Case Report

A Case of Severe Symptomatic Central Nervous System Sarcoidosis Secondary to Treatment with Adalimumab

Table 1

Review of neurosarcoidosis cases in the setting of TNF-alpha inhibitor use.

AuthorAnti-TNF agent usedUnderlying diseaseClinical presentationImaging findingsCerebrospinal (CSF) findingsPathological findingsOutcome

Berrios et al. [15]EtanerceptChronic JRA and refractory uveitisAcute mental status change, fever, headache, joint pain, dizziness, night sweats, chillsFirst brain MRI: diffuse abnormal hyper intensities within sulci of both cerebral hemispheres associated with abnormal leptomeningeal enhancement, 2 small nonspecific foci of fluid-attenuated inversion recovery hyperintense signals in the right thalamus and lower pons. Second brain MRI: small hyperintense cortical lesions. Cervical spine MRI: incidental abnormal lymph nodes in the right upper mediastinum. CT thorax, abdomen and pelvis: hepatosplenomegaly, lymphadenopathy in the axillary, mediastinal, intra-abdominal, iliac chain and inguinal lymph nodes. PET scan: unremarkable.96% lymphocytes, 3% monocytes, 1% lymphocytes, protein: 48 mg/dl, glucose: 49 mg/dl.Noncaseating granulomatous lymphadenitisClinical and imaging improvement.

Sturfelt et al. [16]Infliximab, methotrexateErosive RAHeadache, fever, diplopia, severe papilledema of both eyes, nerve palsy of the left eyeHead CT, brain and spinal cord MRI, MR angiography: unremarkable. Transcranial Doppler analysis: high intracranial pressure.Slight pleocytosis with mononuclear leucocytes of 49 × 106/l and polynuclear leucocytes 4 × 106/l, normal CSF/blood glucose ratio, slightly increased protein of 0.78 g/l. Negative culture results for bacteria, viral, fungal or protozoan infection.Bilateral granulomatous iridocyclitis, retinal periphlebitisResolution of headache, papilledema, eye muscle function. Pathological ophthalmic changes slowly subsided. Symptoms. Returned to work full-time.

Durel et al. [17]EtanerceptErosive RABilateral facial paralysis, anosmia, papilledema, anterior uveitis, weight loss, dyspnea, sicca symptomsBrain MRI: enhanced signal of cranial nerves. Chest CT: bilateral hilar and mediastinal lymph nodes.Elevated protein, normal glucose without oligoclonal bands.No histological confirmationComplete resolution of eye sight and brain MRI findings but not facial paralysis.

Mao-Draayer et al. [18]AdalimumabHLA-B27-positive ASNew seizuresHead CT: hypo density in left frontal subcortical white matter. Brain MRI: leptomeningeal enhancement and several hyperintense lesions.Mild lymphocytic leukocytosis, elevated protein.Noncaseating granulomaResolution of the disease.

CNS, central nervous system; JRA, juvenile rheumatoid arthritis; RA, rheumatoid arthritis; AS, ankylosing spondylitis.