Immunohistochemical Study of Multiple Inflammatory Pseudotumors With Both Brain and Spinal Cord Involvement  -Case Report-

Ishihara, Masahiro; Izumoto, Shuichi; Iwatsuki, Koichi; Yoshimine, Toshiki

doi:10.2176/nmc.50.246

Cited by 18 publications

(15 citation statements)

References 22 publications

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“…Only two cases have involved both the brain and the spinal cord. 1,5 The present report is on a young male with TIDD, in whom progressive lesions involved his brain and spinal cord.…”

Section: Administration Of High-dose Intravenous Immunoglobulinmentioning

confidence: 88%

“…1 TIDD can be difficult to diagnose because the inflammatory lesion clinically and radiologically mimics a tumor in the early stages. 2 Its exact incidence and etiology remain unknown.…”

Section: Introductionmentioning

confidence: 99%

“…TIDD with both brain and spinal cord involvement is even rarer, with only two reported cases. 1,5 Because of the presence of the demyelinating mechanism, corticosteroids may provide effective treatment. Only a small percentage of TIDD patients do not respond to corticosteroid therapy.…”

Section: Introductionmentioning

confidence: 99%

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A case of tumor-like inflammatory demyelinating disease with progressive brain and spinal cord involvement

et al. 2015

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CONTEXT: Tumor-like inflammatory demyelinating disease (TIDD) usually occurs in the brain and rarely occurs in the spinal cord. TIDD appears to be very similar to tumors such as gliomas on imaging, which may lead to incorrect or delayed diagnosis and treatment. CASE REPORT: Because of headache and incoherent speech, a 24-year-old Chinese male presented to our hospital with a two-week history of respiratory infections. After dexamethasone treatment, his symptoms still got worse and surgery was performed for diagnostic purposes. Histological examination revealed that the lesion was inflammatory. Further lesions appeared in the spine (T3 and T4 levels) after two months and in the right occipital lobe after three months. After intravenous immunoglobulin (IVIG) and methylprednisolone treatment, his symptoms improved. CONCLUSION: Progressive lesions may damage the brain and spinal cord, and long-term prednisolone and IVIG therapy are beneficial in TIDD patients. RESUMO CONTEXTO:A doença desmielinizante inflamatória tumoral (DDIT) geralmente ocorre no cérebro e raramente na medula espinhal. A DDIT é muito semelhante a tumores tais como gliomas em exames de imagem, o que pode conduzir a diagnóstico e tratamento tardios e incorretos. RELATO DO CASO: Por causa de dor de cabeça e discurso incoerente, um homem chinês de 24 anos de idade foi ao hospital com história de duas semanas de infecções respiratórias. Após o tratamento com dexametasona, seus sintomas ficaram ainda piores e a cirurgia foi realizada para fins de diagnóstico. O exame histológico revelou que a lesão era inflamatória. Mais lesões apareceram na coluna vertebral (níveis T3 e T4) após dois meses, e no lobo occipital direito depois de três meses. Depois de tratamento com imunoglobulina intravenosa (IGIV) e metilprednisolona, seus sintomas melhoraram. CONCLUSÃO: Lesões progressivas podem danificar o cérebro e a medula espinhal, e prednisolona a longo prazo e terapia de IGIV são benéficas em pacientes DDIT.I MD, MSc. Attending Physician,

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“…Only two cases have involved both the brain and the spinal cord. 1,5 The present report is on a young male with TIDD, in whom progressive lesions involved his brain and spinal cord.…”

Section: Administration Of High-dose Intravenous Immunoglobulinmentioning

confidence: 88%

“…1 TIDD can be difficult to diagnose because the inflammatory lesion clinically and radiologically mimics a tumor in the early stages. 2 Its exact incidence and etiology remain unknown.…”

Section: Introductionmentioning

confidence: 99%

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A case of tumor-like inflammatory demyelinating disease with progressive brain and spinal cord involvement

et al. 2015

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“…showed a case of mass lesions in the brain parenchyma and spinal cord that responded well to steroid treatment (15). In these cases, IgG4-related disease was suspected.…”

Section: Discussionmentioning

confidence: 99%

Inflammatory Pseudotumor of the Brain Parenchyma with IgG4 Hypergammaglobulinemia

et al. 2016

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A 58-year-old woman with a 1-month history of right hand clumsiness and speaking difficulty was admitted to our hospital. A neurological examination revealed sensory aphasia and right hemiparesis. Her laboratory tests showed elevated serum levels of IgG and IgG4, pancytopenia, and liver dysfunction. The results of the imaging studies of her abdomen were compatible with sclerosing cholangitis. Brain MRI showed extensive signal abnormalities in the left hemisphere on T2-weighted and fluid-attenuated inversion recovery (FLAIR) images, extending from left internal capsule to the cerebral peduncle with an irregularly enhancing lesion in the left parietal lobe. A brain biopsy revealed lymphocyte and plasma cell infiltration and reactive gliosis. Most of the plasma cells were IgG positive; however, IgG4-positive plasma cells were sparsely observed. After the initiation of betamethasone treatment, her symptoms and the brain MRI abnormalities showed significant improvement. The brain biopsy results did not meet the current criteria of IgG4-related disease. This is the first reported case of a tumefactive lesion of the brain parenchyma with serum IgG4 elevation, which was responsive to steroid treatment. The accumulation of a greater number of reports on the pathological investigation of cases of possible IgG4-related disease may help to elucidate the exact role of IgG4 in IgG4-related disorders.

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“…Their malignant potential is controversal 5 . The rare entity of multilocality has been described 6,7 and might even be interpreted as metastatic occurrence 8 . Although very rare, even paraneoplastic symptoms have been attributed to IMT 9 .…”

mentioning

confidence: 99%

Intraosseous Inflammatory Myofibroblastic Tumor of the Twelfth Thoracic Vertebra

Farshad

Bode

Min

2013

JBJS Case Connector

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Immunohistochemical Study of Multiple Inflammatory Pseudotumors With Both Brain and Spinal Cord Involvement -Case Report-

Cited by 18 publications

References 22 publications

A case of tumor-like inflammatory demyelinating disease with progressive brain and spinal cord involvement

A case of tumor-like inflammatory demyelinating disease with progressive brain and spinal cord involvement

Inflammatory Pseudotumor of the Brain Parenchyma with IgG4 Hypergammaglobulinemia

Intraosseous Inflammatory Myofibroblastic Tumor of the Twelfth Thoracic Vertebra

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