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doi:10.1111/jog.12386

J. Obstet. Gynaecol. Res. Vol. 40, No. 6: 1598–1602, June 2014

Behçet’s disease and pregnancy: A retrospective analysis of course of disease and pregnancy outcome Cantekin Iskender, Ozlem Yasar, Oktay Kaymak, Selen Taflan Yaman, Dilek Uygur and Nuri Danisman Department of Perinatology, Dr Zekai Tahir Burak Research and Training Hospital, Ankara, Turkey

Abstract Aim: Behçet’s disease (BD) is a rare, chronic, multisystemic disease of unknown cause. BD is characterized by mucocutaneous, ocular, vascular and central nervous system manifestations and is also associated with thrombogenicity. In this retrospective analysis we investigated the relation between gestation and BD. Material and Methods: This retrospective study consisted of 49 pregnancies in 24 patients with BD between January 2008 and June 2013. The following clinical and demographic data were obtained: maternal age, obstetric history, pregnancy outcome, and maternal and neonatal complications during pregnancy following diagnosis of BD. To avoid recall bias, the following data were collected only in the recent pregnancy of each patient: disease activity and use of medications during pregnancy. Results: Sixty-three pregnancies occurred in this group and 52 of them were after the diagnosis. Mean age of the patients at diagnosis was 21.4 years. The duration of BD during pregnancy was 7.2 years. Fourteen patients (58.3%) had no symptoms during recent pregnancy. No change was observed in the disease activity during pregnancy in eight patients. Disease activity was aggravated in two patients. The rate of vascular complications was higher in pregnancies of patients with BD. The rates of stillbirth, pre-eclampsia, preterm delivery and intrauterine growth restriction did not differ between the groups. Perinatal mortality and neonatal intensive care unit admissions as well as low birthweight infants were also similar between groups. Conclusion: Patients with BD had a higher rate of vascular complications during pregnancy; however, other obstetric complications were not increased and neonatal outcomes were not negatively influenced by BD. Key words: autoimmunity, Behçet’s disease, pregnancy, prenatal care, intrauterine growth restriction.

Introduction Behçet’s disease (BD) is a rare, chronic, multisystemic disease of unknown cause.1 BD is characterized by mucocutaneous, ocular, vascular and central nervous system manifestations and is associated with thrombogenicity.2–4 BD typically occurs between 18 and 40 years of age and mainly affects young men; it has a higher prevalence along the ancient ‘silk route’.5,6 The diagnosis of BD is made according to the 1990 International Study Group Criteria for Behçet’s Disease, which

requires the presence of oral ulceration plus any two of the following: genital ulceration, typical defined eye lesions, typical defined skin lesions or a positive pathergy test.7 Because the disease is multisystemic and more prevalent during fertile years, disease activity in pregnancy and obstetric and neonatal outcomes merit special attention. However, only a few studies with relatively low sample sizes have addressed this issue.8–13 In this retrospective analysis, we investigated the relation between BD and pregnancy outcome.

Received: November 13 2013. Accepted: December 16 2013. Reprint request to: Dr Cantekin Iskender, Adalet sitesi-2 Kehribar Sk 19-a, 14 B.esat, Ankara, Turkey. Email: [email protected]

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© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

Behçet’s disease and pregnancy

Methods This retrospective study consisted of pregnant patients with Behçet’s disease who attended the perinatology clinic of Dr Zekai Tahir Burak Research and Training Hospital between January 2008 and June 2013. During this period, 26 patients with BD were diagnosed according to diagnostic criteria suggested by the International Study Group Criteria for Behçet’s Disease. One patient with concomitant connective tissue disease and one patient with insufficient data were excluded from the study; thus, 24 patients who received antenatal care at our institution were found to be eligible for the study. Forty-nine pregnancies following diagnosis of BD were reviewed. The following clinical and demographic data were obtained by reviewing the patients’ medical records: maternal age, obstetric history, pregnancy outcome and maternal and neonatal complications during pregnancy following diagnosis of BD. The BD patients were referred to Behçet’s clinics of other tertiary care centers to monitor disease activity and plan their treatment during the pregnancy. To avoid recall bias, data on BD activity and use of medications during pregnancy were collected for the most recent pregnancy of each patient only. Patients were considered in remission if they were totally symptom-free during pregnancy. Disease exacerbation was defined as the onset of new symptoms or increased frequency of symptoms that required an alteration in the treatment strategy during pregnancy. Three controls per case were randomly selected from the remaining births by using the random table. Thus, 147 patients who delivered at our institution were selected as a control group. Intrauterine growth restriction (IUGR) was defined as estimated fetal weight below the 10th percentile associated with fetal Doppler abnormalities. Deliveries occurring prior to 37 weeks of gestation were recorded as preterm deliveries. Pre-eclampsia was defined as persistent elevation of blood pressure after 20 weeks of gestation with proteinuria. The study was approved by the local ethics committee. Statistical analysis was performed using spss 17. The Student’s t-test was performed for parametric variables between groups, and a χ2-test was performed for non-parametric variables between groups. A P-value less than 0.05 was considered as significant.

Results During the study period there were 99 342 deliveries, 41 of which occurred in patients with BD. The BD

Table 1 Demographic data, reproductive history and disease activity during pregnancy in patients with Behçet’s disease Patients with Behçet’s disease (n = 24) Age at diagnosis 21.4 (12–35) Duration of disease 7.2 (1–17) Total pregnancies 63 Pregnancies before diagnosis 11 Pregnancies after diagnosis 52 Elective termination of pregnancy 3 Miscarriage 8 Live birth 41 Disease activity in recent pregnancy Remission 14 (58.3%) No alteration 8 (33.3%) Exacerbation 2 (8.3%) Symptoms in patients during recent pregnancy Oral ulcer 4 (16.7%) Genital ulcer 1 (4.3%) Oral + genital ulcer 3 (12.5%) Eye inflammation 1 (4.3%) Neurologic manifestation 1 (4.3%) Medications during recent pregnancy Colchicine 11 (45.8%) Corticosteroids 2 (8.3%) Chloroquine 1 (4.3%) Aspirin 7 (29.2%) Low-molecular-weight heparin 4 (16.7%) None 11 (45.8%) Data expressed as number (%), mean (range).

patients’ demographic characteristics, reproductive history and disease activity during pregnancy are shown in Table 1. Sixty-three pregnancies occurred in this group; 52 occurred after the BD diagnosis. The mean age of the patients at diagnosis was 21.4 years and the duration of BD was 7.2 years. Fourteen patients (58.3%) had no symptoms during the most recent pregnancy. In eight patients, there were no changes in disease activity during pregnancy; disease activity was aggravated in two patients. Maternal age, parity, gestational age at delivery, birthweight and pregnancy outcomes in the study and control groups were similar (Table 2). Table 3 shows the pregnancy complications experienced by the BD patients and the control group. The rates of stillbirth, pre-eclampsia, preterm delivery and IUGR did not differ between groups. Perinatal mortality, neonatal intensive care unit admissions and low birthweight were also similar between the two groups.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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Table 2 Pregnancy data of patients with Behçet’s disease and control group Characteristics

Behçet’s disease (n = 49)

Control (n = 147)

Age at pregnancy (mean ± SD) Nulliparity (n (%)) Gestational age (weeks)† 41 Pregnancy outcome Miscarriage Live births Stillbirth Birthweight† 4000

28.6 ± 4.45 17 (35.7%)

28.9 ± 6.11 55 (37.4%)

6 (14.6%) 32 (78.0%) 3 (7.3%)

11 (8.1%) 121 (89.0%) 4 (2.9%)

8 (16.3%) 41 (83.7%) 0

11 (7.5%) 135 (91.8%) 1 (0.7%)

3 (7.3%) 36 (87.8%) 2 (4.9%)

11 (8.1%) 117 (86.0%) 8 (5.9%)

P 0.86 0.73 0.12

0.17

0.96

Data expressed as number (%), mean ± SD. †Excluding patients with miscarriage. SD, standard deviation.

Table 3 Comparison of pregnancy complications in patients with Behçet’s disease and control group Characteristics

Behçet’s disease† (n = 41) (%)

Control (n = 136) (%)

P

Placental dysfunction syndrome‡ Preterm delivery Vascular complications CD Primary CD Cesarean indications Non-progressive labor Fetal distress Repeat CD Malpresentation Low birthweight NICU admission Fetal anomaly Perinatal mortality

8 (19.5%) 6 (14.6%) 2 (4.8%) 17 (41.5%) 12 (33.3%)

14 (10.3%) 10 (7.4%) 0 46 (33.8%) 28 (23.7%)

0.12 0.15