Go Taniguchi

Evaluation of amygdala pathology using 11C-methionine positron emission tomography/computed tomography in patients with temporal lobe epilepsy and amygdala enlargement

OBJECTIVE The association between amygdala enlargement (AE) and temporal lobe epilepsy (TLE) has increasingly been reported. However, the pathology of AE remains poorly understood. The purpose of this study was to explore AE pathology using (11)C-methionine (Met) positron emission tomography (PET)/computed tomography (CT) in patients who have TLE with AE. MATERIALS AND METHODS Twenty-six TLE patients with AE and 18 TLE patients without AE underwent (11)C-Met PET/CT and magnetic resonance imaging (MRI). (11)C-Met uptake in amygdala was evaluated by both visual inspection and semi-quantitative measurement. MRI was assessed visually and semi-quantitatively for AE. Laterality index (LI) was obtained by comparing the amygdala volumes of ipsilateral and contralateral sides. The clinical course and histopathological findings of all patients were also analyzed. RESULTS On (11)C-Met PET/CT images, visual examination detected increased uptake in the enlarged amygdala in 7 of 26 (27%) TLE patients with AE, and the results were confirmed by semi-quantitative analysis. Among six TLE patients with AE who underwent surgery, histopathology revealed neoplasms (low grade astrocytoma and gangliocytoma) in two patients and focal cortical dysplasia in one patient with increased (11)C-Met uptake, but non-neoplastic lesions (focal cortical dysplasia, vacuolar degeneration, and hamartoma) in all three patients with no increased (11)C-Met uptake. On MRI, volume of the affected amygdala was significantly larger compared to the contralateral amygdala. LI was significantly higher in the group with AE than in the group without AE. CONCLUSIONS This study revealed that some TLE patients with AE showed increased (11)C-Met uptake in the enlarged amygdala. (11)C-Met PET/CT is potentially useful for the evaluation of AE pathology, and may provide beneficial information for appropriate decision-making.

PNES around the world: where we are now and how we can close the diagnosis and treatment gaps. An ILAE PNES Task Force report

An international consensus clinical practice statement issued in 2011 ranked psychogenic nonepileptic seizures (PNES) among the top three neuropsychiatric problems. An ILAE PNES Task Force was founded and initially charged with summarizing the current state of the art in terms of diagnosis and treatment, resulting in two publications. The first described different levels of diagnostic certainty. The second summarized current knowledge of management approaches. The present paper summarizes an international workshop of the ILAE PNES Task Force that focused on the current understanding and management of PNES around the world. We initially provide a knowledge update about the etiology, epidemiology, and prognosis of PNES—in adults and in special patient groups, such as children, older adults, and those with intellectual disability. We then explore clinical management pathways and obstacles to optimal care for this disorder around the world by focusing on a number of countries with different cultural backgrounds and at very different stages of social and economic development (United Kingdom, U.S.A., Zambia, Georgia, China, and Japan). Although evidence‐based methods for the diagnosis and treatment of PNES have now been described, and much is known about the biopsychosocial underpinnings of this disorder, this paper describes gaps in care (not only in less developed countries) that result in patients with PNES not having adequate access to healthcare provisions. A range of challenges requiring solutions tailored to different healthcare systems emerges. Continued attention to PNES by the ILAE and other national and international neurologic, psychiatric, and health organizations, along with ongoing international collaboration, should ensure that patients with PNES do not lose out as healthcare services evolve around the world.

A case of autoimmune epilepsy associated with anti-leucine-rich glioma inactivated subunit 1 antibodies manifesting electrical shock-like sensations and transparent sadness.

Autoimmune epilepsy is an isolated phenotype of autoimmune encephalitis, which may be suspected in patients with unexplained adult-onset seizure disorders or resistance to antiepileptic drugs (AEDs). Antibodies against leucine-rich glioma inactivated subunit 1 of the voltage-gated potassium channel (VGKC) complex, recently termed anti-LGI-1 antibodies, are one of the causes of autoimmune epilepsies. Bizarre symptoms with extremely short duration and high frequency are clues to the possible presence of autoimmune epilepsy with anti-LGI-1 antibodies. Precise diagnosis is important because autoimmune epilepsy is treatable and the prognosis can be predicted.

Marked accumulation of oligodendroglia-like cells in temporal lobe epilepsy with amygdala enlargement and hippocampal sclerosis

Although an increasing number of cases of temporal lobe epilepsy (TLE) with ipsilateral amygdala enlargement (AE) have been reported, there are few pathological reports, and no clear consensus has been established. Oligodendroglia or oligodendroglia‐like cells (OLCs) have recently attracted attention in epilepsy studies. Here, we report the clinical and pathological findings of a 40‐year‐old male TLE patient with AE and hippocampal sclerosis, in whom histopathological study demonstrated remarkable clustering of OLCs around the uncus. The patient began to have refractory seizures at the age of 14, and preoperative MRI revealed left amygdala enlargement and left hippocampal atrophy. Other examinations were consistent with left mesial temporal epileptogenicity. He underwent surgical resection and achieved seizure freedom. Histopathological study of the amygdala showed swollen neurons with relatively large bodies and thick neurites, accompanied by vacuolar degeneration in the background. Additionally, there were marked clusters of OLCs with round nuclei and densely stained chromatin around the uncus. The OLCs were Olig2‐positive. In the hippocampus, severe cell loss in CA1 and granule cell dispersion in the dentate gyrus were found. These findings may provide some insights for further pathological investigations of TLE with non‐neoplastic AE.

Improvement in anti-N-methyl-d-aspartate receptor antibody-mediated temporal lobe epilepsy with amygdala enlargement without immunotherapy

Focal neuroinflammation is considered one of the hypotheses for the cause of temporal lobe epilepsy (TLE) with amygdala enlargement (AE). Here, we report a case involving an adult female patient with TLE-AE characterized by late-onset seizures and cognitive impairment. Anti-N-methyl-d-aspartate receptor (NMDAR) antibodies were detected in her cerebrospinal fluid. However, administration of appropriate anti-seizure drugs (ASD), without immunotherapy, improved TLE-AE associated with NMDAR antibodies. In the present case, two clinically significant observations were made: 1) anti-NMDAR antibody-mediated autoimmune processes may be associated with TLE-AE, and 2) appropriate administration of ASD alone can improve clinical symptoms in mild cases of autoimmune epilepsy.

A case of interictal dysphoric disorder comorbid with interictal psychosis: Part of the same spectrum or separate entities?

Depressive disorders in epilepsy often present characteristic clinical manifestations atypical in primary, endogenous depression. Here, we report a case of a 64-year-old woman with right mesial temporal lobe epilepsy, who complained of bizarre, antipsychotic-refractory cenesthetic hallucinations in her interictal phase, and was hospitalized after a suicide attempt. Detailed clinical observations revealed mood symptoms, which led to the diagnosis of interictal dysphoric disorder comorbid with interictal psychosis. Sertraline with low-dose aripiprazole markedly alleviated both depressive and psychotic symptoms. This case suggested that the two diagnostic entities may overlap and that depressive symptoms tend to be concurrent when concurring with psychosis, which hampers the appropriate choice of a treatment option.

Emotional stimuli-provoked seizures potentially misdiagnosed as psychogenic non-epileptic attacks: A case of temporal lobe epilepsy with amygdala enlargement

The association between emotional stimuli and temporal lobe epilepsy (TLE) is largely unknown. Here, we report the case of a depressed, 50-year-old female complaining of episodes of a “spaced out” experience precipitated by emotional stimuli. Psychogenic non-epileptic attacks were suspected. However, video-EEG coupled with emotional stimuli-provoked procedures and MRI findings of amygdala enlargement, led to the diagnosis of left TLE. Accurate diagnosis and explanation improved her subjective depression and seizure frequency. This case demonstrated that emotional stimuli can provoke seizures in TLE and suggested the involvement of the enlarged amygdala and the modulation of emotion-related neural circuits.

Adult-onset refractory epilepsy with hypothalamic hamartoma and no gelastic seizures successfully treated by stereotactic thermocoagulation: A case report

Daichi Sone *, Takanobu Kaido , Masako Watanabe , Yoshiko Murata , Go Taniguchi , Taisuke Otsuki c a Department of Psychiatry, National Center of Neurology and Psychiatry, Tokyo, Japan 4-1-1 Ogawa-Higashi, Kodaira, Tokyo 187-8551, Japan b Department of Neuropsychiatry, Graduate School of Medicine, the University of Tokyo, Tokyo, Japan 7-3-1, Hongo, Bunkyo, Tokyo 113-8654, Japan c Department of Neurosurgery, National Center of Neurology and Psychiatry, Tokyo, Japan d Department of Neuropsychiatry, the University of Tokyo Hospital, Tokyo, Japan