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Calcium stone lithoptysis in promary ciliary dyskinesia
Citation:
GHIO, A. J., M. P. Kennedy, P. G. Noone, J. CARSON, P. L. Molina, M. A. Zariwala, S. L. Minnix, AND M. R. Knowles. Calcium stone lithoptysis in promary ciliary dyskinesia. RESPIRATORY MEDICINE. Elsevier Science Ltd, New York, NY, 101(1):76-83, (2007).
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Description:
BACKGROUND: An association between lithoptysis and primary ciliary dyskinesia (PCD) has not been previously reported. However, reports of lithoptysis from 2 older patients (>60 yr) prompted a study of this association. METHODS: We performed a prospective study of all PCD patients presenting to our institution between August 2003 and March 2006, seeking the symptom of lithoptysis or calcium deposition on radiology. A retrospective analysis of all PCD patients presenting prior to August 2003 was also performed. Patients age > or = 40 previously reviewed were recontacted. If a history of lithoptysis or calcium deposition was present, we further reviewed radiographic, microbiologic, and biochemical data, including serum calcium and phosphate. Broncholiths were analyzed by light and electron microscopy- and electron-dispersive X-ray analysis. RESULTS: In total, 142 patients (n=28 age > or = 40) were included, 41 in the prospective and 91 in the retrospective study. Lithoptysis was reported in 5 patients (all age > or = 40). Chest CT scans identified calcification (4/5), involving bronchiectatic airways in 3 patients and focal nodular calcification in 1 patient. Two other patients (age 46, 59) were identified with airway calcification without lithoptysis. Available broncholiths from 2 of these patients were composed of calcite, whereas a broncholith from 1 patient with focal nodular calcification contained calcium phosphate. Sputum was positive for Pseudomonas aeruginosa in all 7 patients, but negative for mycobacterial and fungal cultures. CONCLUSION: There is an association between lithoptysis and PCD in patients age > or = 40. We hypothesize that calcite stone formation is a biomineralization response to chronic airway inflammation and retention of infected airway secretions in PCD in a subset of PCD patients. PMID: 16757159 [PubMed - indexed for MEDLINE] Related LinksHigh-resolution CT of patients with primary ciliary dyskinesia. [AJR Am J Roentgenol. 2007][Clinical and ultrastructural study on primary ciliary dyskinesia] [Nihon Kyobu Shikkan Gakkai Zasshi. 1990]Effects of airway infection by Pseudomonas aeruginosa: a computed tomographic study. [Thorax. 1997]Markers of airway inflammation in primary ciliary dyskinesia studied using exhaled breath condensate. [Pediatr Pulmonol. 2006]Correlation of presentation and pathologic condition in primary ciliary dyskinesia. [Arch Otolaryngol Head Neck Surg. 2002]See all Related Articles...