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Misao Nishikawa, Hiroaki Sakamoto, Akira Hakuba, Naruhiko Nakanishi and Yuichi Inoue

✓ To investigate overcrowding in the posterior cranial fossa as the pathogenesis of adult-type Chiari malformation, the authors studied the morphology of the brainstem and cerebellum within the posterior cranial fossa (neural structures consisting of the midbrain, pons, cerebellum, and medulla oblongata) as well as the base of the skull while taking into consideration their embryological development. Thirty patients with Chiari malformation and 50 normal control subjects were prospectively studied using neuroimaging. To estimate overcrowding, the authors used a “volume ratio” in which volume of the posterior fossa brain (consisting of the midbrain, pons, cerebellum, and medulla oblongata within the posterior cranial fossa) was placed in a ratio with the volume of the posterior fossa cranium encircled by bony and tentorial structures. Compared to the control group, in the Chiari group there was a significantly larger volume ratio, the two occipital enchondral parts (the exocciput and supraocciput) were significantly smaller, and the tentorium was pronouncedly steeper. There was no significant difference in the posterior fossa brain volume or in the axial lengths of the hindbrain (the brainstem and cerebellum). In six patients with basilar invagination the medulla oblongata was herniated, all three occipital enchondral parts (the basiocciput, exocciput, and supraocciput) were significantly smaller than in the control group, and the volume ratio was significantly larger than that in the Chiari group without basilar invagination.

These results suggest that in adult-type Chiari malformation an underdeveloped occipital bone, possibly due to underdevelopment of the occipital somite originating from the paraxial mesoderm, induces overcrowding in the posterior cranial fossa, which contains the normally developed hindbrain. Basilar invagination is associated with a more severe downward herniation of the hindbrain due to the more severely underdeveloped occipital enchondrium, which further exacerbates overcrowding of the posterior cranial fossa.

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Misao Nishikawa, Hiroaki Sakamoto, Akira Hakuba, Naruhiko Nakanishi and Yuichi Inoue

To investigate overcrowding in the posterior cranial fossa as the pathogenesis of adult-type Chiari malformation, the authors studied the morphology of the brainstem and cerebellum within the posterior cranial fossa (neural structures consisting of the midbrain, pons, cerebellum, and medulla oblongata) as well as the base of the skull while taking into consideration their embryological development. Thirty patients with Chiari malformation and 50 normal control subjects were prospectively studied using neuroimaging. To estimate overcrowding, the authors used a "volume ratio" in which volume of the posterior fossa brain (consisting of the midbrain, pons, cerebellum, and medulla oblongata within the posterior cranial fossa) was placed in a ratio with the volume of the posterior fossa cranium encircled by bony and tentorial structures. Compared to the control group, in the Chiari group there was a significantly larger volume ratio, the two occipital enchondral parts (the exocciput and supraocciput) were significantly smaller, and the tentorium was pronouncedly steeper. There was no significant difference in the posterior fossa brain volume or in the axial lengths of the hindbrain (the brainstem and cerebellum). In six patients with basilar invagination the medulla oblongata was herniated, all three occipital enchondral parts (the basiocciput, exocciput, and supraocciput) were significantly smaller than in the control group, and the volume ratio was significantly larger than that in the Chiari group without basilar invagination.

These results suggest that in adult-type Chiari malformation an underdeveloped occipital bone, possibly due to underdevelopment of the occipital somite originating from the paraxial mesoderm, induces overcrowding in the posterior cranial fossa, which contains the normally developed hindbrain. Basilar invagination is associated with a more severe downward herniation of the hindbrain due to the more severely underdeveloped occipital enchondrium, which further exacerbates overcrowding of the posterior cranial fossa.

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Misao Nishikawa, Hiroaki Sakamoto, Junsuke Katsuyama, Akira Hakuba and Shuro Nishimura

✓ The authors present the case of a patient with ischemic episodes and recurrent intracerebral hemorrhages probably caused by primary angiitis of the central nervous system (CNS). An initial angiogram revealed multiple cerebral artery aneurysms as well as vascular wall irregularity; a second angiogram obtained 2 years later, however, did not demonstrate the previous aneurysms but instead showed new ones together with stenosis. Based on the histopathological findings and clinical course in this case, the multiple aneurysms appear to have been induced by vascular wall fragility and subsequent self-repair resulting from primary angiitis of the CNS. The authors present the histological and clinical characteristics of this unusual case of granulomatous, necrotizing CNS vasculitis.

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Misao Nishikawa, Hiroaki Sakamoto, Junsuke Katsuyama, Akira Hakuba and Shuro Nishimura

The authors present the case of a patient with ischemic episodes and recurrent intracerebral hemorrhages probably caused by primary angiitis of the central nervous system (CNS). An initial angiogram revealed multiple cerebral artery aneurysms as well as vascular wall irregularity; a second angiogram obtained 2 years later, however, did not demonstrate the previous aneurysms but instead showed new ones together with stenosis. Based on the histopathological findings and clinical course in this case, the multiple aneurysms appear to have been induced by vascular wall fragility and subsequent self-repair resulting from primary angiitis of the CNS. The authors present the histological and clinical characteristics of this unusual case of granulomatous, necrotizing CNS vasculitis.

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Masaki Komiyama, Misao Nishikawa, Shouhei Kitano, Hiroaki Sakamoto, Nobuhiro Miyagi, Satoshi Kusuda and Hisakazu Sugimoto

✓ A neonate, in whom a congenital cerebral vascular anomaly had been diagnosed prenatally, exhibited progressive high-output congestive heart failure soon after birth. Cerebral angiography revealed a congenital dural arteriovenous fistula (AVF) with a huge dural lake located at the torcular herophili. In addition to the meningeal blood supply, an unusual pial blood supply from all cerebellar arteries was observed to feed the fistula. The patient was treated by repeated transarterial and transvenous embolization through the umbilical venous route. To the authors' knowledge, neither the existence of a congenital dural AVF at the torcular herophili presenting with an enormous pial blood supply or the technique of transumbilical venous intervention has been reported in the literature.

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Fusiform vertebral artery aneurysms as a cause of dissecting aneurysms

Report of two autopsy cases and a review of the literature

Toshihiro Yasui, Masaki Komiyama, Misao Nishikawa, Hideki Nakajima, Yasutsugu Kobayashi and Takeshi Inoue

✓ Two autopsy cases of angiographically determined fusiform aneurysms of the vertebral arteries (VAs) are reported and the appropriate literature is reviewed to investigate the pathological characteristics of both fusiform and dissecting VA aneurysms and the pathogenesis of dissecting aneurysms. One patient had suffered a subarachnoid hemorrhage (SAH) due to dissection of a previously documented incidental fusiform aneurysm. The other patient had harbored incidental fusiform aneurysms coexistent with a ruptured aneurysm of the posterior inferior cerebellar artery. The location and pathological features of the aneurysms were similar in the two cases. The aneurysms in both cases displayed intimal thickening, disruption of the internal elastic lamina, and degeneration of the media. A mural hemorrhage and patchy calcification were also found in the case that included SAH. Based on their pathological investigation of these two cases and a review of reported cases, the authors propose that incidental fusiform aneurysms in the VAs are characterized by weakness in the internal elastic lamina and, therefore, have the potential to become dissecting aneurysms, resulting in a fatal prognosis. This suggests that long-term control of blood pressure is mandatory in patients with incidental fusiform aneurysms in the VAs.

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Thomas H. Milhorat, Paolo A. Bolognese, Misao Nishikawa, Nazli B. McDonnell and Clair A. Francomano

Object

Chiari malformation Type I (CM-I) is generally regarded as a disorder of the paraxial mesoderm. The authors report an association between CM-I and hereditary disorders of connective tissue (HDCT) that can present with lower brainstem symptoms attributable to occipitoatlantoaxial hypermobility and cranial settling.

Methods

The prevalence of HDCT was determined in a prospectively accrued cohort of 2813 patients with CM-I. All patients underwent a detailed medical and neuroradiological workup that included an assessment of articular mobility. Osseous structures composing the craniocervical junction were investigated morphometrically using reconstructed 3D computed tomography and plain x-ray images in 114 patients with HDCT/CM-I, and the results were compared with those obtained in patients with CM-I (55 cases) and healthy control individuals (55 cases).

Results

The diagnostic criteria for Ehlers–Danlos syndrome and related HDCT were met in 357 (12.7%) of the 2813 cases. Hereditability was generally compatible with a pattern of autosomal dominant transmission with variable expressivity. The diagnostic features of HDCT/CM-I were distinguished from those of CM-I by clinical stigmata of connective tissue disease, a greater female preponderance (8:1 compared with 3:1, p < 0.001), and a greater incidence of lower brainstem symptoms (0.41 compared with 0.11, p < 0.001), retroodontoid pannus formation (0.71 compared with 0.11, p < 0.001), and hypoplasia of the oropharynx (0.44 compared with 0.02, p < 0.001). Measurements of the basion–dens interval, basion–atlas interval, atlas–dens interval, dens–atlas interval, clivus–atlas angle, clivus–axis angle, and atlas–axis angle were the same in the supine and upright positions in healthy control individuals and patients with CM-I. In patients with HDCT/CM-I, there was a reduction of the basion–dens interval (3.6 mm, p < 0.001), an enlargement of the basion–atlas interval (3.0 mm, p < 0.001), and a reduction of the clivus–axis angle (10.8°, p < 0.001), clivus–atlas angle (5.8°, p < 0.001), and atlas–axis angle (5.3°, p < 0.001) on assumption of the upright position. These changes were reducible by cervical traction or returning to the supine position.

Conclusions

The identification of HDCT in 357 patients with CM-I establishes an association between two presumably unrelated mesodermal disorders. Morphometric evidence in this cohort—cranial settling, posterior gliding of the occipital condyles, and reduction of the clivus–axis angle, clivus–atlas angle, and atlas–axis angle in the upright position—suggests that hypermobility of the occipitoatlantal and atlantoaxial joints contributes to retroodontoid pannus formation and symptoms referable to basilar impression.

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Takeo Goto, Kenji Ohata, Toshihiro Takami, Misao Nishikawa, Akimasa Nishio, Michiharu Morino, Naohiro Tsuyuguchi and Mitsuhiro Hara

Object. The authors evaluated an alternative method to avoid postoperative posterior tethering of the spinal cord following resection of spinal ependymomas.

Methods. Twenty-five patients with spinal ependymoma underwent surgery between 1978 and 2002. There were 16 male and nine female patients whose ages at the time of surgery ranged from 14 to 64 years (mean 41.8 years). The follow-up period ranged from 6 to 279 months (mean 112.4 months). In the initial 17 patients (Group A), the procedure to prevent arachnoidal adhesion consisted of the layer-to-layer closure of three meninges and laminoplasty. In the subsequently treated eight patients (Group B), the authors performed an alternative technique that included pial suturing, dural closure with Gore-Tex membrane—assisted patch grafting, and expansive laminoplasty. In Group A, postoperative adhesion was radiologically detected in eight cases (47%), and delayed neurological deterioration secondary to posterior tethering of the cord was found in five cases. In Group B, there was no evidence of adhesive posterior tethering or delayed neurological deterioration. A significant intergroup statistical difference was demonstrated for radiologically documented posterior tethering (p < 0.05, Fisher exact test). Moreover, patients with radiologically demonstrated posterior tethering suffered a significant delayed neurological functional deterioration (p < 0.01, Fisher exact test).

Conclusions. This new technique for closure of the surgical wound is effective in preventing of postoperative posterior spinal cord tethering after excision of spinal ependymoma.

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Takeo Goto, Kenji Ohata, Toshihiro Takami, Misao Nishikawa, Naohiro Tsuyuguchi, Michiharu Morino, Yasuhiro Matusaka, Akimasa Nishio, Yuichi Inoue and Mitsuhiro Hara

Object. The authors describe a new surgical technique for cervical laminoplasty that was performed in 25 patients. The posterior elements along with the various ligaments are removed en bloc and are stabilized in a lift-up position by placing hydroxyapatite (HA) laminar spacers and titanium miniplates and screws. The procedure and clinical results are discussed.

Methods. The posterior spinal elements, including the lamina(e), spinous process(es), and various attached ligaments, are removed en bloc by incising the lamina in its lateral aspect. Trapezoid-shaped HA spacers are placed between the cut ends of the laminae or between the laminae and lateral masses bilaterally at each level. Malleable titanium miniplates and screws are used for fixation of the spacers. The fixation of transected laminae was judged to be successful. Postoperative care included application of a soft neck collar for 1 week but no further restriction of activity. Surgery-related outcome was assessed in the 21 patients who attended more than 6 months of follow up after laminoplasty. There were 18 men and three women who ranged in age from 27 to 81 years. Cervical stenotic myelopathy was demonstrated in 15 patients who underwent decompressive and expansive laminoplasty, and spinal tumors were documented in six patients who underwent a nonexpansive laminoplasty. Postoperative and follow-up computerized tomography scans demonstrated no hardware failure. Bone formation around the spacers was observed either at 6- or at 9-month follow-up examination in all 21 patients. Fusion of the reconstructed laminae was found to be completed at 12 months in all 18 patients able to attend follow up for this duration. Spinal alignment and the range of motion of the cervical spine were well preserved. In patients with stenotic cervical myelopathy, neurological and anatomical outcome of canal expansion were satisfactory.

Conclusions. This technique enables rigid laminoplasty while maintaining anatomical and biomechanical integrity of posterior elements of cervical spine. Expansive and nonexpansive laminoplasty procedures are possible.

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Toshihiro Takami, Kenji Ohata, Misao Nishikawa, Takeo Goto, Yuzo Terakawa, Yuichi Inoue, Kenichi Wakasa and Mitsuhiro Hara

✓ The authors discuss the utility of anterior transposition of the oculomotor nerve from the lateral wall of the cavernous sinus to widen the corridor posterior to the cisternal segment of the oculomotor nerve; this allows exposure of the anterolateral surface of the midbrain. This additional exposure was successfully used for the resection of a large calcified cavernoma in the upper brainstem of a 67-year-old woman who had presented with sudden onset of left hemiparesis and oculomotor palsy. The patient's postoperative course was uneventful and she displayed symptomatic improvement.