Letters to the Editor Am J Dermatopathol  Volume 37, Number 10, October 2015

FIGURE 2. A, Lobular and septal eosinophil-rich granulomatous panniculitis. B, Granulomatous inflammation with numerous
eosinophils and scattered foreign body–type giant cells. C, Nonpolarizable rounded structures corresponding to the injected
material (microspheres of PLG with exenatide). D, An amalgam of microspheres of PLG slightly refractile lowering condensator.
Hematoxylin-eosin stain, original magnifications: A, ·10; B, ·400; C, ·200; D, ·200.

Irene Andrés-Ramos, MD 6. Boysen NC, Stone MS. Eosinophil-rich gran- The most common clinical presenta-
ulomatous panniculitis caused by exenatide tion is a well-defined nodule or
Susana Blanco-Barrios, MD injection. J Cutan Pathol. 2014;41:63–65.
Emilia Fernández-López, MD 7. Sanmartín O, Requena C, Requena L. Fac- papule located on the head or neck of
Ángel Santos-Briz, MD, PhD titial panniculitis. Dermatol Clin. 2008;26: adults.1
Departamento de Dermatología Médico- 519–527. A 61-year-old woman presented
Quirúrgica y Venereología, Complejo 8. Requena L, Requena C, Christensen L, with a 1-year history of an asymptom-
et al. Adverse reactions to injectable soft
Asistencial de Salamanca, Salamanca, Spain
tissue fillers. J Am Acad Dermatol. 2011;
atic, progressively growing nodule on
64:1–34. the mons pubis. The patient was other-
9. Huh SY, Cho S, Kim KH, et al. A case of wise in good health, and family history
REFERENCES complication after Matridex
injection. Ann
1. Shan SJ, Guo Y. Exenatide-induced eosino-
was noncontributory. Physical examina-
Dermatol. 2010;22:81–84.
philic sclerosing lipogranuloma at the injec- 10. Schuller-Petrovic S, Pavlovic MD, Schuller SS,
tion showed a 0.9 · 0.8-cm sized,
tion site. Am J Dermatopathol. 2014;36: et al. Early granulomatous foreign body reactions skin-colored nodule characterized by
510–512. to a novel alginate dermail filler: the system’s a yellowish hyperkeratotic plug in the
2. Kim D, MacConell L, Zhuang D, et al. Effects failure? J Eur Acad Dermatol Venereol. 2013;
of once-weekly dosing of a long-acting release
center (Fig. 1). No bleeding or draining
27:121–123. was observed. Clinical differential diag-
formulation of exenatide on glucose control and
body weight in subjects with type 2 diabetes. nosis included keratoacanthoma and
Diabetes Care. 2007;30:1487–1493. squamous cell carcinoma. The lesion
3. Nikfar S, Abdollahi M, Salari P. The efficacy Warty Dyskeratoma as was completely removed, and histologi-
and tolerability of exenatide in comparison to
placebo; a systematic review and meta- a Cutaneous Horn of cal examination showed 2 adjacent cup-
analysis of randomized clinical trials. J Pharm shaped epidermal invaginations filled
Pharm Sci. 2012;15:1–30. the Mons Pubis with a plug of keratinous material con-
4. DeYoung MB, MacConell L, Sarin V, et al. taining some grains (Fig. 2). The base of
Encapsulation of exenatide in poly-( D ,L -
lactide-co-glycolide) microspheres produced each invagination presented a digitiform
an investigational longacting once-weekly for- To the Editor: proliferation of epithelial cells extending
mulation for type 2 diabetes. Diabetes Technol Warty dyskeratoma (WD) is a rare into the dermis and characterized by
Ther. 2011;13:1145–1154. foci of acantholysis and dyskeratosis
5. Gentilella R, Bianchi C, Rossi A, et al. Exena-
cutaneous lesion histopathologically char-
tide: a review from pharmacology to clinical acterized by acantholytic dyskeratosis. (Fig. 3). A lymphoplasmacytic inflam-
practice. Diabetes Obes Metab. 2009;11: matory infiltrate was observed in the
544–556. The authors declare no conflicts of interest. dermis. The patient had no clinical

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 Am J Dermatopathol  Volume 37, Number 10, October 2015 Letters to the Editor

describe the tumor.4 The connection to

rudimentary pilosebaceous unit and the
presence of multiple infundibular cystic
structures within the epithelial compo-
nent in the majority of the WDs encour-
age this hypothesis.4
Characteristically, most of the
WDs present as a single asymptomatic
nodule or papule located on the head and
neck of adults without predilection for
sun-exposed areas.5 Less commonly re-
ported are the multiple or plaque clinical
FIGURE 1. Solitary, skin-colored nodular lesion with a well-defined hyperkeratotic presentation and the occurrence on the
horn, sited on the mons pubis. The photograph was taken after local anesthesia. trunk and lower and upper extremities.4–7
Histologically similar lesions have also
been described in the oral and genital
mucosa, but they are believed to be a dis-
tinct entity because of the absence of
pilosebaceous units in these regions.8
WD has never been described on
the mons pubis and, to the best of our
knowledge, our case represents the first
occurrence on this site. Although the
clinical presentation was peculiar because
of the presence of a well-defined cutane-
ous horn, histological features reflected the
most common cup-shaped pattern found
FIGURE 2. WD: histological aspect. Cup-shaped epidermal invagination containing in WD. Interestingly, pathological process
parakeratotic material and lined at the base by a digitiform epithelial proliferation involved more than 1 follicle, an histolog-
extending into the dermis. ical finding previously reported only in
a few reports.9 We did not find a clear-cut
connection with pilosebaceous units on
plane of the section, but the proliferation
was located in close vicinity of them.
Pathogenesis of WD is uncertain.
Viral infection and ultraviolet light have
been suggested to play a role. However,
despite some similarities to viral warts,
WD is not a manifestation of human
papillomavirus infection.4 Similarly,
hypothesis of a sun exposure has been
excluded because, despite the cephalic
location of most of the lesions, the pres-
ence of actinic damage is rare among the
reviewed cases.5 Accordingly, the occur-
rence in our case was in a sun-protected
area and showed no relation to actinic
radiation. Because the mons pubis con-
tains a high density of pilosebaceous
units, this case further supports a follicu-
lar origin of the tumor.
FIGURE 3. Prominent dyskeratosis and acantholysis of the base of the lesion. Acantholytic dyskeratosis is not
a histological hallmark of WD. Indeed,
signs of Grover or Darier disease, and tumor microscopically resembling Darier the most important histopathologic dif-
a diagnosis of WD was made. disease.2,3 He also introduced the cur- ferential diagnoses are Darier disease,
WD is a benign tumor originally rently used name Warty dyskeratoma.3 Grover disease, verruca vulgaris, and
described by Helwig in 1954 and later Recently, a follicular infundibular origin acantholytic variant of neoplastic lesions
extensively characterized by Szymanski has been proposed, and the term follicular such as squamous cell carcinoma, kera-
who describe it as a benign cutaneous dyskeratoma has been suggested to better toacanthoma, and actinic keratosis. In

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 Letters to the Editor Am J Dermatopathol  Volume 37, Number 10, October 2015

our case, the absence of other similar 9. Jang EJ, Lee JY, Kim MK, et al. Warty dysker- usually presents during the first or second
clinical lesions has easily permitted to atoma involving two adjoining follicles. Ann decade of life and is typically exacerbated
Dermatol. 2011;23:98–100.
exclude Darier and Grover disease, and 10. Abramovits W, Abdelmalek N. Treatment of by heavy manual labor.3 Causative muta-
the lack of cellular atypia did not warty dyskeratoma with tazarotenic acid. J Am tions in genes encoding desmoglein 1
support the diagnosis of carcinoma. Acad Dermatol. 2002;46:S4. (Dsg1), desmoplakin (DSP), and keratin
Surgical excision is the treatment 1 (KRT1) have been reported in a small
of choice for WD.4 Curettage with elec- number of families of diverse ethnic
trodesiccation and irradiation with x-ray origin.4–9 In a retrospective histopatholog-
had been tried, but these were followed Acantholysis in Striate ical analysis of 4 PPK cases with different
by recurrence. Successful treatment with DSG1 mutations, we demonstrated that
topical tazarotenic acid gel has also been Keratoderma as a Clue the involved epidermis is characterized
reported.10 In our case, because of the
nodular clinical appearance, the lesion
to the Diagnosis of by partial acantholysis.2 Consequently,
we are now using histopathology suc-
was completely removed surgically, a Genetic Abnormality cessfully as an adjunct to the diagnosis
and there were no recurrences after 6 of inherited PPKs. The following is an
months of follow-up. example:
In conclusion, we report the first A 21-year-old man, otherwise
case of WD localized to the mons pubis, To the Editor:
The role of histopathological diag- healthy, presented with PPK. The kera-
presenting as a cutaneous horn. Because toderma has been present since child-
of the rarity of genital WD and similar nosis of inherited palmoplantar kerato-
dermas (PPKs) has been traditionally hood and increased in severity over the
appearance to neoplastic disorders, clin- years. The condition worsened after
ical confusion may occur without biopsy. limited. With the advent of molecular
genetics, this may have changed. An physical effort and was accompanied
example is epidermolytic PPK in which by both pain and cosmetic concern. His
Viviana Lora, MD* the epidermolytic changes along with skin examination revealed thickened,
Alessandra Scarabello, MD* dyskeratosis may lead to the identifica- yellowish hyperkeratotic bands with
tion of the correct KRT gene mutation.1 a verrucous appearance on his palms
Carlo Cota, MD†
*Division of Dermatology, San Gallicano Another example is the PPKs associ- (Fig. 1), and keratotic plaques on the
Dermatological Institute, Rome, Italy ated with DSG1 mutations such as stri- weight-bearing areas of his soles. No
†Unit of Dermatopathology, San Gallicano ate PPK.2 Striate PPKs (OMIM 148700) other skin, nail, hair, or dental abnormal-
Dermatological Institute, Rome, Italy are an uncommon group of genodermato- ities were found. He reported that his
ses with autosomal dominant inheritance. deceased father and grandmother, 2
REFERENCES This group of diseases is characterized by uncles, and 1 aunt were afflicted by
1. Weedon D. Skin Pathology. 3rd ed. Philadelphia, linear hyperkeratosis of the palms running a similar condition. Several cousins
PA: Churchill Livingstone; 2010. the length of each finger and focal to dif- had a milder form of keratoderma
2. Helbig EB. Proceedings of the 20th Seminar on fuse thickening of the soles. The disease involving the soles only.
the Skin Neoplasms and Dermatoses. Interna-
tional Congress of Clinical Pathologists, Wash-
ington, DC, 11 September, 1954. The American
Society of Clinical Pathology; 1955;53.
3. Szymanski FJ. Warty dyskeratoma: a benign
cutaneous tumor resembling Darier’s disease
microscopically. Arch Dermatol. 1957;75:
567–572.
4. Kaddu S, Dong H, Mayer G, et al. Warty dys-
keratoma—“follicular dyskeratoma”: analysis
of clinicopathologic features of a distinctive fol-
licular adnexal neoplasm. J Am Acad Dermatol.
2002;47:423–428.
5. Diallo M, Cribier B, Scrivener Y. Dyskeratome
verruqueus: une tumeur d’origine infundibu-
laire. Etude anatomo-cliniqu de 43 cas. Ann
Dermatol Venereol. 2007;134:633–636.
6. Calatayud AM, Sanmartin-Jimenez O,
Traves V, et al. Numerous umbilicated papules
on the trunk: multiple Warty dyskeratoma. Am J
Dermatopathol. 2012;34:674–675.
7. Omulecki A, Lesiak A, Narbutt J, et al. Plaque
form of warty dyskeratoma—acantholytic dys-
keratotic acanthoma. J Cutan Pathol. 2007;34:
494–496. FIGURE 1. Thick, yellowish hyperkeratotic bands with a verrucous appearance on the
8. Allon I, Buchner A. Warty dyskeratoma/focal palms.
acantholytic dyskeratosis—an update on a rare
oral lesion. J Oral Pathol Med. 2012;41:261–
267. The authors declare no conflicts of interest.

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