BRIEF REPORT Pediatric Dermatology Vol. 32 No. 4 e167–e168, 2015

Congenital Plantar Angiofibroma-Like Hamartoma Abstract: A 2-year-old girl presented with a congenital, slow-growing, exophytic nodule on the plantar surface of the foot. Histopathology revealed a hamartoma consisting of dermal angiofibroma features with overlying epidermal verruciform hyperplasia.

A healthy 2-year-old girl presented to the dermatology clinic for evaluation of a lesion on the left plantar foot. The growth was present as a tiny papule at birth and had continued to slowly enlarge. Putting pressure on the foot while ambulating caused pain. On physical examination, she had a 1.4-cm 9 1-cm exophytic, skin-colored, papilliform nodule of the midplantar foot (Fig. 1). The unique appearance of the lesion resembled a sea anemone. She had no other skin lesions and no personal or family history of tuberous sclerosis, neurofibromatosis, or multiple endocrine neoplasia. A shave excision was performed, and histopathologic examination showed increased dermal vessels with stellate fibroblasts, consistent with the features of an angiofibroma (Fig. 2). There was also overlying verruciform hyperplasia of the epidermis, resembling an epidermal nevus. Within 2 months the lesion began to grow back, and within 1.5 years it had completely recurred. The patient was referred to pediatric plastic surgery for excision. Histopathologic findings were identical to those of the original biopsy. There was no evidence of recurrence at her 6-month follow-up visit.

Figure 1. An exophytic, skin-colored, papilliform nodule of the midplantar surface of the left foot.

A

B

DISCUSSION Cutaneous angiofibromas are morphologically a diverse group of lesions including fibrous papules, pearly penile papules, periungual fibromas, familial myxovascular fibromas and acral fibrokeratomas. Multiple angiofibromas on the face, formerly termed

DOI: 10.1111/pde.12600

© 2015 Wiley Periodicals, Inc.

Figure 2. Histopathology shows (A) a verruciform epidermal architecture (hematoxylin and eosin, 2x) with (B) underlying stellate fibroblasts and an increase in dermal vasculature (hematoxylin and eosin, 20x).

adenoma sebaceum, are associated with tuberous sclerosis. Facial angiofibromas have also been reported in neurofibromatosis and multiple endocrine

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neoplasia type 1 (1). Histopathologically, angiofibromas are characterized by a dermal proliferation of stellate-shaped fibroblasts within a collagenous stroma with an increased number of dilated blood vessels. Collagen bundles tend to be arranged perpendicularly to the epidermis or in a concentric fashion around hair follicles and blood vessels. Elastic fibers are often few in number. We report the first case of a congenital cutaneous angiofibroma-like hamartoma. Congenital soft tissue angiofibromas have been reported within the maxilla (2) and within the soft tissue of the plantar foot (3) but not on the skin. The atypical appearance and discomfort with ambulation that our patient’s lesion caused prompted excision, with histopathology revealing the diagnosis. REFERENCES 1. Darling TN, Skarulis MC, Steinberg SM et al. Multiple facial angiofibromas and collagenomas in patients with

multiple endocrine neoplasia type 1. Arch Dermatol 1997;133:853–857. 2. Manjalay G, Hoare TJ, Pearman K et al. A case of congenital angiofibroma. Int J Pediatr Otorhinolaryngol 1992;24:275–278. 3. Lee JJ, Bredella MA, Springfield DS et al. Soft tissue angiofibroma: a case report. Skeletal Radiol 2014;43:403–407. Elizabeth M. Burgess, B.S.* Laura A. Greyling, M.D.† Loretta S. Davis, M.D.† *College of Medicine, University of Oklahoma, Oklahoma City, Oklahoma, †Section of Dermatology, Medical College of Georgia, Georgia Regents University, Augusta, Georgia Address correspondence to Laura A. Greyling, M.D., Dermatology Resident, PGY-3, Medical College of Georgia, Georgia Regents University, Section of Dermatology, 1004 Chafee Avenue, FH 100, Augusta, GA 30904, or e-mail: lthornsberry@ gmail.com.