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In 25 patients suffering from chronic obstructive bronchitis, it has been shown that the administration of nifedipine brings about a decrease in the beat rate of nasal cilia which, in the individual case, may be very marked. The addition of salbutamol can more than compensate for this decrease in cilial beat rate.
We investigated the relationship between ciliary dyskinesia--commonly referred to as inmotile cilia syndrome--and sterility. In the past two years, we have accurately diagnosed 6 new cases of a total of 20 suspected as having this condition. To make the diagnosis, complete clinical, radiological, ultrastructural and spermatic work up was performed. All males in the fertile age were found to be sterile with spermatozoides with no motility. Sterility may initially go undetected in these patients due to early intense ORL and respiratory symptoms they present. However, mild forms of this disease entity may be asymptomatic and patients may consult for sterility with a clinical picture of scantily florid chronic bronchitis like that of smokers. Coincidental situs inversus may be useful in making the diagnosis.
Malignant lymphoma of the central nervous system in a thirteen-year-old boy with immotile cilia syndrome (ICS) is reported. He had frequent upper respiratory tract infections, chronic sinusitis and pneumonia during in childhood. Bronchiectasis was demonstrated by bronchography. The diagnosis of ICS was confirmed by the lack of dynein arms of cila in the nasal mucosa with electronmicroscopy. In 1987, he complained of headache and vomiting and a space occupied mass lesion in the left frontoparietal lobe was found by head CT scan, which was subtotally resected. Histological studies showed large cell type non-Hodgikin lymphoma of B-cell phenotype. He received radiotherapy (41Gy) to the whole brain and systemic chemotherapy consisting of adriamycin, cyclophosphamide, vincristine, prednisolone, L-asparaginase and intrathecal methotrexate, and the patient maintained complete remission for eight months. However, relapse occurred and the patient died twelve months after the initiation of treatment.
Immotile cilia syndrome is an uncommon disorder, with an autosomal recessive mode of inheritance; the incidence is about 1/25,000 and is a cause of chronic disease of both upper and lower airways, male infertility and, in 50%, situs inversus totalis. It's pathogenesis lies in a variety of genetically determined alteration of ciliary ultrastructure. We report three cases, two in siblings, who had a typical syndrome, all of them with situs inversus totalis (Kartagener syndrome), chronic nasal symptoms, sinusitis and serous otitis with hearing loss. Two had recurrent productive cough with chest X-ray changes suggestive of bronchiectasis. The diagnosis was established in two by bronchial biopsy, showing complete and partial dynein deficiency respectively. In the third case it was based an clinical grounds and familial history. All three children are doing well up to now.
The case of a patient with immotile cilia is reported. This syndrome is characterized by infertility caused by immotile spermatozoa and by bronchiectases owing to inadequate function of ciliated cells. Electron microscope studies showed gross abnormalities of the axial threads in the flagella of the spermatozoa examined.
Melanosis of the choroid plexus, ependyma, subependymal neuroglia of the fourth ventricle, and dentate and trigeminal motor nuclei was found in a 50-year-old man who died with cor pulmonale, hypertensive cardiovascular disease, and intracerebral hemorrhage. This case is the first known example of melanosis not associated with neurocutaneous melanosis to involve multiple structures of the brain. Nonenzymatic conversion of lipofuscin into melanin is suggested as the mechanism of the melanosis. Cilia of the choroidal epithelium and ependyma were identified in this case, despite a long delay in postmortem examination. These observations on choroidal epithelium confirm previous reports that the choroid plexus of man is ciliated, as in other mammals and lower vertebrates.
Atypical basal bodies from the oviductal mucosa of a female, 2.5-years-old cat suffering from immotile-cilia syndrome are described. The microtubular pattern was observed in 250 cross-sectional basal bodies. Four basal bodies (1.6%) had a defective number of microtubular triplets resulting in the appearance of so-called 'half-centriole' type. Two out of 100 longitudinal basal bodies showed duplicating striated rootlets and only one had an additional set of nine subdistal appendages, so that two basal feet arose from a single basal body. Electron-dense material and isolated microtubules into the lumen of some basal apparatuses were occasionally seen. It is known that centrioles and basal bodies show an unusual degree of constancy in size, shape and geometrical configuration, so that their abnormalities are very rare.
Sputum samples were collected from 15 immotile cilia syndrome (ICS) cases, 12 diffuse panbronchiolitis (DPB) cases, and 11 bronchiectasis without ICS (BE) cases, during stable clinical state, to clarify the physicochemical properties of sputum from patients with ICS and to compare them with the properties of sputum from patients with DPB and BE. We measured sputum rheological properties and concentrations of several biochemical components. In ICS, spinnability was higher than that in DPB. No significant difference was seen between ICS and the other cases regarding other rheological properties. Albumin was lower, but fucose, sialic acid, and IgA were higher in ICS than in DPB. Although no significant difference was seen between ICS and BE, the sialic acid/albumin ratio was higher and the sialic acid/fucose ratio was lower in ICS than in BE. These results revealed that sputum from ICS cases was characterized by an absolutely or relatively increased mucus component with high spinnability and by a decreased extravascular transudate component. In view of these rheological properties, sputum from ICS cases was not always indicated to be associated with efficiency in cough clearance. The results suggested that chronic airway inflammation in ICS is not such a serious problem compared with DPB and BE.
The effects of continuous oral administration of antibiotics in mice were investigated. Sulfamerazine, ampicillin, and chlortetracycline were tested at a rate of 500 mg/L of drinking water. Mice were infected by intranasal inoculation with 10(6) bacilli of the SMR strain of cilia-associated respiratory (CAR) bacillus. The mice were treated with the antibiotics starting 1 week before, 1 week after, or 4 weeks after the inoculation, for 5, 3, or 4 weeks respectively, then were examined. The infected mice lost body weight, and this loss was prevented or regained by all of the antibiotic treatments. Serologically no antibodies were detectable in the mice administered sulfamerazine starting 1 week before the inoculation. Mice administered sulfamerazine starting 1 week after the inoculation and ampicillin or chlortetracycline starting 1 week before or after the inoculation yielded a low titer of antibodies compared with nontreated infected mice. Mice administered antibiotics starting 4 weeks after the inoculation yielded the same titer of antibodies as nontreated infected mice. No pathologic respiratory tract lesions were observed in mice administered sulfamerazine starting 1 week before the inoculation. Mice administered sulfamerazine starting 1 week after the inoculation or ampicillin starting 1 week before or after the inoculation had slight peribronchitis without CAR bacillus colonization. Mice administered chlortetracycline, starting either 1 week before or after inoculation, developed peribronchitis, with colonization of the bacillus on the airway mucosa. In mice medicated starting 4 weeks after the inoculation, respiratory tract lesions developed, but their severity was reduced. The airway mucosa in mice treated with chlortetracycline was associated with the CAR bacillus but not in mice treated with sulfamerazine and ampicillin. These findings suggest that prevention and eradication of CAR bacillus infection is possible by treatment with sulfamerazine.
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