European Journal of Obstetrics & Gynecology and Reproductive Biology 103 (2002) 92–94 Case report Homonymous hemianopia following a triplet pregnancy: post-natal cerebral vasculitis or atypical eclampsia Stephen D. Keaya, Stephen G.M. Carrolla, Mark Scruttonb, Anthony Kellya, Ian Ormerodc, David J. Cahilla* a Departments of Obstetrics and Gynaecology, St. Michael’s Hospital, Southwell St., Bristol BS2 8EG, UK b Department of Anaesthetics, St. Michael’s Hospital, Southwell St., Bristol BS2 8EG, UK c Department of Neurology, Bristol Royal Infirmary, Bristol, UK Received 16 August 2001; accepted 21 December 2001 Abstract A 46-year-old woman with a triplet pregnancy developed seizures, an oculogyric crisis and a homonymous hemianopia post-natally. Abnormal neuroimaging studies and lumbar puncture suggested possible cerebral vasculitis. Treatment with aciclovir and prednisolone resulted in a slow resolution of symptoms. This case highlights the difficulty in distinguishing eclampsia from rarer neurological causes of peripartum seizures. # 2002 Elsevier Science Ireland Ltd. All rights reserved. Keywords: Cerebral vasculitis; Eclampsia; Triplet pregnancy 1. Case report A 46-year-old primigravid patient with a triplet pregnancy from ovum donation was admitted at 35 weeks’ gestation in active labour. There was no medical history of epilepsy and blood pressure was mildly elevated at 140/85 and proteinuria (one þ on urine dipstick testing). Clotting studies conducted prior to proposed spinal anaesthesia demonstrated a prolonged activated partial thromboplastin time ratio (APTTr) of 1.4 and consequently caesarean section was performed under general anaesthesia. A total of 6 h following delivery, the patient had two self-limiting episodes of generalised rigidity. Eclampsia was suspected but the second seizure, witnessed by an anaesthetist, appeared to be an oculogyric crisis—the rigidity was associated with eye rolling but no loss of consciousness. Neurological examination revealed hyperreflexia, a left homonymous hemianopia and the patient to be drowsy but orientated. Fundoscopy was normal and blood pressure was 160/90 but did not require treatment and urine dipstick showed þþþ of protein. Laboratory investigations including full blood count, liver function tests, clotting screen (including APTTr), urea and electrolytes and * Corresponding author. Tel.: þ44-117-928-5704; fax: þ44-117-928-5290. E-mail address: d.j.cahill@bristol.ac.uk (D.J. Cahill). uric acid were normal. Magnesium sulphate infusion was commenced (1 g/h; following 4 g loading) for secondary prophylaxis. Computed tomographic (CT) scan of the head showed low density areas in both occipital lobes (Fig. 1) and abnormal filling of the sagittal sinus after intravenous contrast. An echocardiogram, performed to exclude embolic causes, was normal. The unexplained prolongation of the APTTr raised the possibility of antiphospholid syndrome, but the subsequent normal thrombophilia screen (including APTTr and lupus anticoagulant) effectively excluded this. Magnetic resonance angiography (MRA) of the brain demonstrated hyperintensity in the occipital and parietal lobes and pruning of the vertebro-basilar circulation (Fig. 2) and the cerebro-spinal fluid (CSF) from the lumbar puncture contained 264 white blood cells—85% lymphocytes. These findings suggested the possibility of cerebral vasculitis. The patient was treated with intravenous acyclovir and oral prednisolone and made a gradual but full recovery and was discharged after 25 days. 2. Discussion Cerebral vasculitis presenting postpartum in association with pre-eclampsia has been recorded once before [1]. It is an immune complex-mediated process characterised by generalised confusion or multifocal neurological deficits. 0301-2115/02/$ – see front matter # 2002 Elsevier Science Ireland Ltd. All rights reserved. PII: S 0 3 0 1 - 2 1 1 5 ( 0 2 ) 0 0 0 0 8 - 8 S.D. Keay et al. / European Journal of Obstetrics & Gynecology and Reproductive Biology 103 (2002) 92–94 Fig. 1. CT scan of brain showing low density areas in both occipital lobes. This case describes postpartum seizures in association with a homonymous hemianopia, an abnormal lumbar puncture and abnormalities in neuroimaging investigations leading to a provisional diagnosis of cerebral vasculitis. The MRA findings of hyperintensity in the occipital and parietal lobes 93 could result from either a vasculitic process or eclampsia. Hyperperfusion, secondary to vasogenic oedema of the occipital cortex was reported in the case of cortical blindness complicating severe pre-eclampsia but was associated with a normal lumbar puncture [2]. In our case, the diagnosis of cerebral vasculitis is supported by both the CSF lymphocytosis and the cerebral vascular abnormalities detected by MRA. Formal histological confirmation of vasculitis can only be made on biopsy but as vasculitis frequently diffuses, a negative biopsy result does not necessarily exclude the diagnosis [3]. MRA and vascular biopsy have similar sensitivities of approximately 70% for diagnosing vasculitis [4] with biopsy generally reserved for cases where radiological imaging is negative. Other potential causes included a paradoxical embolus through a patent foramen ovale, cerebral venous thrombosis or viral meningo-encephalitis. However, normal echocardiography and the absence of venous thrombosis on MRA effectively excluded the first two. Aciclovir was prescribed because viral meningo-encephalitis was part of the differential diagnosis, supported by the CSF lymphocytosis. There was no other evidence of meningoencephalitis and aciclovir probably did not contribute much to recovery. Corticosteroids were prescribed to improve vasculitis but we acknowledge that the resolution of the neurological signs cannot necessarily be attributed to their use. Medical and obstetric complications are common in women over 45 years, with pre-eclampsia and gestational diabetes occurring in 10 and 13%, respectively [5]. Multiple pregnancies resulting from donor oocytes in women over 45 years carries a greater risk than non-oocyte donation multiple pregnancies of the same age and suggests that a maximum of two embryos should be transferred [6]. Although screening for medical disorders in this case would not have predicted the ophthalmic complication, it is recommended for women over 45 years undergoing assisted conception because their physiological adaptation to pregnancy is relatively compromised. Whilst epilepsy and eclampsia will account for most peripartum seizures all cases require thorough neurological examination and appropriate investigation where atypical signs exist. Early resort to CT scan or more advanced neurological imaging is recommended after specialist neurological opinion. This report emphasises the risks of multiple pregnancy for a 46-year-old woman and the difficulty in differentiating cerebral vasculitis from eclampsia. References Fig. 2. MRA of brain showing pruning of the vertebro-basilar circulation. [1] Farine D, Andreyko J, Lysikiewicz A, Simha S, Addison A. Isolated angiitis of brain in pregnancy and puerperium. Obstet Gynecol 1984;63:586–8. [2] Apollon KM, Robinson JN, Schwartz RB, Norwitz ER. Cortical blindness in severe pre-eclampsia: computed tomography, magnetic resonance imaging, and single-emission computed tomography findings. Obstet Gynecol 2000;95:1017–9. 94 S.D. Keay et al. / European Journal of Obstetrics & Gynecology and Reproductive Biology 103 (2002) 92–94 [3] Berlit P, Moore PM, Bluestein HG. Vasculitis, rheumatic disease and the neurologist: the pathophysiology and diagnosis of neurologic problems is systemic disease. Cerebrovasc Dis 1993; 3:139–45. [4] Fieschi C, Rasura M, Anzini A, Beccia M. Central nervous system vasculitis. J Neurol Sci 1998;153:159–71. [5] Dildy GA, Jackson GM, Fowers GK, Oshiro BT, Varner MW, Clark SL. Very advanced maternal age: pregnancy after age 45. Am J Obstet Gynecol 1996;175:668–74. [6] Sauer MV, Paulson RJ, Lobo RA. Oocyte donation to women of advanced reproductive age: pregnancy results and obstetrical outcomes in patients 45 years and older. Human Reprod 1996;11:540–2543.