Cervical laminectomy and micro resection of the posterior venous plexus in Hirayama disease

Brandicourt, Pierre; Sol, José Cuesta Del; Aldéa, Sorin; Bonneville, Fabrice; Pascal, Pierre; Brauge, David

doi:10.1016/j.neuchi.2018.04.004

Cited by 25 publications

(27 citation statements)

References 21 publications

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“…20 Since males tend to have faster rates of vertebral column growth than females, this disproportionate-growth theory can also explain the male preponderance seen in HD. 20 Non dural-based theories of HD include compression by anteriorly located osteophytic bars, 17 flexion-induced venous stagnation, 16,21,22 and underlying instability. 6,23 The genesis of the latter hypothesis is related to the effectiveness of collar wear and reports of abnormal alignment 24 and range of neck flexion 25 rather than any definite radiological evidence of instability.…”

Section: Pathogenesis Of Hd-theories and Substratesmentioning

confidence: 99%

“…Fusion procedures, anterior or posterior, arrest segmental movement, and prevent the dural migration and cord compression during flexion. Duraplasty after laminectomy or laminoplasty, 21,[36][37][38][39][40] on the other hand, directly addresses the pathology of the tight dural canal. An added advantage of a posterior procedure is that it also addresses the flexion-induced dilatation of the epidural venous plexus seen in HD.…”

Section: Surgical Options In Hdmentioning

confidence: 99%

“…A literature search revealed that while over 300 cases of HD 6,8,26 were managed with anterior or posterior fusion, only 16 patients were treated with duraplasty without fusion (►Table 5). 16,21,[36][37][38][39][40] Ours is the first large series that critically evaluates CD as a surgical option in HD.…”

Section: Selection Of Surgical Proceduresmentioning

confidence: 99%

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Outcomes after Cervical Duraplasty for Monomelic Amyotrophy (Hirayama Disease): Results of a Case–Control Study of 60 Patients

Thakar

Arun

Rajagopal

et al. 2021

JNRP

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Background The optimal management and the role of surgery in monomelic amyotrophy, also known as Hirayama disease (HD), remain controversial. In the largest series of patients with HD managed by cervical duraplasty (CD), this study compares the outcomes between conservative and surgical management. Methods A retrospective case–control study was performed on 60 patients with HD. The cases consisted of 30 patients who underwent CD, and 30 age- and sex-matched controls who managed with long-term collar wear. Clinical improvement was recorded using two patient-reported outcome measures—the Odom's scale and a self-rated hand grip strength score. Median and ulnar compound muscle action potential (CMAP) amplitudes and various magnetic resonance imaging parameters were recorded. Results None of the controls experienced clinical improvement at follow-up; 90% remained status quo at 24.33 ± 9.38 months. Seventy-six percent of the operated patients demonstrated clinical improvement at 29.61 ± 12.78 months, and a majority of them reported moderately better grip strength. The preoperative anteroposterior diameter (APD) of the surgical group improved from 0.74 ± 0.13 to 0.86 ± 0.21 (p = 0.01), and the cord demonstrated a significant decrease in forward migration on flexion (0.20 ± 0.11 and 0.03 ± 0.07 preoperatively and at follow-up, respectively, p < 0.0001). Patients with clinical improvement demonstrated significantly better mean APD and median and ulnar CMAP values than those who did not improve (p < 0.0001). The mean cervical alignment did not change significantly at follow-up (p = 0.13). Conclusions This study shows that CD can significantly alter the clinical course of patients with progressive symptoms of HD. This approach successfully arrests the forward migration of the cervical cord on flexion and promotes morphological recovery of the cord. A majority of the patients undergoing CD demonstrate clinical and electrophysiological improvement at mid-term follow-up.

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Section: Pathogenesis Of Hd-theories and Substratesmentioning

confidence: 99%

Section: Surgical Options In Hdmentioning

confidence: 99%

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Outcomes after Cervical Duraplasty for Monomelic Amyotrophy (Hirayama Disease): Results of a Case–Control Study of 60 Patients

Thakar

Arun

Rajagopal

et al. 2021

JNRP

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“…However, when surgery is indicated, for patients with severe clinical symptoms or rapid progression, the ideal treatment is questionable. Anterior cervical discectomy and fusion is probably the most accepted treatment but cervical decompressive laminectomy and coagulation of the posterior epidural plexus without fixation has also been reported [21,22].…”

Section: Dynamic Mri Studiesmentioning

confidence: 99%

Dynamic Cord Compression Causing Cervical Myelopathy

et al. 2019

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Due to the highly mobile nature of the cervical spine, and the fact that most magnetic resonance imagings (MRIs) and computed tomography scans are obtained only in one single position, dynamic cord compression can be an elusive diagnosis that is often missed and not well-understood. In this context, dynamic MRI (dMRI) has been utilized to improve the diagnostic accuracy of cervical stenosis. We performed a literature review on dynamic cord compression in the context of cervical spondylotic myelopathy (CSM), with particular emphasis on the role of dMRI. Cadaveric studies report that the spinal cord lengthens in flexion and the spinal canal dimension increases, whereas the spinal cord relaxes and shortens in extension and the spinal canal decreases. These changes may lead to biomechanical stress in the spinal cord with movement, especially in patients with critical cervical stenosis. The majority of the studies using dMRI in CSM reported that this imaging modality is more sensitive at detecting cervical cord compression compared to routine MRIs done in a neutral position, especially with the neck in extension. Dynamic MRI was also useful to diagnose dynamic cervical cord compression after laminectomies in patients with clinical deterioration without evident cord compression on neutral static MRI. Finally, dMRI is more sensitive in detecting stenosis in patients with CSM than in those with ossification of the posterior longitudinal ligament (OPLL), likely because OPLL patients often have a more limited range of motion than CSM patients. Thus, dMRI is a promising new tool that can help spine surgeons in diagnosing and treating CSM.

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“…Disproportionate growth between the vertebral column and thecal sac and a tight, inflexible dura have been proposed to explain the loss of dural attachment and forward displacement [2, 3, 5, 6, 8]. Venous stasis and engorgement within the posterior epidural plexus have also been proposed to contribute and/or exacerbate the anterior displacement of the dura [3, 9].…”

Section: Introductionmentioning

confidence: 99%

Hirayama Disease in a Patient with a History of Late-Onset Symptomatic Vein of Galen Aneurysmal Malformation

et al. 2021

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Introduction: The purpose of this report is to present a rare case of Hirayama disease (HD) in a patient with a history of late-onset symptomatic vein of Galen aneurysmal malformation (VGAM). This report may provide new insights into the pathophysiology of HD, a rare disorder consisting of insidious onset of unilateral weakness and atrophy of the forearm and intrinsic hand muscles. These symptoms are believed to result from cervical myelopathy affecting the anterior horn cells due to abnormal compressive forces on the spinal cord from adjacent anatomical structures (i.e., dura and/or epidural veins), but this has not been proven. VGAM is a rare congenital cerebral vascular malformation, consisting of high-flow arteriovenous shunting between a persistent median prosencephalic vein and arterial feeders, which most frequently presents in the early neonatal period with congestive heart failure. Case Presentation: We report the case of an otherwise healthy boy who presented with heart failure due to VGAM at 7 years and subsequently presented at 14 years with left-sided HD. His cervical MRI with neck flexion revealed enlarged epidural veins at the C5–C6 spinal level with anterior, leftward displacement of the posterior dura and spinal cord as well as left hemicord flattening and/or atrophy at this level. He underwent successful surgical treatment by C5–C6–C7 osteoplastic laminotomies and tenting and expansile autologous duraplasty, during which enlarged, engorged epidural veins were confirmed and coagulated. This halted the progression of his left hand weakness and atrophy and allowed significant functional improvement. Postoperative catheter angiogram showed no anatomical connection between the persistent VGAM and the engorged epidural veins, and genetic testing revealed no genetic predisposition of vascular malformations. Discussion/Conclusion: In this patient, a combined surgical technique was performed that included epidural venous plexus coagulation with posterior autologous duraplasty and dural fixation using tenting sutures. This combined approach led to a favorable clinical and radiographic outcome with no complications or limitations and has not been previously proposed in the literature, to our knowledge. Although not completely ruled out, we found no angiographic connection or genetic predisposition to suggest there is a pathophysiological link between HD and VGAM.

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Cervical laminectomy and micro resection of the posterior venous plexus in Hirayama disease

Abstract: Posterior cervical decompression with coagulation of epidural venous plexus is a technique that seems effective in Hirayama disease in young subjects. It effectively treats patients by avoiding permanent cervical fixation.

Cited by 25 publications

References 21 publications

Outcomes after Cervical Duraplasty for Monomelic Amyotrophy (Hirayama Disease): Results of a Case–Control Study of 60 Patients

Outcomes after Cervical Duraplasty for Monomelic Amyotrophy (Hirayama Disease): Results of a Case–Control Study of 60 Patients

Dynamic Cord Compression Causing Cervical Myelopathy

Hirayama Disease in a Patient with a History of Late-Onset Symptomatic Vein of Galen Aneurysmal Malformation

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