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Nobuhito Morota

OBJECTIVE

Despite the surge in the intraoperative use of the bulbocavernosus reflex (BCR) during lumbosacral surgeries, there are as yet no widely accepted BCR warning criteria for use with intraoperative neurophysiological monitoring (IONM). The author’s aim was to find clinically acceptable warning criteria for use in IONM of the BCR.

METHODS

Records of IONM of the BCR in 164 operations in 163 patients (median age 5 months) with a conus spinal lipoma who underwent surgery between August 2002 and May 2016 were retrospectively analyzed. The outcomes of IONM of the BCR were grouped by the residual amplitude at the end of surgery: group 1, ≥ 50%; group 2, 25%–50% (including the lower bound, but not the upper); and group 3, < 25%. Cases in which the BCR was lost were separately assessed as a subgroup of group 3. The postoperative urinary complication rate was used to verify the warning criteria zones.

RESULTS

The BCR could be monitored in 149 surgeries (90.9%). There were 118 surgeries (79.2%) in group 1, 18 (12.1%) in group 2, and 13 (8.7%) in group 3. Two surgeries (11.1%) in group 2 and 6 (46.2%) in group 3 resulted in urinary complications. In the group 3 subgroup (lost BCR), all 5 surgeries resulted in urinary complications. The cutoff value of the BCR amplitude reduction was placed between groups 1 and 2 (zone 1: cutoff 50%), groups 2 and 3 (zone 2: cutoff 25%), and group 3 and its subgroup (zone 3: cutoff zero, present or lost). In zone 1, the positive predictive value (PPV) was 25.8% and the negative predictive value (NPV) was 100%. In zone 2, the PPV was 53.8% and the NPV 98.5%. In zone 3, the PPV was 100% and the NPV 97.9%. The PPV was highest in zone 3. The NPV was highest in zone 1, but its PPV was low (25.8%).

CONCLUSIONS

The “lost or remained” criterion of BCR amplitude (zone 3: cutoff zero) can be used as a predictor of postoperative urinary function. As a warning criterion, the cutoff value of the BCR amplitude reduction at 75% (zone 2) may be used. This preliminary clinical report on the warning criteria for the BCR may contribute to improving the safety of surgery for conus spinal lipoma.

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Goichiro Tamura, Nobuhito Morota, and Satoshi Ihara

OBJECTIVE

Sacrococcygeal dimples in neonates and infants are of uncertain pathological import. Previously they were believed to be rarely associated with intraspinal anomalies. Recent studies using MRI, however, revealed that 6%–7% of pediatric cases of sacrococcygeal dimples were associated with anatomical tethered spinal cord (TSC). Because the prevalence of tethered cord syndrome is still unclear, there is no consensus among pediatric neurosurgeons on the management of children with sacrococcygeal dimples. The authors performed an analysis of MRI and urodynamic studies to validate their management strategy for pediatric cases of sacrococcygeal dimples.

METHODS

A total of 103 Japanese children (49 male and 54 female, median age 4 months, range 8 days–83 months) with sacrococcygeal dimples who were referred to the Division of Pediatric Neurosurgery between 2013 and 2015 were included in this study. The lumbosacral region of all the patients was investigated using MRI. Anatomical TSC was defined as a condition in which the caudal end of the conus medullaris is lower than the inferior border of the L2–3 intervertebral disc. Patients with minor spinal anomalies (e.g., anatomical TSC, filum lipoma, thickened filum, or filar cyst) underwent further urodynamic studies to ascertain the presence of neurogenic bladder (NGB). In this study, the presence of NGB without anatomical TSC but with other minor spinal anomalies was defined as “functional TSC.” The prevalence of anatomical and functional TSC was investigated. The association of the following cutaneous findings with spinal anomalies was also assessed: 1) depth of the dimple, 2) deviation of the gluteal fold, and 3) other skin abnormalities (e.g., discoloration, angioma, or abnormal hair).

RESULTS

The children were classified into 4 groups: Group 1, patients with anatomical TSC; Group 2, patients with functional TSC; Group 3, patients without anatomical or functional TSC but with other minor spinal anomalies; and Group 4, patients with no spinal anomaly. There were 6 patients (5.8%) in Group 1, 8 patients (7.8%) in Group 2, 10 patients (9.7%) in Group 3, and 79 patients (76.7%) in Group 4. Twenty-four patients (23.3%; Groups 1, 2, and 3) showed MRI abnormalities, including filum lipoma (14 cases), filar cysts (5 cases), thickened filum (2 cases), and anatomical TSC without other spinal anomalies (3 cases). Untethering of the spinal cord was indicated for 14 patients (13.6%; Groups 1 and 2) with anatomical and functional TSCs. Preoperative NGB was found in 12 patients and improved postoperatively in 7 (58.3%). None of the associated lumbosacral skin findings predicted the presence of underlying spinal anomalies.

CONCLUSIONS

The prevalence of tethered cord syndrome among children with sacrococcygeal dimples was, for the first time, revealed to be higher than previously thought. MRI and supplemental urodynamic studies may be indicated for children with sacrococcygeal dimples to identify patients with symptomatic TSC.

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Hideki Ogiwara and Nobuhito Morota

Object

Transsphenoidal encephalocele (TSEC) is a rare developmental anomaly with herniation of neural elements including pituitary gland or optic apparatus, through a defect of sphenoid bone. To prevent obstructive dyspnea, endocrine dysfunction, or visual disturbance, surgical correction is usually performed. However, the optimal operative approach has not been determined yet. The authors present their surgical experiences with transpalatal, transcranial, or combined approach.

Methods

The authors retrospectively analyzed surgical results in patients with TSEC. The charts were reviewed for demographic data, clinical presentation, surgical therapy, and clinical outcomes.

Results

Seven consecutive patients with TSEC were surgically treated between March 2004 and February 2012. The mean age was 23.9 months (range 2–69 months). Four patients with TSEC who had a bone defect within the sphenoid bone were successfully treated using a transpalatal approach. Two patients with an extensive sphenoethmoidal bone defect that was treated either by a transpalatal or transcranial approach experienced several relapses with a prolapse of the encephalocele. One patient with an extensive sphenoethmoidal bone defect was successfully treated by combined transpalatal and transcranial approach without relapse for 3.6 years.

Conclusions

The combined transpalatal and transcranial approach is useful for TSEC with an extensive sphenoethmoidal bone defect to dissect the sac and expose the bone defect circumferentially. If dissection is not sufficient, relapse with a prolapse of the encephalocele can ensue. The transpalatal approach is sufficient for TSEC in which a bone defect is limited within the sphenoid bone.

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Hideki Ogiwara and Nobuhito Morota

Object

Endoscopic surgery is generally withheld in patients with small ventricles due to difficulties in ventricular cannulation and intraventricular manipulation. The effectiveness of flexible endoscopy for management of intraventricular brain tumors in patients with small ventricles was evaluated.

Methods

Forty-five patients who underwent endoscopic surgery with a flexible endoscope for intraventricular brain tumors were divided into small-ventricle and ventriculomegaly groups according to the frontal and occipital horn ratio (FOR). Retrospective review of these cases was performed and achievement of surgical goals and morbidity were assessed.

Results

Among the 45 patients, there were 14 with small ventricles and 31 with ventriculomegaly. In the smallventricle group, targeted tumors were located in the suprasellar region in 12 patients and in the pineal region in 2. In the ventriculomegaly group, tumors were located in the pineal region in 15 patients, in the suprasellar region in 9, in the lateral ventricle in 4, in the midbrain in 2, and in the fourth ventricle in 1. In the small-ventricle group, ventricular cannulation was successful and the surgical goals were accomplished in all patients. In ventriculomegaly group, sampling of the tumor was not diagnostic due to intraoperative hemorrhage in 1 patient. There were no significant differences in the rate of achieving the surgical goals or the morbidity between the 2 groups.

Conclusions

Endoscopic surgery using a flexible endoscope is useful for management of intraventricular brain tumors in patients with small ventricles. A flexible endoscope allows excellent maneuverability in introducing the device into the lateral ventricle and manipulating through small ventricles.

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Nobuhito Morota and Satoshi Ihara

Object

Postnatal improvement in Chiari malformation type II (CM-II) following surgical repair of myelomeningocele was evaluated.

Methods

The authors reviewed data obtained in 20 cases in which patients underwent postnatal myelomeningocele repair within the first 48 hours after birth between October 2002 and September 2006. In 14 patients (Group 1), myelomeningocele was diagnosed in utero and the infants were delivered by cesarean section at 35–39 weeks' gestation (mean 36.4). The 6 infants in Group 2 were born after full-term gestation (39–41 weeks), and their myelomeningoceles were diagnosed postnatally. In all 20 patients, the myelomeningoceles were surgically repaired postnatally. Dynamic change of the herniated cerebellar tonsils in CM-II before and after the myelomeningocele repair, associated hydrocephalus, and symptomatic CM-II were analyzed.

Results

In Group 1, the CM-II was confirmed before myelomeningocele repair in 13 cases (93%). The spinal level of the caudal end of the cerebellar tonsils ranged from C-2 to C-7. Ascent of the cerebellar tonsils was observed in 11 patients (range 1–4 spinal levels, mean 2 levels) and continued even after ventriculoperitoneal (VP) shunt placement in most patients. A VP shunt was required for the treatment of hydrocephalus in 12 patients (86%). Symptomatic CM-II developed in 8 of 13 patients (61%), 3 of whom required surgical decompression.

In Group 2, CM-II was confirmed in 5 infants (83%), with the cerebellar tonsils at a spinal level of C-2 to C-4 or C-5. Ascent of the cerebellar tonsils was observed in 4 patients (range 1–1.5 spinal levels, average 1.1 levels), and no patient had symptomatic CM-II. A VP shunt was placed in 5 patients (83%). No patient was lost to follow-up during the 18-month follow-up period. The only statistically significant difference between the 2 groups was the presence of symptomatic CM-II in Group 2 (p = 0.02).

Conclusions

Patients showed ascent of the cerebellar tonsils after postnatal myelomeningocele repair. Placement of a VP shunt helped promote the ascent. However, postnatal myelomeningocele repair in the patients in Group 1 failed to consistently prevent development of symptomatic CM-II. This limited experience suggests that postnatal repair of myelomeningocele can partially reverse the anatomical CM-II, but symptomatic CM-II cannot be prevented in some patients when the repair is performed after 36 weeks' gestation.

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Hideki Ogiwara and Nobuhito Morota

The authors report on 2 cases of subependymal giant cell astrocytoma (SEGA) with intratumoral hemorrhage causing acute hydrocephalus, necessitating emergent resection of the tumor. They review the literature and present their insights on the management of SEGA showing growth on serial imaging.

Intratumoral hemorrhage causing acute hydrocephalus can occur not only in the pediatric ages but also in the early 20s in patients with SEGA. Awareness of this sequela is considered to be important in addressing surgical timing. The authors suggest early resection of the lesions when the evidence of growth has been confirmed, to prevent possible morbidity and mortality.

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Hideki Ogiwara and Nobuhito Morota

Object

Several operative techniques have been reported to increase the cerebral blood flow (CBF) of the anterior cerebral artery (ACA) territory in patients with moyamoya disease. However, the optimal procedure has not yet been determined. This study compared the efficacy of performing bifrontal encephalogaleosynangiosis (EGS) using a craniotomy with performing EGS using bur holes.

Methods

The authors retrospectively analyzed surgical results of pediatric patients with moyamoya disease treated using encephaloduroarteriosynangiosis (EDAS) and bifrontal EGS with a craniotomy, or EGS with bur holes, for the purpose of improving the ischemia of the ACA territory. Patients' demographic data, clinical presentations, surgical therapies, and clinical outcomes were reviewed.

Results

Nine patients underwent EDAS with bifrontal EGS using a craniotomy. Three patients underwent EDAS with EGS using bur holes for 5 cerebral hemispheres (1 bur hole in 2 hemispheres, 2 bur holes in 2 hemispheres, and 3 bur holes in 1 hemisphere). The mean follow-up period was 46.8 months (range 7–96 months). Preoperative symptoms improved in all patients in each group. The collateral circulation in the ACA territory made by EGS was divided into 3 groups according to postoperative angiography results: Group A (revascularization of more than two-thirds of the ACA territory), Group B (revascularization between one-third and two-thirds), and Group C (revascularization less than one-third). Of 5 hemispheres treated with EGS using bur holes, 1 was evaluated as Group B and 4 as Group C. Of 18 hemispheres treated with bifrontal EGS using a craniotomy, 8 were evaluated as Group A, 7 as Group B, and 3 as Group C. The patients who underwent bifrontal EGS using a craniotomy showed significantly better revascularization than those who underwent EGS using bur holes (p = 0.0065). Cerebral blood flow in the ACA territory improved in 13 (92.9%) of 14 hemispheres in patients treated with bifrontal EGS. In contrast, only 3 (60%) of 5 hemispheres demonstrated an increase of CBF in patients treated with EGS using bur holes. A trend was demonstrated for better improvement with bifrontal EGS (p = 0.08).

Conclusions

Encephaloduroarteriosynangiosis with bifrontal EGS using a craniotomy demonstrated better revascularization and improvement of CBF in the ACA territory than with EGS using bur holes. Bifrontal EGS using a craniotomy is considered to be a simple, safe, and effective surgical procedure for improvement of the ischemia of the ACA territory in pediatric patients with moyamoya disease.

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Nobuhito Morota, Satoshi Ihara, and Hideki Ogiwara

OBJECTIVE

Spinal lipomas are generally thought to occur as a result of failed primary neurulation. However, some clinical features cannot be explained by this theory. The authors propose a novel classification of spinal lipomas based on embryonic changes seen during primary and secondary neurulation.

METHODS

A total of 677 patients with occult spinal dysraphism underwent 699 surgeries between August 2002 and May 2015 at the National Center for Child Health and Development and Tokyo Metropolitan Children's Medical Center. This group of patients had 378 spinal lipomas, including 119 conus spinal lipomas, 27 lipomyelomeningoceles, and 232 filum lipomas, which the authors classified into 4 types based on neural tube formation during embryonic development. Type 1 is defined as pure primary neurulation failure; Type 2 ranges from primary to secondary neurulation failure; Type 3 consists of secondary neurulation failure (early phase); and Type 4 is defined as secondary neurulation failure (late phase). The authors also review embryogenesis in secondary neurulation and analyze the clinical utility of the new classification.

RESULTS

There were 55 Type 1 spinal lipomas, 29 Type 2, 62 Type 3, and 232 Type 4. All filum lipomas fell into the Type 4 spinal lipoma category. Association with anorectal and/or sacral anomalies was seen in none of the Type 1 cases, 15 (52%) of Type 2, 35 (56%) of Type 3, and 31 (13%) of Type 4. Urogenital anomalies were observed in none of the Type 1 or Type 2 cases, 1 (2%) of Type 3, and 28 (12%) of Type 4. Anomaly syndromes were present in none of the Type 1 cases, 6 (21%) of Type 2, 3 (5%) of Type 3, and 16 (7%) of Type 4. Associated anomalies or anomaly syndromes were clearly observed only for Type 2–4 spinal lipomas encompassing failed secondary neurulation. Radical resection was feasible for Type 1 spinal lipomas.

CONCLUSIONS

Secondary neurulation of the spinal cord gives rise to the conus medullaris and filum terminale, which are often involved in spinal lipomas. Formation of spinal lipomas seems to be a continuous process overlapping primary and secondary neurulation in some cases. Association with other anomalies was higher in Type 2–4 spinal lipomas, which included failed secondary neurulation, than in Type 1 lipomas, with failed primary neurulation. On the other hand, radical resection was indicated for Type 1, but not for Type 2, spinal lipomas. The new classification of spinal lipomas based on embryonic stage has the potential for clinical use and agrees well with both clinical and surgical findings. The classification proposed here is still preliminary. Further studies and verification are necessary to establish its clinical utility.

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Hideki Ogiwara, Kodai Uematsu, and Nobuhito Morota

Object

Endoscopic choroid plexus coagulation (CPC) with or without endoscopic third ventriculostomy (ETV) has been shown to be effective for selected patients with hydrocephalus. However, whether the effect of the coagulation is temporary and the choroid plexus regenerates or can be obliterated has remained largely unknown. The authors evaluate the effectiveness of CPC and report 3 cases of obliteration demonstrated by direct endoscopic observation.

Methods

The authors retrospectively analyzed the surgical results of patients with hydrocephalus primarily treated by CPC with or without ETV. Charts were reviewed for demographic data, clinical presentations, surgical therapies, and clinical outcomes.

Results

Eighteen patients with hydrocephalus were surgically treated using endoscopic CPC between July 2002 and July 2012. In 12 patients, ETV was concurrently performed. The etiology of hydrocephalus was posthemorrhagic in 5 patients, myelomeningocele in 3, postmeningitis in 2, congenital aqueductal stenosis in 1, hydranencephaly in 1, porencephaly in 1, and idiopathic in 5.

The mean age at surgery was 8 months (range 0.3–24 months). The mean follow-up was 64 months. In 9 cases (50%), control of hydrocephalus was successful and the patients did not require further surgeries. In 9 patients (50%), treatment failed. Of these, 3 patients underwent repeat ETV 2, 3, and 38 months after the initial surgery. Endoscopic observation of the previous coagulation site revealed no regeneration of the choroid plexus in 2 patients, who underwent repeat ETV 2 and 3 months after CPC. In 1 patient who underwent repeat ETV 38 months after CPC, no regeneration of the choroid plexus, except for that in the proximity of the foramen of Monro, was observed.

Conclusions

Endoscopic CPC with or without ETV can be a safe and effective treatment alternative to shunt placement in infantile hydrocephalus. Obliteration of the choroid plexus can persist in the relatively long term following CPC, which may contribute to the long-term control of hydrocephalus in successful cases.

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Nobuhito Morota, Satoshi Ihara, Hideki Ogiwara, and Goichiro Tamura

OBJECTIVE

Chondrodysplasia punctata (CDP), a rare skeletal dysplasia, can lead to cervical spine instability and deformity. However, an optimal neurosurgical intervention has yet to be established. Thus, a retrospective study was conducted to assess the efficacy of various surgical interventions for children with CDP.

METHODS

The authors retrospectively reviewed 9 cases of CDP in which cervical decompression with or without posterior fusion was performed between April 2007 and May 2016. Patient demographics, preoperative clinical conditions, radiographic findings, surgical procedures, and the postoperative course were analyzed in detail.

RESULTS

A total of 12 operations were carried out in 9 patients (8 male, 1 female) during the study period. The patients’ ages at the initial surgery ranged from 2 months to 2 years. Seven of the children had CDPX1, 1 had CDPX2, and 1 had tibia-metacarpal type CDP (CDP-TM). The lesion occurred at the craniovertebral junction (CVJ) in 7 cases and involved a subaxial deformity in 2 cases. The initial surgery was C-1 laminectomy with occipitocervical fusion (OCF) followed by halo external fixation in 5 cases, OCF alone in 1 case, and C-1 laminectomy alone in 3 cases. Three children required additional surgery. In one of these cases, a staged operation was required because the patient’s head was too small to attach a halo ring at the time of the initial procedure (C-1 laminectomy). In another case, OCF was performed 11 months after C-1 laminectomy because of intramedullary signal change on serial MRI, although the child remained asymptomatic. In the third case, additional posterior fusion was performed 17 months after an initial laminectomy and OCF due to newly developed cervical dislocation caudal to the original fusion. This last patient required a third operation 9 months after the second because of deep wound infection. Surgery improved the motor function of all 7 children with CDPX1, but 3 children who had already suffered respiratory failure preoperatively required continued respiratory support. At the time of this report, 7 of the 9 children were alive and in stable condition. One child died due to restrictive respiratory insufficiency, and another died in an accident unrelated to CDP.

CONCLUSIONS

Surgical decompression with or without fusion for CVJ and subaxial cervical lesions in infants and toddlers with CDP generally saves lives and increases the likelihood of motor function recovery. However, in this case series the patients’ preoperative condition had a strong effect on postoperative respiratory function. The surgery was not straightforward, and a second operation was required in some cases. Nevertheless, the findings indicate that early surgical intervention for CDP with cervical involvement is feasible, suggesting that the role of neurosurgery should be reevaluated.