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The great mimicker: Phenotypic overlap between constitutional mismatch repair deficiency and Tuberous Sclerosis complex.
Shapira Rootman, Mika; Goldberg, Yael; Cohen, Rony; Kropach, Nesia; Keidar, Inbal; Friedland, Rivka; Dotan, Gad; Konen, Osnat; Toledano, Helen.
Afiliação
  • Shapira Rootman M; Department of Radiology, Schneider Children's Medical Center of Israel, Petach Tikva, Israel.
  • Goldberg Y; The Sackler faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
  • Cohen R; The Raphael Recanati Genetic institute, Rabin Medical Center, Petach Tikva, Israel.
  • Kropach N; The Sackler faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
  • Keidar I; Neurology and epilepsy center, Schneider Children's Medical Center of Israel, Petach Tikva, Israel.
  • Friedland R; The Sackler faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
  • Dotan G; The Genetics unit, Schneider Children's Medical Center of Israel, Petach Tikva, Israel.
  • Konen O; The Department of Pediatrics "B", Schneider Children's Medical Center of Israel, Petach Tikva, Israel.
  • Toledano H; The Sackler faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.
Clin Genet ; 97(2): 296-304, 2020 02.
Article em En | MEDLINE | ID: mdl-31730237
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.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Esclerose Tuberosa / Síndromes Neoplásicas Hereditárias / Neoplasias Encefálicas / Neoplasias Colorretais / Reparo de Erro de Pareamento de DNA / Neoplasias Tipo de estudo: Diagnostic_studies Idioma: En Revista: Clin Genet Ano de publicação: 2020 Tipo de documento: Article

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Esclerose Tuberosa / Síndromes Neoplásicas Hereditárias / Neoplasias Encefálicas / Neoplasias Colorretais / Reparo de Erro de Pareamento de DNA / Neoplasias Tipo de estudo: Diagnostic_studies Idioma: En Revista: Clin Genet Ano de publicação: 2020 Tipo de documento: Article