Tillman Robert, Guillerman R Paul, Trojan Timothy, Silva-Carmona Manuel, Chinn Ivan K
Pediatric Pulmonary, Baylor College of Medicine, Texas Children's Hospital, Houston, TX, United States.
Pediatric Radiology, Baylor College of Medicine, Texas Children's Hospital, Houston, TX, United States.
Front Pediatr. 2019 Mar 29;7:105. doi: 10.3389/fped.2019.00105. eCollection 2019.
Granulomatous-Lymphocytic Interstitial Lung disease (GLILD) is a granulomatous and lymphoproliferative condition occurring in ~25% of Common Variable Immunodeficiency (CVID) patients with the highest prevalence in the late teen to young adult years. GLILD was first described in adults and carries a poor prognosis with survival estimated to be reduced by half. Here we report a pediatric case of CVID-associated GLILD that presented with rapid deterioration over 3 months and responded to adult-based treatment with dual chemotherapeutic agents (rituximab and azathioprine), resulting in complete resolution of clinical findings and near complete resolution of radiologic findings. This case highlights the opportunity to achieve a favorable outcome in GLILD following appropriate diagnosis and therapy.
肉芽肿性淋巴细胞间质性肺病(GLILD)是一种肉芽肿性和淋巴细胞增殖性疾病,约25%的常见变异型免疫缺陷(CVID)患者会出现这种疾病,在青少年晚期至青年期患病率最高。GLILD最早在成人中被描述,预后较差,估计生存率降低一半。在此,我们报告一例CVID相关的GLILD儿科病例,该病例在3个月内迅速恶化,采用基于成人的双化疗药物(利妥昔单抗和硫唑嘌呤)治疗后有反应,临床症状完全缓解,影像学表现几乎完全缓解。该病例突出了在适当诊断和治疗后GLILD获得良好预后的机会。