Pulmonary alveolar microlithiasis: an interesting case report with systematic review of Indian literature

被引:0
作者
Nidhya Ganesan
Marie Moses Ambroise
Anita Ramdas
King Herald Kisku
Kulwant Singh
Renu G’ Boy Varghese
机构
[1] Pondicherry Institute of Medical Sciences,Department of Pathology
[2] Pondicherry Institute of Medical Sciences,Department of Pulmonary Medicine
[3] BPS Government Medical College for Women,Department of Pathology
来源
Frontiers of Medicine | 2015年 / 9卷
关键词
pulmonary alveolar microlithiasis; alveolar microlithiasis; microliths; rare diseases;
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摘要
Pulmonary alveolar microlithiasis is a rare disease characterized by intra-alveolar presence of microliths. This study reports an interesting case of pulmonary alveolar microlithiasis and provides a systematic review of cases reported from India. A 23-year-old female presented with a history of cough, wheeze, chest pain, and episodic wheeze for five months. Pulmonary function tests demonstrated an obstructive pattern, and chest Xray showed fine micronodular opacities predominantly involving the middle and lower zones of both lungs. Transbronchial lung biopsy revealed the diagnosis. She responded well to inhaled steroid therapy. A systematic review of literature was performed and identified 73 cases of pulmonary alveolar microlithiasis reported from India. The mean (SD) age of the patients was 28.8 (14.9) years, with an almost equal male:female ratio. Many patients were asymptomatic at presentation. Breathlessness and cough were the most common symptoms, and the disease progressed into respiratory failure associated with cor pulmonale. About one-third of the cases were initially misdiagnosed and treated as pulmonary tuberculosis. Extra-pulmonary manifestations and comorbidities were also evident in our series. This systematic review helps to determine epidemiological and clinical characteristics of pulmonary alveolar microlithiasis. Further research is needed to elucidate the etiopathogenesis, diagnosis, and therapeutic options, which are beneficial in developing and identifying costeffective treatment for pulmonary alveolar microlithiasis.
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页码:229 / 238
页数:9
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共 382 条
[1]  
Huqun S(2007)Mutations in the Am J Respir Crit Care Med 175 263-268
[2]  
Izumi H(2003) gene are associated with pulmonary alveolar microlithiasis Respiration 70 549-555
[3]  
Miyazawa K(1962)Pulmonary alveolar microlithiasis. World cases and review of the literature Thorax 17 251-256
[4]  
Ishii B(1993)Pulmonary alveolar microlithiasis Thorax 48 171-173
[5]  
Uchiyama T(1997)Pulmonary alveolar microlithiasis: review of Turkish reports Eur J Epidemiol 13 587-590
[6]  
Ishida S(1965)Pulmonary alveolar microlithiasis: review of Italian reports Indian J Chest Dis 7 51-53
[7]  
Tanaka R(1967)Pulmonary alveolar microlithiasis Indian Heart J 19 47-56
[8]  
Tazawa S(1967)Chronic cor pulmonale due to pulmonary alveolar microlithiasis Indian J Pediatr 34 328-331
[9]  
Fukuyama T(1968)Pulmonary alveolar microlithiasis. Report of a case J Indian Med Assoc 50 427-428
[10]  
Tanaka Y(1971)Pulmonary alveolar microlithiasis J Assoc Physicians India 19 335-337