Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Case Reports
. 2024 Oct;15(28):1975-1988.
doi: 10.1111/1759-7714.15420. Epub 2024 Aug 18.

Bronchiolar adenoma/ciliated muconodular papillary tumor complicated by lymphoid interstitial pneumonia in a patient with Sjögren's disease: A case report and systematic review

Pinar Çağan  1 , Ali Kimiaei  1 , Seyedehtina Safaei  1 , Houssam Eddine Youcefi  1 , Alara Abu Saadeh  1 , Feride Yaman  2 , Özlem Yapıcıer  3 , Cemal Asim Kutlu  1
Affiliations
Case Reports

Bronchiolar adenoma/ciliated muconodular papillary tumor complicated by lymphoid interstitial pneumonia in a patient with Sjögren's disease: A case report and systematic review

Pinar Çağan et al. Thorac Cancer. 2024 Oct.

Abstract

Bronchiolar adenoma (BA)/ciliated muconodular papillary tumor (CMPT) is a rare pulmonary neoplasm, with less than 150 cases documented in the literature. We report a unique case of BA/CMPT complicated by lymphoid interstitial pneumonia (LIP) in a 55-year-old male with Sjögren's disease. This is the first documented instance of such a comorbidity. Through a systematic review of PubMed, we also summarize the demographic, clinical, radiological, histopathological, and treatment characteristics of CMPT.

Keywords: CMPT; Sjögren's disease; ciliated muconodular papillary tumor; lung tumor; lymphoid interstitial pneumonia.

PubMed Disclaimer

Conflict of interest statement

The authors declare no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Figures

FIGURE 1
FIGURE 1
PRISMA diagram illustrating the flow of study selection in the systematic review.
FIGURE 2
FIGURE 2
Computed tomography scans showing widespread ground‐glass opacities, septal thickening, fibro atelectatic changes and traction bronchiectasis.
FIGURE 3
FIGURE 3
X20, hematoxylin and eosin, nodules (arrows) located in the subpleural region.
FIGURE 4
FIGURE 4
X100, hematoxylin and eosin, adenoid and papillary structures with mucin pools.
FIGURE 5
FIGURE 5
X200, Goblet cell (black arrow), scattered immunopositivity for TTF1 (green arrow).
FIGURE 6
FIGURE 6
X100, CK7, diffuse positivity.
FIGURE 7
FIGURE 7
X200, basal cells showing positivity for p63.
FIGURE 8
FIGURE 8
X400, carcinoembryonic antigen (CEA), luminal immunopositivity.
See this image and copyright information in PMC

Similar articles

See all similar articles

Cited by

References

    1. Ishikawa Y. Ciliated muconodular papillary tumor of the peripheral lung: benign or malignant. Pathol Clin Med. 2002;20:964.
    1. Chang JC, Montecalvo J, Borsu L, Lu S, Larsen BT, Wallace WD, et al. Bronchiolar adenoma: expansion of the concept of ciliated Muconodular papillary tumors with proposal for revised terminology based on morphologic, Immunophenotypic, and genomic analysis of 25 cases. Am J Surg Pathol. 2018;42(8):1010–1026. - PMC - PubMed
    1. Nicholson AG, Tsao MS, Beasley MB, Borczuk AC, Brambilla E, Cooper WA, et al. The 2021 WHO classification of lung tumors: impact of advances since 2015. J Thorac Oncol. 2022;17(3):362–387. - PubMed
    1. Wang Y, Wang D, Wang J, Zhao S, Ren D, Chen G, et al. Primary ciliated muconodular papillary tumor: a rare pulmonary disease and literature review of 65 cases. Thorac Cancer. 2021;12(12):1917–1922. - PMC - PubMed
    1. Zhang X, Yuan W, Luo R, Luan L, Huang J, Lu S, et al. Ciliated Muconodular papillary tumors of the lung harboring STRN::ALK fusion: case report and review of the literature. Int J Surg Pathol. 2024;10668969241226707. - PubMed

MeSH terms

Supplementary concepts