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June 1, 1979

Dysmorphology of CiliaAssociation With Chronic Suppurative Respiratory Disease

Author Affiliations

From the Departments of Pathology (Dr Neustein), Pediatrics (Dr Church), and Surgery (Dr Cohen), and Divisions of Allergy (Dr Church) and Ear, Nose, and Throat (Dr Cohen), Childrens Hospital of Los Angeles; and The University of Southern California School of Medicine, Los Angeles (Drs Neustein, Church, and Cohen).

JAMA. 1979;241(22):2423. doi:10.1001/jama.1979.03290480057028

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