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Hypertrophic Spinal Pachymeningitis With Thoracic Myelopathy: The Initial Presentation of ANCA-related Systemic Vasculitis.
MedLine Citation:
PMID:  21436740     Owner:  NLM     Status:  Publisher    
Abstract/OtherAbstract:
STUDY DESIGN: A retrospective case review combined with a review of current literature. OBJECTIVES: We describe a case of antineutrophil cytoplasmic antibodies (ANCA)-related systemic vasculitis, with nearly 4years of clinical and radiographic follow-up, initially presenting as hypertrophic spinal pachymeningitis (HSP). The diagnosis, surgical, and medical treatment of HSP are discussed in the context of a literature review. SUMMARY OF BACKGROUND DATA: HSP is a rare disease characterized by hypertrophic inflammation of the dura mater and clinical symptoms that progress from local pain to myelopathy. HSP has been associated with infectious, inflammatory, autoimmune, and traumatic conditions. Surgical decompression and/or corticosteroid therapy have been shown effective at managing this disorder, but identifying associated diseases can be essential to a favorable patient outcome. METHODS: The medical, pathologic, and radiographic records of this case were reviewed. A computer-based search of the PubMed database was used to perform a comprehensive literature review of HSP. RESULTS: We report a 66-year-old male with a history of renal cell carcinoma and bladder cancer who presented with upper abdominal pain, thoracic myelopathy (progressive weakness and numbness of the lower extremities, and gait disturbances), and weight loss. A diagnosis of HSP was subsequently shown to be the initial presentation of ANCA-related systemic vasculitis. CONCLUSIONS: The possibility of an ANCA-related systemic vasculitis presenting as HSP without systemic signs is a diagnostic and therapeutic challenge for the spinal surgeon. The diagnosis of HSP should initiate a search for an associated disease process and close follow-up after initial treatment. With interdisciplinary collaboration between medicine, radiology, pathology, and orthopedics, the diagnosis was made and a treatment initiated which halted disease progression and has maintained remission for more than 4 years.
Authors:
Joseph D Smucker; Austin J Ramme; Richard F Leblond; Leslie A Bruch; Gholamreza Bakhshandehpour
Publication Detail:
Type:  JOURNAL ARTICLE     Date:  2011-3-22
Journal Detail:
Title:  Journal of spinal disorders & techniques     Volume:  -     ISSN:  1539-2465     ISO Abbreviation:  -     Publication Date:  2011 Mar 
Date Detail:
Created Date:  2011-3-25     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  101140323     Medline TA:  J Spinal Disord Tech     Country:  -    
Other Details:
Languages:  ENG     Pagination:  -     Citation Subset:  -    
Affiliation:
Departments of *Orthopaedics and Rehabilitation ‡Internal Medicine §Pathology †Carver College of Medicine, University of Iowa, Iowa City, IA ∥Department of Radiology, Shahid Beheshti University of Medical Science, Imam Hossein Hospital, Madani St, Tehran, Iran.
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