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DOI 10.4049/jimmunol.1601415

Медицинская библиотека · Journal of immunology (Baltimore, Md. : 1950) · 2017

Dysregulation of B Cell Repertoire Formation in Myasthenia Gravis Patients Revealed through Deep Sequencing.

Vander Heiden Jason A, Stathopoulos Panos, Zhou Julian Q, Chen Luan, Gilbert Tamara J, Bolen Christopher R, Barohn Richard J, Dimachkie Mazen M, Ciafaloni Emma, Broering Teresa J, Vigneault Francois, Nowak Richard J, Kleinstein Steven H, O'Connor Kevin C

СтатьяПоддержка NIH: внешняя программаПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

Myasthenia gravis (MG) is a prototypical B cell-mediated autoimmune disease affecting 20-50 people per 100,000. The majority of patients fall into two clinically distinguishable types based on whether they produce autoantibodies targeting the acetylcholine receptor (AChR-MG) or muscle specific kinase (MuSK-MG). The autoantibodies are pathogenic, but whether their generation is associated with broader defects in the B cell repertoire is unknown. To address this question, we performed deep sequencing of the BCR repertoire of AChR-MG, MuSK-MG, and healthy subjects to generate ∼518,000 unique VH and VL sequences from sorted naive and memory B cell populations. AChR-MG and MuSK-MG subjects displayed distinct gene segment usage biases in both VH and VL sequences within the naive and memory compartments. The memory compartment of AChR-MG was further characterized by reduced positive selection of somatic mutations in the VH CDR and altered VH CDR3 physicochemical properties. The VL repertoire of MuSK-MG was specifically characterized by reduced V-J segment distance in recombined sequences, suggesting diminished VL receptor editing during B cell development. Our results identify large-scale abnormalities in both the naive and memory B cell repertoires. Particular abnormalities were unique to either AChR-MG or MuSK-MG, indicating that the repertoires reflect the distinct properties of the subtypes. These repertoire abnormalities are consistent with previously observed defects in B cell tolerance checkpoints in MG, thereby offering additional insight regarding the impact of tolerance defects on peripheral autoimmune repertoires. These collective findings point toward a deformed B cell repertoire as a fundamental component of MG.

Для цитирования: Vander Heiden Jason A, Stathopoulos Panos, Zhou Julian Q, Chen Luan, Gilbert Tamara J, Bolen Christopher R, Barohn Richard J, Dimachkie Mazen M, Ciafaloni Emma, Broering Teresa J, Vigneault Francois, Nowak Richard J, Kleinstein Steven H, O'Connor Kevin C. Dysregulation of B Cell Repertoire Formation in Myasthenia Gravis Patients Revealed through Deep Sequencing.. Journal of immunology (Baltimore, Md. : 1950). 2017. DOI: 10.4049/jimmunol.1601415.

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