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Recherche de sous-groupes évolutifs et leur impact sur la survie : application à des patients atteints d'ataxie spinocérébelleuse (SCA)

Abstract : In cohort studies, most often the models used assume that the study population follows an average pattern of evolution. However, in many cases, such as for spinocerebellar ataxias (SCA), it is not uncommon for heterogeneity to be suspected. This heterogeneity could also be inuenced by other intercurrent events for example joint evolution of a second phenotype or occurrence of an event such as dropout or death. In the first part of this thesis, we analyzed the evolution of the BMI of SCA patients and looked for different evolution profiles of BMI. We identified 3 subgroups of BMI evolution: decrease (23% of patients), increase (18%) and stable (59%) ; and we have shown that patients who lower their BMI are faster disease progression. In the second part, we studied the survival of SCA patients and developed a prognostic nomogram. We have shown that it is different according on the genotype. Survival is shorter in SCA1, intermediate in SCA2 and SCA3, and longer in SCA6. Finally, we assessed the long-term impact of ataxia progression on survival. We have shown that progression of ataxia is associated with shorter survival regardless of genotype. Only in SCA1 patients, we identified three subgroups of homogeneous patients in terms of disease progression and risk of death.
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Alhassane Diallo. Recherche de sous-groupes évolutifs et leur impact sur la survie : application à des patients atteints d'ataxie spinocérébelleuse (SCA). Santé publique et épidémiologie. Sorbonne Université, 2018. Français. ⟨NNT : 2018SORUS447⟩. ⟨tel-02924998⟩

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