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Treatment of anogenital warts with imiquimod 5% cream in 22-month and
18-month old infants
Kevin H. Kim, MD, PhD, University of Arkansas for Medical Sciences, Little Rock,
AR, United States; Salvador Arias-Santiago, MD, PhD, Virgen de las Nieves
University Hospital, Granada, Spain

Urticaria pigmentosa in a 6-month old Filipino male with G6PD deficiency
Maria Franchesca Quinio, MD, East Avenue Medical Center, Quezon City,
Philippines; Milali Torres, MD, East Avenue Medical Center, Quezon City,
Philippines
Introduction: Glucose-6-phosphate dehydrogenase (G6PD) deficiency is the most
common enzyme defect of red blood cells. Around 200 million people are deficient
in this enzyme. In the Philippines, this X-linked hereditary deficiency has a
prevalence rate of 4.5% to 25.7%. Clinical manifestations of patients with G6PD
deficiency may be induced by trigger factors such as certain drugs like diphenhydramine, an H1 antihistamine. Mastocytosis is a rare group of disorders of mast cell
proliferation. The most common cutaneous manifestation of mastocytosis is
urticaria pigmentosa presenting as multiple pruritic hyperpigmented macules,
papules and patches over the face, trunk, and extremities, usually sparing the palms
and soles. Darier’s sign is usually positive. Histopathologic examinations with both
Giemsa and CD 117 stains will reveal abundance of mast cells. In East Avenue
Medical Center, a tertiary government hospital in the Philippines, only three cases
were noted from 1992-2015. H1 antihistamines such as diphenhydramine are the
first-line treatments for such cases in addition to topical steroids, and mast cell
stabilizers.

Anogenital warts are benign epidermal tumors caused by human papillomavirus
(HPV) acquired in children through vertical, autoinoculation, heteroinoculation,
contaminated fomite, and sexual transmission. Fortunately, most anogenital warts
are self-limited making active nonintervention a reasonable initial approach.
However, for those with symptomatic or persistent anogenital warts, intervention
is usually necessary. Imiquimod has been shown to be an effective treatment option
for anogenital warts in adults and children. However, in infants (less than 2 years of
age), reports of effectively treated cases remain relatively few. Here we present two
cases anogenital warts in infants treated with topical imiquimod 5% cream. Case 1: A
healthy 22-month old girl presented with a 1-month history of rapidly growing
perianal lesions. On physical exam, she had numerous grouped pink verrucous
perianal papules consistent with condyloma. Biopsy and HPV typing were
performed, which were consistent with condylomatous changes and positive for
types 6 and 16, respectively. She was treated with imiquimod 5% cream regimen that
was well tolerated: application to the perianal warts for 8 hours at night, three times
a week for a period of 6-weeks with the use of mupirocin cream when imiquimod
was not used. The patient was followed up every 2-weeks with complete resolution
of lesions by 6-weeks. Case 2: A healthy 18-month old girl presented with a 2-month
history of rapidly growing perivulvar lesions. On physical exam, she had pink
verrucous perivulvar papules consistent with condyloma. Anoscopy and vaginoscopy did not show any internal lesions. Biopsy and HPV typing were consistent
with condylomatous lesions and positive for types 6 and 11, respectively. She was
treated with imiquimod 5% cream regimen for a period of 8-weeks that was well
tolerated with complete response. Our patients presented with rapidly growing
perianal (case 1) and perivulvar warts (case 2) that were treated with imiquimod 5%
cream with no serious adverse effects. Interestingly, marked improvement was
observed by 2 weeks of therapy in both cases. Our report adds to the number of
effectively treated anogenital warts with imiquimod 5% cream in infants reported in
the literature. The ease of use, safety profile, efficacy, lower recurrence rates, and
potential for long-standing immunity make imiquimod a viable alternative treatment
option especially in pediatric patients with larger area of involvement.

Case report: This is a case of a six-month old Filipino boy presenting with multiple
pruritic brown macules, papules, and patches on the face, trunk, extremities
including the palms and soles. Darier’s sign is positive. This patient has mild motor
developmental delay and has G6PD deficiency. Histopathologic examination with
Giemsa and CD 117 staining revealed abundance of mast cells that are the typical
histologic features of mastocytosis. He was given cetirizine instead of diphenhydramine, ketotifen, a mast cell stabilizer, emollients, and triamcinolone lotion, a midpotent topical steroid. Clinical improvement of cutaneous lesions was noted after
one month.
Conclusion: Urticaria pigmentosa and G6PD deficiency are both rare disorders. The
combination of these two together with motor developmental delay have not been
reported previously suggesting that this might be a new syndrome with an unknown
etiology. The symptoms of urticaria pigmentosa may be relieved with the use of H1
antihistamines such as diphenhydramine. But it is important to always do a thorough
history to detect other co-morbidities such as G6PD deficiency so as to avoid giving
medications that might cause hemolytic anemia.

Commercial support: None identified.

Commercial support: None identified.

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Two cases of neonatal lupus
Silvia Marte, MD, Instituto Dermatologico de Cirugia y Piel Dr Huberto Bogaert
Diaz, Santo Domingo, Dominican Republic; Fernanda Nanita de Estevez, MD,
Instituto Dermatologico de Cirugia y Piel Dr Huberto Bogaert Diaz, Santo
Domingo, Dominican Republic; Francia Rosa Rodriguez, MD, Instituto
Dermatologico de Cirugia y Piel Dr Huberto Bogaert Diaz, Santo Domingo,
Dominican Republic
Neonatal lupus erythematous (NLE) is an entity that occurs 0.6-2.2 of every 100,000
children annually with female ratio predominance of 2:1. The etiology is thought to
be caused due to transplacental passage of maternal autoantibodies such as IgG
against Ro (SSA), La (SSB) or U1-ribonucleoprotein. The clinical manifestations
involve dermatologic, cardiac and hepatic involvement. Dermatologic lesions
presents with annular erythematous plaques predominantly in periorbital region,
neck, and scalp. Cardiac involvement occurs in about 65% of patients which can
vary from rhythm abnormalities to complete congenital heart block or heart failure
that generally do not resolve. This is a self-limited disease, therefore the treatment
consists of supportive therapy until resolution in 2-6 months. Overall we describe
two cases with almost identical clinical and histopathologic manifestations. They
presented to the hospital with dermatosis of multiple erythematous plaques varying
in size with well circumscribed elevated borders affecting face, skull, thorax, upper
and lower extremities. One mother had systemic lupus erythematosus (SLE)
diagnosed before pregnancy, while the other had nonrelevant medical history. Lab
work requested for both mothers returned positive for anti-SSA and anti-SSB in both
cases confirming the diagnosis for NLE. Biopsies reported superficial and media
perivascular dermatitis with marked interstitial mucinosis. No cardiovascular lesions
found on either patients. Treatment consisted of hygiene education and sunblock
every 2 hours throughout whole body with follow up at 6 months. This disease is
poorly recognized resulting in misdiagnoses with other entities. Therefore, it is of
great importance to incorporate clinical information in order to provide a more
prompt diagnosis. Although in some cases, the mothers do not progress to SLE, there
is an increased risk in these mothers to debut with SLE along their lifetime.
Nevertheless, they must be followed carefully in order to provide the necessary
treatment and prevent progression of disease.

Vulvar xanthelasmoid mastocytosis in a child with urticaria pigmentosa
Audrey Green, MD, Geisinger Medical Center, Danville, PA, United States;
Bethany Rohr, MD, Geisinger Medical Center, Danville, PA, United States;
Katherine Marks, DO, Geisinger Medical Center, Danville, PA, United States
Background: Cutaneous mastocystosis is a mast cell dependent disease that most
commonly presents in childhood as solitary mastocytomas or urticaria pigmentosa.
Xanthelasmoid mastocytosis represents a rare clinical entity on the spectrum of
cutaneous mastocytoses.
Case description: A five year-old female presented to our pediatric dermatology
clinic with a several month history of papular, pruritic lesions on the buttocks and
genitals. Past medical history included biopsy proven urticaria pigmentosa. A review
of systems was negative for headaches, facial flushing, itching, fatigue, or diarrhea.
No other pertinent family, allergic, or social history was noted. On exam, the patient
had multiple firm, well-circumscribed smooth yellow papules of varying sizes
located on the vulva and perineum. Widespread red-brown patches were also noted
on the neck, chest, and back. The patient’s guardian declined a biopsy of the genital
lesions.
Discussion: Xanthelasmoid mastocytosis is a rare clinical entity, occasionally seen in
conjunction with urticaria pigmentosa. These lesions often present in infants and
arise as small, hard, yellow papules. They are usually widespread, but have rarely
been reported in the genital region. Darier’s sign is inconsistent in such lesions.
Histologically, mast cell infiltrates are seen in the deep dermis, which is thought to
yield their yellow color. Like other childhood forms of cutaneous mastocytoses, the
recommended treatment is with antihistamines and avoidance of mast cell
degranulation stimulators. Since these lesions can last into adulthood, surgical
removal can be contemplated but may not be a viable option due to the risk of
negative physical and psychological sequelae.

Commercial support: None identified.

Commercial support: None identified.

MAY 2016

Conclusions: Here we present a case of vulvar xanthelasmoid mastocytosis in a
young girl with urticaria pigmentosa. Our case is of interest due to the rare nature of
such lesions as well as the genital location and late age of onset in our patient.
Dermatologists should be aware of this unique entity as manifestations of cutaneous
mastocytoses seem to exist on a spectrum.

J AM ACAD DERMATOL

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