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Multiple endocrine neoplasia type 2 and autoimmune polyendocrine syndromes (type 1 diabetes mellitus and Graves' disease) in a 16-year-old male with Kabuki syndrome.
Park, Esther; Kim, Min-Sun; Noh, Eu Seon; Lee, Ji-Eun; Kim, Su Jin; Kwon, Young Se; Cho, Sung Yoon.
Affiliation
  • Park E; Department of Pediatrics, Jeonbuk National University Hospital, Jeonju, Korea.
  • Kim MS; Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.
  • Noh ES; Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.
  • Lee JE; Department of Pediatrics, Inha University Hospital, Inha University College of Medicine, Incheon, Korea.
  • Kim SJ; Department of Pediatrics, Inha University Hospital, Inha University College of Medicine, Incheon, Korea.
  • Kwon YS; Department of Pediatrics, Inha University Hospital, Inha University College of Medicine, Incheon, Korea.
  • Cho SY; Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.
Endocr J ; 69(10): 1211-1216, 2022 Oct 28.
Article in En | MEDLINE | ID: mdl-35676000
Multiple endocrine neoplasia type 2A (MEN2A) is caused by germline pathogenic variants in the RET proto-oncogene and is characterized by medullary thyroid cancer (MTC), pheochromocytoma, and hyperparathyroidism. Autoimmune polyendocrine syndromes (APS) are defined as multiple endocrine gland insufficiency associated with loss of immune tolerance. APS type 2 (APS-2) consists of at least two of the following diseases: type 1 diabetes mellitus (T1DM), autoimmune thyroid disease, and Addison's disease. We describe the clinical, molecular, and biochemical findings of MEN2A, APS-2, and Kabuki syndrome (KS) in a 16-year-old male. Whole exome sequencing was performed to identify the genetic cause of the pheochromocytoma and syndromic features including facial dysmorphism, developmental delay, and epilepsy. RET pathogenic variant and KMT2D pathogenic variant were identified, and he was diagnosed with MEN2A and KS. This is the first case of association between MEN2 and APS in adolescence and the second proven case in humans. In addition, this is the first report of MEN2 and APS in KS.
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Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Pheochromocytoma / Multiple Endocrine Neoplasia / Thyroid Neoplasms / Graves Disease / Polyendocrinopathies, Autoimmune / Adrenal Gland Neoplasms / Multiple Endocrine Neoplasia Type 2a / Diabetes Mellitus, Type 1 Type of study: Diagnostic_studies / Prognostic_studies Limits: Adolescent / Humans / Male Language: En Journal: Endocr J Journal subject: ENDOCRINOLOGIA Year: 2022 Type: Article

Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Pheochromocytoma / Multiple Endocrine Neoplasia / Thyroid Neoplasms / Graves Disease / Polyendocrinopathies, Autoimmune / Adrenal Gland Neoplasms / Multiple Endocrine Neoplasia Type 2a / Diabetes Mellitus, Type 1 Type of study: Diagnostic_studies / Prognostic_studies Limits: Adolescent / Humans / Male Language: En Journal: Endocr J Journal subject: ENDOCRINOLOGIA Year: 2022 Type: Article