Volume 198, Issue 5 , Pages e9-e10, May 2008
A case of recurrent hyperreactio luteinalis
Article Outline
Hyperreactio luteinalis (HL) in normal pregnancy has been reported previously. However, only a few cases of HL recurrence have been reported. The present report describes HL in a normal singleton pregnancy presenting with an acute abdomen requiring surgical intervention. In a subsequent normal singleton pregnancy, HL recurred and was treated conservatively.
Key words: hyperreactio luteinalis, normal singleton pregnancy, recurrence
Hyperreactio luteinalis (HL) is characterized by bilaterally enlarged ovaries containing multiple self-limited theca lutein cysts. HL typically occurs with gestational trophoblastic disease, although about 80 reports of unassociated HL have been published. HL may be diagnosed prenatally by ultrasonography, and requires intervention for maternal indications.
Case Report
A 26 year old woman was seen at our prenatal department for amenorrhea for 7 weeks. She had menarche at 13 years of age and had regular menstrual cycles. Her past history was negative for induction of ovulation. Ultrasonography revealed an intrauterine pregnancy consistent with a gestational age of 7 weeks. Two weeks later, she experienced lower back pain and visited our department. Ultrasonography revealed moderate ascites and a 105 × 55 × 60 mm multilocular cystic mass surrounding the uterus. Laboratory findings including human chorionic gonadotropin levels were in the normal range for the gestational age.
Considering the gestational age and lack of malignant signs, she remained under observation until her symptoms exacerbated at 14 weeks of gestation, and she was admitted to the hospital. She had an ultrasonographic appearance of a mass with bright loculi suggested hemorrhage and/or malignancy. Because of the enlarged mass and acute abdomen, laparotomy was performed at 15 weeks of gestation. Exploration revealed bilateral multicystic ovaries measuring 20 × 15 × 12 cm (Figure 1). A left salpingo-oophorectomy was initially performed for rapid diagnosis, and multiple benign luteinized follicle cysts were revealed (HL) (Figure 2).
FIGURE 1.
Macroscopic view of bilateral ovaries
Ovaries were bilaterally and symmetrically enlarged.
Onodera. A case of recurrent hyperreactio luteinalis. Am J Obstet Gynecol 2008.
FIGURE 2.
Microscopic view of the ovary
Cyst lining comprises granulosa and theca layers, both of which exhibit marked luteinization.
Onodera. A case of recurrent hyperreactio luteinalis. Am J Obstet Gynecol 2008.
After confirmation, a right ovarian cystectomy was performed. Although the right ovary contained numerous lutein cysts filled with hemorrhagic fluid and coagulation, it was thought to be functional. The patient had an uneventful postoperative course, except for severe anemia. After discharge, she had an uneventful pregnancy and spontaneously delivered a healthy female baby weighing 3178 g at 39 weeks of gestation. She was informed that ultrasonographic follow-up was necessary because of the possibility of HL recurrence in subsequent pregnancies.
Three years 2 months after the first delivery, she was again seen at our prenatal department at 6 weeks of gestation. Ultrasonography showed a singleton pregnancy and no adnexal masses. The pregnancy progressed uneventfully until multilocular cysts of the right ovary measuring 60 × 46 mm were detected at 10 weeks of gestation. She experienced mild abdominal pain. Although the cysts enlarged to 90 × 60 × 54 mm at 18 weeks of gestation, the condition had regressed after 22 weeks of gestation. Six weeks later, all cysts disappeared. The patient had no further complications and vaginally delivered a healthy female baby weighing 3772 g at 39 weeks. In these pregnancies, the patient and her infants showed no signs of virilization, and pathological examination of placenta revealed no abnormalities.
Comment
HL is a rare, typically self-limited syndrome with bilaterally enlarged ovaries containing multiple theca lutein cysts. HL most frequently accompanies disorders associated with elevated human chorionic gonadotropin levels, such as trophoblastic disease, fetal hydrops, and multiple gestations. Since Burger described the first case of HL unassociated with trophoblastic disease in 1938, about 80 such cases have been published in English literature. Most cases (54%) are noted in the third trimester, whereas 16% present in the first trimester.1 Symptomatic patients present with maternal virilization (25%), ascites, and abdominal pain. In addition, maternal virilization in HL shares similarities with both luteoma and polycystic ovarian syndrome of pregnancy associated with elevated androgen levels.2
Recurrence of HL in subsequent pregnancies has been reported in only a few cases. In 1 report, the patient aborted both pregnancies,3 and in the other, HL appeared incidentally at the first cesarean section.4 Our case is particularly noteworthy because, in contrast to the first pregnancy in which the course of HL required surgical intervention, the second pregnancy progressed without a surgery; furthermore, the patient remained fertile postoperatively and vaginally delivered healthy babies. The typical course of HL is natural regression; however, it may present with acute abdomen because of hemoperitoneum secondary to rupture and torsion. In our case, hemorrhage into the cysts might have caused pain and severe anemia, although neither torsion nor rupture occurred.
It is necessary to consider HL when ovarian multilocular cysts are noted in early gestation, although HL is an extremely rare condition in normal singleton pregnancies. Exacerbation of symptoms and/or apparent malignancy in HL can lead to unnecessary oophorectomy.
References
- . Bilateral ovarian lutein cysts associated with hydrops of fetus and lacenta. Int Congress Voor Verlaskunde en Gynecol. 1938;2:440–444
- . Recurrent maternal virilization during pregnancy caused by benign androgen-producing ovarian lesions. Am J Obstet Gynecol. 1992;167:1342–1343
- . Bilateral theca lutein cysts associated with an apparently normal pregnancy. Am J Obstet Gynecol. 1961;81:1033–1035
- . Recurrent theca lutein cysts. Obstet Gynecol. 1963;21:339–342
PII: S0002-9378(07)02290-9
doi:10.1016/j.ajog.2007.12.010
© 2008 Mosby, Inc. All rights reserved.
Volume 198, Issue 5 , Pages e9-e10, May 2008
