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  Atypical goblet cell hyperplasia in congenital cystic adenomatoid malformation as a possible preneoplasia for pulmonary adenocarcinoma in childhood: A genetic analysis

Stacher, E., Ullmann, R., Halbwedl, I., Gogg-Kammerer, M., Boccon-Gibod, L., Nicholson, A. G., et al. (2004). Atypical goblet cell hyperplasia in congenital cystic adenomatoid malformation as a possible preneoplasia for pulmonary adenocarcinoma in childhood: A genetic analysis. Human Pathology, 35(5), 565-570. doi:10.1016/j.humpath.2004.01.008.

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Item Permalink: http://hdl.handle.net/11858/00-001M-0000-0010-8853-6 Version Permalink: http://hdl.handle.net/11858/00-001M-0000-0010-8854-4
Genre: Journal Article
Alternative Title : Hum. Pathol.

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 Creators:
Stacher, Elvira, Author
Ullmann, Reinhard1, Author              
Halbwedl, Iris, Author
Gogg-Kammerer, Margit, Author
Boccon-Gibod, Liliane, Author
Nicholson, Andrew G., Author
Sheppard, Mary N., Author
Carvalho, Lina, Author
Franca, Maria Teresa, Author
MacSweeney, Fergus, Author
Morresi-Hauf, Alicia, Author
Popper, Helmut H., Author
Affiliations:
1Molecular Cytogenetics (Reinhard Ullmann), Dept. of Human Molecular Genetics (Head: Hans-Hilger Ropers), Max Planck Institute for Molecular Genetics, Max Planck Society, ou_1479645              

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Free keywords: congenital cystic adenomatoid malformation; atypical goblet cell hyperplasia; preneoplasia; adenocarcinoma; comparative genomic hybridization AAH, atypical adenomatous hyperplasia; AC, adenocarcinoma; AGCH, atypical goblet cell hyperplasia; BAC, bronchioloalveolar adenocarcinoma; CCAM, congenital cystic adenomatoid malformation; CGH, comparative genomic hybridization; DOP-PCR, degenerate oligonucleotide primer polymerase chain reaction
 Abstract: Congenital cystic adenomatoid malformation (CCAM) of the lung is a congenital lesion that is sometimes complicated by bronchioloalveolar adenocarcinoma (BAC). In some cases foci of atypical goblet cell hyperplasia (AGCH) can be found within the cysts. It has been proposed that CCAM and AGCH predispose to the development of BAC. The present study used comparative genomic hybridization (CGH) to screen 22 cases of CCAM (epithelium, surrounding normal lung tissue, and both preneoplastic and neoplastic lesions) for chromosomal imbalances. Of these 22 cases, 10 were CCAM type 1, 10 were type 2, and 2 were type 3. Of the 10 cases of CCAM type 1, 2 were associated with AGCH, 1 was associated with atypical adenomatous hyperplasia (AAH) and associated tubular adenocarcinoma (AC), and 2 were associated with BAC (1 mucinous and 1 predominantly nonmucinous). The present study also involved immunohistochemistry for interleukin (IL)-13, IL-4 receptor-small alpha, Greek (IL-4rsmall alpha, Greek), cytokines involved in the differentiation of goblet cells, and mucin 2 protein (Muc2). Chromosomal aberrations were not detected in the epithelium or the surrounding normal lung tissue, whereas varying aberrations were found in the neoplastic lesions. The most frequent genomic imbalances observed in both AGCH and the carcinomas were gains in chromosomes 2 and 4. Interestingly, a predominance of gains was also reported in AC of nonsmokers. Chromosomal aberrations in AGCHs arising in CCAMs support their preneoplastic status. Nuclear expression of IL-13, IL-4rsmall alpha, Greek, and Muc2 was detected in AGCH, whereas a cytoplasmic and nuclear reaction was seen in normal epithelium. This likely reflects an association with goblet cell differentiation, but it also drives proliferation in AGCH.

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Language(s): eng - English
 Dates: 2004-05-08
 Publication Status: Published in print
 Pages: -
 Publishing info: -
 Table of Contents: -
 Rev. Type: -
 Identifiers: eDoc: 225627
DOI: 10.1016/j.humpath.2004.01.008
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Title: Human Pathology
  Alternative Title : Hum. Pathol.
Source Genre: Journal
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Publ. Info: -
Pages: - Volume / Issue: 35 (5) Sequence Number: - Start / End Page: 565 - 570 Identifier: ISSN: 0046-8177