Intraoral Lymphomatoid Papulosis Type D Showing Scarce/Absent CD30 Expression in A Pediatric Patient: Case Report and Literature Review.

IF 4.1 Q2 PATHOLOGY Head & Neck Pathology Pub Date : 2025-02-05 DOI:10.1007/s12105-024-01749-9

Mariana Paravani Palaçon, Marcelo Borges Marques, Débora Fernandes Mendes Silveira, Karina Helen Martins, Heitor Albergoni Silveira, Valfrido Antonio Pereira-Filho, Andreia Bufalino, Fernando Chahud, Jorge Esquiche León

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Abstract

Purpose: 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.

Methods: 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.

Results: 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.

Conclusions: To our best knowledge, this is the first pediatric case of LyP type D with exclusive intraoral involvement.

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一名小儿患者口腔内淋巴瘤样丘疹病 D 型显示稀少/缺失 CD30 表达：病例报告与文献综述

目的：大约10%的淋巴瘤样丘疹病（LyP）病例影响儿科患者。儿童LyP的临床表现与成人LyP相似，常伴有皮肤受累，而粘膜表面的影响是罕见的。LyP临床病理谱包括A型、B型、C型、D型、E型和伴有DUSP22/IRF4重排的LyP，其中以D型LyP为代表。方法：我们先前报道了2例完全口服内的C型和E型LyP，迄今已报道的D型LyP约50例；其中8例均为小儿患者，除1例外均为皮肤受累。罕见的LyP C型病例缺乏CD30表达，给淋巴瘤的诊断带来困难。结果：在这里，我们报告了一例极其罕见的口腔内LyP D型伴CD30表达缺失的病例，患者为一名12岁的巴西白人男孩。结论：据我们所知，这是第一例单纯口腔内受累的儿童LyP D型病例。

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来源期刊

Head & Neck Pathology PATHOLOGY-

CiteScore

5.70

自引率

9.50%

发文量

期刊介绍： Head & Neck Pathology presents scholarly papers, reviews and symposia that cover the spectrum of human surgical pathology within the anatomic zones of the oral cavity, sinonasal tract, larynx, hypopharynx, salivary gland, ear and temporal bone, and neck. The journal publishes rapid developments in new diagnostic criteria, intraoperative consultation, immunohistochemical studies, molecular techniques, genetic analyses, diagnostic aids, experimental pathology, cytology, radiographic imaging, and application of uniform terminology to allow practitioners to continue to maintain and expand their knowledge in the subspecialty of head and neck pathology. Coverage of practical application to daily clinical practice is supported with proceedings and symposia from international societies and academies devoted to this field. Single-blind peer review The journal follows a single-blind review procedure, where the reviewers are aware of the names and affiliations of the authors, but the reviewer reports provided to authors are anonymous. Single-blind peer review is the traditional model of peer review that many reviewers are comfortable with, and it facilitates a dispassionate critique of a manuscript.