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Clinical outcomes of laminoplasty for patients with lysosomal storage disease including mucopolysaccharidosis and mucolipidoses: a retrospective cohort study.

Authors :
Terai, Hidetomi
Tamai, Koji
Hoshino, Masatoshi
Toyoda, Hiromitsu
Suzuki, Akinobu
Takahashi, Shinji
Hori, Yusuke
Yabu, Akito
Nakamura, Hiroaki
Source :
Orphanet Journal of Rare Diseases; 9/28/2021, Vol. 16 Issue 1, p1-10, 10p
Publication Year :
2021

Abstract

<bold>Background: </bold>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.<bold>Methods: </bold>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.<bold>Results: </bold>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).<bold>Conclusions: </bold>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. [ABSTRACT FROM AUTHOR]

Details

Language :
English
ISSN :
17501172
Volume :
16
Issue :
1
Database :
Complementary Index
Journal :
Orphanet Journal of Rare Diseases
Publication Type :
Academic Journal
Accession number :
152677100
Full Text :
https://doi.org/10.1186/s13023-021-02031-9