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Obstetric Case Reports

services. Rupture can occur following medical management (even with falling β-hCG levels.) • Pregnancy following a cornual resection can cause uterine rupture secondary to myometrial thinning. Suturing of this defective area had been planned for our patient, but she conceived while awaiting surgery. It is thought that the risk of uterine rupture in subsequent pregnancy, without reconstructive surgery is around 10–15%. If reconstructive surgery is undertaken before achieving subsequent pregnancy, the risk is around 1%. This highlights the importance of careful counselling and contraception.

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Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

References Bernoux A, Job-Spira N, Germain E et al. 2000. Fertility outcome after ectopic pregnancy and the use of an intrauterine device at the time of the index ectopic pregnancy. Human Reproduction 15:1173–1177. Jermy K, Thomas J, Doo A et al. 2004. The conservative management of interstitial pregnancy. British Journal of Obstetrics and Gynaecology 111:1283–1288. Lau S, Tulandi T. 1999. Conservative medical and surgical management of interstitial ectopic pregnancy. Fertility and Sterility 72:207–215. Timor-Tritsch I, Monteagudo A, Matera C et al. 1992. Sonographic evolution of cornual pregnancies treated without surgery. Obstetrics and Gynecology 79:1044–1049. Tulandi T, Al-Jaroudi D. 2004. Interstitial pregnancy: results generated from the Society of Reproductive Surgeons registry. Obstetrics and Gynecology 103:47–50.

Clinically isolated syndrome in pregnancy R. Jarvis University Hospital Southampton NHS Foundation Trust, Southampton, UK DOI: 10.3109/01443615.2013.817985 Correspondence: R. Jarvis, University Hospital Southampton NHS Foundation Trust, Southampton, SO16 6YD UK. E-mail: [email protected]

Introduction Clinically isolated syndrome (CIS) is a term that describes a first neurological episode, lasting at least 24 h, caused by demyelination at single or multiple sites within the central nervous system. CIS may be the heralding manifestation of multiple sclerosis (MS). This is the first case of CIS occurring during pregnancy reported in the literature.

Case report A 30-year-old primigravida presented at 8 weeks’ gestation with nausea, vomiting and dizziness of 12 h duration. Her past medical history was of borderline osteopaenia secondary to coeliac disease, for which calcium supplementation was taken. Clinical examination revealed a neurologically intact, unwell, dehydrated patient, with mild tachycardia and hypotension. She was admitted to the gynaecology ward where she received intravenous fluid resuscitation and antiemetics. Twelve hours later, the patient developed diplopia and pronounced dizziness. On examination, bilateral torsional nystagmus, right-sided weakness (MRC power 4⫹/5), cerebellar ataxia and clonus were present. Extrapyrimidal side-effects of antiemetic administration were considered, however, in the context of an evolving brainstem syndrome in a gravid patient, suspicion of

a thrombotic event was high. She was reviewed by neurology and transferred to the neurological unit. She received treatment dose aspirin, followed by prophylaxis for the presumptive diagnosis of thrombotic stroke. Hereditary thrombophilia screen (factor V, Leiden and prothrombin gene mutations, protein C, protein S and antithrombin III levels), echocardiogram, CT head and magnetic resonance angiography were normal. Autoantibodies (cANCA, pANCA, ANA, anticardiolipin) were negative, and inflammatory markers (CRP and ESR) were not raised, as expected in an infection or vasculitis. Full blood count, renal, liver and thyroid function were normal, except for mild hypocalcaemia (2.02 corrected, normal range 2.05–2.60), likely secondary to vitamin D malabsorption due to coeliac disease. An MRI brain scan revealed multiple small bilateral periventricular and left brainstem lesions suggestive of demyelination, which correlated with the clinical findings. Lumbar puncture demonstrated the presence of oligoclonal bands with a raised CSF: serum IgG index, and a normal albumin ratio, which excludes any breakdown between the blood–brain barrier. Neurophysiology demonstrated normal visual-evoked potentials from both eyes, suggesting no current or previous evidence of optic neuritis. Retrospectively, a diagnosis of clinically isolated syndrome was made. An observational management approach was employed as the patient’s symptoms spontaneously resolved within 2 weeks, leaving no permanent neurological deficit. The pregnancy was otherwise uneventful; a healthy infant was delivered at term.

Discussion The combination of clinical and radiological evidence here confirms the diagnosis of CIS according to the McDonald diagnostic criteria (Polman et al. 2010). The abnormal finding of immunoglobulin synthesis within the CSF is caused by inflammatory diseases of the central nervous system. While not specific to MS, oligoclonal bands are seen in 95% of MS patients (Link and Huang 2006). Patients usually recover from their presenting episode, though between 53% and 60% presenting with brainstem syndromes go on to develop MS (Fisniku et al. 2008; Tintore et al. 2010). About 20% of patients with CIS will not convert to MS even after two decades. Of those who go on to develop MS, one-third have minimal disability after 15–20 years, and half will have developed secondary progressive MS (Miller et al. 2012). No further symptoms have been reported by this patient at 1 year follow-up. It is not known whether pregnancy impacts upon clinical conversion from CIS to MS. In a small prospective case–control study, Lebrun et al. (2012) found the risk for conversion from radiological isolated syndrome (radiological findings in the absence of symptoms) to a clinical event and new MRI disease activity was influenced by pregnancy. A significantly shorter time of conversion to the first neurological event and higher numbers of active lesions on subsequent MRI brain scans were observed in the pregnant group. Findings are limited by the small number of pregnancies followed. This finding is contrary to what is known about MS, which typically remits during pregnancy. The Pregnancy in Multiple Sclerosis trial demonstrated a 70% reduction in relapse risk in the 3rd trimester, with a rebound in the 3 months postpartum, which is thought to be due to the immunomodulatory effects of oestrogen and progesterone (Confavreux et al. 1998). Evidence supports early disease modifying treatment for CIS patients, with decreased MS conversion rates and time to conversion in patients receiving beta-interferon (Kappos et al. 2006) and glatiramer acetate (Comi et al. 2009). Over 3–5 years, only a marginal significant gain in terms of disability exists. Current guidance suggests clinicians may consider treatment in CIS patients at high risk of relapse (Association of British Neurologists 2009). The use of disease modifying treatments in pregnancy is prohibited due to concerns over safety (despite no adverse effects reported by observational studies) and the immunosuppressive effects of pregnancy itself (Hutchinson 2011). This is the first case of a clinically isolated syndrome occurring during pregnancy, reported in the literature. As MS and CIS

Obstetric Case Reports 87 predominantly affect women in their childbearing years and are no longer seen as contraindications to pregnancy, obstetricians and gynaecologists should be aware of these conditions. This case highlights the importance of considering alternative diagnoses when diagnosing hyperemesis gravidarum. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

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References Association of British Neurologists. 2009. Revised (2009) guidelines for prescribing in multiple sclerosis. London: Association of British Neurologists. Comi G, Martinelli V, Rodegher M et al. 2009. Effect of glatiramer acetate on conversion to clinically definite multiple sclerosis in patients with clinically isolated syndrome (PreCISe study): a randomised, double-blind, placebocontrolled trial. Lancet 374:1503–1511. Confavreux C, Hutchinson M, Hours MM et al. 1998. Rate of pregnancyrelated relapse in multiple sclerosis. New England Journal of Medicine 339:285–291. Fisniku LK, Brex PA, Altmann DR et al. 2008. Disability and T2 MRI lesions: a 20-year follow-up of patients with relapse onset of multiple sclerosis. Brain 131:808–817. Hutchinson M. 2011. Safety first, efficacy second in disease modifying therapies. Multiple Sclerosis 17:380–381. Kappos L, Polman CH, Freedman MS et al. 2006. Treatment with interferon beta-1b delays conversion to clinically definite and McDonald MS in patients with clinically isolated syndromes. Neurology 67:1242–1249. Lebrun C, Le Page E, Kantarci O et al. 2012. Impact of pregnancy on conversion to clinically isolated syndrome in a radiologically isolated syndrome cohort. Multiple Sclerosis Journal 18:1297–1302. Link H, Huang YM. 2006. Oligoclonal bands in multiple sclerosis cerebrospinal fluid: An update on methodology and clinical usefulness. Journal of Neuroimmunology 180:17–28. Miller D, Chard D, Ciccarelli O. 2012. Clinically isolated syndromes. Lancet Neurology 11:157–169. Polman CH, Reingold SC, Banwell B et al. 2011. Diagnostic criteria for multiple sclerosis: 2010 revisions to the McDonald criteria. Annals of Neurology 69:292–302. Tintore M, Rovira A, Arrambide G et al. 2010. Brainstem lesions in clinically isolated syndromes. Neurology 75:1933–1938.

Successful twin delivery after a radical trachelectomy N. Agarwal1, R. Rana1, M. Gupta2 & A. Ugwumadu1 1St George’s Hospital, London, UK , 2Barts Heath NHS Trust, London, UK

internal cerclage using a nylon suture was inserted into the lower segment of the uterus simultaneously. Histopathology revealed no residual tumour and pelvic lymph nodes were negative. Follow-up cytology and MRI at 6, 8 and 12 months, did not reveal any recurrence or residual disease. Three years later, she conceived spontaneously and a 12-week dating ultrasound scan showed monochorionic diamniotic twins. A trans-vaginal ultrasound at 16 weeks’ gestation showed the cerclage suture at the internal cervical os. The ‘neo-cervical’ length on a snapshot scan was ~1.5 cm. Ultrasound at 19 weeks’ gestation showed no evidence of twin–twin transfusion syndrome. At 21 weeks’ gestation, she presented with intermittent uterine activity and abdominal pains. She declined speculum/fibronectin evaluation and also high vaginal swabs. The patient was admitted for psychological and social comfort, strict bed rest, prescribed thromboprophylaxis and received corticosteroid therapy at 26 weeks’ gestation. Serial scans at 28, 30 and 32 weeks’ gestation showed normal growth for both fetuses. The patient was discharged at 30 weeks’ gestation with a plan for elective caesarean section at 34 weeks. This was performed at 34 ⫹ 3 weeks’ gestation and two female infants weighing 2,360 g and 1,720 g were delivered in good health. The patient is currently under outpatient review with no evidence of recurrence.

Discussion

We report a successful outcome of a twin pregnancy at 34 ⫹ 3 weeks’ gestation after radical trachelectomy, without the use of a second trans-abdominal cervicoisthmic cerclage or Saling procedure. There are no standard recommendations for the management of pregnancy after cervical surgery (Jolley and Wing 2008). Trachelectomy is associated with cervical insufficiency and pre-term delivery. In the six largest published studies, there were three sets of twins in a total of 149 pregnancies. All delivered at between 24 and 28 weeks (Plante et al. 2005). Multiple pregnancy is an independent risk factor for pre-term birth (Bernardini et al. 2003). To the best of our knowledge, this is the first report of twins delivered at 34 weeks’ gestation after a radical vaginal trachelectomy. The use of the vaginal occlusion procedure does not prolong gestation when compared with those women who did not have the procedure (Jolley et al. 2007). Although the placement of another cerclage might be helpful, this is technically difficult because the ‘neo-cervix’ after trachelectomy is fibrotic and markedly shortened and does not prolong gestation (Berghella et al. 2005). In fact, the use of cerclage in multiple gestations has been shown to worsen outcome (Berghella et al. 2005). The purpose of the Saling procedure is to reduce the risk of chorioamnionitis by closing the vaginal mucosa over the cervical os after the 1st trimester. In our case, we did not perform the Saling procedure or place another trans-abdominal cervicoisthmic cerclage. The good outcome achieved in our case may have been due to the non-operative management plan. A viable pregnancy resulting in a live healthy birth is the ultimate goal when choosing trachelectomy, which was successfully achieved in this case, with only the conservative approach of bed rest.

DOI: 10.3109/01443615.2013.822481 Correspondence: N. Agarwal, St George’s Healthcare NHS Trust, Blackshaw Road, London, UK. E-mail: [email protected]

Case report A 36-year-old nulliparous woman was referred with severe dyskaryosis, for colposcopy. Colposcopic biopsy suggested cervical intraepithelial neoplasia (CIN) 3. A large loop excision of the transformation zone (LLETZ) was performed and histology showed moderately differentiated squamous cell carcinoma. Cone biopsy confirmed stage 1B cervical carcinoma with the lesion size of ~16 mm. The patient was counselled as to the options for management of cervical carcinoma (FIGO stage 1 B1). She wished to preserve her fertility if possible and a staging procedure was done, including examination under anaesthesia, hysteroscopy, cystoscopy and sigmoidoscopy for clinical staging, to establish whether trachelectomy was an option. Subsequently, a laparoscopic pelvic lymphadenectomy and vaginal radical trachelectomy was done, during which the cervical branch of the uterine artery was ligated. A prophylactic

Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

References Berghella V, Odibo AO, To MS et al. 2005. Cerclage for short cervix on ultrasonography: meta-analysis of trials using individual patient-level data. Obstetrics and Gynecology 106:181–189. Bernardini M, Barrett J, Seaward G et al. 2003. Pregnancy outcomes in patients after radical trachelectomy. American Journal of Obstetrics and Gynecology 189:1378–1382. Jolley JA, Battista L, Wing DA. 2007. Management of pregnancy after radical trachelectomy: case reports and systematic review of the literature. American Journal of Perinatology 24:531–539. Jolley JA, Wing DA. 2008. Pregnancy management after cervical surgery. Current Opinion in Obstetrics and Gynecology 20:528–533. Plante M, Renaud MC, Hoskins IA et al. 2005. Vaginal radical trachelectomy: a valuable fertility-preserving option in the management of early-stage cervical cancer. A series of 50 pregnancies and review of the literature. Gynecologic Oncology 98:3–10.

Clinically isolated syndrome in pregnancy.

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