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Neurocognitive outcomes and associated clinical factors 5 years after surgery in children with craniosynostosis

Joo Whan Kim, Kyung Hyun Kim, Ji Hoon Phi, Ji Yeoun Lee, Eun Jung Koh, Byung Jun Kim, Jee Hyeok Chung, Min-Sup Shin, and Seung-Ki Kim

OBJECTIVE

Craniosynostosis involves early closure of one or more sutures, which is known to limit normal cranium growth and interfere with normal brain development. Various surgical methods are used, ranging from minimally invasive strip craniectomy to more extensive whole-vault cranioplasty. This study aimed to evaluate neurocognitive outcomes 5 years after surgical treatment in children with craniosynostosis and to evaluate relevant clinical factors.

METHODS

After exclusion of genetically confirmed syndromic craniosynostosis patients, a retrospective review was conducted on 112 nonsyndromic craniosynostosis patients who underwent surgical treatment and follow-up neurocognitive assessment. Ninety-seven patients underwent strip craniectomy with postoperative orthotic helmet therapy, and 15 received other surgical treatment: 4 with distraction osteotomy and 11 with craniofacial reconstruction. Neurocognitive assessment using the Korean Wechsler Preschool and Primary Scale of Intelligence, Fourth Edition (K-WPPSI-IV), was performed 5 years postoperatively. Clinical factors were assessed regarding neurocognitive outcomes.

RESULTS

The mean age at surgery was significantly younger in the strip craniectomy group (strip craniectomy 4.6 months vs other surgical treatment 18.6 months, p < 0.01). Patients with 2 or more sutures involved were more likely to receive more extensive surgical treatment (16.5% in the strip craniectomy group vs 53.8% in the other group, p < 0.01). Four (3.5%) patients who showed evidence of increased intracranial pressure (ICP) also underwent more extensive surgical treatment. Multivariable linear regression revealed a significant correlation of age at neurocognitive testing (−3.18, 95% CI −5.95 to −0.40, p = 0.02), increased ICP (−34.73, 95% CI −51.04 to −18.41, p < 0.01), and the level of maternal education (6.11, 95% CI 1.01–11.20, p = 0.02) with the Full-Scale Intelligence Quotient (FSIQ). Age at surgery, involvement of 2 or more sutures, and type of operation demonstrated no correlation with FSIQ. Among the 97 patients who underwent strip craniectomy, the FSIQ ranged from mean ± SD 100.2 ± 10.2 (bicoronal) to 110.1 ± 12.7 (lambdoid), and there was no significant difference between the suture groups (p = 0.41). The 5 index scores were all within average ranges based on their age norms.

CONCLUSIONS

Age at neurocognitive assessment, increased ICP, and maternal education level showed significant correlations with the neurocognitive function of craniosynostosis patients. Although children with craniosynostosis exhibited favorable 5-year postoperative neurocognitive outcomes across various synostosis sutures, longer follow-up is needed to reveal the incidence of neurocognitive dysfunction in these patients.

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Surgical indication of pediatric Rathke’s cleft cyst based on a 20-year retrospective cohort

Jong Seok Lee, Yong Hwy Kim, Eun Jung Koh, Ji Hoon Phi, Ji Yeoun Lee, Kyung Hyun Kim, Kyu-Chang Wang, Jung-Eun Cheon, Sung-Hye Park, Young Ah Lee, Choong Ho Shin, and Seung-Ki Kim

OBJECTIVE

Rathke’s cleft cyst (RCC) is the most commonly encountered pituitary incidentaloma in children. Because RCC is not frequently diagnosed in children, there are few reports on pediatric RCCs. The natural course of the disease and appropriate treatments are still obscure. The present study aimed to elucidate the natural history and surgical indications of RCCs in children.

METHODS

The authors retrospectively reviewed the clinical presentations, imaging features, ophthalmological evaluations, endocrine evaluations, and surgical outcomes of pediatric RCCs at a single institution from January 2000 to October 2022. Clinical outcomes between the surgery and observation groups were compared.

RESULTS

Among 93 patients, there were 41 patients in the surgery group and 52 patients in the observation group. The mean age at diagnosis was 10.9 years, and the mean follow-up period was 5.6 years. Headache fully or partially improved after surgery (86.2%), but the rate of improvement was not different from that of the observation group (70.0%). Ophthalmological abnormalities were effectively improved by surgical treatment (93.3%). Both the improvement and deterioration rates of endocrine abnormalities were significantly higher in the surgery group (p = 0.026 and p < 0.001, respectively), but the deterioration rate (43.9%) was higher than the improvement rate (14.6%). In the surgery group, the recurrence rate was 17.1% and the reoperation rate was 4.9%. Compared with total cyst wall resection, cyst fenestration with partial wall resection was associated with a higher recurrence rate (26.9%, p = 0.035) but a lower rate of endocrine abnormalities (30.8%, p = 0.049).

CONCLUSIONS

Pediatric RCCs of ≥ 10 mm in size were analyzed. Ophthalmological abnormalities are the major surgical indications for pediatric RCCs. Headache and partial endocrine abnormalities may be improved with surgery, but they are not absolute indications for surgery. Cyst fenestration with partial wall resection via an endoscopic endonasal approach is the most recommended surgical method. Follow-up is essential to monitor for the occurrence of visual field defects and the recurrence of cysts.

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Clinical outcomes of pediatric cerebral cavernous malformation: an analysis of 124 consecutive cases

Youngbo Shim, Ji Hoon Phi, Kyu-Chang Wang, Byung-Kyu Cho, Ji Yeoun Lee, Eun Jung Koh, Kyung Hyun Kim, Eun Jung Lee, Ki Joong Kim, and Seung-Ki Kim

OBJECTIVE

One-fourth of cerebral cavernous malformation (CCM) patients are children, but studies on these patients are scarce. This study aimed to identify the clinical presentation of pediatric CCM patients and to investigate clinical outcomes according to the treatment modalities applied on the basis of our institution’s treatment strategy.

METHODS

The authors performed a retrospective analysis of 124 pediatric CCM patients with a follow-up of more than 1 year from 2000 to 2019. They performed resection (n = 62) on lesions causing intractable seizure, rebleeding, or mass effect and observed the clinical courses of patients with lesions in deep or eloquent areas without persistent symptoms (n = 52). Radiosurgery (n = 10) was performed when the patient refused resection or strongly desired radiosurgery. The authors investigated the clinical characteristics, performance status (modified Rankin Scale [mRS] score), and rebleeding rate at the 1-year and last follow-up examinations and compared these among 3 groups classified on the basis of treatment applied. The authors evaluated seizure outcomes at the 1-year and last follow-up examinations for the surgery (n = 32) and observation (n = 17) groups. Finally, the authors drew cumulative incidence curves for the discontinuation of antiepileptic drugs (AEDs) for patients in the surgery (n = 30) and observation (n = 9) groups.

RESULTS

The 3 groups showed slight differences in initial symptoms, lesion locations, and rates of recent hemorrhage. The proportion of patients with improved mRS score at the 1-year follow-up was significantly greater in the surgery group than in the other groups (67% of the surgery group, 52% observation group, and 40% radiosurgery group; p = 0.078), as well as at the last follow-up (73% surgery group, 54% observation group, and 60% radiosurgery group; p = 0.097). The surgery group also had the lowest rebleeding rate during the follow-up period (2% surgery group, 11% observation group, and 20% radiosurgery group; p = 0.021). At the 1-year follow-up, the proportion of seizure-free patients without AEDs was significantly higher in the surgery group than the observation group (88% surgery group vs 53% observation group, p < 0.001), and similar results were obtained at the last follow-up (91% surgery group vs 56% observation group, p = 0.05). The 5-year AED-free rates for the surgery and observation groups were 94% and 50%, respectively, on the cumulative incidence curve (p = 0.049).

CONCLUSIONS

The clinical presentation of pediatric CCM patients was not significantly different from that of adult patients. Lesionectomy may be acceptable for pediatric CCM patients with indications of persistent seizures despite AED medications, rebleeding, and mass effects.

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Early cranial vault distraction for a more balanced and enhanced expansion: a 3D craniometric analysis of anterior versus posterior distraction osteogenesis in patients with craniosynostosis

Sungmi Jeon, Jee Hyeok Chung, Sukwha Kim, Seung-Ki Kim, Ji Hoon Phi, Ji Yeoun Lee, Kyung Hyun Kim, Kyu-Chang Wang, and Byung Jun Kim

OBJECTIVE

Posterior distraction osteogenesis (DO) is widely accepted for the treatment of craniosynostosis. The aim of this study was to quantitatively compare the effect of DO on the cranial vault according to the age of the patient and direction of distraction.

METHODS

This was a retrospective study of patients with craniosynostosis who underwent DO in the anteroposterior direction. Postdistraction changes in intracranial volume (ICV), anteroposterior distance, biparietal distance, cranial height, and frontal bossing angle were measured using Mimics software on CT scans. Craniometric data were analyzed using a multivariate regression model.

RESULTS

Thirty-two patients (16 anterior and 16 posterior DOs) were included in the study. The mean ICV increase in the anterior and posterior DO group was 211 cm3 (range 142–281 cm3) and 214 cm3 (range 150–279 cm3), respectively. Patients who were aged 1 year or younger showed a greater percentage increase in ICV than patients older than 1 year. In the anterior DO group, a more balanced increase in both anterior and posterior anteroposterior distance was observed in patients aged 1 year or younger when compared to patients older than 1 year. In the posterior DO group, a bigger expansion and smoother contour in the posterior cranial fossa was observed in patients aged 1 year or younger.

CONCLUSIONS

Both anterior and posterior DO are effective surgical options for expanding the cranial vault in patients with craniosynostosis. Early distraction appeared to show greater morphological changes in the growing cranial vault than those predicted with the vector of distraction.

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Dosimetric parameters associated with the long-term oncological outcomes of Gamma Knife surgery for sellar and parasellar tumors in pediatric patients

Eun Jung Lee, Ji Yeoun Lee, Jin-Wook Kim, Ji Hoon Phi, Yong Hwy Kim, Seung-Ki Kim, Hyun-Tai Chung, Kyu-Chang Wang, and Dong Gyu Kim

OBJECTIVE

The authors aimed to investigate the dosimetric parameter and the minimally required dose associated with long-term control of sellar and parasellar tumors after Gamma Knife surgery (GKS) in children.

METHODS

A retrospective analysis was performed on pediatric patients younger than 19 years of age who were diagnosed with sellar and parasellar tumors and received GKS at the authors’ institution from 1998 to 2019. Cox proportional hazards regression analyses were used to investigate the dosimetric parameters associated with treatment outcome. The Kaplan-Meier method was used to analyze tumor control rates after GKS.

RESULTS

Overall, 37 patients with 40 sellar and parasellar tumors, including 22 craniopharyngiomas and 12 pituitary adenomas, had a mean follow-up of 85.8 months. The gross target volume was 0.05 cm3 to 15.28 cm3, and the mean marginal dose was 15.8 Gy (range 9.6–30.0 Gy). Ten patients experienced treatment failure at a mean of 28.0 ± 26.7 months. The actuarial 5- and 10-year tumor control rates were 79.0% and 69.8%, respectively. D98% was an independent predictive factor of tumor control (HR 0.846 [95% CI 0.749–0.956], p = 0.007), with a cutoff value of 11.5 Gy for the entire cohort and 10 Gy for the craniopharyngioma group. Visual deterioration occurred in 2 patients with the maximum point dose of 10.1 Gy and 10.6 Gy to the optic apparatus.

CONCLUSIONS

In pediatric patients, D98% was a reliable index of the minimum required dose for long-term control of sellar and parasellar tumors after GKS. The optimal D98% value for each tumor diagnosis needs to be elucidated in the future.

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Long-term outcome of large sylvian arachnoid cysts: the role of surgery has been exaggerated

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

OBJECTIVE

The surgical indications for some arachnoid cysts (ACs) are controversial. While surgical procedures can be effective when an AC is a definite cause of hydrocephalus or papilledema, most ACs do not cause any symptoms or signs. Some surgeons perform several procedures to treat ACs because of their large size. The purpose of this study was to compare the long-term outcomes of Galassi type III ACs between surgery and nonsurgery groups.

METHODS

The medical records of 60 patients diagnosed with sylvian ACs (Galassi type III) who visited Seoul National University Children’s Hospital from July 1990 to March 2018 were analyzed. The authors compared the outcomes between those treated with surgery and those not treated with surgery.

RESULTS

Of the 60 patients, 27 patients had no symptoms, 19 patients had vague symptoms and signs associated with ACs, and the remaining 14 patients had definite AC-related symptoms and signs. Thirty-eight patients underwent surgery, and 22 patients underwent observation. Some operations were accompanied by complications. Among the 33 patients in the surgery group, excluding 5 with hydrocephalus or papilledema, 8 patients needed 18 additional operations. However, there were no patients in the nonsurgery group who needed surgical intervention during the follow-up period (mean 67.5 months), although the size of the AC increased in 2 patients. Changes in AC size were not correlated with symptom relief.

CONCLUSIONS

When patients with hydrocephalus or papilledema were excluded, there was no difference in the outcomes between the surgery and nonsurgery groups regardless of the size of the sylvian AC. Surgeons should be cautious when deciding whether to operate.

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High prevalence of systemic hypertension in pediatric patients with moyamoya disease years after surgical treatment

Joongyub Lee, Seung-Ki Kim, Hee Gyung Kang, Il-Soo Ha, Kyu-Chang Wang, Ji Yeoun Lee, and Ji Hoon Phi

OBJECTIVE

Although an association between moyamoya disease (MMD) and renovascular hypertension has been acknowledged, the literature on systemic hypertension without renal artery stenosis among patients with MMD is scarce. The authors aimed to evaluate the prevalence and risk factors of hypertension using data from MMD patients who visited an outpatient clinic of a pediatric neurosurgical department in 2016.

METHODS

The authors evaluated the blood pressure (BP) of pediatric MMD patients at their postsurgical appointment following the American Academy of Pediatrics clinical practice guideline on high BP, in which hypertension was defined as BP measurements higher than the value of age-, sex-, and height-specific 95th percentile of the general population from at least 3 separate visits. Growth of patients was determined using 2017 Korean National Growth Charts for children and adolescents. The cutoff value of the 95th percentile of BP was determined by referring to normative BP tables of Korean children and adolescents. A logistic regression model was used to assess the associations between patients’ clinical characteristics and prevalent hypertension.

RESULTS

In total, 131 surgically treated pediatric MMD patients were included, of whom 38.9% were male and the median age at diagnosis was 8.0 years (range 1.2–15.0 years). The definition of hypertension was met in 38 patients, with a prevalence of 29.0% (95% CI 21.2%–36.8%). A tendency was observed for a higher prevalence of hypertension in male patients (31.4%), in patients with posterior cerebral artery (PCA) involvement (47.8%), and in cases in which infarction was shown on initial MRI (37.3%). Age at diagnosis (adjusted OR [aOR] 0.82, 95% CI 0.70–0.97), PCA involvement (aOR 3.81, 95% CI 1.29–11.23), body mass index (aOR 1.30, 95% CI 1.13–1.51), and years of follow-up since surgery (aOR 0.80, 95% CI 0.68–0.94) were related to systemic hypertension.

CONCLUSIONS

A high prevalence of hypertension was demonstrated in pediatric MMD patients. Therefore, adequate attention should be paid to reduce BP and prevent subsequent events.

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Feasibility of intraoperative monitoring of motor evoked potentials obtained through transcranial electrical stimulation in infants younger than 3 months

You Gyoung Yi, Keewon Kim, Hyung-Ik Shin, Moon Suk Bang, Hee-Soo Kim, Jinwoo Choi, Kyu-Chang Wang, Seung-Ki Kim, Ji Yeoun Lee, Ji Hoon Phi, and Han Gil Seo

OBJECTIVE

This study aimed to investigate the feasibility and safety of intraoperative motor evoked potential (MEP) monitoring in infants less than 3 months of age.

METHODS

The authors investigated 25 cases in which infants younger than 3 months (mean age 72.8 days, range 39–87) underwent neurosurgery between 2014 and 2017. Myogenic MEPs were obtained through transcranial electrical stimulation. In all cases, surgery was performed under total intravenous anesthesia, maintained with remifentanil and propofol.

RESULTS

MEPs were documented in 24 infants, the sole exception being 1 infant who was lethargic and had 4-limb weakness before surgery. The mean stimulation intensity maintained during monitoring was 596 ± 154 V (range 290–900 V). In 19 of 24 infants MEP signals remained at ≥ 50% of the baseline amplitude throughout the operation. Among 5 cases with a decrease in intraoperative MEP amplitude, the MEP signal was recovered in one during surgery, and in the other case a neurological examination could not be performed after surgery. In the other 3 cases, 2 infants had relevant postoperative weakness and the other did not show postoperative neurological deficits. Postoperative weakness was not observed in any of the 20 infants who had no deterioration (n = 19) or only temporary deterioration (n = 1) in MEP signal during surgery.

CONCLUSIONS

Transcranial electrical MEPs could be implemented during neurosurgery in infants between 1 and 3 months of age. Intraoperative MEP monitoring may be a safe adjunct for neurosurgical procedures in these very young patients.

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Impaired functional recovery of endothelial colony-forming cells from moyamoya disease in a chronic cerebral hypoperfusion rat model

Seung Ah Choi, Sangjoon Chong, Pil Ae Kwak, Youn Joo Moon, Anshika Jangra, Ji Hoon Phi, Ji Yeoun Lee, Sung-Hye Park, and Seung-Ki Kim

OBJECTIVE

Endothelial colony-forming cells (ECFCs) isolated from pediatric patients with moyamoya disease (MMD) have demonstrated decreased numbers and defective functioning in in vitro experiments. However, the function of ECFCs has not been evaluated using in vivo animal models. In this study, the authors compared normal and MMD ECFCs using a chronic cerebral hypoperfusion (CCH) rat model.

METHODS

A CCH rat model was made via ligation of the bilateral common carotid arteries (2-vessel occlusion [2-VO]). The rats were divided into three experimental groups: vehicle-treated (n = 8), normal ECFC-treated (n = 8), and MMD ECFC-treated (n = 8). ECFCs were injected into the cisterna magna. A laser Doppler flowmeter was used to evaluate cerebral blood flow, and a radial arm maze test was used to examine cognitive function. Neuropathological examinations of the hippocampus and agranular cortex were performed using hematoxylin and eosin and Luxol fast blue staining in addition to immunofluorescence with CD31, von Willebrand factor, NeuN, myelin basic protein, glial fibrillary acidic protein, and cleaved caspase-3 antibodies.

RESULTS

The normal ECFC-treated group exhibited improvement in the restoration of cerebral perfusion and in behavior compared with the vehicle-treated and MMD ECFC-treated groups at the 12-week follow-up after the 2-VO surgery. The normal ECFC-treated group showed a greater amount of neovasculogenesis and neurogenesis, with less apoptosis, than the other groups.

CONCLUSIONS

These results support the impaired functional recovery of MMD ECFCs compared with normal ECFCs in a CCH rat model. This in vivo study suggests the functional role of ECFCs in the pathogenesis of MMD.

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Optimal stimulation parameters for intraoperative bulbocavernosus reflex in infants

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.