We report a female with infantile onset of systemic lupus erythematosus secondary to C1q deficiency, in whom we identified a novel homozygous mutation in C1qB. The patient developed a progressive encephalopathy associated with spasticity, and suffered several arterial ischaemic strokes. Cerebral imaging demonstrated acquired intracranial calcification and a cerebral vasculopathy reminiscent of moyamoya. This case demonstrates overlap with some features of Aicardi-Goutières syndrome which, like C1q deficiency, is a monogenic cause of inflammation involving dysregulation of the innate immune system and stimulation of a type I interferon response.
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http://dx.doi.org/10.1177/0961203313486950 | DOI Listing |
BMC Endocr Disord
December 2024
Department of Biochemistry, Faculty of Medicine, Iran University of Medical Sciences, P.O. Box: 1449614525, Tehran, Iran.
Introduction: Many patients with polycystic ovary syndrome (PCOS) also experience thyroid disorders. There is a notable similarity in energy metabolism among PCOS, C1q/tumor necrosis factor (TNF)-related proteins (CTRP-5)deficiency, C3a/desArg (also known as acylation-stimulating protein (ASP)) deficiency, and hypothyroidism. This study aimed to investigate the relationship between serum levels of these factors and hypothyroidism in patients with PCOS.
View Article and Find Full Text PDFMol Neurodegener
December 2024
Xiamen Key Laboratory of Brain Center, The First Affiliated Hospital of Xiamen University, and Fujian Provincial Key Laboratory of Neurodegenerative Disease and Aging Research, Institute of Neuroscience, School of Medicine, Xiamen University, Xiamen, Fujian, 361102, China.
Background: The CD2-associated protein (CD2AP) was initially identified in peripheral immune cells and regulates cytoskeleton and protein trafficking. Single nucleotide polymorphisms (SNPs) in the CD2AP gene have been associated with Alzheimer's disease (AD). However, the functional role of CD2AP, especially its role in microglia during AD onset, remains elusive.
View Article and Find Full Text PDFInt J Biol Sci
December 2024
Department of Respiratory and Critical Care Medicine, Beijing Institute of Respiratory Medicine and Beijing Chao-Yang Hospital, Capital Medical University, Beijing 100020, China.
Macrophages play a crucial role in malignant pleural effusion (MPE), a frequent complication of advanced cancer. While C1q macrophages have been identified as a pro-tumoral cluster, direct evidence supporting the role of C1q-mediated macrophages remains to be elucidated. This study employed global and macrophage-specific knockout mice to investigate the role of C1q in MPE.
View Article and Find Full Text PDFCurr Opin Allergy Clin Immunol
December 2024
Paediatric Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle upon Tyne, United Kingdom.
Purpose Of Review: C1q deficiency is a rare inborn error of immunity characterized by susceptibility to severe infections and profound immune dysregulation, with a systemic lupus erythematosus-like phenotype. The management of patients with C1q deficiency is challenged by the rarity of this condition and the wide clinical variability. This review aims to emphasize the importance of a thorough immunological and clinical characterization to help guide a personalized and comprehensive approach to patients.
View Article and Find Full Text PDFJ Clin Immunol
October 2024
Paediatric Haematopoietic Stem Cell Transplant Unit, Great North Children's Hospital, Newcastle Upon Tyne, NE1 4LP, UK.
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