Cunha John Lennon Silva, de Carvalho Rejane Andrade, de Almeida Oslei Paes, de Carvalho Maria Goretti Freire, Soares Ciro Dantas
Department of Oral Diagnosis, Piracicaba Dental School, University of Campinas (UNICAMP), Piracicaba, Brazil.
Department of Dentistry, Federal University of Rio Grande do Norte (UFRN), Natal, Brazil.
Oral Oncol. 2022 May;128:105831. doi: 10.1016/j.oraloncology.2022.105831. Epub 2022 Apr 5.
Langerhans cell histiocytosis (LCH) is an uncommon myeloid neoplasm characterized by clonal neoplastic proliferation of Langerhans-type dendritic cells associated with a reactive inflammatory infiltrate composed predominantly of lymphocytes and eosinophils. Only three cases of LCH mimicking periapical lesions have been reported in the English-language literature to date. Herein, we report a rare case of LCH involving the mandible of a 45-years-old woman mimicking microscopically and radiographically a residual cyst. The patient underwent enucleation and curettage of the lesion. Microscopically, the lesion showed fibrous tissue with an intense inflammatory infiltrate and histiocytes with irregular to elongated nuclei with prominent nuclear grooves. The tumor cells were positive for S-100 protein, CD1a, and CD207. After careful evaluation through imaging tests to rule out lesions in other anatomical locations, the diagnosis was solitary LCH of the mandible. After four years of follow-up, the patient remained with no evidence of recurrence. This case emphasizes the importance of a carefully clinical, radiographic, and microscopical evaluation of bone lesions, including periapical or residual cysts, since some neoplasms can mimic common benign lesions of the jaws. Although conservative approaches to treating solitary mandibular bone lesions of LCH can be employed, long-term follow-up is strongly recommended.
朗格汉斯细胞组织细胞增多症(LCH)是一种罕见的髓系肿瘤,其特征为朗格汉斯型树突状细胞的克隆性肿瘤增殖,并伴有主要由淋巴细胞和嗜酸性粒细胞组成的反应性炎症浸润。迄今为止,英文文献中仅报道了3例酷似根尖周病变的LCH病例。在此,我们报告1例罕见的LCH病例,累及一名45岁女性的下颌骨,在显微镜和影像学上酷似残余囊肿。患者接受了病变的摘除和刮除术。显微镜下,病变显示为纤维组织,伴有强烈的炎症浸润以及细胞核不规则至细长且有明显核沟的组织细胞。肿瘤细胞S-100蛋白、CD1a和CD207呈阳性。通过影像学检查仔细评估以排除其他解剖部位的病变后,诊断为下颌骨孤立性LCH。经过4年的随访,患者无复发迹象。该病例强调了对骨病变,包括根尖周或残余囊肿,进行仔细的临床、影像学和显微镜评估的重要性,因为一些肿瘤可能酷似颌骨常见的良性病变。尽管可以采用保守方法治疗下颌骨孤立性LCH病变,但强烈建议进行长期随访。