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以纵隔淋巴结肿大为突出表现的儿童肺隐球菌病3例报告

Case Report: Three Pediatric Pulmonary Cryptococcosis Patients with Prominent Manifestation of Mediastinal Lymphadenopathy

  • 摘要: 本文报道3例以纵隔淋巴结肿大为突出表现的儿童肺隐球菌病。3例患儿既往均体健;2例有鸽子和家禽接触史。3例均表现为长期发热,伴轻咳;肺部无明显阳性体征,1例颈部淋巴结肿大;3例外周血白细胞、中性粒细胞及血清C-反应蛋白(CRP)均升高;2例血隐球菌荚膜多糖抗原阳性;3例体液免疫(IgA,IgM,IgG,IgE)及淋巴细胞亚群均正常,人类免疫缺陷病毒(HIV)抗体均阴性;3例患儿胸片及肺CT均示明显的纵隔淋巴结肿大,1例伴肺内结节。2例纵隔淋巴结活检示肉芽肿性炎,找到大量隐球菌孢子;此3例诊断肺隐球菌病,其中2例为确诊,1例为临床诊断。2例单独使用氟康唑治疗,合并脾播散的1例初始1个月联合两性霉素B治疗,继予氟康唑维持;总疗程5~9个月,均治愈。因此,免疫功能正常儿童肺隐球菌病可以纵隔淋巴结肿大为突出/孤立表现,在鉴别诊断中需考虑此点;氟康唑单用或者必要时加用两性霉素B治疗效果良好。

     

    Abstract: In this study, we report on three immunocompetent children with pulmonary cryptococcosis presenting mediastinal lymphadenopathy as the prominent manifestation. All three children were otherwise healthy previously. Two children had a history of exposure to pigeons and poultry. All three presented persistent fever accompanied by mild cough. There were no obvious positive signs in the lungs. One patient had enlarged cervical lymph nodes. All three had elevated levels of white blood cells, neutrophil count, and C-reactive protein (CRP). The levels of IgG, IgM, IgA, IgE and T cell subsets were normal in all cases, and they were all tested negative for HIV antibody. Two children were tested positive for serum cryptococcal antigen (sCRAG). The chest X-ray and pulmonary CT findings of the three patients all demonstrated marked enlargement of mediastinal lymph nodes, and one patient had nodules in the parenchyma. Surgical biopsies of mediastinal lymph nodes were performed in two children and large numbers of capsule spores were found in the histological examination. In the three cases, definitive diagnosis of pulmonary cryptococcosis were made in two patients, and clinical diagnosis was made in the third patient. Two patients were treated with fluconazole alone. The other patient whose condition was complicated with spleen infection was treated with fluconazole combined with amphotericin B for the first month, and was then given fluconazole for maintenance treatment. The overall treatment course lasted 5-9 months and all three were cured eventually. In conclusion, immunocompetent children with pulmonary cryptococcosis may present mediastinal lymphadenopathy as a prominent or isolated manifestation, which should be considered in differential diagnosis. Treatment with fluconazole alone or in combination with amphotericin B when it was necessary showed good therapeutic outcomes.

     

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