Table of Contents  

Hubaishi, Cherifi, Raza, Adam, Almahdi, Sibai, and Abdulkarim: Postpartum ovarian vein thrombosis – unusual clinical presentation

Introduction

Ovarian vein thrombosis (OVT) is a rare but potentially serious complication mainly occurring in the postpartum period, but also complicating pelvic inflammatory disease, malignancies and pelvic surgery. It can cause thrombosis of the vena cava or renal vein, as well as pulmonary embolism, all of which are potentially life-threatening.13

The clinical findings of OVT are non-specific. Patients usually present with abdominal pain in the lower quadrant, fever and sometimes a palpable mass within the 15 days following delivery. These symptoms may mimic acute appendicitis and may also resemble many other various clinical situations, such as pelvic infection, adnexal torsion, tubo-ovarian abscess, haematoma of the broad ligament, pyelonephritis, volvulus and cholecystitis.4,5

As a result of advances in diagnostic imaging, OVT can now be diagnosed very accurately; ultrasonography may suggest the diagnosis, but computerized tomography (CT) and magnetic resonance imaging (MRI) can confirm it and quantify the extent of the thrombosis.6 However, there is debate regarding the optimal therapy; heparin and antibiotics are currently preferred.

We present a case to illustrate the importance of including OVT as a differential diagnosis in women who present in the postpartum period with a tender lower abdominal and with fever following a vaginal delivery.

Case report

A 33-year-old woman (parity 1, miscarriage 2) presented to the emergency department on day 2 post partum with severe right lower abdominal pain and fever (39°C). There was no associated nausea or vomiting. The patient had a given birth to a pre-term baby by spontaneous vaginal delivery at 26 weeks; the immediate postpartum period was unremarkable. Her medical history included two miscarriages. During her recent pregnancy, the patient was referred to our department at 16 weeks of gestation because of threatened miscarriage due to a short cervix. Rescue cerclage was performed and the patient was discharged home 6 days later in good condition. At 26 weeks, she attended the emergency department and was admitted for premature preterm rupture of membranes (PPROM). The cervical os was closed and cervical cerclage was not under tension. The white blood cell (WBC) count was 11 500 cells/mm3 and C-reactive protein (CRP) 22 mg/l. The cerclage was removed and the patient was treated conservatively; however, 4 days later, she went into spontaneous labour and delivered a male baby weighing 750 g. She was discharged home in good condition but she attended the emergency department on day 2 post partum.

At admission, she was febrile with a temperature of 39°C, blood pressure of 92/52 mmHg and pulse rate of 111 beats per minute. Abdominal examination revealed right lower quadrant tenderness with no masses detected. There were no signs of deep-vein thrombosis in the lower extremities. There was no offensive vaginal smell and no uterine or adnexal tenderness. Laboratory analysis showed haemoglobin 9.7 g/dl, WBC count 7900 cells/mm3 and a CRP level of 150 mg/l; a blood culture, urine culture and high vaginal swab showed no growth. The initial clinical diagnoses were acute appendicitis or endometritis. As the pelvic sonograph showed suspected appendicitis, she was referred to the surgeons, who ruled out the diagnosis. Therefore, in view of PPROM, endometritis was diagnosed despite the absence of clinical signs and she was treated with broad-spectrum antibiotics (cefuroxime, metronidazole and gentamicin). On day 3, she became afebrile. She was discharged home after 8 days’ antibiotics treatment. In addition to the antibiotics, she received prophylactic enoxaparin sodium (Clexane®, Sanofi-Aventis, Paris, France) 40 mg subcutaneously (s.c.), which was discontinued upon discharge. Unfortunately, she was readmitted 5 days later with the same complaints (right lower abdominal pain and high-grade fever). Examination revealed no more than it did previously. Blood tests showed haemoglobin 9.1 g/dl, WBC count 5200 cells/mm3 with high neutrophilia (81.9%), erythrocyte sedimentation rate of 90 mm/h, fibrinogen 581 mg/dl and D-dimer 1060 µg/l. Thrombophilia screening was negative. Pelvic sonography revealed a fixed tubular structure extending from the right iliac fossa superior, lateral to the right ovary, up to the level of the right kidney. The diagnosis of appendicitis was suspected and subsequent abdominal CT showed right OVT extending to the inferior vena cava (Figures 1 and 2).

FIGURE 1

Abdominal CT scan showing a tubular structure corresponding to thrombosed right ovarian vein (arrow).

HMJ-608-fig1.jpg
FIGURE 2

Axial contrast-enhanced CT scan showing right ovarian thrombosis (arrow).

HMJ-608-fig2.jpg

The patient was started on a therapeutic dose of enoxaparin at 60 mg s.c., then 40 mg for 1 week with warfarin at 10 mg daily and piperacillin/tazobactam 4.5 g three times daily for 1 week. She continued to take warfarin only for 3 months. She was rescheduled for repeat CT after she stopped her anticoagulants and was also counselled to use contraception, but she was not keen to do so. Before repeat CT could be carried out the patient was found to be pregnant and was treated with enoxaparin 40 mg weekly throughout her pregnancy. She delivered a healthy baby at 38 weeks of gestation and was discharged with 40 mg of s.c. enoxaparin for 6 weeks.

Discussion

Ovarian vein thrombosis is a rare complication that is reported to occur most commonly in the postpartum period and complicates 0.05–0.18% of deliveries.2,4 In 80–90% of cases the right ovarian vein is affected.7,8 Many hypotheses have been put forward to explain why the right vein is implicated so much more frequently. The most common theory is that the right vein is longer than the left and has many valves along its length, which may act as a nidus for thrombosis.4,9 In addition, the dextrotorsion of the uterus may compress the right vein and increases the risk of thrombosis.2,9,10

The pathophysiology of puerperal OVT is related to the Virchow triad: the venostasis and hypercoagulability that commonly accompany the puerperium with superimposed endothelial trauma during delivery or caused by local uterine infection.1113 In fact, pregnancy is a hypercoagulable state as a result of normal physiological changes to protect women from haemorrhagic complications during placentation and delivery. In addition to these modifications, which limit maternal blood loss at delivery, the relative venostasis at the vena cava due to uterine compression can predispose pregnant women to thromboembolism.12,13

In the literature, most cases are diagnosed on day 2 post partum and 90% occur within 10 days post partum8,1315 Patients with OVT typically present with fever, pelvic pain and sometimes a palpable abdominal mass, described as a firm, rope-like, tender structure varying in diameter from 2 to 8 cm.13,14,16 Our patient complained of high-grade fever and severe lower abdominal quadrant pain; however, no palpable masses were felt in her two postpartum admissions. Laboratory investigations showed a high D-dimer level of 1.6 mg/l.

Despite leucocytosis, described by some authors,13,17 and in one case series found to be > 12 000 cells/mm3 in 70–100%,18 in our case, the WBC count was normal in both admissions; however, high neutrophilia was noticed. The rarity of this clinical entity presents a diagnostic dilemma. The most common differential diagnoses in patients presenting with lower abdominal pain and fever in the postpartum period are endometritis, pelvic inflammatory disease, acute appendicitis, adnexal torsion and pyelonephritis. If the underlying cause of symptoms is, in fact, OVT, treatment may be delayed, as in the case of our patient. We decided to treat the patient for endometritis in view of her history of PPROM, suprapubic tenderness, neutrophilia and high CRP, despite the absence of an abnormal vaginal discharge and a sterile blood and urine culture. She was treated empirically with intravenous antibiotics. Appropriate diagnostic radiological modalities include sonography, CT and MRI, with a sensitivity of 52%, 92% and 100%, respectively.10,19 Ultrasound examination is non-invasive and less expensive. It can reveal the location and extent of the thrombus, but its sensitivity is operator dependent and is frequently limited by overlying bowel gas, impairing the visualization of the thrombus, which can lead to an erroneous diagnosis of appendicitis, as in our case. The diagnosis of OVT is ideally made by pelvic CT, which shows an enlarged vein with a low-density lumen and sharply defined walls.14 It enabled us to make a definitive diagnosis for our patient. MRI can also provide a reliable diagnosis.

Anticoagulants and antibiotics are the cornerstones of treatment. It is important to institute broad-spectrum antibiotics and intravenous heparin as soon as possible; however, there is no consensus about the type, dose or duration of treatment. Many authors have shown that 3–6 months’ treatment is required, or until resolution of the thrombus is confirmed radiologically.16,17,20 The treatment includes a brief course (7–10 days) of antibiotics and anticoagulation with heparin.1 Once thrombolysis has begun, oral anticoagulants, such as warfarin, can be started and continued for 3–4 months.21 Low-molecular-weight heparin can also be used at therapeutic doses but further investigation is necessary to determine if this is appropriate.14 In general, the decision to initiate, continue or discontinue anticoagulation in patients with OVT depends on the anticipated risk of recurrent thrombosis and related morbidity and mortality weighed against the risk of haemorrhagic complications.1 According to Wysokinska et al.,1 it would be reasonable to follow the current treatment guidelines for lower-extremity deep-vein thrombosis and pulmonary embolism. If the condition does not improve with medical support, then endovascular or surgical procedures, such as thrombectomy, cava filters, ovarian or cava vein ligature, may be required.4

Conclusion

Ovarian vein thrombosis is a relatively uncommon but serious postpartum complication that, in most cases, occurs in the right ovarian vein. Clinical signs are not specific but a prompt diagnosis can now be made by CT, although other imaging techniques, such as colour Doppler ultrasonography and MRI, are useful. Heparin and intravenous antibiotics are the mainstays of treatment.

References

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Hafsa C, Golli M, Jerbi OS, et al. A rare aetiology of the post-partum fever: ovarian vein thrombophlebitis. Ann Fr Anesth Réanim 2006; 25:286–20. http://dx.doi.org/10.1016/j.annfar.2005.10.030

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Witlin AG, Mercer BM, Sibai BM. Septic pelvic thrombophlebitis or refractory postpartum fever of undetermined aetiology. J Matern Fetal Med 1996; 5:355–8. http://dx.doi.org/10.1002/(SICI)1520-6661(199611/12)5:6<355::AID-MFM12>3.3.CO;2-T

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