IgG4-related disease presenting with an epidural inflammatory pseudotumor: A case report

被引：19

作者：

Ferreira N.R. ^{[1
]}

Vaz R. ^{[1
]}

Carmona S. ^{[1
]}

Mateus S. ^{[1
]}

Pereira P. ^{[1
]}

Fernandes L. ^{[1
]}

Moreira H. ^{[1
]}

Chorão M. ^{[2
]}

Saldanha L. ^{[3
]}

Carvalho A. ^{[1
]}

Campos L. ^{[1
]}

机构：

[1] Department of Internal Medicine, Hospital São Francisco Xavier, Centro Hospitalar de Lisboa Ocidental, Estrada do Forte do Alto do Duque, Lisboa

[2] Department of Pathology, Hospital Egas Moniz, Centro Hospitalar de Lisboa Ocidental, Rua da Junqueira, 126, Lisboa

[3] Department of Anaesthesiology, Hospital São Francisco Xavier, Centro Hospitalar de Lisboa Ocidental, Estrada do Forte do Alto do Duque, Lisboa

来源：

Journal of Medical Case Reports | / 10卷 / 1期

关键词：

Immunoglobulin G4-related disease; Inflammatory pseudotumor;

D O I：

10.1186/s13256-016-0838-2

中图分类号：

学科分类号：

摘要：

Background: Inflammatory pseudotumor is a rare clinical condition that can be related to immunoglobulin G4 disease. Only a few cases of spinal inflammatory pseudotumors have been reported in the literature and an association with immunoglobulin G4 disease was not conclusive in any of them. We describe what we believe to be the first biopsy-proven case of an epidural inflammatory pseudotumor related to immunoglobulin G4 disease. Case presentation: A 57-year-old Caucasian woman presented to our hospital with severe paraparesis, gait disturbance, and sensory loss secondary to a relapsing epidural mass. Examination of a biopsy specimen revealed a lymphoplasmacytic infiltration with fibrosis and an immunoglobulin G4-positive plasma cell ratio of over 50 %, which are compatible with a diagnosis of immunoglobulin G4-related inflammatory pseudotumor. Our patient was successfully treated with systemic and epidural administration of glucocorticoids. Conclusion: Immunoglobulin G4-related disease is an emerging clinical condition in which central nervous system involvement is still uncommon. We describe the case of a patient with an epidural mass with medullar compression, which was proved to be an immunoglobulin G4-related epidural inflammatory pseudotumor. Our findings suggest a new manifestation of immunoglobulin G4-related disease. This disorder should be considered in the differential diagnosis of spinal tumors as a potentially treatable condition with glucocorticoids. © 2016 Ferreira et al.

引用

共 15 条

[1]

Stone J.H., Zen Y., Deshpande V., IgG4-related disease, N Engl J Med., 366, pp. 539-551, (2012)

[2]

Umehara H., Okazaki K., Masaki Y., Kawano M., Yamamoto M., Saeki T., Et al., A novel clinical entity, IgG4-related disease (IgG4RD): General concept and details, Mod Rheumatol, 22, 1, pp. 1-14, (2012)

[3]

Shimatsu A., Oki Y., Fujisawa I., Sano T., Pituitary and stalk lesions (infundibulo-hypophysitis) associated with immunoglobulin G4-related systemic disease: An emerging clinical entity, Endocr J, 56, 9, pp. 1033-1041, (2009)

[4]

Regev K., Nussbaum T., Cagnano E., Giladi N., Karni A., Central nervous system manifestation of IgG4-related disease, JAMA Neurol, 71, 6, pp. 767-770, (2014)

[5]

Kim S.H., Kang Y., Oh S.H., Paik S., Kim J.S., Paraplegia in a patient with IgG4-related sclerosing disease: A case report, Ann Rehabil Med, 38, 6, pp. 856-860, (2014)

[6]

Wallace Z.S., Carruthers M.N., Khosroshahi A., Carruthers R., Shinagare S., Stemmer-Rachamimov A., Et al., IgG4-related disease and hypertrophic pachymeningitis, Medicine (Baltimore), 92, 4, pp. 206-216, (2013)

[7]

Gilliard C., De Coene B., Lahdou J.B., Cervical epidural pseudotumour and multifocal fibrosis: Case report and review of the literature, J Neurosurg Spine., 93, pp. 152-156, (2000)

[8]

Kanagaraju V., Rai D., Alluri R.V., Prasanna C., Shyam Sundar V., Et Al Sm AKumar, An inflammatory pseudotumor in the thoracic epidural space presenting with progressive paraplegia: A histopathological diagnosis with clinical and radiological uncertainty, Eur Spine J, (2015)

[9]

Kato S., Murakami H., Demura S., Epidural inflammatory pseudotumour in the thoracic spine in a patient with polymialgia rheumatic, Spine J., 12, pp. e1-4, (2012)

[10]

Kim J.S., Park S.Y., Inflammatory pseudotumor in the epidural space of lumbosacral spine on 18F-FDG PET / CT, Asia Oceania J Nucl Med Biol., 2, pp. 138-142, (2014)

← 1 2 →