Uhlving Hilde Hylland, Mathiesen Sidsel, Buchvald Frederik, Green Kent, Heilmann Carsten, Gustafsson Per, Müller Klaus, Nielsen Kim Gjerum
Department of Pediatrics and Adolescent Medicine, Rigshospitalet, University of Copenhagen, Copenhagen, Denmark.
Research Unit Women's and Children's Health, Rigshospitalet, University of Copenhagen, Copenhagen, Denmark.
Pediatr Pulmonol. 2015 Jul;50(7):704-12. doi: 10.1002/ppul.23058. Epub 2014 May 21.
Chronic graft-versus-host disease (cGvHD) in the lungs is a life-threatening complication of allogeneic hematopoietic stem cell transplantation (HSCT). Pulmonary cGvHD is initiated in the peripheral airways, and diagnosis may be delayed by low sensitivity of standard pulmonary function tests. Multiple breath nitrogen washout (MBWN2 ) is a promising, sensitive method to assess small airways function. This is the first report on MBWN2 in survivors of pediatric HSCT.
This cross-sectional study undertaken 3-10 years post-HSCT, included 64 patients and 64 matched controls who all performed spirometry, whole-body plethysmography and MBWN2 . From MBWN2 the lung clearance index (LCI) and indices reflecting ventilation inhomogeneity arising close to the acinar lung zone (Sacin ) and in the conductive airway zone (Scond ) were derived. Subjective respiratory morbidity was assessed using the St. George Respiratory Questionnaire.
LCI, Sacin , and Scond were significantly higher in HSCT-patients compared with controls. Despite few reported symptoms and normal forced expiratory volume in 1 sec (FEV1 ) in 91%, LCI, Scond , and Sacin were abnormal in 34%, 52%, and 25% of HSCT-patients, respectively. LCI and Scond correlated weakly with spirometric findings in HSCT-patients, but not in controls. Scond was abnormal in 82% (9/11) of patients with evidence of cGvHD, and was associated with cGvHD in the multivariate analysis (r(2) = 0.26, P = 0.001).
Small airways dysfunction as measured by MBWN2 was a common finding at long term follow-up of children after allogeneic HSCT and was associated with cGvHD. The majority of these subjects had normal spirometric values and did not report any respiratory symptoms. Prospective studies are required to evaluate the long term clinical consequences of these signs of small airway disease and the value of MBWN2 as an early marker of pulmonary cGvHD.
肺部慢性移植物抗宿主病(cGvHD)是异基因造血干细胞移植(HSCT)的一种危及生命的并发症。肺部cGvHD始于外周气道,标准肺功能测试的低敏感性可能会延迟诊断。多次呼气氮洗脱(MBWN2)是一种有前景的、评估小气道功能的敏感方法。这是关于儿科HSCT幸存者MBWN2的首份报告。
这项在HSCT后3至10年进行的横断面研究纳入了64例患者和64例匹配的对照,所有患者均进行了肺量计检查、全身体积描记法和MBWN2检查。从MBWN2得出肺清除指数(LCI)以及反映肺泡肺区附近(Sacin)和传导气道区(Scond)通气不均匀性的指数。使用圣乔治呼吸问卷评估主观呼吸发病率。
与对照组相比,HSCT患者的LCI、Sacin和Scond显著更高。尽管报告的症状较少,且91%的患者一秒用力呼气量(FEV1)正常,但分别有34%、52%和25%的HSCT患者的LCI、Scond和Sacin异常。在HSCT患者中,LCI和Scond与肺量计检查结果的相关性较弱,但在对照组中并非如此。82%(9/11)有cGvHD证据的患者Scond异常,在多变量分析中Scond与cGvHD相关(r(2) = 0.26,P = 0.001)。
通过MBWN2测量的小气道功能障碍是异基因HSCT后儿童长期随访中的常见发现,且与cGvHD有关。这些受试者中的大多数肺量计值正常,且未报告任何呼吸道症状。需要进行前瞻性研究来评估这些小气道疾病体征的长期临床后果以及MBWN2作为肺部cGvHD早期标志物的价值。