Division of Reproductive Medicine, Department of Obstetrics and Gynecology, University of Ottawa, Ottawa Hospital,1053 Carling Avenue, Ottawa, Ontario K1Y 4E9, Canada
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Abstract |
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Key words: chronic renal failure/haemodialysis/hyperreactio luteinalis
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Introduction |
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Case report |
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A transabdominal pelvic ultrasound showed a single viable intrauterine pregnancy of 10 weeks' gestational age. There was a bilateral ovarian enlargement with multilocular cysts. Massive ascites extending to the diaphragm was noted. Her complete blood count revealed: haemoglobin 122 g/l, haematocrit 0.366, white blood cell concentration 8.2 x 109/l, and platelet concentration 196 x 109/l. Her electrolyte concentrations were: sodium 129 mmol/l, potassium 4.2 mmol/l, urea 13.4 mmol/l, creatinine 794 µmol/l, albumin 34 g/l.
The patient was admitted for conservative treatment. There was concern about thrombosis; therefore she was started on prophylactic heparin 5000 IU s.c. every 12 h, and continued on dialysis. Results of blood tests performed on the day after admission are shown in Table I. Four days later, she had vaginal spotting that was associated with lower abdominal pain. This was followed by the passage of tissue. Subsequently the patient underwent a dilatation and curettage for an incomplete abortion. The pathology report confirmed the presence of products of conception.
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Discussion |
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The aetiology of hyperreactio luteinalis is unknown, but it is believed that the production of high concentrations of HCG and increased ovarian sensitivity to prolonged exposure to HCG (Bradshaw et al., 1986) may be manifested as an exaggerated ovarian response leading to theca lutein cyst formation. This may be associated with gestational trophoblastic disease in 3050% of cases (Clement, 1993
) or hyperplacentation as is seen in multiple pregnancy, diabetes, and rhesus isoimmunization. This patient had a prolonged exposure to high concentrations of HCG because of the decreased clearance of HCG due to anuria resulting from her chronic renal failure.
There was improvement of her symptoms 6 weeks after the spontaneous abortion; however, the ß-HCG did not become negative until 16 weeks after the miscarriage. Because of chronic renal failure, the resolution of symptoms and ß-HCG was very slow.
Hyperreactio luteinalis can mimic ovarian hyperstimulation syndrome (OHSS). Patients with OHSS usually have a history of induction of ovulation. Although it is rare, OHSS may be spontaneous (Zalel et al., 1992; Ayhan et al., 1996
). OHSS is usually manifested in the early first trimester with a rapid increase in ovarian size and fluid shift leading to ascites or hydrothorax in some cases. Patients usually present with a history of rapid weight gain, bloating and abdominal discomfort, and gastro-intestinal symptoms. In severe cases they may present with thrombo-embolic disorders or renal failure. Patients may require admission, hydration and prophylactic heparinization. They may undergo paracentesis or thoracocentesis as needed. OHSS usually resolves spontaneously although resolution may be delayed by up to 3 weeks in pregnant patients.
Hyperreactio luteinalis is usually asymptomatic. It may be discovered during a routine pelvic ultrasound for pregnancy, during Caesarean section or sometimes post partum. In symptomatic cases, patients may present with abdominal bloating or cyst complications, such as torsion and haemorrhage. In some cases it may be associated with fluid shift. Lambers and Rosenn (1996) described a case of hyperreactio luteinalis associated with normal singleton pregnancy where pregnancy termination was carried out for maternal respiratory decompensation secondary to pleural effusion and ascites. O'Loughlin and Brookes (1987) reported a 16 weeks pregnant patient who presented with dyspnoea and abdominal discomfort secondary to massive ascites and pleural effusion and underwent exploratory laparotomy with partial ovarian resection.
In this report, the woman presented with a spontaneous severe OHSS-like picture. Knowing that the patient was on haemodialysis, we were not expecting the usual haemoconcentration and electrolyte imbalance associated with severe OHSS. Subsequent to her abortion, it might have been expected that she would achieve a rapid resolution of her symptoms; however, in this case the ß-HCG concentrations remained high and her symptoms persisted. The pathology report confirmed an intrauterine conception without trophoblastic changes. The most likely diagnosis of this condition is therefore hyperreactio luteinalis which did not resolve subsequent to termination of pregnancy due to the persistence of high concentrations of ß-HCG in circulation. During this period of time, free testosterone was also elevated confirming the hyperandrogenic state that may be associated with hyperreactio luteinalis. Once the hyperreactio luteinalis had resolved, this patient's free testosterone did return to normal values. Hyperprolactinaemia is ongoing in this patient and is associated with chronic renal failure and dialysis (Hochstetler et al., 1994).
Although infrequent, pregnancy can occur in women with chronic renal failure when they undergo haemodialysis. The prognosis for a live birth delivery is poor. These patients may develop hyperreactio luteinalis due to the decreased clearance of ß-HCG.
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Notes |
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3 To whom correspondence should be addressed
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References |
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Submitted on May 12, 1998; accepted on November 4, 1998.