Genetic factors play a crucial role in determining human height. Short stature commonly affects multiple family members and therefore, familial short stature (FSS) represents a significant proportion of growth disorders. Traditionally, FSS was considered a benign polygenic condition representing a subcategory of idiopathic short stature (ISS). However, advancements in genetic research have revealed that FSS can also be monogenic, inherited in an autosomal dominant manner and can result from different mechanisms including primary growth plate disorders, growth hormone deficiency/insensitivity or by the disruption of fundamental intracellular pathways. These discoveries have highlighted a broader phenotypic spectrum for monogenic forms of short stature, which may exhibit mild manifestations indistinguishable from ISS. Given the overlapping features and the difficulty in differentiating polygenic from monogenic FSS without genetic testing, some researchers redefine FSS as a descriptive term that encompasses any familial occurrence of short stature, regardless of the underlying cause. This shift emphasizes the complexity of diagnosing and managing short stature within families, reflecting the diverse genetic landscape that influences human growth.

Download full-text PDF

Source
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC11693446PMC
http://dx.doi.org/10.3389/fendo.2024.1506323DOI Listing

Publication Analysis

Top Keywords

short stature
28
familial short
8
short
7
stature
7
fss
5
monogenic
4
monogenic familial
4
genetic
4
stature genetic
4
genetic factors
4

Similar Publications

Background: Male EBP disorder with neurologic defects (MEND syndrome) is an extremely rare disorder with a prevalence of less than 1/1,000,000 individuals worldwide. It is inherited as an X-linked recessive disorder caused by impaired sterol biosynthesis due to nonmosaic hypomorphic EBP variants. MEND syndrome is characterized by variable clinical manifestations including intellectual disability, short stature, scoliosis, digital abnormalities, cataracts, and dermatologic abnormalities.

View Article and Find Full Text PDF

Monogenic causes of familial short stature.

Front Endocrinol (Lausanne)

January 2025

Department of Pediatrics, 2nd Faculty of Medicine, Charles University in Prague and Motol University Hospital, Prague, Czechia.

Genetic factors play a crucial role in determining human height. Short stature commonly affects multiple family members and therefore, familial short stature (FSS) represents a significant proportion of growth disorders. Traditionally, FSS was considered a benign polygenic condition representing a subcategory of idiopathic short stature (ISS).

View Article and Find Full Text PDF

Functional characterization of novel compound heterozygous missense gene variants causing congenital dyshormonogenic hypothyroidism.

Front Endocrinol (Lausanne)

January 2025

Departamento de Bioquímica Clínica, Facultad de Ciencias Químicas, Universidad Nacional de Córdoba, Córdoba, Argentina.

Introduction: The sodium/iodide symporter (NIS) mediates active iodide accumulation in the thyroid follicular cell. Biallelic loss-of-function variants in the NIS-coding gene cause congenital dyshormonogenic hypothyroidism due to a defect in the accumulation of iodide, which is required for thyroid hormonogenesis.

Objective: We aimed to identify, and if so to functionally characterize, novel pathogenic gene variants in a patient diagnosed with severe congenital dyshormonogenic hypothyroidism characterized by undetectable radioiodide accumulation in a eutopic thyroid gland, as well as in the salivary glands.

View Article and Find Full Text PDF

Genetic variants of accessory proteins and G proteins in human genetic disease.

Crit Rev Clin Lab Sci

January 2025

Institute of Metabolism and Systems Research (IMSR), University of Birmingham, Birmingham, West Midlands, UK.

We present a series of three articles on the genetics and pharmacogenetics of G protein- coupled receptors (GPCR). In the first article, we discuss genetic variants of the G protein subunits and accessory proteins that are associated with human phenotypes; in the second article, we build upon this to discuss "G protein-coupled receptor (GPCR) gene variants and human genetic disease" and in the third article, we survey "G protein-coupled receptor pharmacogenomics". In the present article, we review the processes of ligand binding, GPCR activation, inactivation, and receptor trafficking to the membrane in the context of human genetic disease resulting from pathogenic variants of accessory proteins and G proteins.

View Article and Find Full Text PDF
Article Synopsis
  • Turner syndrome is a genetic disorder caused by the deletion of one X chromosome, leading to diverse karyotypes and phenotypes, but predicting phenotypes remains challenging due to mosaicism.
  • A study included 487 Turner women with non-mosaic X chromosome structural rearrangements and found prevalence rates of short stature (72.4%) and ovarian dysfunction (78.8%) linked to specific deletion groups.
  • Understanding the specific X chromosome breakpoints is crucial for managing Turner syndrome, particularly for predicting and addressing ovarian dysfunction and future fertility issues.
View Article and Find Full Text PDF

Want AI Summaries of new PubMed Abstracts delivered to your In-box?

Enter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!