Recurrent Gianotti-Crosti syndrome

Abstract

To the Editor: Gianotti-Crosti syndrome (GCS) classically presents in children as a self-limited, symmetric erythematous papular eruption affecting the cheeks, extremities, and buttocks. While initial reports implicated hepatitis B virus as the etiologic agent, many other bacterial, viral, and vaccine triggers have since been described. A previously healthy 2-year-old boy presented with a 3-week history of a cutaneous eruption that initially appeared on his legs and subsequently progressed to affect his arms and face. Two weeks after onset of the eruption, he was immunized with intramuscular Vaxigrip influenza vaccination (Sanofi Pasteur), and new lesions appeared at the immunization site on his right upper arm. Physical examination demonstrated an afebrile child with erythematous papules on the cheeks, arms, and legs (Fig 1). He had a localized papular eruption on his right upper arm (Fig 2). There was no lymphadenopathy or hepatosplenomegaly. Laboratory investigations revealed leukocytosis (white cell count, 14,600/mm) with a normal differential, reactive thrombocytosis ( platelet count, 1,032,000/mm), a positive urine culture for cytomegalovirus, and positive IgM serology for Epstein-Barr virus (EBV). Histopathologic examination of a skin biopsy specimen from the right buttock revealed a perivascular and somewhat interstitial lymphocytic infiltrate in the superficial and mid-dermis with intraepidermal exocytosis of lymphocytes, mild spongiosis and papillary dermal edema. He was treated with 2.5% hydrocortisone cream, and the eruption resolved. Twelve months later, he presented with a similar papular eruption localized to the left upper arm at the site of a recent intramuscular influenza vaccination (Vaxigrip). Although an infection represents the most important etiologic agent, a second event involving immunomodulation might lead to further disease accentuation, thus explaining the association of GCS with vaccinations. In our case, there was evidence of both cytomegalovirus (CMV) and EBV infection as well as a recent history of immunization. Localized accentuation of papules at the immunization site was unusual, as previous cases of GCS following immunizations have had a widespread and typically symmetric eruption. It is possible that trauma from the injection or a component of the vaccine elicited a Koebner response, causing local accentuation. There are no previous reports of recurrence of vaccine-associated GCS. One report documented recurrence with two different infectious triggers. As GCS is a mild and selflimiting disease, further vaccinations are not contraindicated. Andrei I. Metelitsa, MD, FRCPC, and Loretta Fiorillo, MD, FRCPC