309
A
utoimmune bullous diseases encompass a
group of blistering diseases characterized by
the presence of autoantibodies that are found
both in the circulation and bound in vivo to perile-
sional epithelial tissues. They are generally classified
into 2 categories, based on the location of blister for-
mation. In the pemphigus group, blisters occur with-
in the epidermis, and in the group of subepidermal
blistering diseases, the lesions occur just below the
epidermis.
1
Within each of these groups, multiple,
distinct clinical entities have been defined based on
clinical, histologic, and immunofluorescence (IF)
findings.
2
More recently, biochemical and molecular
biologic approaches have revealed the identity of the
target antigens in several of the bullous diseases.
2
With the advent of these new techniques, it has also
been possible to describe and characterize new blis-
tering diseases with novel immunologic features. In
this report, we describe a patient with an autoim-
mune subepidermal bullous disease associated with
IgG autoantibodies to a 200 kd dermal protein that
has recently been reported as a novel autoantigen of
the lower lamina lucida of the basement membrane
zone (BMZ).
3-6
CASE REPORT
In December 1996, a consultation was required
for an 81-year-old white man with a bullous eruption
that had first appeared 2 weeks earlier. Past medical
history was relevant for an adult-onset diabetes mel-
litus treated with glibenclamide, hypertension treat-
ed with enalapril, congestive heart failure treated
with furosemide, and hypercholesterolemia treated
with cholestyramine. Cutaneous examination
revealed the presence of tense vesicles and numer-
ous erosions on the dorsum of the hands, as well as
on forearms and knees (Fig 1). The remaining skin
and mucous membranes were unaffected.
Histologic examination of a lesional skin biopsy
specimen showed an area of dermal-epidermal sepa-
ration and dermal inflammatory infiltrates mainly
consisting of neutrophils. Eosinophils and mononu-
clear cells were less frequent. Neutrophilic microab-
A subepidermal bullous eruption associated with
IgG autoantibodies to a 200 kd dermal antigen:
The first case report from the United States
José M. Mascaró Jr, MD,
a,c
Detlef Zillikens, MD,
a,d
George J. Giudice, PhD,
a,b
Fréderic Caux, MD,
a
Matthew G. Fleming, MD,
a
Henry M. Katz, MD,
e
and Luis A. Diaz, MD
a
Milwaukee, Wisconsin; Barcelona, Spain; Würzburg, Germany; and Manitowoc, Wisconsin
We describe an 81-year-old white man in whom a subepidermal bullous eruption developed that clinically
resembled bullous pemphigoid. The eruption promptly responded to oral tetracycline and niacinamide and
topical clobetasol. Histologic examination of perilesional skin revealed neutrophilic infiltration with
formation of papillary microabscesses and subepidermal cleavage. Direct immunofluorescence showed
linear deposition of IgG and C3 along the basement membrane zone. By indirect immunofluorescence,
circulating IgG autoantibodies bound exclusively to the dermal side of salt-split normal human skin.
Immunoblot analysis demonstrated that the patient’s autoantibodies reacted with a 200 kd dermal protein
that was different from type VII collagen, the epidermolysis bullosa acquisita autoantigen. This patient
represents the first confirmed case from the United States with a recently reported novel autoimmune
subepidermal bullous disease associated with IgG autoantibodies to a 200 kd dermal antigen. (J Am Acad
Dermatol 2000;42:309-15.)
This supplement is made possible through an
educational grant from Ortho Dermatological
to the American Academy of Dermatology.
From the Departments of Dermatology
a
and Biochemistry,
b
Medical College of Wisconsin, Milwaukee; the Department of
Dermatology, Hospital Clinic, Barcelona;
c
the Department of
Dermatology, University of Würzburg;
d
in private practice in
Manitowoc.
e
Reprint requests: Luis A. Diaz, MD, Department of Dermatology,
Medical College of Wisconsin, 8701 Watertown Plank Rd,
Milwaukee,WI 53226.E-mail:ldiaz@med.unc.edu
Copyright © 2000 by the American Academy of Dermatology,Inc.
0190-9622/2000/$12.00 + 0 16/4/98954