Abstract
Lymphatic plastic bronchitis (PB) most commonly occurs in children with
congenital heart disease as a result of secondary pulmonary lymphatic
flow disorder (PLFD). However, PB caused by primary PLFD is rare. The
clinical symptoms of lymphocytic PB are nonspecific; thus, the diagnosis
may be long delayed, especially in the absence of chylothorax. We made a
retrospective analysis of two children diagnosed with PB due to primary
PLFD, in order to contribute to further understanding of these
disorders. Patient 1, an eight-year-old boy, presented with chronic
productive cough and expectorated milky-white mucous plugs accompanied
by intermitted wheezing for one year. Patient 2, a nine-month-old girl,
presented with episodes of acute respiratory distress with expectoration
of milky-white bronchial casts for four months. There was no obvious
evidence of infection in either child. Bilateral thickening of
bronchovascular bundles and interlobular septal, as well as multiple
patchy ground-glass opacities were seen on chest computed tomography
(CT) in both patients. Lymphangioscintigraphy demonstrated pulmonary
lymph reflux in both patients and slowed lymphatic drainage of the lower
limbs in patient 1. Primary PLFD was considered for both patients, and a
diagnosis of yellow nail syndrome was made in patient 1. Both patients
received lymphatic interventional treatment, but all experienced
recurrence following the procedure.