Clinical features and treatment of stroke-like episodes in mitochondrial disease: a cohort-based study DOI Creative Commons

Nora Mickelsson,

Jussi Hirvonen, Mika H. Martikainen

и другие.

Journal of Neurology, Год журнала: 2024, Номер 272(1)

Опубликована: Дек. 12, 2024

Abstract Background Stroke-like episode (SLE) is a subacute evolving brain syndrome in patients with primary mitochondrial diseases. Despite previous research, the understanding of clinical spectrum, treatment, and outcomes SLEs far from complete. In this single centre study, we report symptoms radiological findings as well medical treatment disease. Methods This retrospective, observational study during years 2000–2023 was based on cohort diagnosed disease at Turku University Hospital (TUH; Turku, Finland) region Southwest Finland. Data were obtained hospital electronic record system. Results The investigated consisted 76 (37 men, 39 women) diagnosis Among these, 12 had history least one SLE; total number 20. most common genetic aetiology among m.3243A > G (N = 7). mean age first SLE 40 (range: 5–66 years), interval between episodes 4.8 4 months—10 years). duration varied 1 193 days (median 14 days, 37 days); 10 needed intensive care unit (ICU) treatment. survival time death 3.6 0–16 Conclusion Our highlights importance early recognition prompt management symptoms, especially epileptic seizures, life-threatening entity.

Язык: Английский

Mitochondria at the crossroads of health and disease DOI Creative Commons
Anu Suomalainen, Jodi Nunnari

Cell, Год журнала: 2024, Номер 187(11), С. 2601 - 2627

Опубликована: Май 1, 2024

Mitochondria reside at the crossroads of catabolic and anabolic metabolism—the essence life. How their structure function are dynamically tuned in response to tissue-specific needs for energy, growth repair, renewal is being increasingly understood. respond intrinsic extrinsic stresses can alter cell organismal by inducing metabolic signaling within cells distal tissues. Here, we review how centrality mitochondrial functions manifests health a broad spectrum diseases aging.

Язык: Английский

Процитировано

100

Mitochondrial diseases: from molecular mechanisms to therapeutic advances DOI Creative Commons
Hu Wen,

Hui Deng,

Bingyan Li

и другие.

Signal Transduction and Targeted Therapy, Год журнала: 2025, Номер 10(1)

Опубликована: Янв. 9, 2025

Abstract Mitochondria are essential for cellular function and viability, serving as central hubs of metabolism signaling. They possess various metabolic quality control mechanisms crucial maintaining normal activities. Mitochondrial genetic disorders can arise from a wide range mutations in either mitochondrial or nuclear DNA, which encode proteins other contents. These defects lead to breakdown metabolism, such the collapse oxidative phosphorylation, one mitochondria’s most critical functions. diseases, common group disorders, characterized by significant phenotypic heterogeneity. Clinical symptoms manifest systems organs throughout body, with differing degrees forms severity. The complexity relationship between mitochondria diseases results an inadequate understanding genotype-phenotype correlation these historically making diagnosis treatment challenging often leading unsatisfactory clinical outcomes. However, recent advancements research technology have significantly improved our management conditions. translations mitochondria-related therapies actively progressing. This review focuses on physiological mitochondria, pathogenesis potential diagnostic therapeutic applications. Additionally, this discusses future perspectives diseases.

Язык: Английский

Процитировано

4

A safety and absolute activity measurement method for Phi29 DNA polymerase based on chemiluminescent detection of dATP consumption DOI
Xue‐Feng Wang, Ping Gui, Xiao Li

и другие.

Analytica Chimica Acta, Год журнала: 2025, Номер unknown, С. 343952 - 343952

Опубликована: Март 1, 2025

Язык: Английский

Процитировано

0

Genetic Landscape and Mitochondrial Metabolic Dysregulation in Patients Suffering From Severe Long COVID DOI Creative Commons
Kristoffer Skaalum Hansen, Sofie E. Jørgensen, Cagla Cömert

и другие.

Journal of Medical Virology, Год журнала: 2025, Номер 97(3)

Опубликована: Март 1, 2025

ABSTRACT Long COVID represents a significant global health challenge with an unclear etiology. Alongside accumulating evidence of mitochondrial dysfunction in patients acute SARS‐CoV‐2 infection, symptomatic overlap exists between long and disorders. However, the genetic underpinnings have not been previously explored. We employed whole genome sequencing to analyze 13 severe identify defects related function. performed extracellular bioenergetics flux analysis on peripheral blood mononuclear cells proteomics evaluate cellular compared results those healthy controls. Our investigation identified 10 variants classified as pathogenic or likely 83 unknown significance affecting wide range mitochondria‐associated biological functions. Bioenergetics revealed altered ATP production rate four This study presents initial potential underlying predisposition while demonstrating energy capacity subset these patients. These findings open avenues for further research into role pathology suffering from may pave way targeted therapeutic strategies aimed at mitigating dysfunction.

Язык: Английский

Процитировано

0

Interferon Stimulated Gene Expression Is a Biomarker for Primary Mitochondrial Disease DOI Creative Commons
Nandaki Keshavan,

Lana Mhaldien,

Kimberly Gilmour

и другие.

Annals of Neurology, Год журнала: 2024, Номер unknown

Опубликована: Сен. 25, 2024

Objective Mitochondria are implicated in regulation of the innate immune response. We hypothesized that abnormalities interferon signaling may contribute to pathophysiology patients with primary mitochondrial disease (PMD). Methods Expression stimulated genes (ISGs) was measured by real‐time polymerase chain reaction (PCR) whole blood samples from a cohort PMD. Results Upregulated ISG expression observed high proportion (41/55, 75%) PMD on at least 1 occasion, most frequently IFI27 upregulation, seen 50% samples. Some had extremely levels, similar those interferonopathies. A statistically significant correlation between elevated gene and PMD, but not secondary dysfunction, suggesting upregulation is biomarker In some persisted repeat measurement over several years, indicative ongoing chronic inflammation. Subgroup analyses suggested common signatures mechanisms positive correlations severity among identical genetic diagnoses. Interpretation Dysregulated dysregulation contributor pathophysiology. This indicate role for repurposing immunomodulatory therapies treatment PMDs targeting signaling. ANN NEUROL 2024

Язык: Английский

Процитировано

3

An updated review of HSV-1 infection-associated diseases and treatment, vaccine development, and vector therapy application DOI Creative Commons
Dan Su,

Han Li-ping,

Chengyu Shi

и другие.

Virulence, Год журнала: 2024, Номер 15(1)

Опубликована: Ноя. 7, 2024

Herpes simplex virus type 1 (HSV-1) is a globally widespread that causes and associates with wide range of diseases, including herpes encephalitis, keratitis, labialis. The interaction between HSV-1 the host involves complex immune response mechanisms, recognition viral invasion, maintenance latent infection, triggering reactivation. Antiviral therapy core treatment for infections. Meanwhile, vaccine development employs different strategies methods, several promising types have emerged, such as live attenuated, protein subunit, nucleic acid vaccines, offering new possibilities prevention infection. Moreover, can be modified into therapeutic vector gene tumour immunotherapy. This review provides an in-depth summary infection-associated innate adaptive responses, disease pathogenesis, current approaches, recent advances in development, applications cancer treatment. Through systematic multiple aspects HSV-1, this study aims to provide comprehensive detailed reference public on prevention, control, HSV-1.

Язык: Английский

Процитировано

3

Caspase-11 drives macrophage hyperinflammation in models of Polg-related mitochondrial disease DOI Creative Commons

Jordyn J. VanPortfliet,

Yuanjiu Lei, Camila Guerra Martinez

и другие.

bioRxiv (Cold Spring Harbor Laboratory), Год журнала: 2024, Номер unknown

Опубликована: Май 14, 2024

Mitochondrial diseases (MtD) represent a significant public health challenge due to their heterogenous clinical presentation, often severe and progressive symptoms, the lack of effective therapies. Environmental exposures, such bacterial viral infection, can further compromise mitochondrial function exacerbate progression MtD. Infections in MtD patients more frequently progress sepsis, pneumonia, other detrimental inflammatory endpoints. However, underlying immune alterations that enhance immunopathology remain unclear, constituting key gap knowledge complicates treatment increases mortality this population. Here we employ vitro vivo approaches clarify molecular cellular basis for innate hyperactivity models polymerase gamma (Polg)-related We reveal type I interferon (IFN-I)-mediated upregulation caspase-11 guanylate-binding proteins (GBPs) increase macrophage sensing opportunistic microbe

Язык: Английский

Процитировано

2

STING exerts antiviral innate immune response by activating pentose phosphate pathway DOI Creative Commons

Dan‐Hui Wu,

Zilong Zhao,

Wei-Tao Yin

и другие.

Cell Communication and Signaling, Год журнала: 2024, Номер 22(1)

Опубликована: Дек. 18, 2024

The innate immune system serves as the host's first line of defense against invading pathogens. Stimulator interferon genes (STING) is a key component this system, yet its relationship with glucose metabolism, particularly in antiviral immunity, remains underexplored. Metabolomics analysis was used for detecting metabolic alterations spleens from STING knockout (KO) and wild-type (WT) mice. Co-immunoprecipitation employed determining ubiquitination TKT. Mass spectrometry interaction proteins STING. Enzyme activity kits were activities TKT G6PD. In study, we demonstrate that herpes simplex virus (HSV) infection activates pentose phosphate pathway (PPP) host cells, thereby initiating an response. Using STING-manipulated cells systemic mice, show positively regulates PPP, which, turn, limits HSV infection. Inhibition PPP significantly reduced production factors dampened STING-induced responses. Mechanistically, discovered interacts transketolase (TKT), enzyme non-oxidative branch reduces via E3 ubiquitin ligase UBE3A, stabilizing Silencing or inhibiting oxythiamine diminished factor production. Our findings reveal plays synergistic role generating during viral suggest activation could serve adjunct strategy therapy.

Язык: Английский

Процитировано

1

Clinical features and treatment of stroke-like episodes in mitochondrial disease: a cohort-based study DOI Creative Commons

Nora Mickelsson,

Jussi Hirvonen, Mika H. Martikainen

и другие.

Journal of Neurology, Год журнала: 2024, Номер 272(1)

Опубликована: Дек. 12, 2024

Abstract Background Stroke-like episode (SLE) is a subacute evolving brain syndrome in patients with primary mitochondrial diseases. Despite previous research, the understanding of clinical spectrum, treatment, and outcomes SLEs far from complete. In this single centre study, we report symptoms radiological findings as well medical treatment disease. Methods This retrospective, observational study during years 2000–2023 was based on cohort diagnosed disease at Turku University Hospital (TUH; Turku, Finland) region Southwest Finland. Data were obtained hospital electronic record system. Results The investigated consisted 76 (37 men, 39 women) diagnosis Among these, 12 had history least one SLE; total number 20. most common genetic aetiology among m.3243A > G (N = 7). mean age first SLE 40 (range: 5–66 years), interval between episodes 4.8 4 months—10 years). duration varied 1 193 days (median 14 days, 37 days); 10 needed intensive care unit (ICU) treatment. survival time death 3.6 0–16 Conclusion Our highlights importance early recognition prompt management symptoms, especially epileptic seizures, life-threatening entity.

Язык: Английский

Процитировано

0