Abstract：
BACKGROUND:Pemphigus herpetiformis (PH) is a rare subtype of pemphigus that presents challenges in diagnosis. OBJECTIVE:To review the presentation, diagnosis, and management of PH. METHODS:We reviewed the charts of all patients diagnosed and treated for PH in an immunobullous referral center between September 2007 and June 2013. RESULTS:Eight patients were identified with a diagnosis of PH. All presented initially with pruritus. Clinical disease was manifest as either urticated erythematous plaques or a vesiculobullous eruption. Histological evaluation demonstrated eosinophilic spongiosis in all patients with acantholysis in half of cases (n = 4/8). Peripheral eosinophilia was noted in three of eight (37.5%) patients. In all cases, direct immunofluorescence showed intercellular deposition of immunoglobulin G in the epidermis. All patients required high-dose corticosteroid initially. All patients treated with dapsone or sulfasalazine (n = 4) achieved at least partial control. Other effective treatments included intravenous immunoglobulin (n = 2), azathioprine (n = 2), and leflunomide (n = 1). Rituximab was ineffective in two patients. CONCLUSION:The clinical and histological features of PH develop over time and with treatment, making distinction between pemphigus subtypes challenging and delay in diagnosis common. Diagnosis of PH requires a high index of suspicion and is made on clinical grounds (urticated erythema) in the context of compatible histology and immunofluorescence findings. Treatment may be challenging, although efficacy of sulfonamide derivatives appears to offer a therapeutic effect.
journal_name
Int J Dermatoljournal_title
International journal of dermatologyauthors
Laws PM,Heelan K,Al-Mohammedi F,Walsh S,Shear NHdoi
10.1111/ijd.12582subject
Has Abstractpub_date
2015-09-01 00:00:00pages
1014-22issue
9eissn
0011-9059issn
1365-4632journal_volume
54pub_type
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