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Shunya Hanakita, Tomoyuki Koga, Masahiro Shin, Hiroshi Igaki and Nobuhito Saito

OBJECT

Although stereotactic radiosurgery (SRS) has been accepted as a therapeutic option for arteriovenous malformations (AVMs) in children and adolescents, substantial data are still lacking regarding the outcomes of SRS for AVMs in this age group, especially long-term complications. This study aimed to clarify the long-term outcomes of SRS for the treatment of AVM in pediatric patients aged ≤ 18 years.

METHODS

Outcomes of 116 patients who were aged 4–18 years when they underwent SRS between 1990 and 2009 at the study institute were analyzed retrospectively.

RESULTS

The median follow-up period after SRS was 100 months, with 6 patients followed up for more than 20 years. Actuarial obliteration rates at 3 and 5 years after SRS were 68% and 88%, respectively. Five hemorrhages occurred in 851 patient-years of follow-up. The annual bleeding rate after SRS before obliteration was calculated as 1.3%, which decreased to 0.2% after obliteration. Shorter maximum nidus diameter (p = 0.02) and higher margin dose (p = 0.03) were associated with a higher obliteration rate. Ten patients experienced adverse events after SRS. Of them, 4 patients presented with delayed complications years after SRS (range 9–20 years after SRS).

CONCLUSIONS

SRS can reduce the risk of hemorrhage in pediatric and adolescent AVMs, with an acceptable risk of complications in the long term. However, adverse events such as expanding hematoma and radiation necrosis that can occur after substantial follow-up should be taken into account at the time that treatment decisions are made and informed consent is obtained.

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Masahiro Shin, Kenji Kondo, Shunya Hanakita, Keigo Suzukawa, Taichi Kin, Masaaki Shojima, Daichi Nakagawa and Nobuhito Saito

OBJECT

In recent years, application of endoscopic transnasal surgery (ETS) has been expanded to orbital lesions, and preliminary results have started to be published for medially located soft mass lesions. However, reports on experience with endoscopic intraorbital surgery aimed at resection of invasive skull base tumors remains quite limited. This report presents the authors’ experience with ETS for locally aggressive tumors involving the orbit.

METHODS

ETS was performed for 15 cases of aggressive tumors involving the orbit: 5 meningiomas (meningothelial, n = 3; atypical, n = 1; anaplastic, n = 1), 4 chordomas, 2 chondrosarcomas, and 4 others (metastasis from systemic myxofibrosarcoma, schwannoma, inverted papilloma, and acinic cell carcinoma, n = 1 each). Among these, 9 tumors were located outside the periorbita and 6 inside the periorbita. In 6 intraperiosteal tumors, 5 were intraconal lesions, of which 3 arose in the muscle cone (anaplastic meningioma, optic sheath meningioma, and metastatic myxofibrosarcoma), and 2 meningothelial meningioma had invaded from the sphenoid ridge or the cavernous sinus into the muscle cone through the optic canal and the superior orbital fissure. A case of schwannoma originated around the cavernous sinus and pterygopalatine fossa and extended extraconally into the periorbita. Intraoperatively, ethmoid air cells and the lamina papyracea were removed, and extraperiosteal tumors were safely approached. For intraperiosteal tumors, the periorbita was widely opened, and the tumors were approached through the surgical window between the rectus and oblique muscles.

RESULTS

Gross-total resection was achieved for 12 of the 15 tumors, including 2 intraconal lesions. After surgery, exophthalmos resolved in all 8 patients with this symptom, and diplopia resolved in 5 of 6 patients. Improvement of visual symptoms was reported by 4 of 5 patients with loss of visual acuity or constriction of the visual field. Postoperatively, 1 patient showed mild, transient worsening of existing facial dysesthesia, and another showed transient ptosis and mild hypesthesia of the forehead on the affected side. All those symptoms resolved within 3 months. No patients showed enophthalmos, worsening of diplopia or visual function, or impairment of olfaction after surgery.

CONCLUSIONS

ETS appears acceptable as a less-invasive alternative for treating aggressive tumors involving the orbit. For extraperiosteal tumors, gross-total removal can generally be achieved without neurological complications. For intraperiosteal tumors, surgical indications should be carefully discussed, considering the relationship between the tumor and normal anatomy. Wide opening of the periorbital window is advocated to create a sufficient surgical pathway between the extraocular muscles, allowing a balance between functional preservation and successful tumor resection.

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Shunya Hanakita, Atumi Takenobu, Atsushi Kambe, Takashi Watanabe, Masahiro Shin and Akira Teraoka

The authors present a case of germinoma that was initially found in the pineal region and recurred 15 years later in the intramedullary cervical spinal cord after intensive chemo- and radiotherapy and diagnosis of complete remission.

This 28-year-old man initially presented with seizures. Hydrocephalus and a pineal tumor were found on radiological examination, and partial resection of the tumor was performed. Histological diagnosis showed a pure germinoma. Following surgery, the patient received a combination of chemo- and radiotherapy, and a complete remission was shown. However, after 15 years of follow-up, he presented with gait disturbances. Spinal MRI showed an intramedullary mass lesion in the cervical spinal cord. The cervical lesion was biopsied, and histological examination again revealed a pure germinoma.

With germinomas, the possibility of a drop metastasis from an intracranial lesion to the spinal cord must be considered during follow-up. However, in the present case, analysis of a CSF sample showed no abnormalities as in previously published cases. In recent years, multidisciplinary treatments have demonstrated good event-free survival rates in cases of pure germinomas, but long-term outcomes over the decades are not fully known. Continual follow-up of such cases is recommended even after complete remission has been achieved.

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Shunya Hanakita, Tomoyuki Koga, Hiroshi Igaki, Naoya Murakami, Soichi Oya, Masahiro Shin and Nobuhito Saito

Object

Atypical meningioma often recurs even after resection. As a salvage modality, radiotherapy or stereotactic radiosurgery (SRS) is attempted for this aggressive tumor. This retrospective study was performed to evaluate the efficacy of SRS that involved Gamma Knife surgery (GKS) for atypical meningioma.

Methods

The authors reviewed records from 22 patients with histologically proven atypical meningioma who underwent GKS for 28 lesions at the authors' institute. The median patient age was 70 years (range 24–91 years), and the median tumor volume for each procedure was 6.0 cm3 (range 1.6–38.7 cm3). The margin dose ranged from 14 to 20 Gy (median 18 Gy). Follow-up periods ranged from 3 months to 98 months (median 23.5 months).

Results

In total, 39 GKS procedures were performed for 28 lesions. The local control rates at 1, 2, and 5 years were 74%, 39%, and 16%, respectively. Volume less than 6 cm3 (p = 0.01), a margin dose higher than 18 Gy (p = 0.02), and a Karnofsky Performance Scale (KPS) score of 90 or more (p = 0.02) were factors associated with a longer duration of tumor control in the univariate analysis.

Conclusions

Atypical meningioma could be more successfully controlled when a higher margin dose was used to treat patients with a good performance (KPS score of ≥ 90) status and smaller tumor volumes. It would be desired if patients are treated with a relatively higher margin dose, ideally as high as the dose applied for malignant tumor. A boost SRS after fractionated radiotherapy may be effective to achieve better local control.

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Masahiro Shin, Kenji Kondo, Shunya Hanakita, Hirotaka Hasegawa, Masanori Yoshino, Yu Teranishi, Taichi Kin and Nobuhito Saito

OBJECTIVE

Reports about endoscopic endonasal surgery for skull base tumors involving the lateral part of petrous apex remain scarce. The authors present their experience with the endoscopic transsphenoidal anterior petrosal (ETAP) approach through the retrocarotid space for tumors involving the internal auditory canal, jugular fossa, and cavernous sinus.

METHODS

The authors performed the ETAP approach in 10 patients with 11 tumors (bilateral in 1 patient) that extensively occupied the lateral part of petrous apex, e.g., the internal auditory canal and jugular fossa. Eight patients presented with diplopia (unilateral abducens nerve palsy), 3 with tinnitus, and 1 with unilateral hearing loss with facial palsy. After wide anterior sphenoidotomy, the sellar floor, clival recess, and carotid prominence were verified. Tumors were approached via an anteromedial petrosectomy through the retrocarotid triangular space, defined by the cavernous and vertical segments of the internal carotid artery (ICA), the clivus, and the petrooccipital fissure. The surgical window was easily enlarged by drilling the petrous bone along the petrooccipital fissure. After exposure of the tumor and ICA, dissection and resection of the tumor were mainly performed under direct visualization with 30° and 70° endoscopes.

RESULTS

Gross-total resection was achieved in 8 patients (9 tumors). In a patient with invasive meningioma, the tumor was strongly adherent to the ICA, necessitating partial resection. Postoperatively, all 8 patients who had presented with abducens nerve palsy preoperatively showed improvement within 6 months. In the patient presenting with hearing loss and facial palsy, the facial palsy completely resolved within 3 months, but hearing loss remained. Regarding complications, 3 patients showed mild and transient abducens nerve palsy resolving within 2 weeks, 3 months, and 6 months. Postoperative CSF rhinorrhea requiring surgical repair was observed in 1 patient. No patient exhibited hearing deterioration, facial palsy, or symptoms of lower cranial nerve palsy after surgery.

CONCLUSIONS

The ETAP approach can offer a simple, less invasive option for invasive skull base tumors involving petrous regions, including the internal auditory canal, jugular fossa, and cavernous sinus. The ETAP approach can reach more extensive areas in the extradural regions around the petrous bone. The authors' results indicate that the transsphenoidal retrocarotid route is sufficient to approach the petrosal areas in select cases. Further expansion of the surgical field is not always necessary. However, experience with intradural lesions remains limited, and the extent of tumor resection largely depends on tumor characteristics. Application of the ETAP approach should thus be carefully determined in each patient, taking into consideration the size of the retrocarotid window and tumor characteristics.

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Hirotaka Hasegawa, Masahiro Shin, Kenji Kondo, Shunya Hanakita, Akitake Mukasa, Taichi Kin and Nobuhito Saito

OBJECTIVE

Skull base chondrosarcoma is one of the most intractable tumors because of its aggressive biological behavior and involvement of the internal carotid artery and cranial nerves (CNs). One of the most accepted treatment strategies for skull base chondrosarcoma has been surgical removal of the tumor in conjunction with proactive extensive radiation therapy (RT) to the original tumor bed. However, the optimal strategy has not been determined. The goal of this study was to evaluate the early results of endoscopic transnasal surgery (ETS).

METHODS

The authors retrospectively analyzed 19 consecutive patients who underwent ETS at their institution since 2010. Adjuvant stereotactic radiosurgery (SRS) was performed only for the small residual tumors that were not resected to avoid critical neurological complications. Histological confirmation and evaluation of the MIB-1 index was performed in all cases. The Kaplan-Meier method was used to determine the actuarial rate of tumor-free survival.

RESULTS

The median tumor volume and maximal diameter were 14.5 cm3 (range 1.4–88.4 cm3) and 3.8 cm (range 1.5–6.7 cm), respectively. Nine patients (47%) had intradural extension of the tumor. Gross-total resection was achieved in 15 (78.9%) of the 19 patients, without any disabling complications. In 4 patients, the surgery resulted in subtotal (n = 2, 11%) or partial (n = 2, 11%) resection because the tumors involved critical structures, including the basilar artery or the lower CNs. These 4 patients were additionally treated with SRS. The median follow-up duration was 47, 28, and 27 months after the diagnosis, ETS, and SRS, respectively. In 1 patient with an anterior skull base chondrosarcoma, the tumor relapsed in the optic canal 1 year later and was treated with a second ETS. Favorable tumor control was achieved in all other patients. The actuarial tumor control rate was 93% at 5 years. At the final follow-up, all patients were alive and able to perform independent activities of daily living without continuous neurological sequelae.

CONCLUSIONS

These preliminary results suggest that ETS can achieve sufficient radical tumor removal, resulting in comparative resection rates with fewer neurological complications to those in previous reports. Although the follow-up periods of these cases were relatively short, elective SRS to the small tumor remnant may be rational, achieving successful tumor control in some cases, instead of using proactive extensive RT. Thus, the addition of RT should be discussed with each patient, after due consideration of histological grading and biological behavior. To determine the efficacy of this strategy, a larger case series with a longer follow-up period is essential. However, this strategy may be able to establish evidence in the management of skull base chondrosarcoma, providing less-invasive and effective options as an initial step of treatment.

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Shunya Hanakita, Tomoyuki Koga, Masahiro Shin, Masaaki Shojima, Hiroshi Igaki and Nobuhito Saito

Object

The goal of this study was to assess the efficacy of Gamma Knife surgery (GKS) in the management of dural arteriovenous fistulas (dAVFs).

Methods

The authors performed a retrospective analysis of a group of 22 patients who underwent GKS for dAVFs at the University of Tokyo Hospital between 1991 and 2009. The patients underwent CT or MR imaging with contrast enhancement every 6 months after GKS; when obliteration of a dAVF was indicated by these images, patients also underwent angiography. Follow-up in these patients ranged from 12 months to 100 months (median 33 months) after GKS.

Results

Obliteration of the dAVF was confirmed by neuroimaging in 12 patients (55%). According to a Kaplan-Meier analysis, obliteration rates for the dAVFs were 51% at 3 years and 80% at 5 years. The obliteration rate for lesions without cortical venous drainage (CVD) was 86%, which was significantly higher than the rate for dAVFs with CVD (47%) (p = 0.007). Hemorrhage at presentation (p = 0.03), a target volume less than 1.5 cm3 (p = 0.009), and Cognard Type III or IV dAVF (p = 0.005) were factors associated with a higher obliteration rate. Among 10 patients whose dAVFs were not obliterated by the initial GKS, 5 patients underwent additional treatment and complete obliteration was achieved in all. Relief of tinnitus was obtained in 5 (83%) of 6 patients with transverse-sigmoid sinus dAVFs, and ophthalmic symptoms improved in 2 (67%) of 3 patients with cavernous sinus dAVFs. No patient experienced interval hemorrhage or radiation-induced complications after treatment.

Conclusions

Gamma Knife surgery is a safe and effective treatment for dAVF. It can be a first line of therapy in the multidisciplinary treatment strategy for dAVFs, especially when significant morbidity is anticipated with other therapeutic options. One should be very careful about recommending GKS for patients harboring dAVFs with CVD because of the expected natural history of such a lesion and the possibility of other therapeutic options.

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Yuta Fukushima, Soichi Oya, Hirofumi Nakatomi, Junji Shibahara, Shunya Hanakita, Shota Tanaka, Masahiro Shin, Kensuke Kawai, Masashi Fukayama and Nobuhito Saito

Object

Meningiomas treated by subtotal or partial resection are associated with significantly shorter recurrence-free survival than those treated by gross-total resection. The Simpson grading system classifies incomplete resections into a single category, namely Simpson Grade IV, with wide variations in the volume and location of residual tumors, making it complicated to evaluate the achievement of surgical goals and predict the prognosis of these tumors. Authors of the present study investigated the factors related to necessity of retreatment and tried to identify any surgical nuances achievable with the aid of modern neurosurgical techniques for meningiomas treated using Simpson Grade IV resection.

Methods

This retrospective analysis included patients with WHO Grade I meningiomas treated using Simpson Grade IV resection as the initial therapy at the University of Tokyo Hospital between January 1995 and April 2010. Retreatment was defined as reresection or stereotactic radiosurgery due to postoperative tumor growth.

Results

A total of 38 patients were included in this study. Regrowth of residual tumor was observed in 22 patients with a mean follow-up period of 6.1 years. Retreatment was performed for 20 of these 22 tumors with regrowth. Risk factors related to significantly shorter retreatment-free survival were age younger than 50 years (p = 0.006), postresection tumor volume of 4 cm3 or more (p = 0.016), no dural detachment (p = 0.001), and skull base location (p = 0.016). Multivariate analysis revealed that no dural detachment (hazard ratio [HR] 6.42, 95% CI 1.41–45.0; p = 0.02) and skull base location (HR 11.6, 95% CI 2.18–218; p = 0.002) were independent risk factors for the necessity of early retreatment, whereas postresection tumor volume of 4 cm3 or more was not a statistically significant risk factor.

Conclusions

Compared with Simpson Grade I, II, and III resections, Simpson Grade IV resection includes highly heterogeneous tumors in terms of resection rate and location of the residual mass. Despite the difficulty in analyzing such diverse data, these results draw attention to the favorable effect of dural detachment (instead of maximizing the resection rate) on long-term tumor control. Surgical strategy with an emphasis on detaching the tumor from the affected dura might be another important option in resection of high-risk meningiomas not amenable to gross-total resection.

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Hirotaka Hasegawa, Shunya Hanakita, Masahiro Shin, Mariko Kawashima, Taichi Kin, Wataru Takahashi, Yuichi Suzuki, Yuki Shinya, Hideaki Ono, Masaaki Shojima, Hirofumi Nakatomi and Nobuhito Saito

OBJECTIVE

In Gamma Knife radiosurgery (GKS) for arteriovenous malformations (AVMs), CT angiography (CTA), MRI, and digital subtraction angiography (DSA) are generally used to define the nidus. Although the AVM angioarchitecture can be visualized with superior resolution using rotational angiography (RA), the efficacy of integrating RA into the GKS treatment planning process has not been elucidated.

METHODS

Using data collected from 25 consecutive patients with AVMs who were treated with GKS at the authors’ institution, two neurosurgeons independently created treatment plans for each patient before and after RA integration. For all patients, MR angiography, contrasted T1 imaging, CTA, DSA, and RA were performed before treatment. The prescription isodose volume before (PIVB) and after (PIVA) RA integration was measured. For reference purposes, a reference target volume (RTV) for each nidus was determined by two other physicians independent of the planning surgeons, and the RTV covered by the PIV (RTVPIV) was established. The undertreated volume ratio (UVR), overtreated volume ratio (OVR), and Paddick’s conformal index (CI), which were calculated as RTVPIV/RTV, RTVPIV/PIV, and (RTVPIV)2/(RTV × PIV), respectively, were measured by each neurosurgeon before and after RA integration, and the surgeons’ values at each point were averaged. Wilcoxon signed-rank tests were used to compare the values obtained before and after RA integration. The percentage change from before to after RA integration was calculated for the average UVR (%ΔUVRave), OVR (%ΔOVRave), and CI (%ΔCIave) in each patient, as ([value after RA integration]/[value before RA integration] − 1) × 100. The relationships between prior histories and these percentage change values were examined using Wilcoxon signed-rank tests.

RESULTS

The average values obtained by the two surgeons for the median UVR, OVR, and CI were 0.854, 0.445, and 0.367 before RA integration and 0.882, 0.478, and 0.463 after RA integration, respectively. All variables significantly improved after compared with before RA integration (UVR, p = 0.009; OVR, p < 0.001; CI, p < 0.001). Prior hemorrhage was significantly associated with larger %ΔOVRave (median 20.8% vs 7.2%; p = 0.023) and %ΔCIave (median 33.9% vs 13.8%; p = 0.014), but not %ΔUVRave (median 4.7% vs 4.0%; p = 0.449).

CONCLUSIONS

Integrating RA into GKS treatment planning may permit better dose planning owing to clearer visualization of the nidus and, as such, may reduce undertreatment and waste irradiation. Further studies examining whether the observed RA-related improvement in dose planning also improves the radiosurgical outcome are needed.