Literature DB >> 33269527

LZTR1-related spinal schwannomatosis and 7q11.23 duplication syndrome: A complex phenotype with dual diagnosis.

Karthik Muthusamy1, Maciej M Mrugala2,3,4,5, Bernard R Bendok3,6,7, Radhika Dhamija2,8.   

Abstract

BACKGROUND: Dual diagnoses in genetics practice are not uncommon and patients with dual diagnosis often present with complex and challenging phenotypes. A combination of meticulous phenotyping and molecular genetic techniques are essential in solving these diagnostic odysseys.
METHODS: Clinical features and genetic workup of a patient presenting with incidental schwannomatosis.
RESULTS: A 19-year-old male presented with incidental painless schwannomatosis in the background of macrocephaly, distinctive facies, and learning disability. Comprehensive genetic testing with gene panel and chromosomal microarray led to a dual diagnosis of LZTR1-related schwannomatosis and 7q11.23 duplication syndrome.
CONCLUSION: We emphasize the need for high index of suspicion and comprehensive genetic testing in complex phenotypes. Interrogation of the interplay between the pathogenic variants in multiple genes could improve our understanding of the pathophysiologic pathways and contribute to therapeutic discoveries.
© 2020 The Authors. Molecular Genetics & Genomic Medicine published by Wiley Periodicals LLC.

Entities:  

Keywords:  zzm321990LZRT1zzm321990; 7q11.23 duplication syndrome; Schwannomatosis; dual diagnoses; pain

Year:  2020        PMID: 33269527      PMCID: PMC7963420          DOI: 10.1002/mgg3.1560

Source DB:  PubMed          Journal:  Mol Genet Genomic Med        ISSN: 2324-9269            Impact factor:   2.183


  15 in total

1.  Familial 7q11.23 duplication with variable phenotype.

Authors:  Siddaramappa J Patil; Smrithi Salian; Venkaraman Bhat; Katta Mohan Girisha; Yash Shrivastava; Kiran Vs; Anilkumar Sapare
Journal:  Am J Med Genet A       Date:  2015-06-24       Impact factor: 2.802

2.  Debunking Occam's razor: Diagnosing multiple genetic diseases in families by whole-exome sequencing.

Authors:  T B Balci; T Hartley; Y Xi; D A Dyment; C L Beaulieu; F P Bernier; L Dupuis; G A Horvath; R Mendoza-Londono; C Prasad; J Richer; X-R Yang; C M Armour; E Bareke; B A Fernandez; H J McMillan; R E Lamont; J Majewski; J S Parboosingh; A N Prasad; C A Rupar; J Schwartzentruber; A C Smith; M Tétreault; A M Innes; K M Boycott
Journal:  Clin Genet       Date:  2017-03-13       Impact factor: 4.438

3.  Creation of an international registry to support discovery in schwannomatosis.

Authors:  K L Ostrow; A L Bergner; J Blakeley; D G Evans; R Ferner; J M Friedman; G J Harris; J T Jordan; B Korf; S Langmead; G Leschziner; V Mautner; V L Merker; L Papi; S R Plotkin; J M Slopis; M J Smith; A Stemmer-Rachamimov; K Yohay; A J Belzberg
Journal:  Am J Med Genet A       Date:  2016-10-19       Impact factor: 2.802

4.  The Human Phenotype Ontology: a tool for annotating and analyzing human hereditary disease.

Authors:  Peter N Robinson; Sebastian Köhler; Sebastian Bauer; Dominik Seelow; Denise Horn; Stefan Mundlos
Journal:  Am J Hum Genet       Date:  2008-10-23       Impact factor: 11.025

5.  Germline loss-of-function mutations in LZTR1 predispose to an inherited disorder of multiple schwannomas.

Authors:  Arkadiusz Piotrowski; Jing Xie; Ying F Liu; Andrzej B Poplawski; Alicia R Gomes; Piotr Madanecki; Chuanhua Fu; Michael R Crowley; David K Crossman; Linlea Armstrong; Dusica Babovic-Vuksanovic; Amanda Bergner; Jaishri O Blakeley; Andrea L Blumenthal; Molly S Daniels; Howard Feit; Kathy Gardner; Stephanie Hurst; Christine Kobelka; Chung Lee; Rebecca Nagy; Katherine A Rauen; John M Slopis; Pim Suwannarat; Judith A Westman; Andrea Zanko; Bruce R Korf; Ludwine M Messiaen
Journal:  Nat Genet       Date:  2013-12-22       Impact factor: 38.330

6.  7q11.23 Duplication syndrome: Physical characteristics and natural history.

Authors:  Colleen A Morris; Carolyn B Mervis; Alex P Paciorkowski; Omar Abdul-Rahman; Sarah L Dugan; Alan F Rope; Patricia Bader; Laura G Hendon; Shelley L Velleman; Bonita P Klein-Tasman; Lucy R Osborne
Journal:  Am J Med Genet A       Date:  2015-09-03       Impact factor: 2.802

7.  Clinical features of spinal schwannomas in 65 patients with schwannomatosis compared with 831 with solitary schwannomas and 102 with neurofibromatosis Type 2: a retrospective study at a single institution.

Authors:  Peng Li; Fu Zhao; Jing Zhang; Zhenmin Wang; Xingchao Wang; Bo Wang; Zhijun Yang; Jun Yang; Zhixian Gao; Pinan Liu
Journal:  J Neurosurg Spine       Date:  2015-09-25

8.  Resolution of Disease Phenotypes Resulting from Multilocus Genomic Variation.

Authors:  Jennifer E Posey; Tamar Harel; Pengfei Liu; Jill A Rosenfeld; Regis A James; Zeynep H Coban Akdemir; Magdalena Walkiewicz; Weimin Bi; Rui Xiao; Yan Ding; Fan Xia; Arthur L Beaudet; Donna M Muzny; Richard A Gibbs; Eric Boerwinkle; Christine M Eng; V Reid Sutton; Chad A Shaw; Sharon E Plon; Yaping Yang; James R Lupski
Journal:  N Engl J Med       Date:  2016-12-07       Impact factor: 91.245

Review 9.  A retrospective review of multiple findings in diagnostic exome sequencing: half are distinct and half are overlapping diagnoses.

Authors:  Erica D Smith; Kirsten Blanco; Samin A Sajan; Jesse M Hunter; Deepali N Shinde; Bess Wayburn; Mari Rossi; Jennifer Huang; Cathy A Stevens; Candace Muss; Wendy Alcaraz; Kelly D Farwell Hagman; Sha Tang; Kelly Radtke
Journal:  Genet Med       Date:  2019-03-21       Impact factor: 8.822

10.  Pain correlates with germline mutation in schwannomatosis.

Authors:  Justin T Jordan; Miriam J Smith; James A Walker; Serkan Erdin; Michael E Talkowski; Vanessa L Merker; Vijaya Ramesh; Wenli Cai; Gordon J Harris; Miriam A Bredella; Marlon Seijo; Alessandra Suuberg; James F Gusella; Scott R Plotkin
Journal:  Medicine (Baltimore)       Date:  2018-02       Impact factor: 1.889

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  2 in total

1.  Clinical characteristics and genetic testing outcome of suspected hereditary peripheral nerve sheath tumours in a tertiary cancer institution in Singapore.

Authors:  Jerold Loh; Pei Yi Ong; Denise Li Meng Goh; Mark E Puhaindran; Balamurugan A Vellayappan; Samuel Guan Wei Ow; Gloria Chan; Soo-Chin Lee
Journal:  Hered Cancer Clin Pract       Date:  2022-06-13       Impact factor: 2.164

2.  Comparison of the frequency of loss-of-function LZTR1 variants between schwannomatosis patients and the general population.

Authors:  Fanxuan Deng; D Gareth Evans; Miriam J Smith
Journal:  Hum Mutat       Date:  2022-04-14       Impact factor: 4.700

  2 in total

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