Hyper‐IgE Syndrome with Widespread Premalign Oral Papillomas Treated with Interferon α2b

Abstract

We report a case of a 7-year-old girl with hyperimmunoglobulin-E syndrome presenting with widespread oral papillomas which were tested for human papilloma virus DNA and had shown to be at high/intermediate risk group for malignancy. She had elevated levels of IgE, recurrent sinopulmonary infections, atopic-like dermatitis, peripheral eosinophilia and defective neutrophil chemotaxis. Interferon alfa 2b therapy and chemoprophylaxis with sulfamethoxazole-trimethoprim was given. Although the papillomas partially improved with the treatment, sinopulmonary infections continue to occur.

Hyper-IgE syndrome and autoimmunity in Mexican children

“…Our second patient did not present with any of these, but the first patient had disseminated HPV infection in the form of plane warts. HPV infection in patients with hyper IgE syndrome has been described in other cases, but papillomas have been mainly located in mucosal surfaces: ear, nose and throat [12][13][14]. Clark et al report a 41 year-old female with HIES and persistent HPV vulvar warts who later developed an invasive vulvar cancer [14].…”

“…These patients appear to have associated autoimmune manifestations with devastating neurological complications in some cases [4]. Several cases of sporadic HIES associated with autoimmune processes like systemic lupus erythematosus (SLE), idiopathic thrombocytopenic purpura (ITP) and nephritic syndrome have been reported [5][6][7][8][9][10][11][12]. In these article, we present two cases of HIES with autoimmune disease, one with suspected AR-HIES and ITP and the other with sporadic HIES and SLE.…”

Use of Cytokine Therapy in Primary Immunodeficiency

“…This regimen resulted in improvement, but smaller and infrequent new lesions continued to occur. Sinopulmonary infections also continued [39]. Treatment of eczema in HIES with subcutaneous IFN-α demonstrated reductions in eczema, serum IgE levels, and circulating CD23+ cells [40].…”

“…In contrast, treatment with rhG-CSF increased ANC greater than 1,000/μL in all subjects. No severe bacterial infections occurred during therapy with either factor, and all tolerated therapy with the exception of one receiving the highest dose of GM-CSF who Treatment with IL-2 after weaning subject off corticosteroids post brain biopsy [88] Clinic Rev Allerg Immunol (2010) 38: [39][40][41][42][43][44][45][46][47][48][49][50][51][52][53] developed cutaneous necrotic vasculitis [8]. Treatment of SCN in an open label randomized phase III trial of G-CSF in 41 subjects resulted in increased ANC >1,500/mm 3 .…”

Generalized verrucosis: A review of the associated diseases, evaluation, and treatments

“…86,87 Although HIES is most frequently associated with staphylococcal skin abscesses, several cases of widespread or persistent warts have been reported. [87][88][89] Oral papillomas and verruca vulgaris of the hands have been reported in a 7-year-old girl with HIES. This patient underwent skin biopsies of verrucous lesions and HPV DNA typing demonstrating intermediate-to high-risk types.…”