Alveolar adenoma combined with multifocal cysts: Case report and literature review

Journal of International Medical Research, Jun 2013

Alveolar adenoma is an extremely rare and benign pulmonary neoplasm; it is always asymptomatic and is usually detected incidentally on routine chest X-radiography. Typically on imaging examinations, alveolar adenoma exhibits as a peripheral, solitary, cystic nodule in the lung, which may easily imitate other lung lesions, consequently leading to difficulties in the differential diagnosis of this condition. Surgical resection is the primary treatment option. The diagnosis of alveolar adenoma is mainly based on postoperative histopathology, with features of proliferative type 2 alveolar epithelial cells and septal mesenchyme. The present case was a 60-year-old woman with alveolar adenoma, combined with systemic mutifocal cystic lesions. She underwent surgery following the obvious enlargement of this mass and a cystic nodule 7 cm in maximum diameter was resected. Postoperative histopathology confirmed a diagnosis of alveolar adenoma; her prognosis was favourable. In addition to reporting a rare case of alveolar adenoma coexisting with multifocal cysts, the English-language literature was reviewed for similar cases of alveolar adenoma.

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Alveolar adenoma combined with multifocal cysts: Case report and literature review

Xiang Wang 0 2 Wei-Qing Li 0 1 Hong-Zhu Yan 0 1 Yi-Ming Li 0 2 Jin He 0 1 Hui-Min Liu 0 1 Hong-Yu Yu 0 1 0 >> Version of Record - Jun 5, 2013 OnlineFirst Version of Record - May 7, 2013 What is This? 1 Department of Pathology, Changzheng Hospital, The Second Military Medical University , Shanghai, China 2 Department of Neurosurgery, Changzheng Hospital, The Second Military Medical University , Shanghai, China - Alveolar adenoma combined with multifocal cysts: Case report and literature review Journal of International Medical Research 41(3) 895906 ! The Author(s) 2013 Reprints and permissions: sagepub.co.uk/journalsPermissions.nav DOI: 10.1177/0300060513477304 imr.sagepub.com Introduction Alveolar adenoma is a rare pulmonary disease of uncertain histogenesis. Few sporadic cases have been reported since its first description in 1986.1 Alveolar adenoma represents a type of pulmonary adenoma, according to the World Health Organization histological classification of lung tumours.2 Other types of pulmonary adenoma include papillary adenoma, adenomas of salivary gland type (such as mucous gland adenoma *These authors contributed equally to this work. or pleomorphic adenoma) and mucinous cystadenoma.2 Alveolar adenoma typically presents as a solitary, circumscribed and peripheral nodule ,composed of a network of cystic spaces lined by type 2 alveolar epithelial cells.3 Most patients are asymptomatic and are diagnosed incidentally on routine chest X-radiography.4 Alveolar adenoma represents a slow-growing benign neoplasm with a good prognosis.4 The current report presents a case of alveolar adenoma combined with multifocal cysts, along with a review of the relevant Englishlanguage literature on alveolar adenoma. Case report Approval for the publication of this case report was obtained from the Committee on Ethics of Biomedicine Research, The Second Military Medical University, Shanghai, China. Written informed consent was obtained from the patient concerned. A 60-year-old woman presented to the Department of Thoracic Surgery in Changzheng Hospital, Shanghai, China, in May 2012. She informed the doctors that she had a 10-year history of a pulmonary mass of the right lung, with a diameter of 6 cm at initial diagnosis; the mass had shown enlargement on a recent chest X-radiography. At initial diagnosis, the patient was asymptomatic in terms of the respiratory system, therefore she received no treatment. In the only follow-up visit, which took place 3 months before admission to the study hospital, computed tomography (CT) of the patients lung had been undertaken at the outpatient clinic of the Department of Respiratory Medicine, at a local hospital in Hongkou District. This examination showed that the mass had increased in size from her initial diagnosis, and was now 7 cm in maximum diameter. The patient had a history of hypertension (2 years), type 2 diabetes (1 year), and left renal calculus (1 month) before admission to the authors hospital. She had been receiving 0.15 g irbesartan (once daily, orally, for 2 years) and 0.1 g acarbose (three times daily, orally, for 1 year) for the treatment of her hypertension and diabetes, respectively; her blood pressure and blood glucose levels were well controlled. The patient did not smoke and reported no family history of smoking or lung disease. The patient also had no family history of polycystic disease, such as polycystic kidney disease. Her physical examination was unremarkable. Routine urine analyses, (which included white and red blood cell counts, and protein, creatinine and urea nitrogen levels) were all within normal range. Chest X-radiography showed a nodular opacity in the right lower lobe, near the spine. Magnetic resonance imaging (MRI) of the chest and abdomen revealed a well-circumscribed solitary pulmonary nodule in the right lower lobe, with a maximum diameter of 7 cm. The nodule showed low-signal intensity on T1-weighted MRI (Figure 1A), high-signal intensity on T2-weighted MRI (Figure 1B), and thin rimenhancement on gadolinium-enhanced T1-weighted MRI (Figure 1C), all of which are characteristic of cysts. In addition, MRI revealed other cysts involving multiple organs, including a 2-cm cyst in the right breast (Figure 1D), a 1.5-cm cyst in the right lobe of the liver (Figure 1E), and several small cysts in the left kidney (Figure 1F). Considering the apparent enlargement of the pulmonary nodule and its uncertain histopathology, the patient was referred for surgical excision of the mass 3 days after admission, and a huge cyst in the right lower lobe of the lung was removed by thoracotomy. During surgery, a biopsy was taken and immediately underwent cryosection and histological analysis, which revealed a benign lesion with cystic spaces. Following this procedure, a wedge resection of the right lower lobe with the cystic mass was performed. Resected tissues contained a yellowish, soft, globular mass measuring 7 cm in diameter that was well circum (...truncated)


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Xiang Wang, Wei-Qing Li, Hong-Zhu Yan, Yi-Ming Li, Jin He, Hui-Min Liu, Hong-Yu Yu. Alveolar adenoma combined with multifocal cysts: Case report and literature review, Journal of International Medical Research, 2013, pp. 895-906, 41/3, DOI: 10.1177/0300060513477304