Case Presentation
A 44-year-old man with a history of relapsing-remitting multiple sclerosis was referred to our MS Clinic for the evaluation of bilateral edema of the lower limbs. The edema appeared shortly after the patient had received his last rituximab infusion. The patient also complained of headache and generalized arthralgia. Upon physical examination bilateral symmetric pitting edema of the lower limbs was evident, no skin lesions were present, vital signs were stable and patient was not febrile. Lab data revealed an increased serum creatinine level of 1.4 mg/dl, SGPT of 78 U/l (N=6-45), SGOT of 64 U/l (N=8-40), along with a C-reactive protein (CRP) level of 57 mg/dl (N= less than 10 mg/dl).
On 2001 the patient had experienced a right side optic neuritis, one year later he had ataxia and diplopia and magnetic resonance imaging (MRI) of the brain and spinal cord were done, which showed hyperintense lesions at the level of the spinal cord (Figure 1), the patient was diagnosed with MS and interferon-beta 1a was started. 5 years later he had an attack of bilateral lower limb paresis, and 3 years later he experienced a severe attack of quadriparesis. After the acute management of his attack, the patient’s drug was changed to fingolimod. On 2018 when he was on fingolimod for 2 years his drug was changed to rituximab by another neurologist. After the first dose of rituximab he experienced a severe infusion reaction, presented by erythema and urticarial lesions. Two days later he noticed bilateral edema of the lower limbs that had gradually worsened, and somnolence. The patient was evaluated by several specialists regarding his limb edema. His symptoms improved with antihistamines and corticosteroids, but after 6 months, when he receives his second dose of rituximab, he presented again with bilateral limb edema.
Based on the patient’s history and by taking into account lab data, cytokine release syndrome was highly suspected. The patient was managed with antihistamines and corticosteroids.