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Jung Won Choi, Seung-Ki Kim, Kyu-Chang Wang, Ji Yeoun Lee, Jung-Eun Cheon and Ji Hoon Phi

Object

Ventriculoperitoneal (VP) shunt surgery is the most common treatment for hydrocephalus. In certain situations, uncommon complications can occur after shunting procedures. The authors undertook this study to analyze the clinical characteristics of pediatric patients who developed multifocal intraparenchymal hemorrhages (MIPHs) as a complication of shunt surgery. The authors also analyzed the risk factors for MIPH in a large cohort of patients with hydrocephalus.

Methods

This study included all pediatric patients (age < 18 years) who underwent VP shunt surgery at the authors' institution between January 2001 and December 2012. During this period, 507 VP shunt operations were performed in 330 patients. Four of these patients were subsequently diagnosed as having MIPH. The authors analyzed the clinical characteristics of these patients in comparison with those of the entire group of shunt-treated patients.

Results

The incidence of MIPH was 1.2% (4 of 330 cases) for all pediatric patients who underwent VP shunt placement but 2.9% (4 of 140 cases) for infants less than 1 year old. When the analysis was limited to patients whose corrected age was less than 3 months, the incidence was 5.3% (4 of 76 cases). Of the 4 patients with MIPH, 2 were male and 2 were female. Their median age at surgery was 54 days (range 25–127 days), and in all 4 cases, the patients' corrected age was less than 1 month. Three patients were preterm infants, whereas one patient was full-term. None of these patients had a prior history of intracranial surgery (including CSF diversion procedures). All showed severe hydrocephalus during the preoperative period. Their clinical courses as patients with MIPH were comparatively favorable, despite the radiological findings.

Conclusions

MIPH is a rare but not negligible complication of VP shunt surgery. This complication might be a unique phenomenon in infants, especially young, preterm infants with severe hydrocephalus. Moreover, the absence of previous intracranial procedures might be one of the risk factors for this complication. The rapid alteration of brain conditions in the setting of immaturity might cause MIPH. To prevent this complication, the authors recommend that pressure settings of programmable valves should be gradually adapted to the target pressure.

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Hyunho Choi, Ji Yeoun Lee, Ji Hoon Phi, Seung-Ki Kim, Byung-Kyu Cho and Kyu-Chang Wang

Object

Postoperative epidural hematoma (EDH), a blood collection between the inserted galeal flap and the overlying skull flap (epigaleal flap hematoma), is a frustrating complication of the surgical treatment of moyamoya disease (MMD) in pediatric patients. The symptoms of postoperative EDH are often similar to those of postoperative cerebral ischemia, and may cause confusion during clinical decision making. The authors designed this study to evaluate the incidence, clinical presentation, risk factors, and treatment outcomes of postoperative EDH in pediatric patients with MMD.

Methods

A retrospective review of 148 pediatric patients with 250 craniotomies who underwent indirect bypass revascularization surgery between January 2002 and December 2006 was performed. This group consisted of 60 male and 88 female patients, and the mean age at surgery was 7.5 years (range 1–18 years).

Results

Of the 250 craniotomies, postoperative EDH was detected in 32 cases. In 12 cases of EDH, surgical treatment was necessary (4.8% of 250 craniotomies). During the same period, 743 non-MMD craniotomy operations were performed. In 6 of these 743 operations, patients developed postoperative EDH that required surgical treatment, significantly less than the percentage of EDH requiring postoperative treatment in patients with MMD (0.8%, p < 0.001). The average interval between craniotomy surgery and the detection of EDH was 1.8 days (range 0–5 days) in patients with MMD and 0.5 days (range 0–2 days; p = 0.018) in the non-MMD craniotomy group.

Postoperative EDH was observed in significantly fewer cases (17 of 191) when a subcutaneous drain (SCD) was inserted over the bone flap than in cases in which an SCD was not inserted (14 of 55; p = 0.001). The mean international normalized ratio of prothrombin time in the immediate postoperative blood test was 1.27 ± 0.17 in the EDH group and 1.20 ± 0.11 in the non-EDH group (values are expressed as the mean ± SD; p = 0.008). Central galeal flap tenting suture and immediate postoperative platelet count were not related to the incidence of postoperative EDH following pediatric MMD surgery.

Conclusions

Postoperative EDH is more likely to be found following craniotomy in patients with MMD than in those without MMD, and may occur in a delayed fashion. Insertion of an SCD and immediate correction of an abnormal value for international normalized ratio of prothrombin time can decrease the incidence of postoperative EDH following surgery for pediatric MMD.

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Eun Ji Kim, Kyu-Chang Wang, Ji Yeoun Lee, Ji Hoon Phi, Sung-Hye Park, Jung-Eun Cheon, Young Eun Jang and Seung-Ki Kim

Infantile myofibromatosis, a rare mesenchymal disorder that develops in early childhood, is classified by the number of lesions that occur: solitary or multicentric. Involvement of the CNS is unusual in either type. Infantile myofibromatosis in the spine is exceptional, and most published cases represent a secondary invasion. Here, the authors report on an 8-month-old girl presenting with weakness below the ankle and an intraspinal mass extending from T-6 to the conus. The patient underwent only partial surgical removal of the lesion, and the pathology was confirmed as infantile myofibromatosis. After the operation, weakness in the lower extremities gradually improved; however, she could not walk at the time of the final follow-up. On follow-up MRI performed 19 months after the operation, the residual lesion remained unchanged with decreased enhancement.

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Ji Yeoun Lee, Young Ah Lee, Hae Woon Jung, Sangjoon Chong, Ji Hoon Phi, Seung-Ki Kim, Choong-Ho Shin and Kyu-Chang Wang

OBJECTIVE

Due to their distinct location, suprasellar arachnoid cysts are known to cause a wide variety of problems, such as hydrocephalus, endocrine symptoms, and visual abnormalities. The long-term outcome of these cysts has not been elucidated. To find out the long-term outcome of suprasellar arachnoid cysts, a retrospective review of the patients was performed. The neurological and endocrine symptoms were thoroughly reviewed.

METHODS

Forty-five patients with suprasellar arachnoid cysts, with an average follow-up duration of 9.7 years, were enrolled in the study. A comprehensive review was performed of the results of follow-up regarding not only neurological symptoms but also endocrine status. The outcomes of 8 patients who did not undergo operations and were asymptomatic or had symptoms unrelated to the cyst were included in the series.

RESULTS

Surgery was most effective for the symptoms related to hydrocephalus (improvement in 32 of 32), but endocrine symptoms persisted after surgery (4 of 4) and required further medical management. More surprisingly, a fairly large number of patients (14 of 40; 1 was excluded because no pre- or postoperative endocrine evaluation was available) who had not shown endocrine symptoms at the time of the initial diagnosis and treatment later developed endocrine abnormalities such as precocious puberty and growth hormone deficiency. The patients with endocrine symptoms detected during the follow-up included those in both the operated (n = 12 of 32) and nonoperated (n = 2 of 8) groups who had been stable during follow-up since the initial diagnosis.

CONCLUSIONS

This study implies that patients with suprasellar arachnoid cysts can develop late endocrine problems during follow-up, even if other symptoms related to the cyst have been successfully treated. Hence, patients with these cysts need long-term follow-up for not only neurological symptoms but also endocrine abnormalities.

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Jun Hyong Ahn, Ji Hoon Phi, Hyun-Seung Kang, Kyu-Chang Wang, Byung-Kyu Cho, Ji Yeoun Lee, Gi Beom Kim and Seung-Ki Kim

This 13-month-old boy, in whom Kawasaki disease had been diagnosed at the age of 6 months, presented with subarachnoid hemorrhage caused by the rupture of a middle cerebral artery aneurysm. The authors performed an emergency craniectomy and clip occlusion of the aneurysm, which was found to be partially thrombosed. The patient was discharged 4 weeks postoperatively without apparent neurological deficit.

Intracranial saccular aneurysms in the pediatric population are rare, and are occasionally associated with various systemic disorders. Kawasaki disease is a systemic vasculopathy of unknown origin, but cerebral arteries are usually spared from the disease process. This is the second case report of a ruptured cerebral aneurysm in a patient with Kawasaki disease, providing a novel clinical feature that the authors call Kawasaki syndrome.

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Byung Chan Lim, Jong Hee Chae, Seung-Ki Kim, Sung-Hye Park, Kyu-Chang Wang, Ji Yeoun Lee and Ji Hoon Phi

Brainstem glioma is a highly devastating disease, and any mass-like lesion in the brainstem can raise suspicion of this diagnosis. However, other inflammatory, demyelinating, or degenerative diseases can mimic brainstem glioma in clinical presentation and imaging features. Therefore, diagnosis based solely on imaging is often insufficient for brainstem lesions and may lead to incorrect diagnosis and treatment.

This case report is the first description of central nervous system aquaporin-4 (AQP4) autoimmunity confined mainly to the brainstem. It demonstrates the wide spectrum of neuroinflammatory diseases in children and highlights the utility of surgical biopsy for suspicious brainstem lesions with atypical imaging features for glioma.

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Ji Yeoun Lee, Bo Sung Kim, Ji Hoon Phi, Hyoung Jin Kang, Sung-Hye Park, Kyu-Chang Wang, Il Han Kim, Byung-Kyu Cho and Seung-Ki Kim

Primary sarcomas of the CNS are rare and are sometimes associated with chronic subdural effusion (SDE). Approximately 10 patients with such presentations have been reported. The authors report the case of a 5-year-old boy with multiple subdural masses and SDE. An SDE had been diagnosed when the patient was 2 months old, and he received a subduroperitoneal shunt when he was 5 months old. Since then, he had been clinically stable and well for 5 years. When he presented with acute headache, nausea, and vomiting, a newly developed tumor was found. Near-total resection of the tumor was performed, and the mass was diagnosed as an embryonal-type rhabdomyosarcoma. The child was given radiation therapy and 13 cycles of chemotherapy and is still free of disease 13 months after surgery.

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Jung Won Choi, Ji Yeoun Lee, Ji Hoon Phi, Kyu-Chang Wang, Hyun-Tai Chung, Sun Ha Paek, Dong Gyu Kim, Sung-Hye Park and Seung-Ki Kim

Object

Neurofibromatosis Type 2 (NF2) is an autosomal-dominant inherited disease, characterized by multiple neoplasia syndromes, including meningioma, schwannoma, glioma, and ependymoma. In this report, the authors present their clinical experience with pediatric NF2 patients. In particular, they focused on the clinical course of vestibular schwannoma (VS), including the natural growth rate, tumor control, and functional hearing outcomes.

Methods

From May 1988 to June 2012, the authors recruited patients who were younger than 18 years and fulfilled the Manchester criteria. In total, 25 patients were enrolled in this study. The authors analyzed the clinical course of these patients. In addition, they measured the natural growth rate of VS before any treatment in these children with NF2. Then, they evaluated the tumor control rate and functional hearing outcomes after the treatment of VS.

Results

The mean age at the onset of NF2-related symptoms was 9.9 ± 4.5 years (mean ± SD, range 1–17 years). The mean age at the diagnosis of NF2 was 12.9 ± 2.9 years (range 5–17 years). The mean follow-up period was 89.3 months (range 12–311 months). As initial manifestations, nonvestibular symptoms were frequently observed in pediatric patients with NF2. The mean natural growth rate of VS was 0.33 ± 0.41 cm3/year (range 0–1.35 cm3/year). The tumor control rate of VS was 35.3% at 3 years after Gamma Knife surgery (GKS). The actuarial rate of useful hearing preservation was 67% in the 1st year and 53% in the 5th year after GKS.

Conclusions

Clinical manifestations in children with NF2 were highly variable, compared with their adult counterparts. The natural growth rate of VS in children is slow, and this oncological feature may explain the diverse clinical manifestations besides vestibular symptoms in children with NF2. The treatment outcome of GKS for VS in children with NF2 was not favorable compared with previous reports of affected adults.

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Hyeoncheol Hwang, Kyu-Chang Wang, Moon Suk Bang, Hyung-Ik Shin, Seung-Ki Kim, Ji Hoon Phi, Ji Yeoun Lee, Jinwoo Choi, Seungwoo Cha and Keewon Kim

OBJECTIVE

The aim of this study was to establish optimal electric stimulation parameters for intraoperatively monitoring the bulbocavernosus reflexes (BCRs) in infants.

METHODS

The authors retrospectively reviewed the medical records of all infants (age < 24 months) who had undergone an untethering operation for tethered cord syndrome between May 2013 and February 2014 at a single institution and whose baseline BCR had been elicited during surgery. Using different combinations of stimulation parameters—number of stimulation pulses: 4 or 8 pulses, interpulse interval: 1, 2, or 5 msec, and polarity of stimulation: biphasic or monophasic—the authors compared the relative mean amplitude of 10 BCR responses (rmaBCRs) to each combination of parameters.

RESULTS

The rmaBCRs were larger with the 8-pulse stimulations than with the 4-pulse stimulations (p < 0.0001). There was a tendency, though not statistically significant, for larger rmaBCRs to be obtained with the longer interpulse interval in the 8-pulse stimulation (p = 0.1289). The biphasic stimulation produced larger rmaBCRs than the monophasic stimulation (p = 0.0005).

CONCLUSIONS

Biphasic 8-pulse stimulations with 5-msec or 2-msec intervals yield the largest BCR responses. Considering that an 8-pulse stimulation with 5-msec intervals may overlap the onset of the BCR, a biphasic 8-pulse stimulation with 2-msec intervals is recommended as the optimal stimulation paradigm to monitor intraoperative BCRs in infants.

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Ji Yeoun Lee, Sangjoon Chong, Young Hun Choi, Ji Hoon Phi, Jung-Eun Cheon, Seung-Ki Kim, Sung Hye Park, In-One Kim and Kyu-Chang Wang

OBJECTIVE

Since the entity limited dorsal myeloschisis (LDM) was proposed, numerous confusing clinical cases have been renamed according to the embryopathogenesis. However, clinical application of this label appears to require some clarification with regard to pathology. There have been cases in which all criteria for the diagnosis of LDM were met except for the presence of a neural component in the stalk, an entity the authors call “probable” LDM. The present study was performed to meticulously review these cases and suggest that a modified surgical strategy using limited laminectomy is sufficient to achieve the surgical goal of untethering.

METHODS

The authors retrospectively reviewed the imaging findings, operative notes, and pathology reports of spinal dysraphism patients with subcutaneous stalk lesions who had presented to their institution between 2010 and 2014.

RESULTS

Among 33 patients with LDM, 13 had the typical nonsaccular lesions with simple subcutaneous stalks connecting the skin opening to the spinal cord. Four cases had “true” LDM meeting all criteria for diagnosis, including pathological confirmation of CNS tissue by immunohistochemical staining with glial fibrillary acidic protein. There were also 9 cases in which all clinical, imaging, and surgical findings were compatible with LDM, but the “neural” component in the resected stalk was not confirmed. For all the cases, limited exposure of the stalk was done and satisfactory untethering was achieved.

CONCLUSIONS

One can speculate based on the initial error of embryogenesis that if the entire stalk were traced to the point of insertion on the cord, the neural component would be proven. However, this would require an extended level of laminectomy/laminotomy, which may be unnecessary, at least with regard to the completeness of untethering. Therefore, the authors propose that for some selected cases of LDM, a minimal extent of laminectomy may suffice for untethering, although it may be insufficient for diagnosing a true LDM.