Isolated spinal cord granulomatous angiitis: a case report and review of the literature

被引:1
作者
Cagan, Cansu Ayvacioglu [1 ]
Temucin, Cagri Mesut [1 ]
Arslan, Doruk [1 ]
Gocmen, Rahsan [2 ]
Bolek, Ertugrul Cagri [3 ]
Soylemezoglu, Figen [4 ]
Topcuoglu, M. Akif [1 ]
机构
[1] Hacettepe Univ, Hacettepe Univ Hosp, Dept Neurol, Fac Med, TR-06230 Ankara, Turkey
[2] Hacettepe Univ, Dept Radiol, Fac Med, Ankara, Turkey
[3] Hacettepe Univ, Dept Internal Med Rheumatol, Fac Med, Ankara, Turkey
[4] Hacettepe Univ, Dept Pathol, Fac Med, Ankara, Turkey
关键词
Medulla spinalis; Spinal cord; Angiitis; Vasculitis; Arteriopathy; CENTRAL-NERVOUS-SYSTEM; VASCULITIS;
D O I
10.1007/s00415-021-10912-z
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Introduction Isolated spinal cord angiitis (ISCA) is very rare disease. But, it is frequently encountered in the differential diagnosis of atypical spinal cord syndromes. Case presentation and review of the literature We present a 31-year-old male who presented with progressive paraparesis, and diagnosed with pathologically confirmed ISCA. Longitudinal cystic transverse myelitis was documented in spinal MRI. He responded well to cyclophosphamide and steroid combination, and no relapse was noted during the 4-year follow-up. A standard systematic analysis of the germane literature disclosed 15 more ISCA cases. In total 16 cases (mean age: 46.5, 10 males), ISCA was diagnosed with pathological evaluation in all (Biopsy in 11, Autopsy in 5). MRI lesion is characterized by usually multisegmental longitudinal and sometimes cystic expansile lesions. In seven cases, it was described as "(pseudo)tumoral" by the authors. Albeit absence of elevation of CSF protein/WBC or "compatible" spinal MRI lesion may aid to exclude ISCA to some extent, pathological confirmation is currently necessary for the diagnosis. In 11 cases, ISCA was treated similar to primary supratentorial vasculitis. Mortality rate is 31%. Discussion ISCA diagnosis, a typical example of which we have presented here, can only be established by tissue examination. However, noninvasive diagnostic criteria are critically needed. Our data suggest that this can only be possible with multinational multicenter prospective registry.
引用
收藏
页码:3175 / 3179
页数:5
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