Literature DB >> 31730237

The great mimicker: Phenotypic overlap between constitutional mismatch repair deficiency and Tuberous Sclerosis complex.

Mika Shapira Rootman1,2, Yael Goldberg3,2, Rony Cohen4,2, Nesia Kropach5,6,2, Inbal Keidar3, Rivka Friedland7,2, Gad Dotan8,2, Osnat Konen1,2, Helen Toledano9,2.   

Abstract

Constitutional mismatch repair deficiency is a rare cancer predisposition syndrome caused by biallelic mutations in one of the four mismatch repair genes. Patients are predisposed to various tumors including hematological malignancies, brain tumors and colorectal carcinomas. Phenotypic overlap with Neurofibromatosis-1 is well known, with most patients presenting with café-au-lait macules. Other common features include axillary and/or inguinal freckling and intracranial MRI foci of high T2W/FLAIR signal intensity similar to the typical FASI seen in Neurofibromatosis-1. In this cohort of eight patients with constitutional mismatch repair deficiency we describe overlapping phenotypical features with Tuberous Sclerosis complex. In addition to "ash-leaf like" hypomelanotic macules (five patients), we detected intracranial tuber-like lesions (three patients), renal cysts (three patients) and renal angiomyolipomas (two patients). All our patients also had Neurofibromatosis-1 like features, mainly café-au-lait macules. This study suggests that features of Tuberous sclerosis especially when overlapping with those of Neurofibromatosis 1 or malignancies atypical for these syndromes should raise the possibility of constitutional mismatch repair deficiency. Correct diagnosis is essential for appropriate genetic counseling and pre-emptive cancer surveillance.
© 2019 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  CMMRD; Neurofibromatosis-1; cancer syndromes; tuberous sclerosis

Mesh:

Year:  2019        PMID: 31730237     DOI: 10.1111/cge.13656

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  1 in total

1.  Performance of simultaneous multi-slice accelerated diffusion-weighted imaging for assessing focal renal lesions in pediatric patients with tuberous sclerosis complex.

Authors:  Azadeh Tabari; Fedel Machado-Rivas; John E Kirsch; Katherine Nimkin; Michael S Gee
Journal:  Pediatr Radiol       Date:  2020-08-26
  1 in total

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