Cervical spinal cord injury following osteophyte excision for respiratory distress caused by diffuse idiopathic skeletal hyperostosis associated with ossification of posterior longitudinal ligament: a case report and literature review

Abstract Background Diffuse idiopathic skeletal hyperostosis (DISH) is a systemic non-inflammatory disorder characterized by enthesopathy and osteophyte formation. DISH can also cause several other symptoms. Limited range of motion (ROM) is the most common symptom; however, dysphagia and respiratory...

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Main Authors: Keisuke Masuda, Hideki Shigematsu, Akinori Okuda, Sachiko Kawasaki, Yusuke Yamamoto, Takahiro Mui, Yasuhito Tanaka
Format: Article
Language:English
Published: BMC 2024-12-01
Series:BMC Musculoskeletal Disorders
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Online Access:https://doi.org/10.1186/s12891-024-08134-1
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author Keisuke Masuda
Hideki Shigematsu
Akinori Okuda
Sachiko Kawasaki
Yusuke Yamamoto
Takahiro Mui
Yasuhito Tanaka
author_facet Keisuke Masuda
Hideki Shigematsu
Akinori Okuda
Sachiko Kawasaki
Yusuke Yamamoto
Takahiro Mui
Yasuhito Tanaka
author_sort Keisuke Masuda
collection DOAJ
description Abstract Background Diffuse idiopathic skeletal hyperostosis (DISH) is a systemic non-inflammatory disorder characterized by enthesopathy and osteophyte formation. DISH can also cause several other symptoms. Limited range of motion (ROM) is the most common symptom; however, dysphagia and respiratory distress are clinically important symptoms. Dysphagia caused by cervical DISH is initially treated conservatively, but surgical treatment is performed when conservative treatment is ineffective. Although there are many reports on the surgical excision of osteophytes for refractory dysphagia, only a few reports on surgery for dysphagia caused by DISH associated with ossification of the posterior longitudinal ligament (OPLL) exist. Here, we report a rare case of cervical spinal cord injury following osteophyte excision for a respiratory distress and dysphagia caused by DISH associated with OPLL. Case presentation A 76-year-old male with hypertension and diabetes presented with dysphagia, respiratory insufficiency, and palpitations. Four months later, he experienced severe dyspnea and was hospitalized. His vital signs indicated respiratory distress, which led to intubation and tracheotomy due to his worsening condition. Imaging revealed massive anterior cervical osteophytes and multisegmental OPLL that caused spinal canal stenosis and tracheal compression. Surgical excision of the osteophytes was performed, but the patient later developed tetraplegia attributed to C5/C6 instability. Posterior fusion and laminoplasty were performed, resulting in neurological improvement but persistent dysphagia and motor deficits. He was transferred to another hospital for rehabilitation but died of aspiration pneumonia. Conclusions Patients with cervical OPLL and spinal cord compression may experience spinal cord injury when intervertebral mobility is slightly increased due to osteophyte excision. If dysphagia or respiratory distress occur in patients with DISH and OPLL, decompression and fusion surgery at the mobile segment is required, in addition to osteophyte excision surgery. Posterior decompression and fusion surgery should be performed before anterior osteophyte excision surgery to avoid implant infection, particularly in patients with respiratory distress who have undergone tracheostomy. Patients receiving long-term mechanical ventilation are less likely to recover their swallowing function and should undergo a total laryngectomy.
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spelling doaj-art-ee7cf1db13cb4b8ea1aea584bbeac2e52025-08-20T01:59:47ZengBMCBMC Musculoskeletal Disorders1471-24742024-12-012511710.1186/s12891-024-08134-1Cervical spinal cord injury following osteophyte excision for respiratory distress caused by diffuse idiopathic skeletal hyperostosis associated with ossification of posterior longitudinal ligament: a case report and literature reviewKeisuke Masuda0Hideki Shigematsu1Akinori Okuda2Sachiko Kawasaki3Yusuke Yamamoto4Takahiro Mui5Yasuhito Tanaka6Department of Emergency and Critical Care Medicine, Nara Medical UniversityDepartment of Orthopedic Surgery, Nara Medical UniversityDepartment of Emergency and Critical Care Medicine, Nara Medical UniversityDepartment of Orthopedic Surgery, Nara Medical UniversityDepartment of Orthopedic Surgery, Nara Prefecture General Medical CenterDepartment of Orthopedic Surgery, Nara Medical UniversityDepartment of Orthopedic Surgery, Nara Medical UniversityAbstract Background Diffuse idiopathic skeletal hyperostosis (DISH) is a systemic non-inflammatory disorder characterized by enthesopathy and osteophyte formation. DISH can also cause several other symptoms. Limited range of motion (ROM) is the most common symptom; however, dysphagia and respiratory distress are clinically important symptoms. Dysphagia caused by cervical DISH is initially treated conservatively, but surgical treatment is performed when conservative treatment is ineffective. Although there are many reports on the surgical excision of osteophytes for refractory dysphagia, only a few reports on surgery for dysphagia caused by DISH associated with ossification of the posterior longitudinal ligament (OPLL) exist. Here, we report a rare case of cervical spinal cord injury following osteophyte excision for a respiratory distress and dysphagia caused by DISH associated with OPLL. Case presentation A 76-year-old male with hypertension and diabetes presented with dysphagia, respiratory insufficiency, and palpitations. Four months later, he experienced severe dyspnea and was hospitalized. His vital signs indicated respiratory distress, which led to intubation and tracheotomy due to his worsening condition. Imaging revealed massive anterior cervical osteophytes and multisegmental OPLL that caused spinal canal stenosis and tracheal compression. Surgical excision of the osteophytes was performed, but the patient later developed tetraplegia attributed to C5/C6 instability. Posterior fusion and laminoplasty were performed, resulting in neurological improvement but persistent dysphagia and motor deficits. He was transferred to another hospital for rehabilitation but died of aspiration pneumonia. Conclusions Patients with cervical OPLL and spinal cord compression may experience spinal cord injury when intervertebral mobility is slightly increased due to osteophyte excision. If dysphagia or respiratory distress occur in patients with DISH and OPLL, decompression and fusion surgery at the mobile segment is required, in addition to osteophyte excision surgery. Posterior decompression and fusion surgery should be performed before anterior osteophyte excision surgery to avoid implant infection, particularly in patients with respiratory distress who have undergone tracheostomy. Patients receiving long-term mechanical ventilation are less likely to recover their swallowing function and should undergo a total laryngectomy.https://doi.org/10.1186/s12891-024-08134-1Forestier’s diseaseDiffuse idiopathic skeletal hyperostosisOssification of posterior longitudinal ligamentOsteophyte excision
spellingShingle Keisuke Masuda
Hideki Shigematsu
Akinori Okuda
Sachiko Kawasaki
Yusuke Yamamoto
Takahiro Mui
Yasuhito Tanaka
Cervical spinal cord injury following osteophyte excision for respiratory distress caused by diffuse idiopathic skeletal hyperostosis associated with ossification of posterior longitudinal ligament: a case report and literature review
BMC Musculoskeletal Disorders
Forestier’s disease
Diffuse idiopathic skeletal hyperostosis
Ossification of posterior longitudinal ligament
Osteophyte excision
title Cervical spinal cord injury following osteophyte excision for respiratory distress caused by diffuse idiopathic skeletal hyperostosis associated with ossification of posterior longitudinal ligament: a case report and literature review
title_full Cervical spinal cord injury following osteophyte excision for respiratory distress caused by diffuse idiopathic skeletal hyperostosis associated with ossification of posterior longitudinal ligament: a case report and literature review
title_fullStr Cervical spinal cord injury following osteophyte excision for respiratory distress caused by diffuse idiopathic skeletal hyperostosis associated with ossification of posterior longitudinal ligament: a case report and literature review
title_full_unstemmed Cervical spinal cord injury following osteophyte excision for respiratory distress caused by diffuse idiopathic skeletal hyperostosis associated with ossification of posterior longitudinal ligament: a case report and literature review
title_short Cervical spinal cord injury following osteophyte excision for respiratory distress caused by diffuse idiopathic skeletal hyperostosis associated with ossification of posterior longitudinal ligament: a case report and literature review
title_sort cervical spinal cord injury following osteophyte excision for respiratory distress caused by diffuse idiopathic skeletal hyperostosis associated with ossification of posterior longitudinal ligament a case report and literature review
topic Forestier’s disease
Diffuse idiopathic skeletal hyperostosis
Ossification of posterior longitudinal ligament
Osteophyte excision
url https://doi.org/10.1186/s12891-024-08134-1
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