Cytokine release syndrome: case reportA 44-year-old man developed cytokine release syndrome during treatment with rituximab for relapsing-remitting multiple sclerosis.The man, who had a history of relapsing-remitting multiple sclerosis, was referred to a clinic in Iran for the evaluation of bilateral oedema of the lower limbs. The oedema appeared shortly after he had received his last rituximab infusion. He also complained of headache and generalised arthralgia. Upon physical examination, bilateral symmetric pitting oedema of the lower limbs was found evident, no skin lesions were present, vital signs were stable and he was not febrile. Lab data showed an increased level of serum creatinine, SGPT, SGOT and C-reactive protein. He had normal haemoglobin, haematocrit, white blood cell count, and mildly decreased platelet counts. On 2001, he had experienced a right side optic neuritis and one year later he had ataxia and diplopia. A MRI of the brain and spinal cord were done, which revealed hyperintense lesions at the level of the cervical spinal cord. He was diagnosed with multiple sclerosis (MS) and interferon-beta 1a was initiated. Five 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, his drug was changed to fingolimod. On 2018, when he was on fingolimod for 2 years his drug was again changed to rituximab by another neurologist. After the first dose of IV infusion of rituximab [Zytux] 1 gram, he experienced a severe infusion reaction, presented by erythema and urticarial lesions. Two days later, he noticed to have bilateral oedema of the lower limbs, that had gradually worsened and somnolence was observed. He was evaluated by several specialists regarding his limb oedema.The man's symptoms improved with unspecified antihistamines and corticosteroids. After 6 months, when the man again received his second dose of IV infusion of rituximab 1 gram, he presented again with bilateral limb oedema. Consult with an infectious disease specialist was done to rule out any infectious causes or sepsis. Based on his history and by taking into account lab data, cytokine release syndrome was diagnosed secondary to rituximab [exact duration of treatment to reaction onset not stated]. He was managed with unspecified antihistamines and corticosteroids. Prednisolone was started and titrated over the course of 4 weeks. His symptoms were improved in 2 weeks, and limb oedema was completely resolved on week four of follow-up.