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Keisuke Takai and Makoto Taniguchi

OBJECTIVE

Dorsal root entry zone (DREZ) lesioning has been the most effective surgical treatment for the relief of intractable pain due to root avulsion injury, but residual pain and a decrease in pain relief in the follow-up period have been reported in 23%–70% of patients. Based on pain topography in the most recent studies on neuropathic pain, the authors modified the conventional DREZ lesioning procedure to improve clinical outcomes. The presumed rationale for this procedure is to eliminate the spontaneous discharges of neurons in the superficial spinal dorsal horn as well as wide dynamic range neurons in the deep spinal dorsal horn.

METHODS

Ten patients with avulsion-related pain underwent surgery between 2011 and 2015. The surgical procedure was described and postoperative pain relief was assessed as follows: excellent (residual pain never exceeded 3 on the visual analog scale [VAS] without medication), good (residual pain never exceeded 5 on the VAS with medication), and poor (residual pain was greater than 5 with medication). Specific perioperative complications were assessed.

RESULTS

The aim of this surgical procedure was to destroy the deeper layers of the posterior horn of spinal gray matter, which was in contrast to the procedures of Nashold and Sindou, which were to destroy the superficial layers. All patients achieved excellent (n = 7, pain relief without medication) or good (n = 3, pain relief with medication) pain relief postoperatively, and the recurrence of pain was not reported in any patients (median 29 months after surgery, range 12–64 months). Nine patients (90%) achieved complete pain relief (a score of 0 or 1 on the VAS) with or without medication. No surgical site complications such as infection or CSF leakage were noted. No motor deficit was observed in any patient. A sensory deficit was observed in 2 patients and disappeared within 1 month in 1 patient. New pain at the adjacent level of DREZ lesioning was observed in 3 patients and disappeared within 1 month in 2 patients. In the other patient, new pain persisted and required analgesics.

CONCLUSIONS

These preliminary results demonstrated that total and persistent global pain relief was achieved with the modified DREZ lesioning procedure in 90% of patients without major neurological deficits. The clinical improvements achieved by this modified surgical procedure support the hypothesis that not only the superficial layers, but also deeper layers of the spinal dorsal horn are associated with intractable pain due to root avulsion injury.

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Keisuke Takai, Takashi Komori and Makoto Taniguchi

OBJECT

The microvascular anatomy of spinal dural arteriovenous fistulas (AVFs), especially the relationships of the vessels with the dura mater, has yet to be angiographically demonstrated in detail and proven histologically.

METHODS

From January 2012 through April 2014, a total of 7 patients with spinal dural AVFs in the thoracic region underwent open microsurgical obliteration at Tokyo Metropolitan Neurological Hospital. The microvascular anatomy of spinal dural AVFs was comprehensively assessed by using advanced microangiography, including 3D computer graphics and intraoperative indocyanine green video angiography, and by histological findings.

RESULTS

The 2 microangiography techniques revealed the spatial course and in vivo blood flow of the meningeal vessels and their relationships with the dura mater in sufficient detail. The meningeal branch of the intercostal artery split into multiple meningeal vessels on the outer dural surface adjacent to the root sleeve. After crossing the dura mater to the inner dural surface, these vessels gathered and joined a single intradural draining vessel. On the inner dural surface, the single draining vessel was fed by the surrounding multiple meningeal vessels, which appeared to be caput medusae.

Histological findings revealed that the structure of the meningeal branch of the intercostal artery corresponded to that of a normal artery. The structure of intradural draining vessels corresponded to that of a vein modified by retrograde arterial inflow. On the inner dural surface, more than 1 meningeal artery gathered and joined with the proximal radiculomedullary vein.

CONCLUSIONS

Spinal dural AVFs are located on the inner dural surface, where multiple direct AV connections between more than 1 meningeal feeding artery and a single proximal radiculomedullary vein occur at the site where the vein connects to the dura mater.

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Hirotaka Ito, Keisuke Takai and Makoto Taniguchi

Object

Hirayama disease, juvenile muscular atrophy of the distal upper extremity, is a rare type of cervical flexion segmental myelopathy and its etiology is still being debated. Two theories have been proposed: a “contact pressure” theory and “tight dural canal in flexion” theory. Previously reported treatments, including conservative neck collar therapy and surgical spinal fusion, used fixation of the cervical spine with the aim of avoiding contact pressure between the cord and anterior structures. On the other hand, treatment by duraplasty without spinal fusion has also been used, which aims at decompressing a tight dural canal in flexion by preventing abnormal forward displacement of the posterior dura mater without restricting cervical motion in young patients.

The authors developed a new surgical approach for treating a tight dural canal in flexion in patients with Hirayama disease: cervical duraplasty with tenting sutures via laminoplasty without spinal fusion. With this treatment they aimed to both decompress the spinal cord and preserve as much cervical motion as possible. The purpose of this study was to assess the clinical outcomes of patients who underwent this new surgical procedure and to investigate the etiology of Hirayama disease.

Methods

Six male patients (age range 17–23 years) with Hirayama disease underwent surgery between 2006 and 2012. The pre- and postoperative anteroposterior diameters of the dural canal in the flexed neck position, grip strength of the bilateral upper extremities, cervical alignment (C2–7), and cervical local flexion range of motion were compared. The presence or absence of surgical complications was assessed. To investigate the comparison group of Hirayama disease treated with spinal decompression, the PubMed database was searched for all relevant Englishlanguage case reports and series published between 1990 and 2013.

Results

The postoperative anteroposterior diameters of the dural canal were significantly expanded in the flexed neck position (7.2 ± 2.2 mm preoperatively vs 9.8 ± 1.7 mm postoperatively, p = 0.001). Grip strength of the upper extremities significantly improved bilaterally (20 ± 14 kg preoperatively vs 26 ± 15 kg postoperatively, p = 0.001). No significant difference was observed between pre- and postoperative cervical alignment in the neutral neck position (7.7° ± 8.1° preoperatively vs 9.0° ± 7.7° postoperatively, p = 0.74) or the cervical local flexion angle in the flexed neck position at the corresponding level of laminoplasty (16.6° ± 5.1° preoperatively vs 15.0° ± 9.4° postoperatively, p = 0.8). No surgical complications were noted, except for transient CSF leakage, which was resolved after lumbar drainage. The systematic review identified 37 cases from 7 reports: 26 with spinal fusion only, 5 with duraplasty without fusion, and 6 with combined duraplasty and fusion. In the largest series, in which 12 cases were treated with anterior fusion, cervical alignment was maintained, but local flexion motion was significantly decreased as a result of fixation. Although significant improvements in or stabilization of grip strength occurred in all 7 reported studies regardless of decompression procedures, one major delayed surgical complication was noted in a patient treated with anterior fusion. The patient developed severe kyphotic changes, which required reconstruction surgeries.

Conclusions

Cervical duraplasty with tenting sutures via laminoplasty prevented abnormal forward displacement of the posterior dura mater while preserving normal anterior structures and flexion motion of the cervical spine without major surgical complications. The clinical improvements achieved by the authors' method support evidence that a tight dural canal in flexion largely contributes to segmental myelopathy in patients with Hirayama disease.

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Hiroki Yoshida, Keisuke Takai and Makoto Taniguchi

Object

The purpose of this study was to describe significant CT myelography findings for determination of the leak site and outcome of targeted epidural blood patch (EBP) in patients with spontaneous CSF leaks.

Methods

During 2005–2013, spontaneous CSF leaks were diagnosed for 12 patients with orthostatic headaches. The patients received targeted EBP on the basis of CT myelography assessments.

Results

Computed tomography myelograms revealed ventral extradural collection of contrast medium distributed over multiple spinal levels (average 16 levels). Intraforaminal contrast medium extravasations were observed at multiple spinal levels (average 8.2 levels). For 8 (67%) of 12 patients, spinal lesions were noted around the thecal sac and included calcified discs with osteophytes, an ossified posterior longitudinal ligament, and an ossified yellow ligament; lesions were mostly located ventral to the thecal sac and were in close contact with the dura mater. The levels of these spinal lesions were considered potential leak sites and were targeted for EBP. For the remaining 4 patients who did not have definite spinal lesions around the thecal sac, leak site determination was based primarily on the contrast gradient hypothesis. The authors hypothesized that the concentration of extradural contrast medium would be the greatest and the same as that of intradural contrast medium at the leak site but that it would decrease with increased distance from the leak site according to the contrast gradient.

Epidural blood patch was placed at the level of spinal lesions and/or of the greatest and same concentration of contrast medium between the intradural and extradural spaces. For 10 of the 12 patients, the orthostatic headaches decreased significantly within a week of EBP and disappeared within a month. For the remaining 2 patients, headaches persisted and medical treatment was required for several months. For 3 patients, thick chronic subdural hematomas caused severe headaches and/or disturbed consciousness because of the mass effect of the hematomas, which were removed by bur hole drainage surgery. For 1 patient, bur hole drainage before EBP on the day of admission to hospital resulted in subdural tension pneumocephalus. The patient's headache immediately disappeared after EBP, and the hematoma did not recur. The other 2 patients underwent EBP followed by bur hole drainage, which resulted in improvements and disappearance of the hematomas. Over the follow-up period (mean 39 months), no CSF leaks or chronic subdural hematomas had recurred in any patient after EBP; by the final follow-up visit, all patients had returned to their jobs.

Conclusions

The most significant finding of this study was that spinal ventral calcified or ossified lesions, which may be associated with a dural tear, were present in approximately 70% of patients. Targeted EBP to these lesions resulted in good outcomes.

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Katsushige Watanabe, Nobuhito Saito, Makoto Taniguchi, Takaaki Kirino and Tomio Sasaki

Object. The frequency, nature, and history of subjective taste disturbance before and after vestibular schwannoma (VS) surgery was investigated.

Methods. Personal interviews were conducted in 108 patients with unilateral VS. Abnormalities in taste perception, either a significant reduction or a change in character, were experienced by 31 patients (28.7%) before surgery and by 37 (34.3%) after tumor removal. Preoperative taste disturbance worsened after surgery in five (16.1%) of the 31 patients, remained unchanged in eight (25.8%), improved in two (6.5%), and became normal in 16 (51.6%). Taste disturbance occurred postoperatively in 22 (28.6%) of 77 patients who had experienced no preoperative taste disturbance. The mean onset of the abnormality after resection was 1.1 ± 1.7 months. Postoperative taste disturbance resolved in 24 of the 37 patients (64.9%) within 1 year after onset.

Conclusions. Subjective taste disturbance was common before and after VS removal, and the natural history of this condition was very variable in the pre- and postoperative periods. All patients who undergo surgery for VS should receive appropriate counseling about the likelihood and course of postoperative complications, including dysfunction of the sensory component of the facial nerve.

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Keisuke Takai, Hiroki Kurita, Takayuki Hara, Kensuke Kawai and Makoto Taniguchi

OBJECTIVE

The microvascular anatomy of spinal perimedullary arteriovenous fistulas (AVFs) is more complicated than that of dural AVFs, and occlusion rates of AVF after open microsurgery or endovascular embolization are lower in patients with perimedullary AVFs (29%–70%) than they are in those with dural AVF (97%–98%). Reports of intraoperative blood flow assessment using indocyanine green (ICG) video angiography in spinal arteriovenous lesions have mostly been for spinal dural AVFs. No detailed reports on spinal perimedullary AVFs are available.

METHODS

Participants were 11 patients with spinal perimedullary AVFs (Type IVa in 5 patients, Type VIb in 4, and Type IVc in 2). Intraoperative ICG video angiography was assessed by measuring the number of cases in which this modality was judged essential by the surgeon to correctly occlude the fistula.

RESULTS

In all patients, arterial feeders were identified and intravenous ICG video angiography was performed before and after blocking the feeders. In one patient, selective intraarterial ICG video angiography was also performed. The findings provided by ICG video angiography significantly changed the surgical procedure in 4 of 11 patients (36%). Postoperatively, complete occlusion of the AVF was achieved in 10 of the 11 patients (91%).

CONCLUSIONS

Intraoperative ICG video angiography can have a significant impact on deciding surgical strategy in the microsurgical treatment of spinal perimedullary AVF.

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Keisuke Takai, Takashi Komori, Manabu Niimura and Makoto Taniguchi

In most patients with superficial siderosis of the CNS, the exact source of bleeding remains unknown because of a lack of objective surgical data. The authors herein describe the case of a 58-year-old man with superficial siderosis of the CNS. The patient also had spinal CSF leakage due to a spinal dural defect. Repair surgery for the dural defect was performed using posterior laminoplasty with a transdural approach without spinal fixation. During repair surgery, the bleeding source was found to be the epidural vein around the defect. The intraoperative and histological results of the present case suggest that epidural veins exposed to CSF represent a chronic bleeding source in patients with superficial siderosis of the CNS complicated by CSF leakage. Dural repair surgery may result in discontinuation of the CSF leaks, resolution of the epidural CSF collection, and cessation of chronic epidural bleeding.

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Junichi Yokosuka, Keisuke Takai, Takashi Komori and Makoto Taniguchi

Superficial siderosis of the CNS is a rare disease characterized by the deposition of hemosiderin in the subpial layers of the CNS as the result of chronic subarachnoid bleeding. The arrest of bleeding is important for preventing the progression of this disease; however, the exact source of bleeding remains unknown in most cases because of a lack of objective surgical data. The authors of this report have described a unique case of superficial siderosis following cervical laminectomy and autograft fusion for the removal of a spinal schwannoma; the bleeding source was verified by intraoperative and histopathological findings. The patient exhibited no obvious neurological deficits, such as hearing loss or cerebellar ataxia, when the superficial siderosis was diagnosed, although there were the social impairments associated with schizophrenia. During repair surgery, the bleeding source was revealed as the bone marrow vasculature of the remaining vertebral arch, and not the fragile vessels at the dural defect or the residual tumor. Chronic bleeding was stopped before the onset of irreversible neurological deficits in this case. Bone marrow exposed to the intrathecal space may represent a chronic bleeding source in patients with superficial siderosis following CNS surgery including laminectomy or craniotomy. The following recommendations have been proposed for superficial siderosis of the CNS from both a preventative and a therapeutic perspective: 1) During CNS surgery, neurosurgeons should make every effort to prevent exposing bone marrow to the intrathecal space to avoid the risk of chronic subarachnoid bleeding. 2) In the case of a large dural defect and pseudomeningocele following CNS surgery, bone marrow around the dural defect should be considered as the bleeding source of superficial siderosis, and such cases should undergo revision surgery before the progression of this disease.

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Shoko Yoshimoto, Keisuke Takai, Koichi Takahashi, Toshio Yasui and Makoto Taniguchi

Gorham-Stout disease (GSD) is an intractable disease characterized by massive osteolysis caused by abnormal lymphangiogenesis in bone. In rare cases of GSD, CSF abnormalities develop. The authors present the case of a 19-year-old woman with GSD presenting with orthostatic headache due to intracranial hypotension (5 cm H2O). The clinical course of this case was very unusual. Orthostatic headache was associated with a CSF leak from the thigh after pathological fractures of the femur and pelvis. The chronic CSF leak led to acquired Chiari malformation (CM) with syringomyelia. After an epidural blood patch, her neurological status improved; however, after the complete arrest of the CSF leak from the thigh, she presented with severe nonpostural headache and progressive visual acuity loss with optic papilledema. A ventriculoperitoneal shunt was placed to treat intracranial hypertension (50 cm H2O). Headache improved and optic papilledema decreased after shunt surgery. This case shows that dynamic CSF abnormalities may lead to reversible CM in patients with GSD. Sealing a CSF leak rather than performing suboccipital decompression is recommended for acquired CM resulting from a CSF leak.

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Tomio Sasaki, Makoto Taniguchi, Ichiro Suzuki and Takaaki Kirino

✓ The authors report a new technique for en bloc petrosectomy using a Gigli saw as an alternative to drilling the petrous bone in the combined supra- and infratentorial approach or the transpetrosal—transtentorial approach. It is simple and easy and avoids postoperative cosmetic deformity. This technique has been performed in 11 petroclival lesions without injuring the semicircular canals, the cochlea, or the facial nerve.