Symmetrical Giant Facial Plaque-Type Juvenile Xanthogranuloma: A Case Report with a Successful Response to Fractional CO₂Laser Treatment

Abstract

Symmetrical giant facial plaque-type juvenile xanthogranuloma (SGFP-JXG) is a rare variant of juvenile xanthogranuloma, reported only in two cases in the literature. We report a case of a 3-year-old Korean boy who developed bilateral yellowish indurated plaques on both cheeks since 1 year after birth. A skin biopsy revealed numerous foam cells and Touton type giant cells throughout the upper dermis, and its immunohistochemical studies resulted positive for CD68 and negative for S-100. The boy was therefore diagnosed as a persistent SGFP-JXG. As the lesion did not show any signs of spontaneous regression, we performed a single session of fractional ablative CO2 laser, which resulted in a significant reduction of the lesion. This is the first case report of a persistent SGFP-JXG on which a single ablative laser therapy was performed with a successful outcome. (Ann Dermatol 31(2) 209∼212, 2019

Symmetrical Facial Giant Plaque-Type Juvenile Xanthogranuloma: Case Report and Review of the Literature

“…Our patient's lesions are smaller in size and the multiple flat plaques are composed of macular, papular, and nodular qualities, which form an inhomogeneous surface. Similar to the other cases, spared areas are well-demarcated [ 4 , 12 , 13 , 14 ].…”

“…Atypical clinical forms including keratotic, lichenoid, pedunculated, subcutaneous, clustered, intramuscular, plaque-type, and giant lesions seem to be very rare [ 7 , 8 , 9 ]. To the best of our knowledge, only eight plaque-type JXGs have been reported in the literature [ 4 , 8 , 9 , 10 , 11 , 12 , 13 , 14 ], of which four cases have been reported as SGFP-JXG [ 4 , 12 , 13 , 14 ]. The present case is the fifth SGFP-JXG.…”

“…In SGFP-JXG, extrafacial cutaneous sites with JXG were described, in 1 case on the elbow [ 12 ], and in other case body regions, such as abdomen, back, and upper extremities were affected [ 14 ]. Furthermore, none of the SGFP-JXG cases observed internal organ involvements [ 4 , 12 , 13 , 14 ].…”

“…Our patient had no spontaneous resolution 2 years after the disease started, although we observed a slight regression of the lesions over time. The four cases in the literature describing SGFP-JXG showed no regression over time for the patients aged 2-and-a-half [ 14 ], 3 [ 13 ], 7 [ 4 ], and 10 [ 12 ] years. Plaque and cluster types of JXG at extrafacial sites usually regress within 1 year with scarring and pigmentation despite the atypically large lesions [ 24 , 25 ].…”

“…In unresectable symptomatic visceral lesions, high dose steroids, LCH-based chemotherapy with vinca alkaloids, cyclosporine, and radiotherapy or a combination of the two have been used successfully [6, 7, 15]. Lee and Oh [13] described the first case report of a persistent SGFP-JXG on which a single fractional ablative laser therapy was performed with a significant reduction of the lesion.…”