Literature DB >> 12120419

A review of the literature of Bardet-Biedl disease and report of three cases associated with metabolic syndrome and diagnosed after the age of fifty.

S Iannello1, P Bosco, A Cavaleri, M Camuto, P Milazzo, F Belfiore.   

Abstract

Bardet-Biedl syndrome (BBS) is a genetic autosomal-recessive disease (formerly grouped with Laurence-Moon-Biedl syndrome but considered today as a separate entity) characterized by abdominal obesity, mental retardation, dysphormic extremities (syndactyly, brachydactyly or polydactyly), retinal dystrophy or pigmentary retinopathy, hypogonadism or hypogenitalism (limited to male patients) and kidney structural abnormalities or functional impairment. The expression and severity of the various clinical BBS features show inter- and intrafamilial variability. This study focuses on three cases of familial BBS--two sisters and one brother (66, 64 and 51 years of age, respectively)--with the main cardinal findings of the disease plus a classic 'metabolic syndrome' (characterized by abdominal obesity, atherogenic dyslipidaemia, raised blood pressure, insulin resistance with or without glucose intolerance, and prothrombotic risk and proinflammatory states). One female patient (not affected by reproductive dysfunction) had three healthy offspring, while the other two patients were unmarried. Another severely affected brother died at 70 years of age; two other brothers are lean but affected by nephropathy, retinopathy, slight mental retardation, polydactyly, hypertension and thrombotic diseases, and had healthy offspring. BBS is a rather rare but severe syndrome that is often mis- or undiagnosed. Ophthalmologists, endocrinologists and nephrologists should be aware of BBS because of its adverse prognosis--early onset of blindness, associated findings of metabolic syndrome and increased vascular risk, and severe renal impairment (the most frequent cause of reduced survival and death early in life).

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Year:  2002        PMID: 12120419     DOI: 10.1046/j.1467-789x.2002.00055.x

Source DB:  PubMed          Journal:  Obes Rev        ISSN: 1467-7881            Impact factor:   9.213


  12 in total

1.  Management of a 10-month-old child with a rare combination of Bardet-Biedl syndrome and ano-rectal malformation undergoing anterior sagittal ano-rectoplasty.

Authors:  Harihar V Hegde; Rohini Bhat Pai; Vijay G Yaliwal; Venkatesh M Annigeri; Anil B Halgeri; P Raghavendra Rao
Journal:  J Anesth       Date:  2011-10-15       Impact factor: 2.078

2.  Bardet-Biedl syndrome: a study of the renal and cardiovascular phenotypes in a French cohort.

Authors:  Olivier Imhoff; Vincent Marion; Corinne Stoetzel; Myriam Durand; Muriel Holder; Sabine Sigaudy; Pierre Sarda; Christian P Hamel; Christian Brandt; Hélène Dollfus; Bruno Moulin
Journal:  Clin J Am Soc Nephrol       Date:  2010-09-28       Impact factor: 8.237

3.  Sporadic familial ulnar hexadactyly of all four limbs.

Authors:  Uwe Wollina; Shyam B Verma
Journal:  J Dermatol Case Rep       Date:  2010-04-11

4.  Patients with Bardet-Biedl syndrome have hyperleptinemia suggestive of leptin resistance.

Authors:  Penelope P Feuillan; David Ng; Joan C Han; Julie C Sapp; Katie Wetsch; Emma Spaulding; Yuqian C Zheng; Rafael C Caruso; Brian P Brooks; Jennifer J Johnston; Jack A Yanovski; Leslie G Biesecker
Journal:  J Clin Endocrinol Metab       Date:  2011-01-05       Impact factor: 5.958

5.  Inactivation of Bardet-Biedl syndrome genes causes kidney defects.

Authors:  Deng-Fu Guo; Andreas M Beyer; Baoli Yang; Darryl Y Nishimura; Val C Sheffield; Kamal Rahmouni
Journal:  Am J Physiol Renal Physiol       Date:  2010-11-24

6.  Recurrence risks for Bardet-Biedl syndrome: Implications of locus heterogeneity.

Authors:  Julie C Sapp; Darryl Nishimura; Jennifer J Johnston; Edwin M Stone; Elise Héon; Val C Sheffield; Leslie G Biesecker
Journal:  Genet Med       Date:  2010-10       Impact factor: 8.822

7.  Phenotypic characterization of Bbs4 null mice reveals age-dependent penetrance and variable expressivity.

Authors:  Erica R Eichers; Muhammad M Abd-El-Barr; Richard Paylor; Richard Alan Lewis; Weimin Bi; Xiaodi Lin; Thomas P Meehan; David W Stockton; Samuel M Wu; Elizabeth Lindsay; Monica J Justice; Philip L Beales; Nicholas Katsanis; James R Lupski
Journal:  Hum Genet       Date:  2006-06-23       Impact factor: 4.132

8.  Adjustable gastric banding and sleeve gastrectomy in Bardet-Biedl syndrome.

Authors:  S Mujahid; M S B Huda; P Beales; P V Carroll; B M McGowan
Journal:  Obes Surg       Date:  2014-10       Impact factor: 4.129

9.  Obesity control and low protein diet preserve or even improve renal functions in Bardet-Biedl syndrome: a report of two cases.

Authors:  Erkan Dervisoglu; Serkan Isgoren; Derya Kasgari; Hakan Demir; Ahmet Yilmaz
Journal:  Med Sci Monit       Date:  2011-01

Review 10.  Alstrom syndrome (OMIM 203800): a case report and literature review.

Authors:  Tisha Joy; Henian Cao; Graeme Black; Rayaz Malik; Valentine Charlton-Menys; Robert A Hegele; Paul N Durrington
Journal:  Orphanet J Rare Dis       Date:  2007-12-21       Impact factor: 4.123

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