Abstract:
BACKGROUND:Neonatal autosomal-recessive polycystic kidney disease (ARPKD) is associated with pulmonary hypoplasia and severe respiratory distress. There is no published information on long-term lung function in ARPKD survivors. METHODS:Pulmonary function tests, including spirometry and diffusion capacity, were performed in a nationwide cohort of Finnish paediatric patients with ARPKD. The annual incidence of respiratory infections and the need for permanent asthma medication were also evaluated in this population. RESULTS:Pulmonary function in 11 children surviving the neonatal period was good when measured after a median follow-up time of 10.4 years (range 5.4-16.1 years). None of the patients required oxygen supplementation, and only one patient had asthma. Patients who had received ventilator therapy during infancy had significantly lower maximal instantaneous forced expiratory flow (MEF%) (66%; 43-93% vs 105%; 63-110%; p=0.048) and forced expiratory volume/forced vital capacity (0.76; 0.7-0.81 vs 0.89; 0.77-0.91; p=0.03) than patients without a history of mechanical ventilation, suggesting tendency for airway obstruction in the former group of patients. The frequency of respiratory infections did not differ from Finnish paediatric population in general. CONCLUSIONS:The results of pulmonary function tests were within reference values for most patients with ARPKD, which suggested good long-term lung prognosis. Lung function tests should be considered for patients with ARPKD with a history of mechanical ventilation during infancy.
journal_name
Arch Dis Childjournal_title
Archives of disease in childhoodauthors
Jahnukainen T,Kirjavainen T,Luoto T,Ylinen E,Linkosalo L,Arikoski P,Pakarinen M,Jalanko Hdoi
10.1136/archdischild-2015-308451subject
Has Abstractpub_date
2015-10-01 00:00:00pages
944-7issue
10eissn
0003-9888issn
1468-2044pii
archdischild-2015-308451journal_volume
100pub_type
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