Literature DB >> 19768645

LEOPARD syndrome with recurrent PTPN11 mutation Y279C and different cutaneous manifestations: two case reports and a review of the literature.

Ingrid Kalev1, Kai Muru, Rita Teek, Riina Zordania, Tiia Reimand, Kristel Köbas, Katrin Ounap.   

Abstract

LEOPARD syndrome (LS) is a heterogeneous disease characterised mainly by cutaneous manifestations. LEOPARD is the acronym for its major features-multiple lentigines, electrocardiographic conduction defects, ocular hypertelorism, pulmonary stenosis, abnormalities of (male) genitalia, retardation of growth and sensorineural deafness. As clinical manifestations are variable, molecular testing is supportive in the diagnosis of LS. We describe two unrelated LS cases with a common PTPN11 mutation Y279C and with completely different clinical features including distinct changes in skin pigmentation. In patient 1, the first complaint was hyperactive behaviour. First lentigines were presented at birth, but intensive growth began at the age of 2-4 years. Multiple dark lentigines were located mainly on the face and the upper part of the trunk, but the oral mucosa was spared. Patient 2 was born from induced labour due to polyhydramnion, and in the second week of life, mitral valve insufficiency and hypertrophic cardiomyopathy were diagnosed. Rapid growth of lentigines began at the age of 3 years. These are mostly located in the joint areas in the lower extremities; the face and upper trunk are spared from lentigines. In both cases, the rapid growth of lentigines made it possible to shift the diagnosis towards LS. Clinicians should give more consideration to rare genetic syndromes, especially in the case of symptoms from different clinical areas.

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Year:  2009        PMID: 19768645     DOI: 10.1007/s00431-009-1058-1

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  18 in total

1.  Clinical and molecular analysis of 30 patients with multiple lentigines LEOPARD syndrome.

Authors:  A Sarkozy; E Conti; M Cristina Digilio; B Marino; E Morini; G Pacileo; M Wilson; R Calabrò; A Pizzuti; B Dallapiccola
Journal:  J Med Genet       Date:  2004-05       Impact factor: 6.318

2.  Two novel and one recurrent PTPN11 mutations in LEOPARD syndrome.

Authors:  Rie Yoshida; Toshiro Nagai; Tomonobu Hasegawa; Eiichi Kinoshita; Toshiaki Tanaka; Tsutomu Ogata
Journal:  Am J Med Genet A       Date:  2004-11-01       Impact factor: 2.802

3.  Multiple lentigenes syndrome.

Authors:  R J Gorlin; R C Anderson; M Blaw
Journal:  Am J Dis Child       Date:  1969-06

4.  Mutations in PTPN11, encoding the protein tyrosine phosphatase SHP-2, cause Noonan syndrome.

Authors:  M Tartaglia; E L Mehler; R Goldberg; G Zampino; H G Brunner; H Kremer; I van der Burgt; A H Crosby; A Ion; S Jeffery; K Kalidas; M A Patton; R S Kucherlapati; B D Gelb
Journal:  Nat Genet       Date:  2001-12       Impact factor: 38.330

5.  Neurofibromatosis type I gene mutation in a patient with features of LEOPARD syndrome.

Authors:  R Wu; E Legius; W Robberecht; M Dumoulin; J J Cassiman; J P Fryns
Journal:  Hum Mutat       Date:  1996       Impact factor: 4.878

6.  PTPN11 mutations in LEOPARD syndrome.

Authors:  E Legius; C Schrander-Stumpel; E Schollen; C Pulles-Heintzberger; M Gewillig; J-P Fryns
Journal:  J Med Genet       Date:  2002-08       Impact factor: 6.318

7.  Grouping of multiple-lentigines/LEOPARD and Noonan syndromes on the PTPN11 gene.

Authors:  Maria Cristina Digilio; Emanuela Conti; Anna Sarkozy; Rita Mingarelli; Tania Dottorini; Bruno Marino; Antonio Pizzuti; Bruno Dallapiccola
Journal:  Am J Hum Genet       Date:  2002-06-07       Impact factor: 11.025

8.  LEOPARD syndrome: clinical diagnosis in the first year of life.

Authors:  M Cristina Digilio; Anna Sarkozy; Andrea de Zorzi; Giuseppe Pacileo; Giuseppe Limongelli; Rita Mingarelli; Raffaele Calabrò; Bruno Marino; Bruno Dallapiccola
Journal:  Am J Med Genet A       Date:  2006-04-01       Impact factor: 2.802

9.  Genotype-phenotype analysis and natural history of left ventricular hypertrophy in LEOPARD syndrome.

Authors:  Giuseppe Limongelli; Anna Sarkozy; Giuseppe Pacileo; Paolo Calabrò; Maria Cristina Digilio; Valeria Maddaloni; Giulia Gagliardi; Giovanni Di Salvo; Maria Iacomino; Bruno Marino; Bruno Dallapiccola; Raffaele Calabrò
Journal:  Am J Med Genet A       Date:  2008-03-01       Impact factor: 2.802

Review 10.  Leopard syndrome.

Authors:  Anna Sarkozy; Maria Cristina Digilio; Bruno Dallapiccola
Journal:  Orphanet J Rare Dis       Date:  2008-05-27       Impact factor: 4.123

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  6 in total

1.  Leopard syndrome: a report of five cases from one family in two generations.

Authors:  Fatima Begić; Husref Tahirović; Mediha Kardašević; Ingrid Kalev; Kai Muru
Journal:  Eur J Pediatr       Date:  2014-01-09       Impact factor: 3.183

Review 2.  Genetics of Short Stature.

Authors:  Youn Hee Jee; Anenisia C Andrade; Jeffrey Baron; Ola Nilsson
Journal:  Endocrinol Metab Clin North Am       Date:  2017-02-23       Impact factor: 4.741

Review 3.  Genetics of inherited cardiocutaneous syndromes: a review.

Authors:  Tara Bardawil; Samar Khalil; Christina Bergqvist; Ossama Abbas; Abdul Ghani Kibbi; Fadi Bitar; Georges Nemer; Mazen Kurban
Journal:  Open Heart       Date:  2016-11-22

4.  Do you know this syndrome? Leopard syndrome.

Authors:  Flávio Heleno da Silva Queiroz Cançado; Luis Candido Pinto da Silva; Paulo Franco Taitson; Ana Carolina Dias Viana de Andrade; Matheus Melo Pithon; Dauro Douglas Oliveira
Journal:  An Bras Dermatol       Date:  2017 Jan-Feb       Impact factor: 1.896

Review 5.  Mystery behind labial and oral melanotic macules: Clinical, dermoscopic and pathological aspects of Laugier-Hunziker syndrome.

Authors:  Ning Duan; Yang-Heng Zhang; Wen-Mei Wang; Xiang Wang
Journal:  World J Clin Cases       Date:  2018-09-26       Impact factor: 1.337

6.  Whole-exome sequencing identified a novel variant in an Iranian patient affected by pycnodysostosis.

Authors:  Ehsan Razmara; Homeyra Azimi; Amirreza Bitaraf; Mohammad Ali Daneshmand; Mohammad Galehdari; Maryam Dokhanchi; Elika Esmaeilzadeh-Gharehdaghi; Masoud Garshasbi
Journal:  Mol Genet Genomic Med       Date:  2020-01-15       Impact factor: 2.183

  6 in total

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