Literature DB >> 24124115

Multiple tumor types including leiomyoma and Wilms tumor in a patient with Gorlin syndrome due to 9q22.3 microdeletion encompassing the PTCH1 and FANC-C loci.

Livia Garavelli1, Maria Rosaria Piemontese, Alberto Cavazza, Simonetta Rosato, Anita Wischmeijer, Chiara Gelmini, Enrico Albertini, Giuseppe Albertini, Francesca Forzano, Fabrizia Franchi, Massimo Carella, Leopoldo Zelante, Andrea Superti-Furga.   

Abstract

Gorlin syndrome or nevoid basal cell carcinoma syndrome (NBCCS) is an autosomal dominant condition mainly characterized by the development of mandibular keratocysts which often have their onset during the second decade of life and/or multiple basal cell carcinoma (BCC) normally arising during the third decade. Cardiac and ovarian fibromas can be found. Patients with NBCCS develop the childhood brain malignancy medulloblastoma (now often called primitive neuro-ectodermal tumor [PNET]) in 5% of cases. The risk of other malignant neoplasms is not clearly increased, although lymphoma and meningioma can occur in this condition. Wilms tumor has been mentioned in the literature four times. We describe a patient with a 10.9 Mb 9q22.3 deletion spanning 9q22.2 through 9q31.1 that includes the entire codifying sequence of the gene PTCH1, with Wilms tumor, multiple neoplasms (lung, liver, mesenteric, gastric and renal leiomyomas, lung typical carcinoid tumor, adenomatoid tumor of the pleura) and a severe clinical presentation. We propose including leiomyomas among minor criteria of the NBCCS.
© 2013 Wiley Periodicals, Inc.

Entities:  

Keywords:  9q22.3 microdeletion; FANC-C; PTCH1; Wilms tumor; gorlin syndrome; leiomyoma; nevoid basal cell carcinoma syndrome (NBCCS)

Mesh:

Substances:

Year:  2013        PMID: 24124115     DOI: 10.1002/ajmg.a.36259

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  4 in total

1.  Deficient expression of aldehyde dehydrogenase 1A1 is consistent with increased sensitivity of Gorlin syndrome patients to radiation carcinogenesis.

Authors:  Aaron T Wright; Thierry Magnaldo; Ryan L Sontag; Lindsey N Anderson; Natalie C Sadler; Paul D Piehowski; Yannick Gache; Thomas J Weber
Journal:  Mol Carcinog       Date:  2013-11-27       Impact factor: 4.784

2.  Improvement of Basal Cell Carcinomas in Patients with Nevoid Basal Cell Carcinoma Syndrome Following by 5-Aminolevulinic Acid Photodynamic Therapy: A Case Report.

Authors:  Yan Jing Chen; Qin Yi; Yi Ming Li; Li Li
Journal:  Clin Cosmet Investig Dermatol       Date:  2021-12-16

3.  Microdeletion of 9q22.3: A patient with minimal deletion size associated with a severe phenotype.

Authors:  Adam D Ewing; Seth W Cheetham; James J McGill; Michael Sharkey; Rick Walker; Jennifer A West; Malcolm J West; Kim M Summers
Journal:  Am J Med Genet A       Date:  2021-05-07       Impact factor: 2.802

Review 4.  Update on Genetic Conditions Affecting the Skin and the Kidneys.

Authors:  Antonia Reimer; Yinghong He; Cristina Has
Journal:  Front Pediatr       Date:  2018-03-02       Impact factor: 3.418

  4 in total

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