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Dysmorphology of Cilia Association With Chronic Suppurative Respiratory Disease

Harry B. Neustein, MD; Joseph Church, MD; Seymour Cohen, MD
JAMA. 1979;241(22):2423. doi:10.1001/jama.1979.03290480057028.
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RECENTLY attention has been called to a small group of patients who were sterile, who had chronic lung disease, and whose sperm tails and respiratory tract cilia were abnormal when examined by electron microscopy.1,2 Cilia showed an absence of dynein arms, which contain adenosine triphosphatase (ATPase), and thus account for the lack of movement of the cilia. This absence of dynein arms (ATPase) was considered to be hereditary.1 Additional reports have noted abnormal cilia from the respiratory tracts of patients with several diseases of the lung.3,4 These descriptions led us to examine mucosa of the respiratory tract in selected patients with chronic lung disease. In one such patient with normal immunologic function, a bizarre morphological condition of cilia was found.

Report of a Case  An 8-year-old boy had chronic suppurative respiratory disease, which had its onset at age 1 month. He also had chronic purulent otitis media


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