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Neuropathological findings in a patient with epilepsy and the Parry-Romberg syndrome.

Abstract：

PURPOSE:The Parry-Romberg syndrome is an unusual disorder frequently associated with epilepsy. The origin of this disease, and the cause of epilepsy, are unknown. This study is the first reported case of the Parry-Romberg syndrome, with intractable temporal lobe epilepsy, in which detailed microanatomic analyses have been performed on resected brain tissue obtained after surgical intervention. METHODS:Standard histopathologic methods and correlative light and electron microscopy, combined with immunocytochemical techniques, were used to study in detail the synaptic microorganization of the resected hippocampal formation. RESULTS:After surgery, the patient was seizure free (follow-up period of 4 years and 7 months). The resected temporal lobe showed a variety of dramatic microanatomic alterations (small groups of ectopic cells, neuronal loss, gliosis, and activated microglial cells) in mesial structures, including the entorhinal cortex, subiculum, and dentate gyrus. At the electron-microscopic level, we found that in the dentate gyrus, the number of synapses in the cell-sparse region adjacent to the ectopic mass of neurons was almost twice that found in the molecular and polymorph cell layers, indicating the intrusion of neuritic processes and synapse formation. In addition, the symmetrical axosomatic synapses characteristically found on granule cells, which are likely derived from gamma-aminobutyric acid (GABA)ergic inhibitory basket cells, were not observed. CONCLUSION:The complete seizure relief after surgery suggests that the pacemaker region(s) of seizure activity were within the resected tissue. However, we do not know which of the multiple neuropathologic findings reported here were the primary cause of seizure activity. Nevertheless, the changes found in the dentate gyrus circuitry appear to be among the most important alterations that would lead to epilepsy.

journal_name

Epilepsia

journal_title

Epilepsia

authors

DeFelipe J,Segura T,Arellano JI,Merchán A,DeFelipe-Oroquieta J,Martín P,Maestú F,Ramón y Cajal S,Sánchez A,Sola RG

doi

10.1046/j.1528-1157.2001.45800.x

subject

Has Abstract

pub_date

2001-09-01 00:00:00

pages

1198-203

issue

eissn

0013-9580

issn

1528-1167

pii

45800

journal_volume

pub_type

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Neuropathological findings in a patient with epilepsy and the Parry-Romberg syndrome.