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Long-term outcome of idiopathic hypertrophic thoracic pachymeningitis.

Abstract
Idiopathic hypertrophic spinal pachymeningitis (IHSP) is a comparatively rare disease characterized by hypertrophic inflammation of the dura mater and clinical symptoms that progress from local pain to myelopathy. We report a case of IHSP followed up for 20 years in a 46-year-old man. Expansive laminoplasty was performed in 1991, and this case has been previously reported by a co-author. After 17 years, the patient's gait disturbance returned. Physical examination and imaging confirmed IHSP that had developed into syringomyelia at the T2-L1 conus level. This case was diagnosed as adhesive spinal arachnoiditis due to pachymeningitis caused by syringomyelia. T1-T4 laminectomy, a syringo-subarachnoid shunt (S-S shunt), and L2-L3 laminectomy were performed. The patient again developed dysesthesia and gait disturbance 3 years after the second operation. Most reports of IHSP have limited their focus to short-term follow-up after initial treatment with no long-term results. At present, there are only five reports referring to long-term results of greater than 5 years. All but one case needed additional surgery. To the best of our knowledge, this is the first case in which syringomyelia occurred in a patient with IHSP. It is important to note that syringomyelia may be a cause of symptom recrudescence during long-term follow-up in IHSP patients.
AuthorsMio Tsutsui, Taketoshi Yasuda, Masahiko Kanamori, Takeshi Hori, Tomoatsu Kimura
JournalEuropean spine journal : official publication of the European Spine Society, the European Spinal Deformity Society, and the European Section of the Cervical Spine Research Society (Eur Spine J) Vol. 21 Suppl 4 Issue Suppl 4 Pg. S404-7 (Jun 2012) ISSN: 1432-0932 [Electronic] Germany
PMID21594749 (Publication Type: Case Reports, Journal Article)
Topics
  • Adult
  • Dura Mater (surgery)
  • Follow-Up Studies
  • Humans
  • Hypertrophy (complications, surgery)
  • Laminectomy
  • Male
  • Meningitis (complications, surgery)
  • Syringomyelia (etiology, surgery)
  • Thoracic Vertebrae (surgery)
  • Treatment Outcome

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