Palaçon Mariana Paravani, Marques Marcelo Borges, Silveira Débora Fernandes Mendes, Martins Karina Helen, Silveira Heitor Albergoni, Pereira-Filho Valfrido Antonio, Bufalino Andreia, Chahud Fernando, León Jorge Esquiche
Department of Diagnosis and Surgery, Araraquara Dental School, FOAr/UNESP, Araraquara, São Paulo, Brazil.
Oral Pathology, Department of Stomatology, Public Oral Health, and Forensic Dentistry, Ribeirão Preto Dental School (FORP/USP), University of São Paulo, Ribeirão Preto, São Paulo, Brazil.
Head Neck Pathol. 2025 Feb 5;19(1):14. doi: 10.1007/s12105-024-01749-9.
Approximately 10% of all lymphomatoid papulosis (LyP) cases affect pediatric patients. Pediatric LyP is clinically similar to adult LyP, frequently with cutaneous involvement, whereas the affectation of mucosal surface is rare. The LyP clinicopathological spectrum includes type A, B, C, D, E, and LyP with DUSP22/IRF4 rearrangement, with LyP type D representing < 5% of all LyP cases.
We have previously reported 2 cases of exclusively intraoral LyP type C and E. To date, about 50 cases of LyP type D have been reported; of them, 8 cases, all but one with exclusive skin involvement, corresponded to pediatric patients. Rare LyP type C cases can lack CD30 expression, creating diagnostic difficulties with lymphoma.
Here, we report an extremely rare case of intraoral LyP type D with scarce/absent CD30 expression affecting a 12-year-old white Brazilian boy.
To our best knowledge, this is the first pediatric case of LyP type D with exclusive intraoral involvement.
所有淋巴瘤样丘疹病(LyP)病例中约10%累及儿科患者。儿科LyP在临床上与成人LyP相似,常累及皮肤,而累及黏膜表面的情况罕见。LyP的临床病理谱包括A、B、C、D、E型以及伴有DUSP22/IRF4重排的LyP,其中D型LyP占所有LyP病例的比例不到5%。
我们之前报告过2例仅累及口腔内的C型和E型LyP。迄今为止,已报告约50例D型LyP;其中8例为儿科患者,除1例之外均仅累及皮肤。罕见的C型LyP病例可能缺乏CD30表达,给淋巴瘤的诊断带来困难。
在此,我们报告1例极其罕见的口腔内D型LyP病例,该病例影响一名12岁的巴西白人男孩,其CD30表达稀少/缺失。
据我们所知,这是首例仅累及口腔的儿科D型LyP病例。