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1例儿童朗格汉斯细胞组织细胞增多症病例短期随访后血嗜酸性粒细胞增多症消退及张口受限情况

Resolution of blood eosinophilia and limited mouth opening after short-term follow-up in a pediatric Langerhans cell histiocytosis case.

作者信息

Reyes Magdalena Raquel Torres, Javaroni Julia Biliato, Nogueira Gustavo Milhomens, Zelaya Florence Juana Maria Cuadra, de León Anyeli Soraya Alas, Silveira Heitor Albergoni, León Jorge Esquiche

机构信息

Department of Pediatric Dentistry - Riberão Preto Dental School (FORP/USP), University of São Paulo, Ribeirão Preto, São Paulo Brazil.

Department of Pathology and Forensic Medicine, Ribeirão Preto Medical School (FMRP/USP), University of São Paulo, Ribeirão Preto, São Paulo Brazil.

出版信息

Indian J Otolaryngol Head Neck Surg. 2023 Sep;75(3):2568-2571. doi: 10.1007/s12070-023-03741-1. Epub 2023 May 2.

Abstract

INTRODUCTION

Langerhans cell histiocytosis (LCH) is an inflammatory myeloid neoplasia that often affects children, presenting a broad clinical spectrum.

METHODS

Here, we report a 13-year-old male Salvadorian patient who was referred presenting a nodular swelling at the mandibular angle region, mildly symptomatic, few weeks ago, which relevantly was associated with limited mouth opening. Intraoral examination was unremarkable. Imaginological exams revealed an osteolytic lesion affecting the vestibular cortex at the right mandibular angle. The blood test results were normal, except for eosinophilia (21%; absolute eosinophil count 4 × 10/L). After an incisional biopsy, microscopical and immunohistochemical analyses were consistent with LCH diagnosis, which corresponded to a single system-single site category. After a few weeks, the mandibular movements were re-established, and complete resolution of blood eosinophilia was observed.

CONCLUSION

LCH with blood eosinophilia is rarely reported. To our knowledge, 3 cases have been previously published.

摘要

引言

朗格汉斯细胞组织细胞增多症(LCH)是一种炎症性髓系肿瘤,常影响儿童,临床表现广泛。

方法

在此,我们报告一名13岁的萨尔瓦多男性患者,几周前因下颌角区域出现结节性肿胀前来就诊,症状轻微,伴有张口受限。口腔内检查未见明显异常。影像学检查显示右下颌角前庭皮质有溶骨性病变。血液检查结果正常,但嗜酸性粒细胞增多(21%;绝对嗜酸性粒细胞计数4×10/L)。切开活检后,显微镜和免疫组化分析结果与LCH诊断一致,属于单系统单部位类型。几周后,下颌运动恢复正常,血液嗜酸性粒细胞增多症完全消退。

结论

伴有血液嗜酸性粒细胞增多的LCH鲜有报道。据我们所知,此前已发表过3例。

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