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1.
Ann Dermatol Venereol ; 140(8-9): 531-4, 2013.
Article in French | MEDLINE | ID: mdl-24034638

ABSTRACT

BACKGROUND: It has been reported that D-penicillamine causes pemphigus that is typically superficial. Immunostaining with monoclonal anti-32-2B antibody targeting desmoglein 1 and 3 can help differentiate between drug-induced and classical auto-immune pemphigus. Absence of specific staining militates in favour of drug-induced pemphigus whilst positive staining suggests an auto-immune aetiology that is ongoing despite discontinuation of drug therapy. PATIENTS AND METHODS: A 59-year-old male patient was referred for management of superficial pemphigus 1 year after starting D-penicillamine treatment for scleroderma. The diagnosis of pemphigus was confirmed histologically (intra-epidermal cleavage, acantholysis and perikeratinocytes, deposition of IgG and complement C3). Immunochemical staining with anti-32-2B antibody was initially normal, in keeping with drug-induced pemphigus. Despite discontinuation of D-penicillamine, pemphigus recurred in 2008. A further skin biopsy was undertaken and anti-32-2B staining was abnormal, which is consistent with auto-immune pemphigus. DISCUSSION: Numerous cases of drug-induced pemphigus have been described in the literature. In approximately half of all cases, the pemphigus recedes after cessation of the causative drug. However, there have been no previous reports that changes over time in the immunostaining with anti-32-2B antibodies can mirror a change in form of pemphigus from a drug-induced type to an idiopathic type as well as the associated clinical feature of persistence after drug withdrawal. CONCLUSION: Normal staining with anti-32-2B antibody is associated with a favourable prognosis as regards resolution of drug-induced pemphigus. When, as in this case, status changes to abnormal staining, there is a risk that the pemphigus may become chronic despite discontinuation of therapy.


Subject(s)
Antibodies, Monoclonal , Autoantigens/analysis , Desmoglein 1/analysis , Desmoglein 3/analysis , Pemphigus/chemically induced , Penicillamine/adverse effects , Acantholysis/chemically induced , Acantholysis/pathology , Autoantibodies/analysis , Autoantigens/immunology , Betamethasone/analogs & derivatives , Betamethasone/therapeutic use , Biopsy , Complement C3/analysis , Dermatologic Agents/therapeutic use , Desmoglein 1/immunology , Desmoglein 3/immunology , Disease Progression , Drug Combinations , Fluorescent Antibody Technique, Direct , Humans , Immunoglobulin G/analysis , Male , Middle Aged , Pemphigus/diagnosis , Pemphigus/drug therapy , Pemphigus/immunology , Pemphigus/pathology , Penicillamine/immunology , Penicillamine/therapeutic use , Recurrence , Scleroderma, Systemic/drug therapy
2.
Clin Exp Dermatol ; 38(7): 745-7, 2013 Oct.
Article in English | MEDLINE | ID: mdl-23962308

ABSTRACT

We report a case of chronic leg ulceration occurring in a patient with necrobiosis lipoidica (NL). After many topical treatments had failed to achieve healing, treatment with topical becaplermin was started, which resulted in rapid improvement and ultimately complete healing of the ulceration. Treatment of ulcerated NL is often disappointing. Many topical and systemic drugs have been tried. Becaplermin is a platelet-derived growth factor indicated for the treatment of neuropathic diabetic ulcers measuring < 50 mm in size. To our knowledge, this is only the second case reported in the literature of ulcerated NL successfully treated with becaplermin.


Subject(s)
Angiogenesis Inducing Agents/administration & dosage , Leg Ulcer/drug therapy , Necrobiosis Lipoidica/drug therapy , Proto-Oncogene Proteins c-sis/administration & dosage , Administration, Topical , Adult , Becaplermin , Chronic Disease , Female , Humans , Leg Ulcer/pathology , Treatment Outcome
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