ArticleVirchows Archiv : an international journal of pathology2006
Natural history of the Nihon (Bhd gene mutant) rat, a novel model for human Birt-Hogg-Dubé syndrome.
Article in Virchows Archiv : an international journal of pathology, 2006. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers.
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Who cites it
12 citing papers in PubMed, 32 citations in OpenAlex.
- Choosing The Right Animal Model for Renal Cancer Research.Translational oncology · 2020Review
- Adult-Type Rhabdomyoma of the Larynx in Birt-Hogg-Dubé Syndrome: Evidence for a Real Association.Head and neck pathology · 2019Article
- Splice-site mutation causing partial retention of intron in the FLCN gene in Birt-Hogg-Dubé syndrome: a case report.BMC medical genomics · 2018Article
- Folliculin regulates ampk-dependent autophagy and metabolic stress survival.PLoS genetics · 2014Article
- Birt-Hogg-Dube syndrome is a novel ciliopathy.Human molecular genetics · 2013Article
- Birt-Hogg-Dube syndrome: clinicopathological features of the lung.Journal of clinical pathology · 2013Review
- Spontaneous renal tumors suspected of being familial in sprague-dawley rats.Journal of toxicologic pathology · 2012Article
- Loss of heterozygosity at the FLCN locus in early renal cystic lesions in dogs with renal cystadenocarcinoma and nodular dermatofibrosis.Mammalian genome : official journal of the International Mammalian Genome Society · 2009Article
- Article
- Interaction of folliculin (Birt-Hogg-Dubé gene product) with a novel Fnip1-like (FnipL/Fnip2) protein.Oncogene · 2008Article
- Spontaneous occurrence of a distinctive renal tubule tumor phenotype in rat carcinogenicity studies conducted by the national toxicology program.Toxicologic pathology · 2008Article
- Diagnostic biomarker of asbestos-related mesothelioma: example of translational research.Cancer science · 2007Review
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Authors and funding
6 authors at 2 institutions in 1 country.
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No grant is acknowledged in the PubMed record.
Abstract
In the Nihon rat, an established model of hereditary renal cell carcinoma (RCC), the propensity for tumor development, is inherited as an autosomal dominant trait due to a single germline nucleotide insertion mutation in the rat Bhd ortholog. The Birt-Hogg-Dubé (BHD) syndrome is a rare autosomal dominant disease characterized by fibrofolliculoma, pulmonary cysts, spontaneous pneumothorax, and renal neoplasm. The renal lesions of the Nihon rat are characterized, and extrarenal lesions are also described in this work. The earliest lesion of the RCC was identified as an altered tubule at as early as 3 weeks of age and rapidly progressed through adenoma to carcinoma with the primary cell type being clear/acidophilic where some similarities were evident to RCCs in BHD syndrome. The Nihon rats demonstrate a heterotopic ossification within RCCs and three extrarenal lesions, clear cell hyperplasia/adenoma of the endometrium, clear cell change of the epithelium of striated portions of salivary glands, and cardiac rhabdomyomatosis. This rat model of hereditary RCC provides a useful tool for analyzing the series of events leading to renal tumorigenesis and for studying BHD gene functions.
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