Surgical Outcome of Children and Adolescents with Tethered Cord Syndrome

Toshitaka Seki,Kazutoshi Hida,Shunsuke Yano,Toru Sasamori,Shuji Hamauchi,Izumi Koyanagi,Kiyohiro Houkin 대한척추외과학회 2016 Asian Spine Journal Vol.10 No.5

Study Design: Retrospective cohort study. Purpose: To compare long-term results of surgery with the outcomes of symptomatic and asymptomatic tethered cord syndrome (TCS) in children and adolescents and to assess the surgery duration for those with TCS. Overview of Literature: Pediatric patients with TCS continue to pose significant diagnostic and management challenges. Methods: We retrospectively analyzed the outcomes of 31 patients (16 males, 15 females) with TCS, including 21 with lumbosacral lipoma. All were surgically treated between 1989 and 2015. They were divided into symptomatic and asymptomatic TCS groups. The results of the treatment were summarized and analyzed using a non-parametric Mann–Whitney U test. Results: Nineteen patients had symptomatic TCS and 12 had asymptomatic TCS. Patients had a median age of 34 months (range, 0–201 months). The median follow-up period was 116 months (range, 7–223 months). Of the 19 symptomatic TCS patients, preoperative deficits improved after surgery in two (10.5%) and remained stable in 17 (89.5%) patients. One of the 12 asymptomatic TCS (8.3%) patients showed an exacerbated illness after surgery, and one in 11 patients remained stable (11.7%). There were significant differences in monthly age at surgery, preoperative bowel and bladder dysfunction, neurological function, presence/absence of clean intermittent catheterization introduction, and presence/absence of motor disorder at final follow-up (all p <0.05). In the univariate analysis, the presence/absence of preoperative bowel and bladder dysfunction, and symptoms were strongly associated with the risk of children and adolescents with TCS (p <0.05). Conclusions: Early accurate diagnosis and adequate surgical release might lead to successful outcomes in children and adolescents with TCS. Surgical untethering is a safe and effective method for treatment of children with TCS.

Surgical Outcomes of Pediatric Patients with Asymptomatic Tethered Cord Syndrome

Toshitaka Seki,Kazutoshi Hida,Shunsuke Yano,Kiyohiro Houkin 대한척추외과학회 2018 Asian Spine Journal Vol.12 No.3

Study Design: A retrospective cohort study. Purpose: To examine the validity of prophylactic surgery for children with tethered cord syndrome (TCS). Overview of Literature: Prophylactic surgery for pediatric patients with TCS remains controversial. Methods: We retrospectively analyzed the surgical outcomes of 14 children (nine boys and five girls) with asymptomatic TCS who were surgically treated at Hokkaido University Hospital between 1989 and 2015. Results: The median age at the time of initial surgery for asymptomatic TCS was 28.6 months (range, 0–66 months). The median final follow-up period was 142 months (range, 7–232 months). Of the 14 children with asymptomatic TCS, 12 had lumbosacral lipoma and two had meningocele. According to the classification of spinal lipoma, two children had dorsal type, four had caudal type, two had transitional type, and four had filar type. There were no children with lipomyelomeningocele. All children were free of neurological symptoms until 94 months after the initial surgery. Subsequently, one child exhibited delayed neurological deficits and underwent a second surgery because of motor and sensory disturbances; slight sensory disturbance was noted at the final follow-up examination. Another child later showed bowel and bladder dysfunction. However, a second surgery was not performed for this child because his motor and sensory functions were normal; hence, we chose to avoid nerve injury in the case of dissecting adhesion. Conclusions: All 14 children with asymptomatic TCS were free of neurological symptoms until 94 months after the initial surgery. However, two children exhibited delayed neurological deficits at 94 months and 177 months. We believe that prophylactic surgery for asymptomatic TCS is effective for a certain period. However, because the natural history of TCS is poorly understood, strict follow-up after surgery is necessary.

Clinical Factors for Prognosis and Treatment Guidance of Spinal Cord Astrocytoma

Toshitaka Seki,Kazutoshi Hida,Shunsuke Yano,Takeshi Aoyama,Izumi Koyanagi,Toru Sasamori,Shuji Hamauchi,Kiyohiro Houkin 대한척추외과학회 2016 Asian Spine Journal Vol.10 No.4

Study Design: Retrospective study. Purpose: To obtain information useful in establishing treatment guidelines by evaluating baseline clinical features and treatment outcomes of patients with spinal cord astrocytoma (SCA). Overview of Literature: The optimal management of SCA remains controversial, and there are no standard guidelines. Methods: The study included 20 patients with low-grade and 13 with high-grade SCA surgically treated between 1989 and 2014. Patients were classified according to the extent of surgical resection. Survival was assessed using Kaplan–Meier plots and compared between groups by log-rank tests. Neurological status was defined by the modified McCormick scale and compared between groups by Mann–Whitney U tests. Results: Surgical resection was performed for 19 of 20 low-grade (95%) and 10 of 13 high-grade (76.9%) SCA patients. Only nine patients (27.3%) underwent gross total resection, all of whom had low-grade SCA. Of all patients, 51.5% showed deteriorated neurological status compared to preoperative baseline. Median overall survival was significantly longer for low-grade SCA than that for high-grade SCA (91 months, 78% at 5 years vs. 15 months, 31% at 5 years; p =0.007). Low-grade SCA patients benefited from more aggressive resection, whereas high-grade SCA patients did not. Multivariate analysis revealed histology status (hazard ratio [HR], 0.30; 95% confidence interval [CI], 0.09–0.98; p <0.05) and postoperative neurological status (HR, 0.12; CI, 0.02–0.95; p <0.05) as independent predictors of longer overall survival. Adjuvant radiotherapy had no significant impact on survival rate. However, a trend for increased survival was observed with radiation cordotomy (RCT) in high-grade SCA patients. Conclusions: Aggressive resection for low-grade and RCT for high-grade SCA may prolong survival. Preservation of neurological status is an important treatment goal. Given the low incidence of SCA, establishing strong collaborative, interdisciplinary, and multiinstitutional study groups is necessary to define optimal treatments.

Surgical Outcomes of High-Grade Spinal Cord Gliomas

Toshitaka Seki,Kazutoshi Hida,Syunsuke Yano,Takeshi Aoyama,Izumi Koyanagi,Kiyohiro Houkin 대한척추외과학회 2015 Asian Spine Journal Vol.9 No.6

Study Design: A retrospective study. Purpose: The purpose of this study was to obtain useful information for establishing the guidelines for treating high-grade spinal cord gliomas. Overview of Literature: The optimal management of high-grade spinal cord gliomas remains controversial. We report the outcomes of the surgical management of 14 high-grade spinal glioma. Methods: We analyzed the outcomes of 14 patients with high-grade spinal cord gliomas who were surgically treated between 1989 and 2012. Survival was charted with the Kaplan-Meier plots and comparisons were made with the log-rank test. Results: None of the patients with high-grade spinal cord gliomas underwent total resection. Subtotal resection was performed in two patients, partial resection was performed in nine patients, and open biopsy was performed in three patients. All patients underwent postoperative radiotherapy and six patients further underwent radiation cordotomy. The median survival time for patients with high-grade spinal cord gliomas was 15 months, with a 5-year survival rate of 22.2%. The median survival time for patients with World Health Organization grade III tumors was 25.5 months, whereas the median survival time for patients with glioblastoma multiforme was 12.5 months. Both univariate and multivariate Cox proportional hazards models demonstrated a significant effect only in the group that did not include cervical cord lesion as a factor associated with survival (p =0.04 and 0.03). Conclusions: The surgical outcome of patients diagnosed with high-grade spinal cord gliomas remains poor. Notably, only the model which excluded cervical cord lesions as a factor significantly predicted survival.

Totally Ossified Metaplastic Spinal Meningioma

Ju, Chang Il,Hida, Kazutoshi,Yamauchi, Tomohiro,Houkin, Kiyohiro The Korean Neurosurgical Society 2013 Journal of Korean neurosurgical society Vol.54 No.3

A 61-year-old woman with a very rare case of totally ossified large thoracic spinal metaplastic meningioma, showing progressing myelopathy is presented. Computed tomographic images showed a large totally ossfied intradural round mass occupying the spinal canal on T9-10 level. Magnetic resonance imaging revealed a large T9-10 intradural extramedullary mass that was hypointense to spinal cord on T1- and T2-weighted sequences, partial enhancement was apparent after Gadolinium administration. The spinal cord was severely compressed and displaced toward the right at the level of T9-10. Surgical removal of the tumor was successfully accomplished via the posterior midline approach and the histological diagnosis verified an ossified metaplastic meningioma. The clinical neurological symptoms of patient were improved postoperatively. In this article we discuss the surgical and pathological aspects of rare case of spinal totally ossified metaplastic meningioma.

Intramedullary Schwannoma of the Spinal Cord: A Nationwide Analysis by the Neurospinal Society of Japan

Takeshi Hara,Masaki Mizuno,Kazutoshi Hida,Toru Sasamori,Yasuyuki Miyoshi,Hisaaki Uchikado,Hiroki Ohashi,Taku Sugawara,Yasuhiro Takeshima,Yukoh Ohara,Akihide Kondo,Toshiki Endo 대한척추신경외과학회 2023 Neurospine Vol.20 No.3

Objective: This study was aimed to report the clinical characteristics of intramedullary schwannomas and discuss imaging findings and treatment strategies. Methods: The inclusion criterion was consecutive patients with intramedullary schwannomas who were surgically treated at 8 centers between 2009 and 2020. Clinical characteristics included age, sex, clinical presentation, disease duration, and follow-up period. The modified McCormick scale was used to compare the preoperative and postoperative conditions. Pre- and postoperative magnetic resonance images (MRI) of each case were analyzed. Results: The mean age of the total 11 patients at the operation was 50.2 years. The mean duration of the symptoms was 23 months, with limb paresthesia being the most common clinical presentation. The cervical spine was the most common localization level of the tumor in 6 cases. The mean follow-up duration was 49.4 months. Gross total resection (GTR) and subtotal resection (STR) was achieved in 9 and 2 cases, respectively. According to the modified McCormick scale at 6 months postoperatively, 7 cases (63.6%) had improved and 4 cases (36.3%) had unchanged grades. Typical MRI findings of the intramedullary schwannoma included ring-like enhancement, syringomyelia, cystic formation, intramedullary edema, and hemosiderin deposition. Gadolinium enhancement was homogenous in 8 cases (72.7%). The tumor margins were well demarcated in all cases. Conclusion: Intramedullary schwannoma should be considered when sharp margins and well-enhanced tumors are present at the cervical spine level and the initial symptoms are relatively mild, such as dysesthesia. When GTR cannot be achieved, STR for tumor decompression is recommended.

Investigation of the Neuropathic Pain Caused by Syringomyelia Associated with Chiari I Malformation

Toshitaka Seki,Shuji Hamauchi,Masayoshi Yamazaki,Kazutoshi Hida,Shunsuke Yano,Kiyohiro Houkin 대한척추외과학회 2019 Asian Spine Journal Vol.13 No.4

Study Design: Retrospective cohort study. Purpose: To investigate the correlation between the syrinx morphology and neuropathic pain caused by syringomyelia associated with Chiari I malformation. Overview of Literature: Neuropathic pain caused by syringomyelia is refractory and markedly impairs the patient. Methods: We examined 24 patients with neuropathic pain caused by syringomyelia associated with Chiari I malformation. We statistically analyzed the illness duration and age at surgery between patients with and without neuropathic pain. Additionally, we classified the morphology of the syringes into deviated (D), enlarged (E), central (C), and bulkhead (B) types using T2-weighted axial imaging. Moreover, we investigated the correlation between syrinx morphology and neuropathic pain. A Mann–Whitney U-test was performed to compare between the presence or absence of neuropathic pain and the presence or absence of type D syringes. Results: The median age at surgery was 27.5 years, and the median illness duration was 24 months. Among the 24 patients, 11 had preoperative neuropathic pain, one of which was free of neuropathic pain during the final follow-up period. Among patients with neuropathic pain, the syringes’ preoperative morphology was type D in nine patients and types E and C in one patient each. No patient exhibited type B morphology. Among patients without neuropathic pain, the preoperative morphology of the syringes was type D in three patients, type E in seven patients, and types C and B in two patients each. For types D and E, a correlation between neuropathic pain and syrinx morphology was observed. Moreover, type D was associated with significant neuropathic pain in both preoperative and postoperative states. Conclusions: This study showed a correlation between the morphological features of the syringes and the occurrence of neuropathic pain in patients with syringomyelia associated with Chiari I malformation.

Comparison of the Recurrence and Surgical Outcome of Spinal Hemangioblastoma in Sporadic and Von Hippel-Lindau Diseases: A Subanalysis of a Nationwide Study by the Neurospinal Society of Japan

Yasuhiro Takeshima,Hirokazu Takami,Toshiki Endo,Masaki Mizuno,Kazutoshi Hida 대한척추신경외과학회 2023 Neurospine Vol.20 No.3

Objective: This study aimed to clarify the relationship between recurrence and the extent of resection in surgery for intramedullary spinal hemangioblastoma (sHB) and its impact on von Hippel-Lindau (vHL) disease. Methods: Data on sHB cases followed up for at least 6 months after surgery were extracted from a nationwide registry of 1,033 consecutive spinal intramedullary tumors surgically treated between 2009 and 2020, and were retrospectively categorized into a sporadic or vHL group. The diagnosis of vHL disease was made at each institution based on clinical findings. Results: A total of 168 patients (sporadic group, 101; vHL group, 67) were included in the study. Compared with the sporadic group, the vHL group had a younger onset (45.4 ± 16.8 years vs. 39.6 ± 14.1 years, p = 0.02), more preoperative motor (47.5% vs. 68.7%, p < 0.01) and gait (37.6% vs. 61.2%, p < 0.01) impairments, and more patients with worsening neurological symptoms at discharge (p = 0.02). The gross total resection (GTR) rates and the recurrence rates were not statistically different between the sporadic and the vHL groups. GTR significantly improved recurrence-free survival compared to non-GTR in all patient analysis (p < 0.01) but this trend was not observed in the sporadic group. Physical functional improvement from discharge to 6 months after surgery was observed in the sporadic group (p < 0.01) but not in the vHL group. Conclusion: A high GTR rate may sufficiently decrease susceptibility to recurrence, especially in patients with sHB with vHL. In sporadic sHB, postoperative functional improvement can be expected, and the long-term functional prognosis is favorable.

Efficacy of Two-Stage Surgery for Spinal Cord Ependymomas

Takamiya Soichiro,Seki Toshitaka,Yamazaki Kazuyoshi,Yano Shunsuke,Hida Kazutoshi 대한척추외과학회 2022 Asian Spine Journal Vol.16 No.4

Study Design: Retrospective cohort study.Purpose: This study aimed to elucidate cases for which staged surgeries are effective by a retrospective review of previous operative cases of spinal ependymomas.Overview of Literature: Patients with spinal ependymomas are expected to have a good prognosis following total resection. However, forcible dissection of spinal ependymomas will lead to neurological deterioration. Moreover, resection is sometimes difficult when the tumor is large. We have performed two-stage surgeries for large spinal ependymomas, but the indication of staged surgery is unclear.Methods: We retrospectively reviewed patients diagnosed with spinal ependymomas who underwent tumor resection in our institution. We obtained data regarding patients’ clinical characteristics, tumoral radiological characteristics, and surgical factors and compared them to clear prognostic factors. Two-stage surgery was performed in 11 patients (36.7%), and single surgery was performed in 19 patients (63.3%).Results: Thirty patients were included in the analyses and divided into two groups: single surgery and two-stage surgery groups. In the single surgery group, high tumor–cord ratio (TCR) and intraoperative motor evoked potential (MEP) reduction were significantly correlated with unfavorable outcomes, which were defined as deterioration of the modified McCormick scale grades 2 months and 1 year postoperatively. Alternatively, these factors were not significantly correlated with postoperative unfavorable outcomes in the two-stage surgery group. Receiver operating characteristic curves indicated that TCR of 0.866 yielded 85.7% sensitivity and 83.3% specificity 2 months postoperatively.Conclusions: The results suggested that high TCR might be an indication of two-stage surgery and that its cutoff value is 0.866. Moreover, switching from single surgery to two-stage surgery may prevent postoperative neurological deterioration when intraoperative MEP is decreasing.

Current Trends in the Surgical Management of Intramedullary Tumors: A Multicenter Study of 1,033 Patients by the Neurospinal Society of Japan

Toshiki Endo,Tomoo Inoue,Masaki Mizuno,Ryu Kurokawa,Kiyoshi Ito,Shigeo Ueda,Toshihiro Takami,Kazutoshi Hida,Minoru Hoshimaru,Investigators of intramedullary spinal cord tumors in the Neurospinal Socie 대한척추신경외과학회 2022 Neurospine Vol.19 No.2

Objective: We performed a retrospective observational study to demonstrate the surgical risks and long-term prognoses of intramedullary tumors in Japan using a multicenter registry authorized by the Neurospinal Society of Japan. Methods: Data from 1,033 consecutive patients with intramedullary tumors, treated between 2009 and 2020, were collected from 58 centers. Patients with spinal lipomas or myxopapillary ependymomas were excluded. Patient characteristics, clinical presentations, imaging characteristics, treatments, and outcomes were analyzed. The modified McCormick scale was used to classify functional status. Survival was described using Kaplan-Meier curves, and multivariable logistic regression analyses were performed. Results: The mean age of the patients was 48.4 years. Data of 361 ependymomas, 196 hemangioblastomas, 168 astrocytic tumors, 160 cavernous malformations, and the remaining 126 cases including subependymomas, metastases, schwannomas, capillary hemangiomas, and intravascular B-cell lymphomas were analyzed. Twenty-two patients were undiagnosed. The mean follow-up duration was 46.1 ± 38.5 months. Gross total tumor removal was achieved in 672 tumors (65.1%). On the modified McCormick scale, 234 patients (22.7%) had worse postoperative grades at the time of discharge. However, neurological status gradually improved. At 6 months postoperatively, 251 (27.5%), 500 (54.9%), and 160 patients (17.6%) had improved, unchanged, and worsened grades, respectively. Preoperative functional status, gross total tumor removal, and histopathological type were significantly associated with mortality and functional outcomes. Conclusion: Our findings demonstrate better postoperative functional outcomes in patients with fewer preoperative neurological deficits. Degree of resection, postoperative treatments, and prognoses are closely related to the histology of intramedullary tumors.