Interactive Cardiovascular and Thoracic Surgery 2:345-347(2003)
© 2003 European Association of Cardio-Thoracic Surgery
Alveolar adenoma: an unusual lung tumor
Alpaslan Cakana,
Ozgur Samancilara,
Deniz Nartb and
Ufuk Cagiricia,*
a Department of Thoracic Surgery, Ege University Medical Faculty, Izmir, Turkey
b Department of Pathology, Ege University Medical Faculty, Izmir, Turkey
* Corresponding author. Ege Universitesi Tip Fakultesi, Gogus Cerrahisi Anabilim Dali, 35100 Bornova, Izmir, Turkey. Tel.: +90-232-3434343; fax: +90-232-3390002
cagirici{at}med.ege.edu.tr
Received October 2, 2002;
received in revised form March 14, 2003;
accepted March 17, 2003
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Abstract
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A 34-year-old woman presented with chest pain and a solitary pulmonary nodule on chest roentgenogram. She was diagnosed with a rare benign lung tumor, alveolar adenoma of the lung, through pathological examination of the surgical specimen resected from the left upper lobe. Clinicians should also keep this entity in mind when making the differential diagnosis of a solitary pulmonary nodule.
Key Words: Alveolar adenoma; Pulmonary nodule
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1. Case report
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A 34-year-old housewife was admitted with the complaint of chest pain. She had no history of smoking or any significant pulmonary disease. A posteroanterior chest roentgenogram showed a coin lesion in the left middle zone (Fig. 1a). Subsequently, a thoracic computed tomography (CT) scan confirmed a 1.5-cm solitary pulmonary nodule in the left upper lobe (Fig. 1b). There were no pathologically enlarged mediastinal or hilar lymph nodes. Her physical examination was unremarkable. Routine blood chemistry was within normal limits while serological tests for hydatid disease were negative. A 12-mm induration was measured on tuberculin skin test. On flexible bronchoscopy, no abnormalities were detected, and the bronchial aspirate was reported to be cytologically benign. Thus, depending on the above diagnostic results, clinical and radiological observation of the nodule was decided to be suitable. However, when a CT scan showed enlargement of the nodule from 1.5 to 1.8 cm and appearance of the adjacent pleural thickening after an 8-month follow-up, surgical intervention was considered necessary for both diagnostic and therapeutic purposes.
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Fig. 1 (a) Posteroanterior spot radiograph shows a coin lesion in the left mid-zone of the thorax (arrows). (b) CT of the chest depicts a solitary nodule 1.5 cm in diameter, in the left upper lobe with sharp margins (arrow).
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Since video assisted thoracoscopic surgery (VATS) could not be undertaken because of intense pleural adhesions, a limited lateral thoracotomy was performed. After pleural adhesions were dissected, a well-circumscribed, ovoid solitary pulmonary nodule in the left upper lobe was excised by wedge resection. Frozen-section analysis indicated a benign lesion. The chest wall was closed after the insertion of a 32-Fr chest tube. After an uneventful 7-day postoperative period, the patient was discharged in cure. Twelve months after surgery, she was asymptomatic and had no recurrence or other abnormality on chest roentgenogram and CT scan.
Macroscopically, the lesion was 1.6 cm in diameter, and the sectioned surface was smooth, glistening in appearance. It had a gray-white color and some foci of hemorrhage. Under low power microscopic magnification, there was a multicystic lesion with no surrounding normal parenchyma (Fig. 2). The cysts were of varying size, and some of the cysts were filled with an eosinophilic, Periodic acid-Schiff (PAS)-positive granular material. The cysts were lined by an epithelium varying from flattened to cuboidal or hobnailed appearance. The prominent interstitial component contained collagen fibrils and displayed hipocellular thickened septa with few spindle-oval shaped cells. Staining for PAS and periodic acid-Schiff with diastase predigestion were negative, confirming the absence of cytoplasmic mucin in epithelial cells. In the interstitium, the collagen bundles were highlighted by Masson trichrome stain. In immunohistochemical investigations, the epithelial cells were diffusely positive for cytokeratin, epithelial membrane antigen, thyroid transcription factor-1 (TTF-1) and focally positive for carcinoembryogenic antigen. The interstitial cellular component was negative for cytokeratin. There was focal positivity for vimentin. CD 34 stained the vascular network but not the epithelial component.
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Fig. 2 Alveolar adenoma. The lesion consists of irregular shaped spaces lined with flat-cuboidal epithelium. (Hematoxylin and eosin stain; original magnification 100x).
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2. Discussion
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Alveolar adenoma is an unusual pulmonary neoplasm first described by Yousem and Hochholzer in 1986 [1]. It is a rare, benign tumor that is usually determined as a solitary pulmonary nodule in asymptomatic patients, more frequently in the middle-aged women [2]. Although most of the cases in a previously reported series were asymptomatic [3], the present case had chest pain. It was probably due to the pleural irritation caused by the peripherally located tumor. All the preoperative diagnostic procedures indicated a benign lesion; thus, a clinical and radiological follow-up was a reasonable option at the initial evaluation. Nevertheless, when enlargement of the lesion was detected, thoracotomy was performed to rule out malignancy. VATS could also be performed for the diagnostic and therapeutic management of the current case; however, massive pleural adhesions precluded its use, and the procedure was converted to open thoracotomy.
Alveolar adenoma represents a benign proliferation of both the alveolar epithelium and septal mesenchyma [4]. It is well demarcated with multiple cystic spaces containing a granular material. Most of the epithelial cells lining the cystic spaces are type 2 pneumocytes [5]. Positive staining of type 2 cells for TTF-1 supports this impression. The interstitial cellular component is consistently negative for cytokeratin.
Papillary adenoma, sclerosing hemangioma, atypical adenomatous hyperplasia (AAH), lymphangioma and hamartoma should be considered in the differential diagnosis. Prominent papillary pattern and heterogeneous epithelial component distinguish alveolar adenoma from papillary adenoma. Lymphangiomas contain lining cells that are cytokeratin-negative and endothelial marker-positive. Sclerosing hemangiomas may also form cystic spaces, but they contain blood, not an eosinophilic material. AAH represents an atypical bronchioloalveolar proliferation whereas alveolar adenomas lack cellular atypia [3].
In conclusion, alveolar adenoma of the lung is a rare benign tumor that should be added to the differential diagnosis of a solitary pulmonary nodule.
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Appendix A.
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ICVTS on-line discussion
Author: Sameh Sersar, Cardiothoracic surgeon, Mansoura University, Cardiothoracic Surgery, Mansoura 123, Egypt
Date: 03-Jun-2003 12:39
Message: A very interesting presentation of a difficult problem. I do not know why I have the suspicion of malignancy process. This may be due to the rapid rate of growth (20%) within 8 months. Does this immune stain profile not give you the impression of malignancy or a localized fibrous pleural mesothelioma? Have you repeated the pathological examination? Again, it is a very interesting subject to go through.
doi:10.1016/S1569-9293(03)00067-7
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References
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- Yousem SA, Hochholzer L. Alveolar adenoma. Hum Pathol. 1986;17:1066–1071[CrossRef][Medline]
- Menet E, Etchandy-Laclau K, Corbi P, Levillain P, Babin P. Alveolar adenoma: a rare peripheral pulmonary tumor. Ann Pathol. 1999;19:325–328[Medline]
- Burke LM, Rush WI, Khoor A, Mackay B, Oliveira P, Whitsett JA, Singh G, Turnicky R, Fleming MV, Koss MN, Travis WD. Alveolar adenoma: a histochemical, immunohistochemical, and ultrastructural analysis of 17 cases. Hum Pathol. 1999;30:158–167[CrossRef][Medline]
- Bohm J, Fellbaum C, Bautz W, Prauer HW, Hofler H. Pulmonary nodule caused by an alveolar adenoma of the lung. Virchows Arch. 1997;430:181–184[CrossRef][Medline]
- Koppl H, Freudenberg N, Berwanger I, Frenzer K, Bohm N. Alveolar adenoma of the lung. Immunohistochemical characterization of type II pneumocytes. Pathologe. 1996;17:150–153[CrossRef][Medline]
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Abstract
1. Case report