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)