Journal of Pathology and Translational Medicine

Case Report

Coexistence of Intrapulmonary Bronchogenic Cyst and Congenital Cystic Adenomatoid Malformation: A Case Report.: Mee Hye Oh, Eun Ah Jung, Ji Hye Lee, Hyun Deuk Cho, Ki Hyun Seo, Seock Yeol Lee, Young Tong Kim; Korean J Pathol. 2011;45(1):92-95.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.1.92

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Congenital cystic lesions of the lung are uncommon and a conjunction of two or more lesions is very rare. We report here on a case of coexisting intrapulmonary bronchogenic cyst and congenital cystic adenomatoid malformation in a 13-year-old female with a cystic mass in the right upper lobe of the lung. Computed tomography showed a cystic lesion measuring 2.5 cm with an air fluid level and surrounding multicystic lesions in the right upper lobe. On gross examination, the cut surface showed a cystic mass containing inspissated mucinous material, and the cystic mass was surrounded by multiple small cysts. Microscopically, the larger cystic cavity was lined with pseudostratified ciliated columnar epithelium. The submucosal tissue contained mucinous glands and plates of cartilage. The surrounding smaller cysts or irregular spaces were lined with bronchiolar-type respiratory epithelium. We propose that this hybrid lung lesion may represent the missing link in a common embryologic pathway determined by the timing of mesenchymal and epithelial interactions.

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Case 2: Coexisting Cystic Lesions of Lung in a Term Neonate: A Management Dilemma
Bichitrananda Raut, Aakriti Soni, Susanta Kumar Badatya, Satish Saluja, Manoj Modi, Arun Soni
NeoReviews.2018; 19(9): e542. CrossRef

Original Article

Immunoexpressions of Thyroid Transcription Factor-1 and bcl-2 in Congenital Cystic Adenomatoid Malformation.: Na Rae Kim, Dong Hoon Kim, Gou Young Kim, Dae Shick Kim, Joungho Han; Korean J Pathol. 2003;37(1):10-14.

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Abstract PDF: BACKGROUND
Congenital cystic adenomatoid malformation (CCAM) is a congenital abnormality of branching morphogenesis of the lung. Thyroid transcription factor-1 (TTF-1) is detected in human respiratory epithelial cells from 11 weeks of gestation, and at full term, TTF-1 expression is confined within type II epithelial cells and in some respiratory nonciliated bronchiolar epithelial cells. Immunoexpression of bcl-2 is intimately related to apoptosis during the development.
METHODS
To elucidate the nature of the lesion, TTF-1 expression was evaluated in twenty-four cases of CCAM (eight cases of type 1 and sixteen cases of type 2) along with immunostaining for bcl-2. For the control group, four cases of fetal lungs (19 week-, 21 week-, 27 week- and 40 week-gestational age) were also evaluated. In all cases of CCAM, TTF-1 was detected in the nuclei of epithelial cells lining the cysts.
RESULTS
TTF-1 was expressed in the majority of the bronchiolar-like epithelial cells of the cysts in CCAM types 1, and 2, where almost 100% of the lining cells of the cysts were TTF-1 positive with variable intensity, while negative TTF-1 expressions were found in the alveolar-like epithelium of the adjacent alveoli or distal nonciliated bronchi. For bcl-2 immunostaining, no lining epithelial cells of the cysts were stained except for the infiltrating lymphocytes. In the control group, strong immunoreactivities found in early fetal stages were absent in the full-term aged lung (40 gestational weeks).
CONCLUSION
These results support the hypothesis that CCAM types 1 and 2 reflect the abnormalities in lung morphogenesis and differentiation that are distinct from those for normally developed alveolar epithelium or adjacent bronchial epithelium, thus retaining the abnormal TTF-1 immunoreactions. Though restricted to CCAM types 1 and 2 in this study, CCAM might be related to TTF-1 rather than apoptosis in the morphogenesis of the developing lung.

Case Reports

Well Differentiated Adenosquamous Carcinoma of Lung Mimicking Benign Lesions in Fine Needle Aspiration Cytology: Report of a Case .: Jong Yup Bae, Hoon Kyu Oh, Jae Bok Park; J Pathol Transl Med. 2004;15(2):101-105.

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Abstract PDF: Fine needle aspiration (FNA) cytological examination is an appropriate method for the evaluation of pulmonary nodules. In major types of lung cancer, its diagnostic accuracy is quite high. However, it is sometimes difficult, using this technique, to differentiate between some unusual phenotypes including adenosquamous carcinoma, bronchioloalveolar carcinoma (BAC), neuroendocrine tumor, mucoepidermoid carcinoma, and sclerosing hemangioma. Here, we present a case involving extremely well differentiated adenosquamous carcinoma, mimicking benign lesions, such as pulmonary scar and adenomatoid malformation with squamous metaplasia. The patient was a 68-year-old man presenting with a solitary pulmonary nodule (1.6x1.6 cm), which was incidentally found at the periphery of the right lower lobe. FNA revealed some clusters of glandular cells with minimal atypia, in addition to squamous cells at a nearly full maturational state. Histological examination verified the cytological diagnosis on a lobectomy specimen. The tumor exhibited a well differentiated adenocarcinoma component, mimicking the bronchioles in scarred lung tissue, and a well differentiated squamous cell carcinoma component, mimicking the squamous cell nests of adenoacanthoma, in the other organs. In the present case, the possibility of adenosquamous carcinoma should have been considered if squamous cells were seen in the FNA from the peripheral pulmonary nodule, even though they appeared to be benign.

Adenocarcinoma Arising in Type 1 Congenital Cystic Adenomatoid Malformation: A Case Report and Review of the Literature.: Jinyoung Yoo, Sun Mi Lee, Ji Han Jung, Myeong Im Ahn, Deog Gon Cho, Seok Jin Kang, Kyo Young Lee; Korean J Pathol. 2008;42(6):396-400.

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Abstract PDF: Malignancies in congenital cystic adenomatoid malformations (CCAMs) of the lung are rare. We report a 41-year-old male patient with a pulmonary cystic lesion suspicious for CCAM, unrecognized until the patient was 40 years of age, and which subsequently became more consolidated during the interval between initial presentation and surgery. Microscopic examination of the resected specimen revealed features of type 1 CCAM with a mucinous adenocarcinoma, metastatic to the mediastinal lymph nodes. This case illustrates the importance of prompt surgical resection for all suspected CCAMs, especially those discovered in adulthood.

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