Case series of volar juvenile xanthogranuloma: Clinical observation of a peripheral rim of hyperkeratosis

Abstract

Juvenile xanthogranuloma is a benign histiocytic tumor predominantly occurring in children as yellowish papules on the head and trunk. Presentations on the volar surfaces are rare and may cause diagnostic confusion with pyogenic granuloma, eccrine poroma and digital fibrokeratoma. We report two patients with unusual presentations of solitary juvenile xanthogranuloma on the palm or sole. Both had lesions lacking the classic yellowish color and demonstrating a well-defined, peripheral hyperkeratotic rim. Histopathological evaluation revealed prominent orthokeratosis corresponding to the rim. Additional histological features, including dermal histiocytes and Touton giant cells, were consistent with the diagnosis of juvenile xanthogranuloma. Given the unusual locations and colors of the lesions, we conclude that histopathological evaluation is central to diagnosing volar juvenile xanthogranuloma. We additionally suggest that juvenile xanthogranuloma should be included in the differential diagnoses of volar lesions displaying a peripheral hyperkeratotic rim.

Solitary Tumor on the Palm of a Young Boy

“…The differential diagnosis of a solitary nodule on the palm includes eccrine poroma, pyogenic granuloma, tufted angioma, dermatofibroma, nevi, verruca, amelanotic melanoma, and solitary reticulohistiocytoma . This case demonstrates that although volar JXGs are rare, they should be added to the differential for solitary acral nodules in the appropriate clinical setting.…”

“…Numerous clinical forms of JXG beyond the classic description are being reported, making diagnosis difficult without knowledge of such variants . Occurrence of JXGs on acral surfaces is rare and can cause diagnostic confusion because of the absence of the classic yellowish hue with characteristic “setting sun” dermoscopy pattern and the unfamiliar presence of a well‐defined, hyperkeratotic rim . Among reported cases of JXG on the palm, the presence of a hyperkeratotic rim is a consistent finding.…”

A case of plantar localization of juvenile xanthogranuloma and review of the literature

“…Lezyonlar en sık baş ve boyunda lokalize olup bunu gövde, üst ekstremite ve alt ekstremite tutulumu izler 2 . Literatürde dudak, dil, göz kapağı, penis, tırnak yatağı, skrotum gibi atipik yerleşim alanları sunulmuş olup plantar yerleşim gösteren olgu sayısı PubMed veri tabanında yaptığımız taramaya göre sadece beş tanedir [3][4][5][6][7] . Literatürdeki olgularla beraber burada sunduğumuz kendi hastamızın özellikleri Tablo 1'de özetlenmiştir.…”

“…Mervak ve ark. 7 plantar tutulumlu olgularda bu bulgunun tanı koymaya yardımcı olabileceğini vurgulamışlardır. JKG, atipik lokalizasyon ve klinik görünümle seyrettiğinde kesin tanısını koyabilmek için histopatolojik inceleme ve özellikle JKG'si diğer histiyositozlardan ayırmak için yapılabilecek immünohistokimyasal boyamalar oldukça önemlidir.…”

Solitary palmar adult xanthogranuloma

“… 2 Case reports including cases of JXG on the volar surfaces (palms and soles) have described unusual colors appearing in this location, such as dull red or flesh-colored, and a well-defined hyperkeratotic peripheral rim upon dermoscopy. 3 Typical dermoscopic findings of adult and juvenile xanthogranuloma include a yellow/orange and red/pink homogeneous background, often referred to as the ‘setting sun’ pattern. 4 Clinical differential diagnosis includes several benign and malignant tumors, such as Spitz nevus, dermatofibroma, adnexal neoplasms, molluscum contagiosum, hemangioma, basal cell carcinoma, amelanotic melanoma, and lymphomas.…”