Case Report and Review of the Literature: Bullous Skin Eruption After the Booster-Dose of Influenza Vaccine in a Pediatric Patient With Polymorphic Maculopapular Cutaneous Mastocytosis

Abstract

Vaccination is a well-known trigger for mast cell degranulation in subjects affected by mastocytosis. Nevertheless, there is no exact standardized protocol to prevent a possible reaction after a vaccine injection, especially for patients who have already presented a previous vaccine-related adverse event, considering that these patients frequently tolerate future vaccine doses. For this reason, we aim to share our experience at Meyer Children’s University Hospital in Florence to raise awareness on the potential risk for future vaccinations and to discuss the valuable therapeutic strategies intended to prevent them, taking into account what is proposed by experts in literature. We describe the case of an 18-month-old female affected by a polymorphic variant of maculopapular cutaneous mastocytosis that presented an extensive bullous cutaneous reaction 24 hours after the second dose (booster dose) of inactivated-tetravalent influenza vaccine, treated with a single dose of oral corticosteroid therapy with betamethasone (0.1 mg/kg) and an oral antihistamine therapy with oxatomide (1 mg/kg/daily) for a week, until resolution. To the best of our knowledge, in the literature, no documented case of reaction to influenza vaccine in maculopapular cutaneous mastocytosis is described. Subsequently, the patient started a background therapy with ketotifen daily (0.05 mg/kg twice daily), a non-competitive H1-antihistamine, and a mast cell stabilizer (dual activity). A non-standardized pharmacological premedication protocol with an H1-receptor antagonist (oxatomide, 0.5 mg/kg) administered 12 hours before the immunizations, and a single dose of betamethasone (0.05 mg/kg) together with another dose of oxatomide (0.5 mg/kg) administered 2 hours before the injections was followed to make it possible for the patient to continue with the scheduled vaccinations. Indeed, no reactions were subsequently reported. Thus, in our experience, a background therapy with ketotifen associated with a premedication protocol made by two doses of oxatomide and a single dose of betamethasone was helpful to make possible the execution of the other vaccines. We suggest how in these children, it could be considered the idea of taking precaution when vaccination is planned, regardless of the kind of vaccine and if a dose of the same vaccine was previously received. However, international consensus needs to be reached to manage vaccinations in children with mastocytosis and previous adverse reactions to vaccines.

Challenges in Drug and Hymenoptera Venom Hypersensitivity Diagnosis and Management in Mastocytosis

“…The National Institutes of Health conducted a retrospective chart review of 94 children with mastocytosis who received 2136 vaccines, finding that 4% of children had urticaria, anaphylaxis, and exacerbated skin lesions [23]. Case studies have reported children developing bullous skin eruptions and a mastocytoma at the site of vaccination [44,48,49]. The frequency and severity of mast cell activation and adverse reactions to vaccines is higher in pediatric mastocytosis patients with greater skin involvement, diffuse cutaneous mastocytosis, and elevated basal serum tryptase [44,50].…”

“…Children with cutaneous mastocytosis are often advised to avoid a variety of medications including NSAIDs, antibiotics, RCM, and vaccines. There are few case reports describing adverse reactions in children with mastocytosis although antibiotics and vaccines have both been reported [44,48,96].…”

Prophylactic vaccination in children with mastocytosis

“…Data from the literature mainly refer to children with CM, which is the most common type in this age group. Most studies have been conducted on small groups of patients, while severe VARs occur only as case reports (20,23,24) . Some studies have reported a higher incidence of adverse reactions in paediatric patients with all types of mastocytosis…”

Drugs and Vaccines Hypersensitivity in Children with Mastocytosis

“…Vaccines for infectious diseases may trigger MC mediator release in patients with mastocytosis, but the rate of such HRs is unknown. In small population samples, vaccines have been reported to trigger adverse reactions in 6–13% of children with mastocytosis [ 71 , 72 , 73 ]. According to National Institute Health (NIH) retrospective data, the incidence of adverse reactions to vaccination was 1.8% and the prevalence 8% in 75 children with mastocytosis [ 1 ].…”