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Thrombophilic factors in idiopathic intracranial hypertension:
a report of 51 patients and a meta-analysis
Anat Kesler
a
, Efrat Kliper
a
, Einor Ben Assayag
b
, Eti Zwang
c
, Varda Deutsch
c
,
Uriel Martinowitz
e
, Aharon Lubetsky
e,1
and Shlomo Berliner
d,1
Several studies have suggested that thrombophilic risk
factors are more prevalent in individuals with idiopathic
intracranial hypertension (IIH), and that a prothrombic state
may be involved in the etiopathogenesis of this disease. We
examine thrombophilic factors in a group of patients with
IIH in relation to obesity. In addition, we reviewed the
relevant literature and performed a meta-analysis.
Thrombophilia work-up was performed on 51 patients with
IIH at least 1 month following their first episode. Samples
for the analysis of factor V Leiden (FVL), prothrombin gene
variant (PGV) G20210A and methylenetetrahydrofolate
reductase (MTHFR) were available in an additional 30
patients, that is 81 patients in all. Meta-analysis was
performed. Of the 51 patients 40 were obese. Increased
concentrations of fibrinogen, D-Dimer, factor VIII, factor IX
and factor XI were found in 15, 7, 7, 6 and 2 patients,
respectively, all obese. The circulating anticoagulant,
measured by dilute Russell’s viper venom time (dRVVT
assay), found mainly in obese. All 51 patients were negative
for the anticardiolipin antibody (IgG immunoglobulin G) and
IgG anti-b2 glycoprotein I. In the meta-analysis
antiphospholipid antibodies were significantly associated
with IIH [odds ratio (OR) of 4.25 (1.68 – 12.60)], similar to the
association with high factor VIII [OR U 16.17 (2.87–91.01)],
higher plasminogen activator inhibitor-1 (PAI-1) levels
[OR U 6.91 (2.28–20.91)], and high lipoprotein (a) [LP(a)]
[OR U 3.54 (1.54–8.70)]. Obesity often observed in IIH
patients is frequently linked with thrombophilic factors.
Thus, we believe that dysmetabolism could be the
thrombophilic target for treatment in patients with IIH. Blood
Coagul Fibrinolysis 21:328–333 ß 2010 Wolters Kluwer
Health | Lippincott Williams & Wilkins.
Blood Coagulation and Fibrinolysis 2010, 21:328–333
Keywords: coagulation, idiopathic intracranial hypertension, obesity
a
Department of Ophthalmology,
b
Department of Neurology,
c
Department of
Hematology,
d
Medicine ‘E’, Tel Aviv Sourasky Medical Center and
e
Institute of
Thrombosis and Hemostasis, and the National Hemophilia Center, The Chaim
Sheba Medical Center, Tel Hashomer, and Sackler Faculty of Medicine, Tel Aviv
University, Tel Aviv, Israel
Correspondence to Anat Kesler, Neuro-Ophthalmology Unit, Department of
Ophthalmology, Tel Aviv -Sourasky Medical Center, Tel Aviv, Israel
Tel: +972 3 6973408; fax: +972 3 9340520; e-mail: kesler@netvision.net.il
Received 7 July 2009 Revised 12 January 2010
Accepted 19 January 2010
Introduction
Idiopathic intracranial hypertension (IIH) is a disorder
associated with increased intracranial pressure without
any clinical, laboratory, or radiological evidence of a
space-occupying lesion. The cause and mechanism of this
syndrome is obscure, and it has been suggested that
cerebrospinal fluid (CSF) drainage is impeded by a venous
occlusive disorder. This is supported by the higher preva-
lence of thrombophilic risk factors in cohorts of IIH
patients compared to the normal population [1–6]. In
addition, our recent finding of increased red blood cell
aggregation in IIH provides additional support and mech-
anism for veno-occlusive cause [7]. The present study was
designed to further investigate the association between
thrombophilic risk factors and IIH. Due to the relatively
small cohorts in our study (51 patients) and in the litera-
ture, a meta-analysis was performed on the available
literature.
Patients and methods
Patients
Fifty-one consecutive patients (50 females and 1 male,
mean age ¼ 31.9 years, range ¼ 18–57 years), with well
established IIH according to the modified Dandy criteria
[8], were recruited to the neuro-ophthalmology unit, Tel
Aviv Sourasky Medical Center (excluding the patients
from our previous study [7]). All patients were in the
stable state of their disease, and undergone a full exam-
ination by a senior neuro-ophthalmologist (A.K.).
Inclusion criteria were age above 18 and below 68 years,
normal brain neuroimaging, intracranial pressure greater
than 250 mm H
2
O with normal CSF, bilateral papillo-
dema, and nonfocal neurologic examination, except for
isolated abducens paresis. Exclusion criteria included
sinus vein thrombosis and pregnancy. The study was
approved by the Tel Aviv Sourasky Medical Center
Ethics Committee. A written informed consent was
obtained from all participants. In addition to the present
cohort, we used previous data for the three coagulation
polymorphisms from additional 30 patients with IIH (26
females and 4 males, mean age ¼ 39.5 years, range ¼
23–68 years) who gave their consents.
Blood sampling and plasma preparation
Venous blood samples were obtained from fasting
patients at least 1 month after the clinical event. Blood
was collected into a 0.109 mol/l buffered citrate in a ratio
328 Original article
1
A.L. and S.B. contributed equally to the writing of this article.
0957-5235 ß 2010 Wolters Kluwer Health | Lippincott Williams & Wilkins DOI:10.1097/MBC.0b013e328338ce12