Download Pregnancy complicated by spontaneous ovarian hyperstimulation

Archives of Perinatal Medicine 15(3), 233-235, 2009
CASE REPORT
Pregnancy complicated by spontaneous ovarian
hyperstimulation syndrome and hyperthyroidism
BEATA MARCINIAK1, GRZEGORZ PIETRAS1, LECHOSŁAW PUTOWSKI2, ŻANETA KIMBER-TROJNAR1,
BOŻENA LESZCZYŃSKA-GORZELAK1, JAN OLESZCZUK1
Abstract
Ovarian hyperstimulation syndrome (OHSS) is potentially life-threatening complication associated with ovarian
induction. OHSS is a very rare event in spontaneous pregnancies, most often in situations of multiple or molar
gestations, hypothyroidism or polycystic ovary syndrome. We describe a patient with naturally conceived pregnancy associated with spontaneous OHSS complicated by hyperthyroidism. The patient was admitted at 17 weeks
of gestation with abdominal pain because of bilateral multilobulated cystic ovaries to the Chair and Department
of Obstetrics and Perinatology of Medical University of Lublin. The woman was treated with antithyroid drug and
successfully delivered at 36 weeks of gestation.
Key words: spontaneous ovarian hyperstimulation syndrome, pregnancy, hyperthyroidism
Introduction
Ovarian hyperstimulation syndrome (OHSS) is a rare iatrogenic complication of ovarian stimulation that
occurs during either the luteal phase or early pregnancy
[1]. It has been described to arise after treatment with
exogenous gonadotropins, clomiphene citrate and gonadotropin-releasing hormone agonists [1, 2]. Women without any pharmacological intervention are hardly ever
diagnosed with spontaneous OHSS [3,4]. Patients with
polycystic ovaries are more sensitive to the endogenous
gonadotropins and more likely develop OHSS [2,5].
The association of OHSS and endocrinopathies in
pregnant women may create a clinical problem. We describe a case of spontaneous OHSS in a patient who became pregnant naturally and suffered from hyperthyroidism.
Case presentation
A 38-year-old woman, gravida 3, was admitted to the
Chair and Department of Obstetrics and Perinatology of
Medical University of Lublin at 17 weeks of gestation with
abdominal pain, nausea and vomiting. She had a history
of two unremarkable normal pregnancies. The patient
conceived spontaneously and denied having ever taken
any ovulation inducing agent. Hyperthyroidism was reported six years before but antithyroid therapy was
ended two years later.
Vomiting, nausea and abdominal disturbances started at 10 weeks of gestation and increased up to admitting to our unit.
1
2
Based on her history of endocrinopathy, thyroid
function was measured. Thyrotropin (TSH) was < 0.04
mIU/l (reference range 0.4-4.9 mIU/l), free triiodothyronine (FT3) 33.2 pmol/l (3.0-7.0 pmol/l) and free thyroxine (FT4) 76.4 pmol/l (12.0-22.0 pmol/l). Treatment
with 150 mg propylthiouracil per day was started.
Abdominal and pelvic ultrasound examination confirmed a single, normal intrauterine pregnancy of 17
weeks and bilaterally enlarged multicystic ovaries measuring 120 × 108 mm on the right and 120 × 117 mm on
the left (Fig. 1). Placenta was normal. There was no fluid
in the cul-de-sac.
The levels of human chorionic gonadotropin (hCG)
were markedly elevated between 16 and 19 weeks of
gestation. Serum levels of hCG were 609.964 mIU/ml
(normal values: 5.730-54.585 mIU/ml), 548.883 mIU/ml
(5.267-57.465 mIU/ml), 458.111 mIU/ml (3.180-51.360
mIU/ml) and 442.763 mIU/ml (5.879-55.183 mIU/ml) at
16, 17, 18 and 19 weeks of gestation respectively. No
signs of virilization were observed.
A complete blood count, electrolytes, liver function
and coagulation profile gave results within normal limits.
A chest x-ray revealed no abnormalities. Conservative
management consisted of bed rest, close monitoring
(blood pressure, pulse and urinary output) and antithyroid therapy. Ultrasound-guided fine needle aspiration of the largest ovarian cysts (400 ml) was performed
at 18 weeks of gestation because of increasing abdominal pain. The procedure was repeated at 21 weeks of
gestation and 200 ml of fluid was obtained. No compli-
Chair and Department of Obstetrics and Perinatology, Medical University of Lublin, Poland
II Chair and Department of Gynecology, Medical University of Lublin, Lublin, Poland
234
B. Marciniak, G. Pietras, L. Putowski, Ż. Kimber-Trojnar, B. Leszczyńska-Gorzelak, J. Oleszczuk
cations were observed. Aspirated fluid was negative for
malignant cells, bacteria and fungi.
Fig. 1. Ultrasound examination of left ovary
During therapy, at 26 weeks of gestation thyroid
function was normalized (TSH 0.04 mIU/l, FT3 16.5
pmol/l, FT4 5.4 pmol/l). Treatment with propylthiouracil
(100 mg/day) was continued. Ultrasound examination
performed at 20 weeks of gestation showed normal fetal
growth and reduction of left ovarian size to 64 × 36 mm
and right to 33 × 24 mm.
At 36 weeks of gestation the patient went into labour and underwent an uncomplicated vaginal delivery
of male newborn weighing 2340 g, but showed hypospodiasis. Histological examination showed normal placenta.
The hCG level at delivery was 4 mIU/ml and ultrasound
examination revealed normal ovaries.
Discussion
Spontaneous OHSS is very infrequent and is always
reported during pregnancy. Only a few cases have been
described in singleton gestation [2, 5-10]. Some of them
were associated with primary hypothyroidism [3, 4, 11].
A series of cases of recurrent OHSS can be found in the
literature [5, 8, 12].
The woman referred to us conceived spontaneously.
Previous two pregnancies were normal; she was never
diagnosed polycystic ovarian syndrome. Abdominal pain,
nausea, vomiting started at 10 weeks of gestation were
reported by other authors [3, 5, 6]. On account of the
fact that she had weight loss, distress, palpitations, tachycardia and a history of thyroid dysfunction, thyroid
hormones were measured. Laboratory data showed hyperthyroidism. Because normal values of TSH were associated with very high FT3 and FT4 levels, hCG was
checked. Markedly elevated hCG noted in our case can
usually be connected with multiple gestation, molar
pregnancy, choriocarcinoma or various germ cell tumors
[13, 14]. In our patient ultrasound examination showed
singleton gestation with normal fetus and placenta.
Similar findings were noted in cases classified as
hyperreactio luteinalis [15]. That uncommon condition
complicating pregnancy is characterized by ovarian
enlargement caused by presence of multiple luteinized
follicular cysts formed in response to hCG stimulation and
is usually observed in the third trimester of pregnancy.
The clinical presentation of hyperreactio luteinalis can
mimic OHSS, but this syndrome is observed in the first
trimester [16]. Virilization is seen in up to 25% of patients
[15]. Our patient developed her signs in the second
trimester and no androgenization was observed.
High levels of hCG are not observed in all cases of
OHSS [2, 17]. In hypothyroid women, elevated concentrations of TSH may stimulate follicular growth and
mediate OHSS because of the presence of nuclear thyroid receptors in the granulosa cells [4, 10, 11, 18]. The
casual relationship between hypothyroidism and OHSS
is suggested by the constant regression of the ovarian
cysts after the institution of thyroid hormone replacement therapy [3]. In contrast to citied reports, our case
shows appearance of hyperthyroidism. Ludwig et al. [14]
suggested that hyperthyroidism which occurred neither
before nor after pregnancy in their patient with a partial
hydatidiform mole and a triploidy of the fetus was due to
the high hCG concentration and spontaneous OHSS. It
has been shown recently that hCG was able to stimulate
TSH and FSH receptors in vitro in conditions that mimic
high ligand concentrations [19-22]. It seems probable
that identification of hypothetical mutation in receptors of
glycoprotein hormones, i.e. hCG, LH, FSH and TSH, could
help us to explain the problem of coexisting hyperthyroidism and spontaneous OHSS in our patient [19, 23].
Women with OHSS require treatment consisting of
bed rest, close monitoring and fluid therapy. In connection with the fact that our patient’s symptoms, i.e. nausea, vomiting, abdominal distension, enlarged ovaries
and neither ultrasound nor clinical evidence of ascites
corresponded to grade 2 of OHSS according to the classification proposed by Golan et al. [24]. Absence of haemoconcentration in our patient may be due to the counteracting effect of haemodilution in pregnancy.
Aspiration of the large cysts was done under ultrasonographic guidance twice during pregnancy in our
patient. Abu-Louz et al. [17] noted decrease of ovarian
and abdominal pain after ultrasound-guided needle aspiration. Additionally, histological examination of aspirated
fluid can have important consequences for diagnosis and
OHSS and pregnancy
management [17, 25, 26]. Some patients with OHSS require exploratory laparotomy [15] or ultrasonography-guided
paracentesis [6] in order to exclude ovarian neoplasms
[27]. Bidus et al. [15] reported the case of unilateral
adnexectomy in case of severe hyperreactio luteinalis.
Our case, as well as literature data [3], indicates the
importance of assessing thyroid function in women with
OHSS.
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J
Żaneta Kimber-Trojnar
Chair and Department of Obstetrics and Perinatology
Medical University of Lublin
Jaczewskiego 8, 20-954 Lublin, Poland
e-mail: [email protected]