Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study
Abstract Background Although the clinical efficacy of laminoplasty in adult cervical spondylotic myelopathy or ossification of posterior longitudinal ligament has been frequently reported, there are only few reports of laminoplasty for patients with lysosome storage diseases (LSDs). Therefore, this...
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doaj-035a053417bf47f9bb03cc0fe52456002021-10-03T11:37:27ZengBMCOrphanet Journal of Rare Diseases1750-11722021-09-0116111010.1186/s13023-021-02031-9Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort studyHidetomi Terai0Koji Tamai1Masatoshi Hoshino2Hiromitsu Toyoda3Akinobu Suzuki4Shinji Takahashi5Yusuke Hori6Akito Yabu7Hiroaki Nakamura8Department of Orthopaedic Surgery, Osaka City University Graduate School of MedicineDepartment of Orthopaedic Surgery, Osaka City University Graduate School of MedicineDepartment of Orthopaedic Surgery, Osaka City University Graduate School of MedicineDepartment of Orthopaedic Surgery, Osaka City University Graduate School of MedicineDepartment of Orthopaedic Surgery, Osaka City University Graduate School of MedicineDepartment of Orthopaedic Surgery, Osaka City University Graduate School of MedicineDepartment of Orthopaedic Surgery, Osaka City University Graduate School of MedicineDepartment of Orthopaedic Surgery, Osaka City University Graduate School of MedicineDepartment of Orthopaedic Surgery, Osaka City University Graduate School of MedicineAbstract Background Although the clinical efficacy of laminoplasty in adult cervical spondylotic myelopathy or ossification of posterior longitudinal ligament has been frequently reported, there are only few reports of laminoplasty for patients with lysosome storage diseases (LSDs). Therefore, this study aimed to report the midterm clinical and radiological outcomes of patients with LSDs after cervical laminoplasty. Methods Six patients with LSD who underwent laminoplasty with/without C1 laminectomy for cervical myelopathy were enrolled. Clinical evaluations, including the cervical Japanese Orthopedic Association (cJOA) score and visual analog scale (VAS) scores for upper extremity numbness, and radiographic parameters, including C2–C7 lordotic angle, atlanto-dens interval (ADI), and ⊿ADI, were evaluated preoperatively, at 2 years postoperatively, and at the final follow-up. Results Five patients had mucopolysaccharidoses (type I: n = 1, II: n = 3, VII: n = 1) and one patient had mucolipidoses type III. The mean age of patients at surgery was 27.5 years, and the mean postoperative follow-up period was 61 months. All mucopolysaccharidoses cases required C1 posterior arch resection with C2–C7 laminoplasty. No critical complications were observed postoperatively. There were no significant differences in C2–C7 angle (p = 0.724) and ⊿ADI (p = 0.592) between the preoperative and final follow-ups. The cJOA score and VAS for numbness significantly improved at the final follow-up (p = 0.004 and p = 0.007, respectively). Conclusions The cervical myelopathy in patients with LSD could be safely and effectively treated with laminoplasty with/without C1 posterior arch resection after excluding patients with atlantoaxial instability. Atlantoaxial stability and symptom improvement could be maintained at an average of 5 years postoperatively.https://doi.org/10.1186/s13023-021-02031-9MucopolysaccharidosesMucolipidosesLaminoplastyCervicalMyelopathyLysosome storage diseases |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Hidetomi Terai Koji Tamai Masatoshi Hoshino Hiromitsu Toyoda Akinobu Suzuki Shinji Takahashi Yusuke Hori Akito Yabu Hiroaki Nakamura |
spellingShingle |
Hidetomi Terai Koji Tamai Masatoshi Hoshino Hiromitsu Toyoda Akinobu Suzuki Shinji Takahashi Yusuke Hori Akito Yabu Hiroaki Nakamura Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study Orphanet Journal of Rare Diseases Mucopolysaccharidoses Mucolipidoses Laminoplasty Cervical Myelopathy Lysosome storage diseases |
author_facet |
Hidetomi Terai Koji Tamai Masatoshi Hoshino Hiromitsu Toyoda Akinobu Suzuki Shinji Takahashi Yusuke Hori Akito Yabu Hiroaki Nakamura |
author_sort |
Hidetomi Terai |
title |
Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study |
title_short |
Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study |
title_full |
Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study |
title_fullStr |
Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study |
title_full_unstemmed |
Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study |
title_sort |
clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study |
publisher |
BMC |
series |
Orphanet Journal of Rare Diseases |
issn |
1750-1172 |
publishDate |
2021-09-01 |
description |
Abstract Background Although the clinical efficacy of laminoplasty in adult cervical spondylotic myelopathy or ossification of posterior longitudinal ligament has been frequently reported, there are only few reports of laminoplasty for patients with lysosome storage diseases (LSDs). Therefore, this study aimed to report the midterm clinical and radiological outcomes of patients with LSDs after cervical laminoplasty. Methods Six patients with LSD who underwent laminoplasty with/without C1 laminectomy for cervical myelopathy were enrolled. Clinical evaluations, including the cervical Japanese Orthopedic Association (cJOA) score and visual analog scale (VAS) scores for upper extremity numbness, and radiographic parameters, including C2–C7 lordotic angle, atlanto-dens interval (ADI), and ⊿ADI, were evaluated preoperatively, at 2 years postoperatively, and at the final follow-up. Results Five patients had mucopolysaccharidoses (type I: n = 1, II: n = 3, VII: n = 1) and one patient had mucolipidoses type III. The mean age of patients at surgery was 27.5 years, and the mean postoperative follow-up period was 61 months. All mucopolysaccharidoses cases required C1 posterior arch resection with C2–C7 laminoplasty. No critical complications were observed postoperatively. There were no significant differences in C2–C7 angle (p = 0.724) and ⊿ADI (p = 0.592) between the preoperative and final follow-ups. The cJOA score and VAS for numbness significantly improved at the final follow-up (p = 0.004 and p = 0.007, respectively). Conclusions The cervical myelopathy in patients with LSD could be safely and effectively treated with laminoplasty with/without C1 posterior arch resection after excluding patients with atlantoaxial instability. Atlantoaxial stability and symptom improvement could be maintained at an average of 5 years postoperatively. |
topic |
Mucopolysaccharidoses Mucolipidoses Laminoplasty Cervical Myelopathy Lysosome storage diseases |
url |
https://doi.org/10.1186/s13023-021-02031-9 |
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