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World J Diabetes. Jun 15, 2014; 5(3): 316-327
Published online Jun 15, 2014. doi: 10.4239/wjd.v5.i3.316
12q24 locus association with type 1 diabetes: SH2B3 or ATXN2?
Georg Auburger, Suzana Gispert, Suna Lahut, Özgür Ömür, Ewa Damrath, Melanie Heck, Nazlı Başak
Georg Auburger, Suzana Gispert, Ewa Damrath, Melanie Heck, Experimental Neurology, Goethe University Medical School, 60590 Frankfurt am Main, Germany
Suna Lahut, Özgür Ömür, Nazlı Başak, NDAL, Kuzey Park Building, Boğaziçi University, Bebek, 34342 Istanbul, Turkey
Author contributions: Auburger G proposed the manuscript concept and surveyed the literature; Gispert S, Lahut S, Ömür Ö, Damrath E, Heck M, Başak N generated relevant background data and expanded the manuscript.
Correspondence to: Dr. Georg Auburger, Professor, Experimental Neurology, Goethe University Medical School, Building 89, Theodor Stern Kai 7, 60590 Frankfurt am Main, Germany. auburger@em.uni-frankfurt.de
Telephone: +49-69-63017428 Fax: +49-69-63017142
Received: October 29, 2013
Revised: March 13, 2014
Accepted: April 11, 2014
Published online: June 15, 2014
Processing time: 229 Days and 22.6 Hours
Abstract

Genetic linkage analyses, genome-wide association studies of single nucleotide polymorphisms, copy number variation surveys, and mutation screenings found the human chromosomal 12q24 locus, with the genes SH2B3 and ATXN2 in its core, to be associated with an exceptionally wide spectrum of disease susceptibilities. Hematopoietic traits of red and white blood cells (like erythrocytosis and myeloproliferative disease), autoimmune disorders (like type 1 diabetes, coeliac disease, juvenile idiopathic arthritis, rheumatoid arthritis, thrombotic antiphospholipid syndrome, lupus erythematosus, multiple sclerosis, hypothyroidism and vitiligo), also vascular pathology (like kidney glomerular filtration rate deficits, serum urate levels, plasma beta-2-microglobulin levels, retinal microcirculation problems, diastolic and systolic blood pressure and hypertension, cardiovascular infarction), furthermore obesity, neurodegenerative conditions (like the polyglutamine-expansion disorder spinocerebellar ataxia type 2, Parkinson’s disease, the motor-neuron disease amyotrophic lateral sclerosis, and progressive supranuclear palsy), and finally longevity were reported. Now it is important to clarify, in which ways the loss or gain of function of the locally encoded proteins SH2B3/LNK and ataxin-2, respectively, contribute to these polygenic health problems. SH2B3/LNK is known to repress the JAK2/ABL1 dependent proliferation of white blood cells. Its null mutations in human and mouse are triggers of autoimmune traits and leukemia (acute lymphoblastic leukemia or chronic myeloid leukemia-like), while missense mutations were found in erythrocytosis-1 patients. Ataxin-2 is known to act on RNA-processing and trophic receptor internalization. While its polyglutamine-expansion mediated gain-of-function causes neuronal atrophy in human and mouse, its deletion leads to obesity and insulin resistance in mice. Thus, it is conceivable that the polygenic pathogenesis of type 1 diabetes is enhanced by an SH2B3-dysregulation-mediated predisposition to autoimmune diseases that conspires with an ATXN2-deficiency-mediated predisposition to lipid and glucose metabolism pathology.

Keywords: Diabetes mellitus type 1; 12q24; ATXN2; Obesity; SH2B3; Autoimmune

Core tip: Within the multifactorial pathogenesis of type 1 diabetes mellitus (T1D), a genetic risk mediated by the chromosome 12q24 locus was consistently observed. Mutations in the ATXN2 gene there trigger the pathogenesis of obesity, while mutations in the SH2B3 gene there trigger the pathogenesis of autoimmune processes. Given that both genes show co-regulated expression, their combined effects may drive these two core aspects of T1D. Tissue and phenotype studies of mouse mutants will identify molecular targets for causal therapies.