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Abstract:  We present the case of a 3-month-old child with probable catastrophic antiphospholipid syndrome who, after initial successful management with immunomodulary therapies including rituximab, experienced a cutaneous relapse. This rare event was successfully re-treated with repeated administration of rituximab, supporting its role in the control of this disorder. Dermatologic manifestations may be the main clinical presentation of antiphospholipid syndrome, a possible underdiagnosed but potentially fatal pathology.