January/February - West Virginia State Medical Association

| Scientific Article
Melkersson-Rosenthal Syndrome with Migraine-Like
Headaches Treated With Minocycline: A Case Report
and Review of the Literature
Matthew A. Hazey, BS
Medical Student
Anthony J. Van Norman, MD
Resident, Section of Dermatology
Drury L. Armistead, MD
Assistant Professor, Section of
Dermatology
West Virginia University School of Medicine,
Morgantown
Abstract
Cheilitis granulomatosa (CG), which
presents clinically as persistent lip
swelling, is characterized histologically by
noncaseating granulomatous
inflammation of unknown origin. CG may
also be part of the classic triad of the
Melkersson-Rosenthal Syndrome (MRS)
and alone is considered by some to be an
oligosymptomatic form of MRS. We report
a case of CG associated with migrainelike
headaches in a 44-year-old woman.
The clinical presentation, histologic
findings, and subsequent treatment are
described. A brief review of the literature
also is provided.
left side (Figure 1). The patient’s
symptoms were initially intermittent
but became persistent. She gave a
history of severe headaches that
were temporally related to increased
swelling of her upper lip. She was
otherwise well and denied symptoms
in any other organ system. Further,
the patient denied a history of any
drug ingestion, nor could she recall
any provocative factors such as foods,
contactants, or activities. Physical
examination revealed soft, nonpitting
edema of the upper lip, left
greater than right, with overlying
erythema. The tongue was normal
in appearance, and there were no
findings consistent with facial nerve
paralysis. Serologic evaluation
including complete blood count,
C1 esterase inhibitor, C3, C4, serum
angiotensin-converting enzyme
level, and antinuclear antibody were
within normal limits. Chest x-ray and
magnetic resonance imaging of the
brain and sinuses were performed
and found to be within normal limits.
A 4-mm punch biopsy specimen
from the cutaneous upper lip showed
scattered noncaseating granulomas,
mild edema, lymphectasia, as well
as superficial and deep perivascular
lymphocytic inflammation (Figures
2 and 3). Special stains were
negative for evidence of fungi or
acid-fast bacilli. No foreign bodies
were seen on examination of the
specimen under polarized light.
Treatment was initiated with
minocycline hydrochloride,100mg,
Figure 2.
Noncaseating granulomas, mild edema, lymphectasia, as well as superficial and deep
perivascular lymphocytic inflammation (H&E, original magnification x 20).
Case Report
A 44-year-old white female
presented with persistent, painless,
non-pruritic swelling of the upper
lip for 8 months. The swelling, which
was associated with an overlying
erythema, was more severe on the
Figure 1.
Before treatment. Upper lip with firm
edema accompanied by erythema.
January/February, 2009, Vol. 105 15