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  • Author or Editor: Chang Kyu Park x
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Eun Jung Koh, Ji Hoon Phi, Sung-Hye Park, In-One Kim, Jung-Eun Cheon, Kyu-Chang Wang, Byung-Kyu Cho and Seung-Ki Kim

This 14-year-old boy presented with left hemiparesis, gait disturbance, and multiple cranial nerve palsies. Magnetic resonance imaging revealed a multicystic mass with hemorrhagic fluid–fluid levels in the right midbrain, suggesting the presence of a cavernous malformation. Diffusion tensor imaging showed the mass to be close to the right corticospinal tract and ipsilateral medial lemniscus. Subtotal removal of the mass was performed via a right subtemporal approach. The histopathological diagnosis was of a mixed germ cell tumor (GCT) comprising mature teratoma and germinoma cells with syncytiotrophoblastic giant cells. The patient underwent postoperative chemotherapy and radiotherapy, and no tumor progression was found during 1 year of follow-up.

Intracranial GCTs arise mainly in the pineal and the suprasellar area. Germ cell tumors in the brainstem are rare, with only 12 reported cases. Among these, 4 were in the midbrain and histologically were pure germinomas. To the authors' knowledge, this is the first reported case of a mixed GCT in the midbrain combining mature teratoma and germinoma cells.

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Seong-Cheol Park, Seung-Ki Kim, Byung-Kyu Cho, Hyun Jib Kim, Jeong Eun Kim, Ji Hoon Phi, In-One Kim and Kyu-Chang Wang

Object

Sinus pericranii (SP) is a rare venous varix in an extracranial location connected to the intracranial venous system. The aim of this retrospective study was to report on 16 pediatric cases of SP with consideration of the preoperative evaluation of surgical risk.

Methods

The study population consisted of 10 patients who had undergone surgery for SP and 6 patients with concomitant craniosynostosis and SP. The mean age of the patients at presentation was 3.7 years. To identify characteristics of SP with high operative risk, 8 cases in this report and 11 previously reported cases of SP with sufficient information were categorized on the basis of the number and size of SP, the number and size of transcranial channels, the venous drainage type, and the amount of blood loss. Hemorrhage amounts were classified into 3 grades based on the description of intraoperative blood loss.

Results

Sinus pericranii not associated with craniosynostosis were resected without any postoperative morbidity. Sinus pericranii associated with craniosynostosis were preserved. After craniofacial reconstruction, 2 cases of SP with craniosynostosis regressed, completely in one patient and partially in another. These 2 patients with SP were confirmed to have compromised intracranial sinus before craniofacial reconstruction. Among a total of 19 patients, multiplicity or size (> 6 cm) of SP (p = 0.036) and multiplicity (> 3) or size (> 3 mm) of transcranial channels (p = 0.004) was associated with more severe hemorrhage grade. Sinus pericranii with peripheral venous drainage (drainer type) was not associated with hemorrhage grade after classification into 3 grades (p = 0.192). However, all 3 cases of SP with massive Grade 3 hemorrhage were the drainer type. Hemorrhage grade was correlated with the number of risk factors for SP (r = 0.793, p < 0.001).

Conclusions

Three risk factors of SP and the presence of compromised intracranial sinus are markers for highrisk SP. “Squeezed-out sinus syndrome” is suggested as a concept for SP associated with compromised intracranial sinus, mainly caused by craniosynostosis. Sinus pericranii in squeezed-out sinus syndrome probably serves as a crucial alternative to venous drainage of the brain with intracranial venous compromise. Conservative treatment for such patients with SP is recommended.

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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.

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Ji Hoon Phi, Byung-Kyu Cho, Kyu-Chang Wang, Ji Yeoun Lee, Yong Seung Hwang, Ki Joong Kim, Jong-Hee Chae, In-One Kim, Sung-Hye Park and Seung-Ki Kim

Object

The long-term surgical outcome of pediatric patients with epilepsy accompanied by focal cortical dysplasia (FCD) is not clear. The authors report on the long-term surgical outcomes of children with FCD, based on longitudinal analyses.

Methods

The authors retrospectively analyzed the records of 41 children who underwent epilepsy surgery for pathologically proven FCD. Twenty of these patients were male and 21 were female. The median age at surgery was 9 years (range 1–17 years).

Results

The actuarial seizure-free rates were 49, 44, and 33% in the 1st, 2nd, and 5th years after surgery, respectively. There was no seizure recurrence after 3 years. Three patients with initial failure of seizure control experienced late remission of seizures (the so-called running-down phenomenon). Eventually, 19 patients (46%) were seizure free at their last follow-up visit. Absence of a lesion on MR imaging and incomplete resection were significantly associated with seizure-control failure. Concordance of presurgical evaluation data was a marginally significant variable for seizure control in patients with lesional epilepsy. Three patients with seizure-control failure became seizure free as a result of the running-down phenomenon. The actuarial rate of antiepileptic drug discontinuation was 91% in the 5th year in the seizure-free patients.

Conclusions

The seizure-free rate after surgery in children with FCD was 49% in the 1st year; however, it declined thereafter. The running-down phenomenon could be an important mechanism of seizure alleviation for patients with FCD during long-term follow-up. Because a complete resection of FCD has a strong prognostic implication for seizure control, a better method to define the extent of FCD is required to assist with resection, especially in nonlesional epilepsy.

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Ji Hoon Phi, Seung-Ki Kim, Joongyub Lee, Chul-Kee Park, Il Han Kim, Hyo Seop Ahn, Hee Young Shin, In-One Kim, Hee-Won Jung, Dong Gyu Kim, Sun Ha Paek and Kyu-Chang Wang

Object

Intracranial germ cell tumors (GCTs) frequently present with bifocal lesions in both the suprasellar and pineal areas. The pathogenesis of these bifocal GCTs has been the subject of controversy. Bifocal GCTs may be caused by synchronous tumors or by metastatic spread of tumor cells from one site to the other. The prognosis associated with bifocal GCTs has also been a cause of concern.

Methods

The authors constructed a single-institution patient cohort comprising 181 patients with intracranial GCTs. The clinical characteristics of bifocal GCTs were compared with those of suprasellar and pineal GCTs.

Results

Bifocal GCTs were observed in 23 patients (12.8%). Eighteen patients presented with bifocal GCTs that were diagnosed as germinomas, but 5 patients exhibited mixed GCTs. Analyses of age distributions and comparisons of tumor sizes were compatible with a model of a metastatic origin of bifocal GCTs. Eleven patients (47.8%) presenting with bifocal GCTs exhibited tumor seeding at presentation. Tumor seeding was significantly associated with bifocal lesions (p < 0.001). Patients with bifocal germinomas showed significantly shorter event-free survival and overall survival than did those presenting with germinomas from a single site of origin.

Conclusions

Bifocal GCTs are not restricted to germinomas, as had been previously reported, but do include mixed GCTs. The authors hypothesize that bifocal GCTs may result from the metastatic spread of suprasellar or pineal GCTs. The bifocal presentation of germinomas may be a poor prognostic sign and should alert clinicians to the possibility of a disseminated disease.