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June Ho Lee, Chae-Yong Kim, Dong Gyu Kim and Hee-Won Jung

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Kihwan Hwang, Yong Hwy Kim, Jung Hee Kim, Jung Hyun Lee, Hee Kyung Yang, Jeong-Min Hwang, Chae-Yong Kim and Jung Ho Han

OBJECTIVE

The authors investigated the natural history of asymptomatic nonfunctioning pituitary adenomas (NFPAs) with optic nerve compression.

METHODS

This study retrospectively analyzed the natural history of asymptomatic NFPAs with documented optic nerve compression on MRI diagnosed between 2000 and 2016 from 2 institutions. The patients were followed up with regular endocrinological, ophthalmological, and radiological evaluations, and the endpoint was new endocrinopathy or neurological deficits.

RESULTS

The study comprised 81 patients. The median age at diagnosis was 58.0 years and the follow-up duration was 60.0 months. As the denominator of overall pituitary patients, 2604 patients were treated with surgery after diagnosis at the 2 institutions during the same period. The mean initial and last measured values for tumor diameter were 23.7 ± 8.9 mm and 26.2 ± 11.4 mm, respectively (mean ± SD). Tumor growth was observed in 51 (63.0%) patients; however, visual deterioration was observed in 14 (17.3%) patients. Ten (12.3%) patients experienced endocrine deterioration. Fourteen (17.3%) patients underwent surgery for either visual deterioration (in 12 patients) or endocrine dysfunction (in 2 patients). After surgery, all patients experienced improvements in visual or hormonal function. The actuarial rates of treatment-free survival at 2, 3, and 5 years were 96.1%, 93.2%, and 85.6%, respectively. In the multivariate analysis, initial cavernous sinus invasion (HR 4.985, 95% CI 1.597–15.56; p = 0.006) was the only independent risk factor for eventual treatment.

CONCLUSIONS

The neuroendocrinological deteriorations were not frequent and could be recovered by surgery with early detection on regular follow-up in asymptomatic NFPAs with documented optic nerve compression on MRI. Therefore, conservative management could be an acceptable strategy for these tumors. Careful follow-up is required for tumors with cavernous sinus invasion.

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Matthew C. Tate, Chae-Yong Kim, Edward F. Chang, Mei-Yin Polley and Mitchel S. Berger

Object

The morbidity associated with resection of tumors in the cingulate gyrus (CG) is not well established. The goal of the present study is to define the short- and long-term morbidity profile associated with resection of gliomas within this region.

Methods

Ninety consecutive patients with gliomas involving the CG were analyzed. Resections were classified by zones corresponding to functionally defined regions of the CG as follows: Zone I (perigenual, anterior), Zone II (midcingulate), Zone III (posterior), and Zone IV (retrosplenial). Basic demographic, imaging, operative details, and pre- and postoperative neurological examinations were recorded for each patient. Patients in whom neurological morbidity was documented during their initial postoperative examination who did not completely improve by the 6-month follow-up examination were considered to have a permanent deficit. For each patient with surgery-related morbidity, postoperative MR imaging and operative notes were reviewed, and the cortical regions incorporated in the surgical trajectory were recorded. The analysis was carried out for tumors confined to the CG (> 90% of tumor contained within the CG) as well as those involving the CG but extending into adjacent cortical structures.

Results

Analysis of the entire patient cohort demonstrated that 29% of patients experienced a new or worsened neurological deficit immediately after surgery. The most common deficits were supplementary motor area (SMA) syndrome (20%), weakness (6%), and sensory changes (2%). All patients with an SMA syndrome in our series had intentional resection of SMA as part of the surgical approach. Patients with resections including Zone II or III had a higher rate of total morbidity and SMA syndrome than patients with Zone I resections (p < 0.05). Only 4% of patients had a persistent neurological deficit at 6 months postoperatively. A similar morbidity profile was observed in the subset analysis of patients with tumors confined to the CG, with no additional morbidity related to known cingulate-specific functions.

Conclusions

Resection of gliomas involving the CG can be performed with minimal, predictable long-term morbidity (< 5%). Surgical morbidity is primarily a function of surgical trajectory rather than the particular cingulate region resected.

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Young-Hoon Kim, Chi Heon Kim, June Sic Kim, Sang Kun Lee, Jung Ho Han, Chae-Yong Kim and Chun Kee Chung

Object

Supplementary motor area (SMA) resection often induces postoperative contralateral hemiparesis or speech disturbance. This study was performed to assess the neurological impairments that often follow SMA resection and to assess the risk factors associated with these postoperative deficits.

Methods

The records for patients who had undergone SMA resection for pharmacologically intractable epilepsy between 1994 and 2010 were gleaned from an epilepsy surgery database and retrospectively reviewed in this study.

Results

Forty-three patients with pharmacologically intractable epilepsy underwent SMA resection with intraoperative cortical stimulation and mapping while under awake anesthesia. The mean patient age was 31.7 years (range 15–63 years), and the mean duration and frequency of seizures were 10.4 years (range 0.1–30 years) and 14.6 per month (range 0.1–150 per month), respectively. Pathological examination of the brain revealed cortical dysplasia in 18 patients (41.9%), tumors in 16 patients (37.2%), and other lesions in 9 patients (20.9%). The mean duration of the follow-up period was 84.0 months (range 24–169 months). After SMA resection, 23 patients (53.5%) experienced neurological deficits. Three patients (7.0%) experienced permanent deficits, and 20 (46.5%) experienced symptoms that were transient. All permanent deficits involved contralateral weakness, whereas the transient symptoms patients experienced were varied, including contralateral weaknesses in 15, apraxia in 1, sensory disturbances in 1, and dysphasia in 6. Thirteen patients recovered completely within 1 month. Univariate analysis revealed that resection of the SMA proper, a shorter lifetime seizure history (< 10 years), and resection of the cingulate gyrus in addition to the SMA were associated with the development of neurological deficits (p = 0.078, 0.069, and 0.023, respectively). Cingulate gyrus resection was the only risk factor identified on multivariate analysis (p = 0.027, OR 6.530, 95% CI 1.234–34.562).

Conclusions

Resection of the cingulate gyrus in addition to the SMA was significantly associated with the development of postoperative neurological impairment.

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Seung-Ki Kim, Kyu-Chang Wang, Yong-Seung Hwang, Ki Joong Kim, Jong Hee Chae, In-One Kim and Byung-Kyu Cho

Object

Ideal epilepsy surgery would eliminate seizures without causing any functional deficits. The aim of the present study was to assess seizure outcomes and complications after epilepsy surgery in children with intractable epilepsy.

Methods

Data obtained in 134 children (75 boys and 59 girls) age 17 years or younger who underwent epilepsy surgery at Seoul National University Children's Hospital between 1993 and 2005 were retrospectively reviewed. Epilepsy surgery included temporal resection (59 cases), extratemporal resection (56 cases), functional hemispherectomy (7 cases), callosotomy (9 cases), multiple subpial transection (1 case), and disconnection of a hamartoma (2 cases). The mean follow-up duration was 62.3 months (range 12–168 months).

Results

The overall seizure-free rate was 69% (93 of 134 cases). The seizure-free rate was significantly higher in children who underwent temporal resection than in those in whom extratemporal resection was performed (88 vs 55%, p < 0.05). The most frequent causes of treatment failure were related to the absence of structural abnormality demonstrated on magnetic resonance imaging, development-associated disease, widespread disease documented by postoperative electroencephalography, and limited resection due to the presence of functional cortex. There were no postoperative deaths. Visual field defects were the most common complication after temporal resection (22% [13 of 59 cases]), whereas hemiparesis (mostly transient) was the most common morbidity after extratemporal resection (18% [10 of 56 cases]).

Conclusions

Epilepsy surgery is an effective and safe therapeutic modality in childhood. In children with extratemporal epilepsy, more careful interpretation of clinical and investigative data is needed to achieve favorable seizure outcome.

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Young-Hoon Kim, Young Jin Lee, Jung Ho Han, Soyeon Ahn, Jaebong Lee, Jae Hyoung Kim, Byung Se Choi, Jae Seung Bang, Chae-Yong Kim, Gyojun Hwang, O-Ki Kwon and Chang Wan Oh

OBJECT

The authors aimed to assess whether the prevalence of intracranial aneurysms in patients with intracranial meningiomas was higher than that in a healthy population.

METHODS

The authors performed a hospital-based case-control study of 300 patients with newly diagnosed intracranial meningiomas and 900 age- and sex-matched controls without a history of brain tumors to evaluate any associations between intracranial aneurysms and intracranial meningiomas. Unconditional multivariate logistic regression models were used for case-control comparisons.

RESULTS

Intracranial aneurysms were identified in 23 patients (7.7%) and 24 controls (2.7%; p < 0.001). There was a significant association between intracranial aneurysms and intracranial meningiomas (OR 2.913, 95% CI 1.613–5.261) and hypertension (OR 1.905, 95% CI 1.053–3.446). In a subgroup analysis of the patients with newly diagnosed intracranial meningiomas, there was a significant association between intracranial aneurysms and hypertension (OR 2.876, 95% CI 1.125–7.352) and tumor volume (OR 1.012, 95% CI 1.001–1.024). These patients were also more likely than controls to have other intracranial vascular diseases (p < 0.001), such as isolated occlusion of the intracranial vessels, excluding intracranial aneurysms.

CONCLUSIONS

The prevalence of intracranial aneurysms was higher in patients with intracranial meningiomas. Hypertension and tumor volume appear to be associated with the formation of intracranial aneurysms in these patients.

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Jin Wook Kim, Sung-Hye Park, Sung-Sup Park, Kyu-Chang Wang, Byung-Kyu Cho, So Yeon Kim, Eun Kyung Ra, Chae-Yong Kim and Seung-Ki Kim

✓Fetus-in-fetu is a very rare condition in which one fetus is contained within another. About 100 cases have been reported, and in most of these the fetus was located in the retroperitoneum. The authors describe an extremely rare case of an intracranial fetus-in-fetu in an extraaxial location. This is the eighth intracranial fetus-in-fetu to be reported, the first intracranial extraaxial case, and involves the oldest documented patient with this condition.

Histopathological analysis of the mass revealed a degenerated amnionic membranelike tissue, well-differentiated extremities (including fingerlike structures), skin, matured lungs, well-formed intestines, cerebellar and cerebral tissue, and a notochord with ganglion cells. DNA analysis using short tandem repeat polymorphisms confirmed that the fetus-in-fetu mass and the host infant had heterozygous alleles and were of identical sex and genotype.

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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.