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Cold induced urticaria (ColdU) is a common chronic induced urticaria
.
It features will wheal when skin is exposed to cold air or liquid object, and sometimes vascular edema
There will be wind group when exposed to cold air, liquid or objects skin, and sometimes vascular edema vascular quality of life
Here, we describe a case of severe refractory cold-induced urticaria associated with cryoglobulinemia that was successfully treated with rituximab
.
Case introduction:
A 60-year-old woman was referred to our Urticaria Reference and Center of Excellence2 because of ColdU.
She has been suffering from this disease since she was 30 years old
.
When the ambient temperature is lower than about 18°C and the duration exceeds 3-4 minutes, local itchy wind masses will appear when exposed to cold wind or contact with cold objects
The TempTest results of the baseline (T0 ) showthat there are obvious wind clusters and flare reactions between 4 °C (A) and 16-18 °C (B ) .
After three months of rituximab treatment, TempTest was negative (C)
After three months of rituximab treatment, TempTest was negative ( C )
Omalizumab and cyclosporine are not
Due to the severity and refractory of her ColdU, further examination found that it was related to type I cryoglobulin (monoclonal IgG) and monoclonal B-cell lymphocytosis
.
Whole-exome sequencing did not find mutations related to auto-inflammation
We hypothesized that in this patient, the detected cryoglobulin was of pathophysiological importance to her ColdU and started treatment with rituximab
.
Rituximab is a therapeutic monoclonal antibody against CD20, which can reduce the production of autoantibodies by depleting memory B lymphocytes
.
The patient received 2 intravenous injections of 1000 mg of rituximab, 2 weeks apart
Laboratory results before and after rituximab treatment
Laboratory results before and after rituximab treatmentCryoglobulin is an immunoglobulin that precipitates at a temperature below 37°C , which can cause vascular occlusion, sometimes leading to immune complex vasculitis and tissue damage
.
ColdU is a very rare manifestation of cryoglobulinemia, and its pathophysiology has not yet been elucidated
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