Ni Maomei, Yang Xiuhai
Department of Otorhinolaryngology, Guizhou Provincial People's Hospital, Guiyang, Guizhou 550002, P.R. China.
Exp Ther Med. 2017 Mar;13(3):1051-1053. doi: 10.3892/etm.2017.4072. Epub 2017 Jan 20.
Langerhansmporal histiocytosis (LCH) refers to a group of diseases that are characterized by the primary pathogenesis of an abnormal polyclonal proliferation of Langerhans cells that affect different structures of the human body, including the temporal bone. Thus far, the etiology of LCH remains unclear. Diagnosis of LCH is based on a synthetic analysis of the clinical presentations, as well as on features of imaging and histopathology. The clinical features, diagnosis, treatment and prognosis of LCH remain obscure, and temporal LCH is often confused with ear inflammatory lesions and malignant tumors. There are several therapeutic modalities for temporal bone LCH that include surgery, chemotherapy, radiotherapy and steroidal injections. The present study reports the case of an infant presenting a 1-month history of worsening left facial paralysis and a slowly enlarging post auricular mass followed by a 1-month history of postauricular swelling in the left ear. Computed tomography demonstrated a large mass of organized tissue. Moreover, the diagnosis of LCH was confirmed by histopathological and immunohistochemical examinations. The patient also suffered from multiple organ failure, including the liver, kidney, lymphatic system, skin, hematopoietic system and lungs. Following surgical intervention with mastoidotympanectomy, the parents of the patient refused further chemotherapy, and the patient succumbed to the disease ~6 months later.
朗格汉斯细胞组织细胞增多症(LCH)是指一组以朗格汉斯细胞异常多克隆增殖为主要发病机制的疾病,可累及人体不同结构,包括颞骨。迄今为止,LCH的病因仍不清楚。LCH的诊断基于临床表现、影像学和组织病理学特征的综合分析。LCH的临床特征、诊断、治疗和预后仍不明确,颞骨LCH常与耳部炎性病变和恶性肿瘤相混淆。颞骨LCH有多种治疗方式,包括手术、化疗、放疗和类固醇注射。本研究报告了一例婴儿病例,该婴儿有1个月逐渐加重的左侧面瘫病史和左耳后肿物缓慢增大病史,随后左耳出现耳后肿胀1个月。计算机断层扫描显示有一大块有组织的肿物。此外,通过组织病理学和免疫组织化学检查确诊为LCH。该患者还患有多器官功能衰竭,包括肝脏、肾脏、淋巴系统、皮肤、造血系统和肺部。在进行乳突鼓室切除术的手术干预后,患者父母拒绝进一步化疗,患者在约6个月后死于该疾病。