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Jiro Nudeshima and Takaomi Taira

In Japan, there has been no neurosurgical treatment for psychiatric disorders since the 1970s. Even deep brain stimulation (DBS) has not been studied or used for psychiatric disorders. Neurosurgery for psychiatric disorders has been thwarted by social taboos for many years, and psychiatrists today seem to simply ignore modern developments and therapies offered by neurosurgery such as DBS. As a result, most patients and their families do not know such “last-resort” options exist.

Historically, as in other countries, frontal lobotomies were widely performed in Japan in the 1940s and 1950s, and some Japanese neurosurgeons used stereotactic methods for the treatment of psychiatric disorders until the 1960s. However, in the 1960s and 1970s such surgical treatments began to receive condemnation based on political judgment, rather than on medical and scientific evaluation. Protest campaigns at the time hinged on the prevailing political beliefs, forming a part of the new “left” movement against leading authorities across a wide range of societal institutions including medical schools. Finally, the Japanese Society for Psychiatry and Neurology banned the surgical treatment for psychiatric disorders in 1975. Even today, Japan’s dark history continues to exert an enormous negative influence on neurosurgery for psychiatric disorders.

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Takaomi Taira, Tomonori Kobayashi and Tomokatsu Hori

✓ Lesch—Nyhan syndrome (LNS) is an X-linked hereditary disorder caused by a deficiency of hypoxanthine-guanine phosphoribosyltransferase. Patients with this syndrome are characterized by hyperuricemia, self-mutilation, developmental retardation, and movement disorders such as spasticity and dystonia. The authors performed bilateral chronic stimulation of the globus pallidus internus for control of dystonic movements in a 19-year-old man with LNS. His self-mutilating behavior unexpectedly disappeared after chronic stimulation. This is the first case of LNS that has been successfully treated with deep brain stimulation. The findings indicate that neurobehavioral features of this syndrome are either mediated in the basal ganglia pathways or secondary to the dystonia.

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Takaomi Taira, Tomonori Kobayashi, Kenji Takahashi and Tomokatsu Hori

Object. The Bertrand selective peripheral denervation for cervical dystonia (CD) has been well described, and its effectiveness and safety are established. It is, however, always accompanied by postoperative sensory loss in the C-2 region. Intraoperative bleeding from epidural venous plexuses may also be problematic. The authors developed a new denervation procedure with which to avoid such complications and compared the surgery-related results with those of the traditional Bertrand operation.

Methods. The new procedure consists of intradural rhizotomy of the anterior C-1 and C-2 nerve roots, extradural peripheral ramisectomy from C-3 to C-6, and selective section of peripheral branches of the accessory nerve to the sternocleidomastoid muscle. This procedure was performed in 30 patients (Group A). The results of this procedure were compared with those obtained in a matched group of 31 patients in the authors' series who underwent Bertrand denervation (Group B).

Changes of CD rating score at 6-month follow up did not differ between the two groups. In one patient in Group A a C-2 sensory deficit was found, whereas C-2 sensory deficits were demonstrated in all the patients in Group B. No patients in Group A and four patients in Group B experienced occipital neuralgia. The operative time was significantly shorter in Group A. The mean intraoperative blood loss was 115 ± 30 ml (± standard deviation) in Group A and 233 ± 65 ml in Group B (p < 0.005).

Conclusions. Although symptomatic improvement is the same after the Bertrand operation, the authors' new procedure for CD was associated with a lower incidence of complications and significant decrease of intraoperative blood loss.

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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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Hiroki Hori, Toshio Yamaguchi, Yoshiyuki Konishi, Takaomi Taira and Yoshihiro Muragaki

OBJECTIVE

This study evaluated changes of fractional anisotropy (FA) in the ventral intermediate nucleus (VIM) of the thalamus after transcranial MR-guided focused ultrasound (TcMRgFUS) thalamotomy and their associations with clinical outcome.

METHODS

Clinical and radiological data of 12 patients with medically refractory essential tremor (mean age 76.5 years) who underwent TcMRgFUS thalamotomy with VIM targeting were analyzed retrospectively. The Clinical Rating Scale for Tremor (CRST) score was calculated before and at 1 year after treatment. Measurements of the relative FA (rFA) values, defined as ratio of the FA value in the targeted VIM to the FA value in the contralateral VIM, were performed before thalamotomy, and 1 day and 1 year thereafter.

RESULTS

TcMRgFUS thalamotomy was well tolerated and no long-term complications were noted. At 1-year follow-up, 8 patients demonstrated relief of tremor (improvement group), whereas in 4 others persistent tremor was noted (recurrence group). In the entire cohort, mean rFA values in the targeted VIM before treatment, and at 1 day and 1 year after treatment, were 1.12 ± 0.15, 0.44 ± 0.13, and 0.82 ± 0.22, respectively (p < 0.001). rFA values were consistently higher in the recurrence group compared with the improvement group, and the difference reached statistical significance at 1 day (p < 0.05) and 1 year (p < 0.01) after treatment. There was a statistically significant (p < 0.01) positive correlation between rFA values in the targeted VIM at 1 day after thalamotomy and CRST score at 1 year after treatment. Receiver operating characteristic curve analysis revealed that the optimal cutoff value of rFA at 1 day after thalamotomy for prediction of symptomatic improvement at 1-year follow-up is 0.54.

CONCLUSIONS

TcMRgFUS thalamotomy results in significant decrease of rFA in the targeted VIM, at both 1 day and 1 year after treatment. Relative FA values at 1 day after treatment showed significant correlation with CRST score at 1-year follow-up. Therefore, FA may be considered a possible imaging biomarker for early prediction of clinical outcome after TcMRgFUS thalamotomy for essential tremor.

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Shiro Horisawa, Taku Ochiai, Shinichi Goto, Takeshi Nakajima, Nobuhiko Takeda, Takakazu Kawamata and Takaomi Taira

OBJECTIVE

Meige syndrome is characterized by blepharospasm and varied subphenotypes of craniocervical dystonia. Current literature on pallidal surgery for Meige syndrome is limited to case reports and a few small-scale studies. The authors investigated the clinical outcomes of deep brain stimulation (DBS) of the globus pallidus internus (GPi) in patients with Meige syndrome.

METHODS

Sixteen patients who underwent GPi DBS at the Tokyo Women’s Medical University Hospital between 2002 and 2015 were included in this study. Burke-Fahn-Marsden Dystonia Rating Scale (BFMDRS) movement subscale (BFMDRS-M) scores (range 0–120) obtained at the following 3 time points were included in this analysis: before surgery, 3 months after surgery, and at the most recent follow-up evaluation.

RESULTS

The patients’ mean age (± SD) at symptom onset was 46.7 ± 10.1 years, and the mean disease duration at the time of the authors’ initial evaluation was 5.9 ± 4.1 years. In 12 patients, the initial symptom was blepharospasm, and the other 4 patients presented with cervical dystonia. The mean postoperative follow-up period was 66.6 ± 40.7 months (range 13–150 months). The mean total BFMDRS-M scores at the 3 time points were 16.3 ± 5.5, 5.5 ± 5.6 (66.3% improvement, p < 0.001), and 6.7 ± 7.3 (58.9% improvement, p < 0.001).

CONCLUSIONS

The results indicate long-term efficacy for GPi DBS for the majority of patients with Meige syndrome.

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Shiro Horisawa, Toshio Yamaguchi, Keiichi Abe, Hiroki Hori, Masatake Sumi, Yoshiyuki Konishi and Takaomi Taira

Musician’s dystonia (MD) is a type of focal hand dystonia that develops only while playing musical instruments and interferes with skilled and fine movements. Lesioning of the ventro-oral (Vo) nucleus of the thalamus (Vo-thalamotomy) using radiofrequency can cause dramatic improvement in MD symptoms. Focused ultrasound (FUS) can make intracranial focal lesions without an incision. The authors used MRI-guided FUS (MRgFUS) to create a lesion on the Vo nucleus to treat a patient with MD. Tubiana’s MD scale (TMDS) was used to evaluate the condition of musical play ranging from 1 to 5 (1: worst, 5: best). The patient was a 35-year-old right-handed man with involuntary flexion of the right second, third, and fourth fingers, which occurred while playing a classical guitar. Immediately after therapeutic sonications of FUS Vo-thalamotomy, there was dramatic improvement in the MD symptoms. The TMDS scores before; at 0 and 1 week after; and at 1, 3, 6, and 12 months after MRgFUS Vo-thalamotomy were 1, 4, 4, 5, 5, 5, and 5, respectively. No complications were observed. Focused ultrasound Vo-thalamotomy can be an effective treatment for MD.