Surgical Treatment for Central Sleep Apnea due to Occipitocervical Compression Myelopathy in a Patient with Klippel-Feil Syndrome.
Central sleep apnea
Klippel-Feil syndrome
Occipitocervical compression myelopathy
Surgical treatment
Journal
World neurosurgery
ISSN: 1878-8769
Titre abrégé: World Neurosurg
Pays: United States
ID NLM: 101528275
Informations de publication
Date de publication:
09 2020
09 2020
Historique:
received:
11
05
2020
revised:
05
06
2020
accepted:
07
06
2020
pubmed:
20
6
2020
medline:
29
12
2020
entrez:
20
6
2020
Statut:
ppublish
Résumé
Central sleep apnea (CSA) due to occipitocervical compression myelopathy is an extremely rare condition. Here we report a case of surgical treatment for CSA due to occipitocervical compression myelopathy in a patient with Klippel-Feil syndrome. A 60-year-old man had become aware of a gradually progressive clumsiness and gait disturbance without any cause of injury 5 years before. He had complicated respiratory discomfort during sleep for the previous month and visited our hospital. Neurologic examination revealed severe myelopathy. Polysomnography showed CSA and Cheyne-Stokes respiration. Imaging findings showed C2-3 vertebral fusion and severe spinal cord compression caused by hypoplasia of the C1 posterior arch complicated by an anomaly of the vertebral artery. We diagnosed the patient with CSA due to occipitocervical compression myelopathy complicated by Klippel-Feil syndrome. After a simulation using a full-scale 3-dimensional model, resection of the C1 posterior arch and C4-5 laminoplasty was performed. After surgery, both clumsiness and gait disturbance gradually improved. Polysomnography 1 month after surgery showed that the CSA and the Cheyne-Stokes respiration disappeared. Although a recent report has indicated the cause of sleep apnea in patients with rheumatoid arthritis and occipitocervical disorders as obstructive sleep apnea, a significant improvement of CSA was observed with decompression surgery in this case. Appropriate surgical planning resulted in a favorable outcome.
Sections du résumé
BACKGROUND
Central sleep apnea (CSA) due to occipitocervical compression myelopathy is an extremely rare condition. Here we report a case of surgical treatment for CSA due to occipitocervical compression myelopathy in a patient with Klippel-Feil syndrome.
CASE DESCRIPTION
A 60-year-old man had become aware of a gradually progressive clumsiness and gait disturbance without any cause of injury 5 years before. He had complicated respiratory discomfort during sleep for the previous month and visited our hospital. Neurologic examination revealed severe myelopathy. Polysomnography showed CSA and Cheyne-Stokes respiration. Imaging findings showed C2-3 vertebral fusion and severe spinal cord compression caused by hypoplasia of the C1 posterior arch complicated by an anomaly of the vertebral artery. We diagnosed the patient with CSA due to occipitocervical compression myelopathy complicated by Klippel-Feil syndrome. After a simulation using a full-scale 3-dimensional model, resection of the C1 posterior arch and C4-5 laminoplasty was performed. After surgery, both clumsiness and gait disturbance gradually improved. Polysomnography 1 month after surgery showed that the CSA and the Cheyne-Stokes respiration disappeared.
CONCLUSIONS
Although a recent report has indicated the cause of sleep apnea in patients with rheumatoid arthritis and occipitocervical disorders as obstructive sleep apnea, a significant improvement of CSA was observed with decompression surgery in this case. Appropriate surgical planning resulted in a favorable outcome.
Identifiants
pubmed: 32553602
pii: S1878-8750(20)31317-6
doi: 10.1016/j.wneu.2020.06.059
pii:
doi:
Types de publication
Case Reports
Langues
eng
Sous-ensembles de citation
IM
Pagination
232-235Informations de copyright
Copyright © 2020 Elsevier Inc. All rights reserved.