Objective We examined the prevalence and clinical features of primary exercise headache (PEH) in middleaged Japanese population. Methods A headache specialist interviewed middle-aged subjects serially on health check-up. The primary headaches were diagnosed according to the International Classification of Headache Disorders (ICHD-III beta). Cardiovascular disease (CVD) risk and radiological findings were analyzed. Prevalence of PEH and clinical features were assessed. Results Among 2,546 subjects (1,588 men and 958 women), thirty subjects (13 men and 17 women) were diagnosed with PEH. The prevalence of PEH was 1.19%, 0.82% in men and 1.77% in women. The mean age [standard deviation (SD)] of the subjects was 44.3 (8.8) years and their mean duration (SD) of PEH was 4.5 (7.0) months. Headache occurred bilaterally (23 patients) or unilaterally (7 patients), and in the occipital (16 patients), frontal (10 patients) or diffuse region (4 patients). The persistent headache time ranged from 5 minutes to 12 hours. The degree of headache severity was classified as mild (13 patients), moderate (5 patients) or severe degree (12 patients). PEH was triggered by gym training (16 patients), swimming (6 patients), running (6 patient) and skiing (2 patients). All patients were exercise beginners or played a sport occasionally. No patients visited physicians for headache consultation. Other primary headaches coexisted in 20 patients (67%). Twenty patients had migraine without aura (MO). Seven patients had headache associated with sexual activity. Five patients had cough headache. Two patients had CVD risk factors. Conclusion The present study of PEH indicated the prevalence of 1.2% and the female/male ratio of 2.1 in middle-aged Japanese. The comorbidity rate of MO was high. PEH may not be an uncommon headache in middle-aged MO sufferers and sport beginners.
BackgroundCerebrotendinous xanthomatosis (CTX) is a rare autosomal recessive sterol storage disease caused by a mutated sterol 27-hydroxylase (CYP27A1) gene. Patients with typical CTX show neurological dysfunction including bilateral cataracts, paresis, cerebral ataxia, dementia, and psychiatric disorders, and magnetic resonance imaging (MRI) has revealed symmetrical lesions in the cerebellar white matter.Case presentationWe report the case of a patient with late-onset spinal form CTX without brain lesion. He showed pyramidal tract signs, and impaired joint position and vibration sensation in the lower limbs. Cervical sagittal MRI demonstrated a longitudinally extensive white matter abnormality in the dorsal column of the C2-C7 spinal cord; however, a brain MRI revealed an absence of lesions, including in the cerebellar white matter. Genetic analysis of CYP27A1 revealed that the patient was compound heterozygous for p.Gln85Arg in exon 1, a novel mutation, and p.Arg405Gln in exon 7, a previously reported mutation.ConclusionThis is the first report of late-onset spinal form CTX without typical neurological symptoms, and the first report of p.Gln85Arg in CYP27A1. We speculate that spinal form CTX without brain lesion is a clinically and radiologically rare variation of CTX. Therefore, spinal xanthomatosis should be included in the differential diagnosis of chronic myelopathy even with late-onset and/or no other typical neurological findings.
Background Little is known about single administration of zonisamide in Parkinson's disease patients. Aim We aimed to evaluate the therapeutic effects of zonisamide monotherapy in early Parkinson's disease patients. Methods A total of 10 untreated (de novo) patients with Yahr stage I or II participated. Zonisamide was given at a daily dose of 25 mg for 1 month and increased to 50 mg for the next 2 months. Unified Parkinson's Disease Rating Scale parts I–IV and tremor‐related Unified Parkinson's Disease Rating Scale (items 16, 20 and 21) were examined every month. Serum zonisamide concentrations and urinary homovanillic acid concentrations were measured. Three patients had rapid eye movement sleep behavior disorder. Sleep condition was asked to patients? spouses. Results At 1 month after zonisamide treatment, Unified Parkinson's Disease Rating Scale scores did not differ from the baseline scores statistically. At 2 and 3 months after zonisamide treatment, Unified Parkinson's Disease Rating Scale part III and tremor‐related scores were reduced significantly compared with pretreatment (P < 0.01). Sleep state improved markedly in three patients with rapid eye movement sleep behavior disorder at 1–2 months after zonisamide treatment. The mean serum concentrations of zonisamide (standard deviation) were 2.0 μg/mL (0.9) at 25 mg/day and 6.6 μg/mL (2.3) at 50 mg/day. Urinary homovanillic acid concentrations were not altered before and after zonisamide administration. Conclusions The present study showed that zonisamide monotherapy improved motor and sleep dysfunction in de novo patients with early Parkinson's disease. Significant therapeutic benefits were present within 1–2 months. Finally, the data highlight a therapeutic potential of zonisamide monotherapy in tremor‐predominant patients with early Parkinson's disease.
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