Oral Presentation ESA-SRB-ANZBMS 2024 in conjunction with ENSA

Decoding the genomic architecture of endocrinopathies: a path to precision endocrinology (#32)

Sunita De Sousa 1
  1. Royal Adelaide Hospital, Adelaide, SA, Australia

[abstract to accompany Clinical Endocrinology Journal Early-Career Research Award application]

The precision medicine revolution with genotype-based prognostication and management compels us to improve the molecular diagnosis of endocrinopathies in our patients. This may be achieved by finding new causative genes and/or by refined genetic testing to improve variant detection in known genes. Through a number of recently completed and ongoing multicentre interdisciplinary Australian collaborations, we are decoding the genomic architecture of endocrinopathies using contemporary next-generation sequencing and bioinformatic methodologies.

Recent major outcomes include the identification and ongoing characterisation of novel pituitary tumorigenesis genes (e.g., PAM, CHEK2 ) (1, 2), new applications of known gene-disease relationships (e.g., BRAF in craniopharyngiomas, GCK in suspected monogenic diabetes), demonstration of novel variant types in established predisposition genes (e.g., copy number variation, deep intronic variants) (3, 4), and examination of the interplay between ethnicity and inherited endocrinopathies (e.g., white European vs. non-white European ethnicity in monogenic diabetes, hereditary pancreatitis in Indigenous Australians) (4, 5). New gene-disease relationships continue to be explored through whole exome/genome projects in the monogenic diabetes and hereditary pancreatitis settings.

To translate these scientific advances into improved diagnostic pathways, these data are being actively disseminated through the broad endocrinology, genetic pathology and clinical genetics communities via the development of cross-specialty guidelines (6) and national endocrine genetics multidisciplinary meetings run by the EndoGen network. Enhancing the molecular diagnosis of endocrinopathies at the basic scientific and clinical levels will be vital in delivering precision endocrinology to affected individuals and their families.

  1. De Sousa SMC, McCormack A, Orsmond A, et al. Increased prevalence of germline pathogenic CHEK2 variants in individuals with pituitary adenomas. J Clin Endocrinol Metab. 2024.
  2. De Sousa SMC, Shen A, Yates CJ, et al. PAM variants in patients with thyrotrophinomas, cyclical Cushing's disease and prolactinomas. Front Endocrinol (Lausanne). 2023;14:1305606.
  3. De Sousa SMC, Toubia J, Hardy TSE, et al. Aberrant Splicing of SDHC in Families With Unexplained Succinate Dehydrogenase-Deficient Paragangliomas. Journal of the Endocrine Society. 2020;4(12):bvaa071-bvaa.
  4. De Sousa SMC, Wu KHC, Colclough K, et al. Identification of monogenic diabetes in an Australian cohort using the Exeter maturity-onset diabetes of the young (MODY) probability calculator and next-generation sequencing gene panel testing. Acta Diabetol. 2024;61(2):181-8.
  5. Wu D, Bampton TJ, Scott HS... De Sousa SMC et al. The clinical and genetic features of hereditary pancreatitis in South Australia. Med J Aust. 2022;216(11):578-82.
  6. Milat F, Ramchand SK, Herath M... De Sousa SMC et al. Primary hyperparathyroidism in adults-(Part I) assessment and medical management: Position statement of the endocrine society of Australia, the Australian & New Zealand endocrine surgeons, and the Australian & New Zealand bone and mineral society. Clin Endocrinol (Oxf). 2024;100(1):3-18.