Chemiluminescent enzyme immunoassay failed to detect anti-desmoglein 3 antibodies in a case of pemphigus vulgaris Dear Editor, Pemphigus vulgaris (PV) is an autoimmune blistering disease which affects the mucous membranes and the skin. 1 In PV, the desmosomal adhesion molecules desmoglein (Dsg)3 and Dsg1 are target autoantigens. As Dsg3 is highly expressed in the oral mucosa, anti-Dsg3 antibodies can cause oral lesions. 2 Enzyme-linked immunosorbent assay (ELISA) has been widely used for detecting these autoantibodies, and chemiluminescent enzyme immunoassay (CLEIA) has been newly developed for the same purpose with a wider dynamic range and a shorter assay time. 3 However, a few cases have shown different results between CLEIA and ELISA. Here, we report a case of PV with anti-Dsg3 antibodies which was detected by ELISA and immunoblotting but not by CLEIA.A 50-year-old Japanese woman presented with oral erosions followed by flaccid blisters on the skin (Fig. 1a-c). Histopathological examination of the gingival lesion and the skin lesion revealed suprabasal acantholysis (Fig. 1d). Direct immunofluorescence of the gingiva and the skin demonstrated immunoglobulin (Ig)G and C3 deposits in intercellular spaces of the epithelium. Indirect immunofluorescence using normal human skin showed that IgG autoantibodies in the patient's serum reacted to intracellular spaces in all layers of the epidermis (Fig. 1e). CLEIA (STACIA MEBLux test Dsg1, 3; MBL, Nagoya, Japan) was positive for anti-Dsg1 IgG (index value, 123.0; cut-off, 20), but negative for anti-Dsg3 IgG (index value, 12.5; cut-off, 20). From these findings, the diagnosis of PV was made. Based on the oral erosive lesions and the immunofluorescence results, we suspected the presence of anti-Dsg3 antibodies in the patient's serum and performed immunoblotting and Dsg3 ELISA (Mesacup-2 Desmoglein Test Dsg3; MBL). Immunoblotting using normal human epidermal extracts showed a 130-kDa band, corresponding to Dsg3 (Fig. 1f), and
A 6-year-old boy presented to our department with a 1-week history of generalized pustules with a high fever. It was suspected that he had pustular psoriasis when he was 8 months old. He had been treated with a topical dry distillation tar of delipidated soybean. The lesions were recurrent but had been under recent control. He had no recent history of infection or any new medication. Physical examination revealed multiple coalescing creamy-white pustules on a background of erythema over the whole body (Figure 1). Bacterial and fungal cultures of the pus were negative. Histologically, the erythematous pustules on the abdomen showed marked aggregates of neutrophils between degenerated keratinocytes in the uppermost portion of the spinous layer (spongiform pustule of Kogoj, Figure 2; available at www.jpeds.com). Based on these findings, the diagnosis of generalized pustular psoriasis was made. Topical corticosteroids and systemic cyclosporine (5 mg/kg/day) resolved the lesions and fever within 4 weeks.Generalized pustular psoriasis is an uncommon subtype of psoriasis that manifests as acute or subacute generalized sterile pustules appearing on erythema. Generalized pustular psoriasis is observed most frequently in middle-aged adults; however, it can also occur in children. 1 Differential diagnoses include acute generalized exanthematous pustulosis, severe impetigo, and extensive tinea corporis. Drug history, bacterial and fungal tests on the pustules, and histologic findings are valuable diagnostic tools for ruling them out.Complications of childhood generalized pustular psoriasis include hepatic and renal failure, cholestasis, and severe infections, and these occasionally lead to a fatal outcome. Therefore, early systemic therapies, such as cyclosporine or methotrexate, are often required. 2-5 Pediatricians should be aware of this potentially fatal disease, which necessitates prompt, appropriate management. ■
The effectiveness of biologics has changed therapeutic strategies for psoriasis dramatically, but biologics are known to have various adverse effects. We report a 63-year-old woman with psoriatic arthritis who suddenly developed a subcutaneous hematoma after being successfully treated with adalimumab. As she had also suffered from alcoholic cirrhosis, we speculated that she had developed thrombocytopenia severe enough to cause a subcutaneous hematoma. Furthermore, we investigated the changes of platelet counts in 65 psoriatic patients treated with biologics at a single institute from 2010 to 2016. Platelet counts were found to have decreased by 17.4 ± 2.8% during adalimumab therapy (n = 16), 18.5 ± 3.8% during infliximab therapy (n = 17), 14.8 ± 2.1% during ustekinumab therapy (n = 20) and 18.5 ± 5.1% during secukinumab therapy (n = 12). Platelet counts decrease after the administration of biologics in accordance with disease activity, and there is the potential risk of subcutaneous hematoma and other adverse effects. When administrating biologics to psoriatic patients, especially to those with chronic liver disease, dermatologists should carefully monitor for thrombocytopenia.
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