Literature DB >> 17211573

Multiple/bilateral renal tumors in patients with Birt-Hogg-Dubé syndrome.

Wahib Fahmy1, Ahmed S Safwat, Nabil K Bissada, Nancy Curry, Nabil Guirguis, Harry S Clarke, Mostafa Fraig, Alex Finkbeiner.   

Abstract

OBJECTIVE: To describe our findings in four patients with multiple/bilateral renal cell carcinoma (RCC) and Birt-Hogg-Dubé (BHD) syndrome. PATIENTS AND METHODS: A series of four patients with BHD syndrome and RCC is analyzed. Patient charts were reviewed for age, sex, presentation, various clinical manifestations, imaging, management and outcome.
RESULTS: Patients included 2 males and 2 females. Age ranged from 40 to 65 years (mean 56 years). The interval between the diagnosis of skin lesions characteristic of the disease and the development of renal tumors ranged between 1 and 35 years. Three of the patients had bilateral renal tumors (2 synchronous and one metachronous), one patient had multiple renal tumors in one kidney. In one patient the renal mass was diagnosed with a screening CT scan of the abdomen after the diagnosis of BHD syndrome. One patient had associated spontaneous pneumothorax and thyroid tumor. Only one of the 4 patients had prior family history of BHD syndrome. Renal tumors were clear cell type in 3 patients, and chromophobe tumor in one. Tumor size ranged from 2 to 9 cm.
CONCLUSION: BHD syndrome is associated with multiple diseases and tumors. We describe four patients with BHD syndrome with multiple or bilateral RCC. Two of the patients were asymptomatic. A high index of suspicion should be present in patients who present with the characteristic skin lesions of BHD syndrome and screening for the presence of renal tumors should be done in those patients. Long term follow up is necessary after treating renal tumors in these patients.

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Year:  2007        PMID: 17211573     DOI: 10.1007/s11255-006-9129-y

Source DB:  PubMed          Journal:  Int Urol Nephrol        ISSN: 0301-1623            Impact factor:   2.370


  11 in total

1.  Flecked chorioretinopathy associated with Birt-Hogg-Dubé syndrome.

Authors:  P Walter; B Kirchhof; B Korge; K Heimann
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  1997-06       Impact factor: 3.117

2.  Multiple lipomas, angiolipomas, and parathyroid adenomas in a patient with Birt-Hogg-Dube syndrome.

Authors:  J Y Chung; F A Ramos-Caro; B Beers; M J Ford; F Flowers
Journal:  Int J Dermatol       Date:  1996-05       Impact factor: 2.736

3.  Renal cancer in families with hereditary renal cancer: prospective analysis of a tumor size threshold for renal parenchymal sparing surgery.

Authors:  M M Walther; P L Choyke; G Glenn; J C Lyne; W Rayford; D Venzon; W M Linehan
Journal:  J Urol       Date:  1999-05       Impact factor: 7.450

4.  Bilateral renal cell carcinoma in the Birt-Hogg-Dubé syndrome.

Authors:  J S Roth; A D Rabinowitz; M Benson; M E Grossman
Journal:  J Am Acad Dermatol       Date:  1993-12       Impact factor: 11.527

5.  Hereditary multiple fibrofolliculomas, trichodiscomas and acrochordons: syndrome of Birt-Hogg-Dubè.

Authors:  M Scalvenzi; G Argenziano; E Sammarco; M Delfino
Journal:  J Eur Acad Dermatol Venereol       Date:  1998-07       Impact factor: 6.166

6.  Evaluation and management of renal tumors in the Birt-Hogg-Dubé syndrome.

Authors:  Christian P Pavlovich; Robert L Grubb; Kathleen Hurley; Gladys M Glenn; Jorge Toro; Laura S Schmidt; Carlos Torres-Cabala; Maria J Merino; Berton Zbar; Peter Choyke; McClellan M Walther; W Marston Linehan
Journal:  J Urol       Date:  2005-05       Impact factor: 7.450

7.  Birt-Hogg-Dubé syndrome: a novel marker of kidney neoplasia.

Authors:  J R Toro; G Glenn; P Duray; T Darling; G Weirich; B Zbar; M Linehan; M L Turner
Journal:  Arch Dermatol       Date:  1999-10

8.  Risk of renal and colonic neoplasms and spontaneous pneumothorax in the Birt-Hogg-Dubé syndrome.

Authors:  Berton Zbar; W Gregory Alvord; Gladys Glenn; Maria Turner; Christian P Pavlovich; Laura Schmidt; McClellan Walther; Peter Choyke; Gregor Weirich; Stephen M Hewitt; Paul Duray; Fathia Gabril; Cheryl Greenberg; Maria J Merino; Jorge Toro; W Marston Linehan
Journal:  Cancer Epidemiol Biomarkers Prev       Date:  2002-04       Impact factor: 4.254

9.  Parenchymal sparing surgery in patients with hereditary renal cell carcinoma: 10-year experience.

Authors:  J C Herring; E G Enquist; A Chernoff; W M Linehan; P L Choyke; M M Walther
Journal:  J Urol       Date:  2001-03       Impact factor: 7.450

10.  Mutations in the fumarate hydratase gene cause hereditary leiomyomatosis and renal cell cancer in families in North America.

Authors:  Jorge R Toro; Michael L Nickerson; Ming-Hui Wei; Michelle B Warren; Gladys M Glenn; Maria L Turner; Laveta Stewart; Paul Duray; Ousman Tourre; Nirmala Sharma; Peter Choyke; Pamela Stratton; Maria Merino; McClellan M Walther; W Marston Linehan; Laura S Schmidt; Berton Zbar
Journal:  Am J Hum Genet       Date:  2003-05-22       Impact factor: 11.025

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  5 in total

Review 1.  Diffuse Cystic Lung Disease. Part II.

Authors:  Nishant Gupta; Robert Vassallo; Kathryn A Wikenheiser-Brokamp; Francis X McCormack
Journal:  Am J Respir Crit Care Med       Date:  2015-07-01       Impact factor: 21.405

2.  Staged, open, no-ischemia nephron-sparing surgery for bilateral-multiple kidney tumors in a patient with birt-hogg-dubé syndrome.

Authors:  Ahmet Tefekli; Ayşe Deniz Akkaya; Kamil Peker; Terman Gümüş; Metin Vural; Fatin Cezayirli; Ahmet Musaoglu; Tarık Esen
Journal:  Case Rep Med       Date:  2012-04-24

3.  Appendiceal diverticulosis in a patient with family history of Birt-Hogg-Dubé syndrome--a case report.

Authors:  Alan Alexander; Kyle Hunter; Stephen Passerini; Roopa Bhat; Ambarish P Bhat
Journal:  Radiol Case Rep       Date:  2020-06-24

4.  Case Report of Birt-Hogg-Dubé Syndrome: Germline Mutations of FLCN Detected in Patients With Renal Cancer and Thyroid Cancer.

Authors:  Li Dong; Ming Gao; Wei-Jing Hao; Xiang-Qian Zheng; Yi-Gong Li; Xiao-Long Li; Yang Yu
Journal:  Medicine (Baltimore)       Date:  2016-05       Impact factor: 1.889

5.  Birt-Hogg-Dubé syndrome in Korean: clinicoradiologic features and long term follow-up.

Authors:  Joo Hee Lee; Min Joo Jeon; Joon Seon Song; Eun Jin Chae; Jin-Ho Choi; Gu-Hwan Kim; Jin Woo Song
Journal:  Korean J Intern Med       Date:  2018-10-26       Impact factor: 2.884

  5 in total

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