Genome-wide analysis implicates inner ear development in Ménière's disease
Zhuozheng Shi1,2, Ravi Mandla1,2, Jingjing Li1
1Department of Genetics, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
Abstract:
Ménière's disease (MD) is a chronic inner ear disorder characterized by recurrent vertigo, fluctuating sensorineural hearing loss, and tinnitus. Despite these distinctive symptoms, its etiology remains poorly understood. We performed a genome-wide meta-analysis of 8,969 cases and 1,962,542 controls across five large biobanks, identifying five independent genome-wide significant loci and estimating an observed-scale SNP heritability of 7% (SE 0.8%), consistent with a modest but significant genetic contribution to MD risk. Fine-mapping and integrative functional analyses implicate two convergent biological processes - developmental regulation of the inner ear, involving EYA4, EYA1, and LMO4 - and retinoic acid metabolism, with loci near CYP26A1/C1 and ALDH1A2 suggesting disrupted RA signaling in sensory and fluid-pressure homeostasis. These developmental regulator genes are robustly expressed in fetal and adult human inner ear cell types, supporting a model in which altered developmental programs predispose to adult vestibular and auditory dysfunction. Phenome-wide and genetic correlation analyses further reveal shared genetic architecture between MD and related traits, including vertigo, tinnitus, hearing loss, migraine, and sleep apnea, situating MD within a broader spectrum of sensory and neurological disorders. Collectively, these findings establish a genetic framework for Ménière's disease risk and implicate developmental regulators and retinoic acid signaling as key contributing pathways.
Insights
Ménière
Area of Science:
- Genetics
- Inner Ear Disorders
- Neuroscience
Background:
- Ménière's disease (MD) is a chronic inner ear condition with unknown causes.
- Symptoms include vertigo, hearing loss, and tinnitus.
- Understanding MD's genetic basis is crucial for diagnosis and treatment.
Purpose of the Study:
- To identify genetic loci associated with Ménière's disease risk.
- To explore the biological pathways contributing to MD pathogenesis.
- To investigate the genetic overlap between MD and related neurological and sensory disorders.
Main Methods:
- Genome-wide meta-analysis of over 8,900 MD cases and 1.9 million controls.
- Fine-mapping and integrative functional analyses of significant genetic loci.
- Phenome-wide and genetic correlation analyses with related traits.
Main Results:
- Identified five independent genome-wide significant loci for MD.
- Estimated SNP heritability at 7%, indicating a significant genetic contribution.
- Implicated inner ear developmental regulation (EYA4, EYA1, LMO4) and retinoic acid metabolism (CYP26A1/C1, ALDH1A2) pathways.
- Revealed shared genetic architecture with vertigo, hearing loss, migraine, and sleep apnea.
Conclusions:
- Established a genetic framework for Ménière's disease risk.
- Highlighted the role of developmental regulators and retinoic acid signaling in MD.
- Positioned MD within a broader spectrum of sensory and neurological disorders with shared genetic underpinnings.
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