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.