Dupilumab Provides Meaningful Clinical Responses in Harlequin Ichthyosis (ABCA12-nEDD): A Report of Two Cases.

Harlequin ichthyosis (HI) is a rare epidermal differentiation disorder characterized by thick hyperkeratotic plates, fissuring, and high neonatal morbidity. Long-term management remains challenging. We report two children with genetically confirmed HI who demonstrated meaningful improvement in pruritus, erythema, scaling, and overall well-being following dupilumab therapy, suggesting a potential role for IL-4/IL-13 blockade as an adjunct or alternative treatment in HI.