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Atsushi Okano and Hideki Ogiwara

OBJECTIVE

Shunt surgery is the most common treatment for hydrocephalus, but it is associated with several long-term complications. Endoscopic choroid plexus coagulation (CPC) and endoscopic third ventriculostomy (ETV) are alternative surgeries that may avoid the need for shunt surgery. Although the short-term efficacy and safety of CPC have been reported in previous studies, long-term outcome, including not only avoiding shunt placement but also intellectual development, remains to be elucidated. The purpose of the present study was to investigate the long-term outcome of CPC.

METHODS

The study population comprised patients who had infantile hydrocephalus treated by endoscopic CPC before the age of 24 months and who were followed until at least 5 years of age. Retrospective review was performed using the medical charts. The authors assessed educational status and the full-scale intelligence quotient (FSIQ) using the Wechsler Intelligence Scale for Children (WISC) IV as the means to evaluate the intellectual development.

RESULTS

Fourteen patients with infantile hydrocephalus underwent CPC with or without ETV as a primary surgery. There were no intraoperative complications. In 7 patients (50%), hydrocephalus was successfully controlled without shunt placement. Six patients (43%) eventually required shunt placement. In one patient hydrocephalus was controlled by additional ETV. In the shunt-independent group, 4 patients went to age-appropriate school or achieved age-appropriate development according to intelligence quotient (IQ), 1 patient went to specialized school, and 2 patients had disabilities. In the shunt-dependent group, 4 patients went to an age-appropriate school or achieved age-appropriate development by IQ, 1 patient went to specialized school, and 1 patient had disabilities. The mean FSIQ score in 3 patients without shunts was 90 (range 89–91) and the mean FSIQ score in 4 patients with shunts was 80 (range 48–107). There was no significant difference in the rate of normal development between the shunt-independent group and the shunt-dependent group (p = 0.72).

CONCLUSIONS

The CPC with or without ETV can be a safe and effective treatment in children with infantile hydrocephalus. Long-term control of hydrocephalus and normal intellectual development can be achieved in successful cases. Further prospective studies should be required to elucidate appropriate indications.

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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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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

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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John R. W. Kestle

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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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Ako Matsuhashi, Kenichi Usami, Eitaro Ishisaka, and Hideki Ogiwara

OBJECTIVE

Although the association between Chiari malformation (CM) and craniosynostosis is well recognized, management remains controversial. There are differences in the clinical course of CM in syndromic craniosynostosis (SC) patients and nonsyndromic craniosynostosis (NSC) patients. Still unclear is whether cranial expansion surgery, foramen magnum decompression (FMD), or both should be conducted and when the appropriate timing of surgery should be. Here, the authors retrospectively reviewed the clinical data at their institution to investigate the optimal management of CM associated with SC and NSC.

METHODS

The authors retrospectively analyzed the clinical records of 163 children with craniosynostosis who underwent surgical treatment at the National Center for Child Health and Development between April 2002 and May 2018.

RESULTS

Twelve (10%) of 119 children with NSC and 14 (31.8%) of 44 children with SC were radiologically diagnosed with CM. The median age at which CM was radiologically diagnosed was 7 months. Of the 12 NSC patients with CM, 1 (8%) was symptomatic with gait disturbance. This patient underwent FMD following expansion surgery. Of the 14 SC patients with CM, 8 (57.1%) were symptomatic with central sleep apnea. For SC patients with symptomatic CM, cranial expansion surgery alone was conducted in 2 cases, FMD was conducted after cranial expansion in 4 cases, and FMD was conducted first and additional expansion surgery was then conducted in 2 cases. In all cases, symptoms of CM improved after completing this series of surgeries. Regarding patients with asymptomatic CM, cranial expansion surgery alone was performed, and none of the patients showed symptoms of CM thereafter in both SC and NSC cases.

CONCLUSIONS

Multiple surgeries tend to be necessary for symptomatic CM in both SC and NSC, while no patient with asymptomatic CM became symptomatic after cranial expansion surgery.

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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.