Epilepsy Associated with Temporal Pole Encephaloceles
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ORIGINAL ARTICLE
Epilepsy Associated with Temporal Pole Encephaloceles An Unrecognized Manifestation of Idiopathic Intracranial Hypertension? Javier Martinez-Poles1,2 · Rafael Toledano1,3 · Adolfo Jiménez-Huete4 · Irene García-Morales1,2 · Ángel Aledo-Serrano3 · Carla Anciones3 · Pablo Campo5 · Juan Álvarez-Linera6 · Antonio Gil-Nagel1 Received: 26 August 2020 / Accepted: 26 September 2020 © Springer-Verlag GmbH Germany, part of Springer Nature 2020
Abstract Purpose We hypothesized that epilepsy associated with temporal pole encephaloceles (ETPE) could be the consequence and an unrecognized manifestation of idiopathic intracranial hypertension (IIH). To test this hypothesis in patients with ETPEs we evaluated: 1) the frequency of two radiological signs of IIH and 2) whether these patients develop over time clinical manifestations suggestive of elevated intracranial pressure (ICP). Methods Case-control study comparing two cardinal radiological signs of IIH pituitary gland height (PGH) and the diameter of the two optic nerve sheaths (ONS) between 29 patients with ETPEs (TPE group) and 29 patients with focal epilepsy of other etiologies (control group), adjusted by age, sex, body mass index (BMI), age at epilepsy onset and epilepsy duration. Analysis was performed using conventional and ordinal logistic regression. The measurements in both groups were compared with validated radiological criteria of IIH. Results Of the patients 17 (63%) in the TPE group had all three measurements over the cut-off values for IIH, while no patients in the control group had all three findings. The TPE group patients had lower PGH (3.2 ± 1.0 mm vs. 4.9 ± 1.3 mm, p < 0.001) and larger diameter of ONS than controls (p < 0.001), being similar to validated data of IIH. No patient with TPE had clinical manifestations of elevated ICP (mean follow-up 15.1 ± 11.7 years). Conclusion Patients with ETPEs frequently had radiological signs of IIH while not developing typical manifestations of elevated ICP over time. In this way, ETPEs could be an unrecognized manifestation of IIH, and temporal lobe seizures the only clinical expression of this epilepsy syndrome.
Keywords MRI · Temporal lobe epilepsy · Veno-glymphatic system · Epileptic syndromes · Drug resistant epilepsy
Introduction Temporal pole encephaloceles (TPEs) have been recently recognized as a relatively common cause of temporal lobe epilepsy (TLE) [1], being found in around 5% of patients with TLE and in up to 24% of patients with TLE in whom
an initial magnetic resonance imaging (MRI) was informed as normal [2, 3]. Besides recent research characterizing the main clinical features of epilepsy associated with TPEs [2, 4], the pathophysiology of this new epilepsy syndrome is not fully elucidated. Some authors have found that patients with TPEs are
Javier Martinez-Poles and Rafael Toledano contributed equally. 3
Epilepsy Unit, Department of Neurology, Hospital Ramón y Cajal, Madrid, Spain
4
Epilepsy Unit, Department of Neurology, Hospital Ruber Internacional, c/La Masó, 38, 28034 Madr
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