Congenital double-level cervical spondylolysis: a case report and review of the literature

Norio Yamamoto, Takaaki Miki, Yoshihisa Nasu, Akihiro Nishiyama, Tomoyuki Dan’ura, Yuzuru Matsui, Toshihumi Ozaki

Research output: Contribution to journalArticle

Abstract

Purpose: We report a rare Japanese female who was affected with three genetic-linked diseases: double-level cervical bilateral spondylolysis in association with spina bifida occulta, cleft lip and monostotic fibrous dysplasia of the right proximal femur. The case was considered to be congenital in origin. We also review the pertinent literature of cervical spondylolysis, with a focus on the pathogenesis of multiple-level cervical spondylolysis. Methods: A 40-year-old female presented with progressive clumsiness and numbness of the hands. Japanese Orthopedic Association (JOA) score for the cervical spine was 14.5. Plain radiographs of the cervical spine showed bilateral spondylolysis of the articular mass portion, with an adjacent dysplastic change and spina bifida occulta of C4 and C5. Cervical laminoplasty from C4 to C6 was performed. Results: The postoperative course was uneventful, and the patient had some recovery of muscle power and sensation, with JOA score improving to 15.5. At the 8-year follow-up, the patient had no recurrence of symptoms, but did show kyphotic and degenerative changes at the C4/5 and C5/6 level with no apparent instability. Conclusions: This case is a rare presentation of bilateral cervical spondylolysis involving C4 and C5, presumably congenital, accompanied by combined dysplastic changes of the cervical spine, cleft lip, and fibrous dysplasia, possibly through an error involving an ossification center during the embryonic stage.

Original languageEnglish
Pages (from-to)1-5
Number of pages5
JournalEuropean Spine Journal
DOIs
Publication statusAccepted/In press - Feb 28 2017

Fingerprint

Spondylolysis
Spina Bifida Occulta
Spine
Cleft Lip
Orthopedics
Monostotic Fibrous Dysplasia
Inborn Genetic Diseases
Hypesthesia
Osteogenesis
Femur
Hand
Joints
Recurrence
Muscles

Keywords

  • Cervical spondylolysis
  • Cleft lip
  • Congenital
  • Fibrous dysplasia
  • Multiple-level cervical spondylolysis

ASJC Scopus subject areas

  • Surgery
  • Orthopedics and Sports Medicine

Cite this

Congenital double-level cervical spondylolysis : a case report and review of the literature. / Yamamoto, Norio; Miki, Takaaki; Nasu, Yoshihisa; Nishiyama, Akihiro; Dan’ura, Tomoyuki; Matsui, Yuzuru; Ozaki, Toshihumi.

In: European Spine Journal, 28.02.2017, p. 1-5.

Research output: Contribution to journalArticle

Yamamoto, Norio ; Miki, Takaaki ; Nasu, Yoshihisa ; Nishiyama, Akihiro ; Dan’ura, Tomoyuki ; Matsui, Yuzuru ; Ozaki, Toshihumi. / Congenital double-level cervical spondylolysis : a case report and review of the literature. In: European Spine Journal. 2017 ; pp. 1-5.
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AB - Purpose: We report a rare Japanese female who was affected with three genetic-linked diseases: double-level cervical bilateral spondylolysis in association with spina bifida occulta, cleft lip and monostotic fibrous dysplasia of the right proximal femur. The case was considered to be congenital in origin. We also review the pertinent literature of cervical spondylolysis, with a focus on the pathogenesis of multiple-level cervical spondylolysis. Methods: A 40-year-old female presented with progressive clumsiness and numbness of the hands. Japanese Orthopedic Association (JOA) score for the cervical spine was 14.5. Plain radiographs of the cervical spine showed bilateral spondylolysis of the articular mass portion, with an adjacent dysplastic change and spina bifida occulta of C4 and C5. Cervical laminoplasty from C4 to C6 was performed. Results: The postoperative course was uneventful, and the patient had some recovery of muscle power and sensation, with JOA score improving to 15.5. At the 8-year follow-up, the patient had no recurrence of symptoms, but did show kyphotic and degenerative changes at the C4/5 and C5/6 level with no apparent instability. Conclusions: This case is a rare presentation of bilateral cervical spondylolysis involving C4 and C5, presumably congenital, accompanied by combined dysplastic changes of the cervical spine, cleft lip, and fibrous dysplasia, possibly through an error involving an ossification center during the embryonic stage.

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