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Shiro Horisawa, Atsushi Fukui, Kotaro Kohara, Takakazu Kawamata, and Takaomi Taira

OBJECTIVE

The objective of this study was to assess the efficacy of unilateral pallidotomy in patients with asymmetrical cervical dystonia.

METHODS

This study retrospectively included 25 consecutive patients with asymmetrical cervical dystonia refractory to botulinum toxin injections, who underwent unilateral pallidotomy between January 2015 and April 2017. Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) scores were evaluated preoperatively and 1 week, 3 months, and 6 months postoperatively. The clinical responses were defined as good responders, exhibiting > 50% improvement in the TWSTRS score at 6 months postsurgery, or poor responders, exhibiting < 50% improvement in TWSTRS scores at 6 months postsurgery.

RESULTS

Twelve and 9 patients showed right- and left-side rotation, respectively; 1 and 3 patients had right- and left-side laterocollis, respectively. The mean age of onset and duration of the disease were 40.2 ± 13.9 and 8.9 ± 10.9 years, respectively. Mean TWSTRS scores were 38.4 ± 12.6 (p < 0.001), 17.3 ± 12.4 (p < 0.001), 19.5 ± 13.4 (p < 0.001), and 20.0 ± 14.7 (p < 0.001), preoperatively and 1 week, 3 months, and 6 months postoperatively, respectively. Fourteen patients (56%) demonstrated > 50% improvement in their TWSTRS total score (mean improvement of TWSTRS total score = 70.5%) 6 months postsurgically. Furthermore, preoperative TWSTRS severity score was a prognostic factor (odds ratio 1.37, 95% confidence interval 1.06–1.78, p = 0.003).

CONCLUSIONS

These results suggest that unilateral pallidotomy is an acceptable treatment option for asymmetrical cervical dystonia. Further investigations with a larger number of cases and longer follow-up period are required to confirm these data.

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Shiro Horisawa, Takashi Arai, Naoki Suzuki, Takakazu Kawamata, and Takaomi Taira

Cerebellar neuromodulation could influence the pathological abnormalities of movement disorders through several connections between the cerebellum and the basal ganglia or other cortices. In the present report, the authors demonstrate the effects of cerebellar deep brain stimulation (DBS) on a patient with severe generalized fixed dystonia (FD) that was refractory to bilateral pallidotomy and intrathecal baclofen therapy. A previously healthy 16-year-old girl presented with generalized FD. Bilateral pallidotomy and intrathecal baclofen therapy had failed to resolve her condition, following which she received DBS through the bilateral superior cerebellar peduncle (SCP) and dentate nucleus (DN). Ipsilateral stimulation of the SCP or DN improved the FD, and the ability of DBS administered via the SCP to relax muscles was better than that of DN DBS. A considerable improvement of generalized FD, from a bedridden state to a wheelchair-bound state, was observed in the patient following 6 months of chronic bilateral DBS via the SCP; moreover, the patient was able to move her arms and legs. The findings in the present case suggest that neuromodulation of deep cerebellar structures is a promising treatment for FD that is refractory to conventional treatments.

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Shiro Horisawa, Mieko Oka, Kotaro Kohara, Takakazu Kawamata, and Takaomi Taira

Camptocormia is a rare, involuntary movement disorder, presenting as truncal flexion while standing or walking, and is mainly observed as a feature of Parkinson’s disease (PD) and primary dystonia. Deep brain stimulation (DBS) of the globus pallidus internus is effective for refractory camptocormia observed with PD or dystonia. However, the effectiveness of pallidotomy for camptocormia has not been investigated. The authors report the case of a 38-year-old man with anterior truncal bending that developed when he was 36 years old. Prior to the onset of the symptom, he had been taking antipsychotic drugs for schizophrenia. There were no features of PD; the symptom severely interfered with his walking and daily life. He was given anticholinergics, clonazepam, and botulinum toxin injections, which did not result in much success. Because of the patient’s unwillingness to undergo implantation of a hardware device, he underwent staged bilateral pallidotomy with complete resolution for a diagnosis of tardive dystonic camptocormia. The Burke-Fahn-Marsden dystonia rating scale subscore for the trunk before and after bilateral pallidotomy was 3 and 0, respectively. No perioperative adverse events were observed. Effects have persisted for 18 months. Bilateral pallidotomy can be a treatment option for medically refractory dystonic camptocormia without the need for device implantation.

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Shunsuke Nomura, Akitsugu Kawashima, Hiroyuki Akagawa, and Takakazu Kawamata

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Taku Yoneyama, Takakazu Kawamata, Masahiko Tanaka, Koji Yamaguchi, and Yoshikazu Okada

In carotid endarterectomy (CEA), the traditional retractors are often difficult to use because they tend to obstruct surgical manipulations, especially in the deep operative field on the rostral side. The authors have invented a new omnidirectional retractor-supporting ring (OD ring) to solve the problems of traditional retractors. The OD ring has an ellipsoid-shaped frame (major axis: 275 mm, minor axis: 192 mm) with 22 equally spaced outward protrusions. Rubber bands from which blunt mini-hooks are hung are twisted around the protrusions. The OD ring was placed on the operative area, and the skin edges were retracted by mini-hooks placed symmetrically. The hooks were moved gradually from the shallow to the deep operative field as surgical dissection continued to expose the carotid bifurcation and distal internal carotid artery (ICA). The OD ring was used in 158 consecutive CEAs in the authors' institute between July 2010 and October 2013. The OD ring provided a flatter surgical field and was less obstructive than traditional retractors, thereby facilitating surgical manipulation in the deep operative field such as at the distal ICA. Furthermore, because of its simpler shape, angiorrhaphy could be conducted more smoothly, with less tangled thread during closure of the arteriotomy. There were no technical complications related to the OD ring. As a new retractor system for CEA, the OD ring is less obstructive and provides a flatter surgical field than traditional retractors, thereby facilitating surgical manipulations in the deep operative field around the distal ICA.

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Takakazu Kawamata, John W. Peterson, Takao Bun, and Nicholas T. Zervas

✓ Although phorbol esters, synthetic activators of protein kinase C (PKC), can stimulate large increases in the binding of cytosolic PKC to form membrane-bound PKC (PKCm, an indicator of PKC activation), the authors report that even small increases in PKCm induced by phorbol esters (8–12% of total PKC content) can be associated with significant PKC-mediated contractions in vitro (50–85% of maximum) in normal canine cerebral arteries. Increases in PKCm of similarly small magnitude were found in vitro when control artery segments were exposed to hemolysate, but only if the arterial smooth-muscle cells were first slightly depolarized by increased extracellular potassium to values of membrane potential similar to those observed in canine cerebral arteries during chronic cerebral vasospasm. These increases in PKCm (6–8% of total PKC content) coincided with a greatly augmented contractile response to hemolysate. These results show that the previous observation of only a small increase in PKCm (approximately 7% of total PKC content) after experimental subarachnoid hemorrhage in the canine model does not preclude a potentially important role for PKC-mediated contraction in the pathogenesis of cerebral vasospasm.

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Hiroyuki Kurihara, Koji Yamaguchi, Tatsuya Ishikawa, Takayuki Funatsu, Go Matsuoka, Yoshihiro Omura, Yoshikazu Okada, and Takakazu Kawamata

Surgical treatments for moyamoya disease (MMD) include direct revascularization procedures with proven efficacy, for example, superficial temporal artery (STA) to middle cerebral artery (MCA) bypass, STA to anterior cerebral artery bypass, occipital artery (OA) to MCA bypass, or OA to posterior cerebral artery bypass. In cases with poor development of the parietal branch of the STA, the posterior auricular artery (PAA) is often developed and can be used as the bypass donor artery. In this report, the authors describe double direct bypass performed using only the PAA as the donor in the initial surgery for MMD.

In the authors’ institution, MMD is routinely treated with an STA-MCA double bypass. Some patients, however, have poor STA development, and in these cases the PAA is used as the donor artery. The authors report the use of the PAA in the treatment of 4 MMD patients at their institution from 2013 to 2016. In all 4 cases, a double direct bypass was performed, with transposition of the PAA as the donor artery. Good patency was confirmed in all cases via intraoperative indocyanine green angiography and postoperative MRA or cerebral angiography. The mean blood flow measurement during surgery was 58 ml/min. No patients suffered a stroke after revascularization surgery.

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Fujun Liu, Wei Chen, and Jing Chen

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Tetsuryu Mitsuyama, Takakazu Kawamata, Fumitaka Yamane, Akira Awaya, and Tomokatsu Hori

Object. A synthetic heterocyclic pyrimidine compound, MS-818 (2-piperadino-6-methyl-5-oxo-5,6-dihydro-(7H) pyrrolo-[3,4-d] pyrimidine maleate) is reported to have a variety of biological activities including neurite outgrowth, astrocyte differentiation, suppression of neuronal apoptosis, regeneration of injured peripheral nerves, fracture repairs, angiogenesis, and superovulation. To be able to explicate the neurotrophic effects of MS-818, the authors evaluated its effect on the reduction of infarct volume and amelioration of sensorimotor dysfunction in a rat model of focal ischemia.

Methods. Forty male Sprague—Dawley rats were subjected to right middle cerebral artery occlusion and assigned to one of four treatment groups (10 animals in each group). The MS-818 (1, 5, or 10 mg/kg) or phosphate-buffered saline (control group) was administered intraperitoneally at onset of ischemia and again 24 hours later. The rats were killed 48 hours after they underwent surgery to induce stroke, and infarct volume was determined using an image-analysis computer software program following staining with 2,3,5-triphenyltetrazolium chloride. Postischemic neurological deficit and body weight were also assessed.

Conclusions. Significant reductions in infarct volume (total and cortical infarction) were found in all the MS-818—treated groups compared with the control group. Furthermore, MS-818 induced significant amelioration of sensorimotor dysfunction, as indicated by the results of forelimb and hindlimb placing tests. The present findings suggest that MS-818, which has a much smaller molecular weight than neurotrophic peptides, represents a new approach to the treatment of focal cerebral ischemia.