses are unaffected.[4] Rarely it may present with acute abdomen due to torsion and rupture associated with pregnancy, uterine or ectopic.[5] There are rare case reports of PL masquerading malignant tumor, presenting with massive ascites and raised CA 125.[6]

Grossly the lesion is typically well-circumscribed, measurement ranging from microscopic lesion to up to 20 cm, however usually measures between 5 and 10 cm. They are often bilateral; Burandt et al.[3] reported four out of 20 cases to be bilateral. They may be intact/ruptured, soft brown to focally hemorrhagic masses. Our case was irregular cystic with areas of hemorrhage and rupture.

Microscopically PLs are well-circumscribed masses with diffused, sheet-like growth of cells. The cells are polygonal, medium-sized with abundant eosinophilic cytoplasm, regular round nucleoli, and easily visible nucleolus. Stroma is minimal with barely perceptible delicate thin-walled vessels. There may be interspersed follicle-like spaces containing an eosinophilic colloid-like material. Cytologic atypia is rare and mitotic figures are infrequent. Malinac and Miller reported rare regressive changes in the form of alteration of cytoplasm from eosinophilic to pale and frothy in postpartum cases.[3]

PL may have many differential diagnoses clinically and histopathologically These may include Corpus luteum of pregnancy, Granulosa cell tumor, Hyperreaction leuteinalis, large solitary luteinized follicular cyst of pregnancy and puerperium, Steroid cell tumor, and Thecoma.[7] However, with the typical macroscopic and microscopic appearance of the tumor in an appropriate clinical setting, a correct diagnosis may be made. A detailed discussion of individual differentials is beyond the scope of this report.

The treatment of PL depends on presentation. The incidentally detected ones may be left as such for spontaneous regression to occur, close clinical monitoring and follow-up are suggested. Whereas cases presenting acutely with rupture/torsion may require urgent intervention in the form of emergency laparotomy, as in our case.[4]

To summarize, our case presented with acute abdomen, clinically and radiologically thought to be ruptured tubal ectopic because of suspected right adnexal mass in a setting of raised beta HCG; however, per operatively fallopian tubes appeared unremarkable with evidence of ruptured ovarian cyst, histologically confirmed to be ruptured PL. As there was no evidence of in utero/tubal gestation, a final diagnosis of ruptured PL associated with missed abortion was made.

To conclude, though PLs are incidental; usually an innocuous condition associated with gestation, these rare complications must be kept in mind. A high index of suspicion of this entity clinically and radiologically may on many occasions spare the patient of unwarranted surgeries, and the patient may be given a trial of more conservative management.

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