bims-tyki2d Biomed News
on Thymidine kinase 2 deficiency
Issue of 2025–12–28
four papers selected by
Zoya Panahloo, UCB



  1. Biochemistry (Mosc). 2025 Dec;90(12): 1849-1861
      The quantitative content of mitochondrial DNA (mtDNA) - a multicopy circular genome - is an important parameter relevant for function of mitochondrial oxidative phosphorylation (OxPhos) in cells, since mtDNA encodes 13 essential OxPhos proteins, 22 tRNAs, and 2 rRNAs. In contrast to the nuclear genome, where almost all lesions have to be repaired, the multicopy nature of mtDNA allows the degradation of severely damaged genomes. Therefore, cellular mtDNA maintenance and its copy number not only depend on replication speed and repair reactions. The speed of intramitochondrial mtDNA degradation performed by a POLGexo/MGME1/TWNK degradation complex and the breakdown rate of entire mitochondria (mitophagy) are also relevant for maintaining the required steady state levels of mtDNA. The present review discusses available information about the processes relevant for turnover of mitochondrial DNA, which dysbalance leads to mtDNA maintenance disorders. This group of mitochondrial diseases is defined by pathological decrease of cellular mtDNA copy number and can be separated in diseases related to decreased mtDNA synthesis rates (due to direct replication defects or mitochondrial nucleotide pool dysbalance) or diseases related to increased breakdown of entire mitochondria (due to elevated mitophagy rates).
    Keywords:  determinants of cellular mtDNA content; mtDNA degradation; mtDNA maintenance; mtDNA maintenance disorders; mtDNA replication
    DOI:  https://doi.org/10.1134/S0006297925602485
  2. J Transl Med. 2025 Dec 23. 23(1): 1415
      Mitochondria function as the primary energy hubs of cells and possess semi-autonomous genetic characteristic. Mutations in mitochondrial DNA (mtDNA) frequently lead to severe illness and even premature death. The rapid advancement of gene therapy offers promising potential for correcting such disorders. This review first aims to delineate the mechanisms of gene therapy strategies applicable to mitochondrial diseases, including the allotopic expression of mtDNA in the nucleus, mitochondrial-targeted nuclease cleavage, and mtDNA-targeted base editing. It also discusses in detail the clinical efficacy of mtDNA allotopic expression and the preclinical progress of other strategies. Furthermore, the unique physiological features of mitochondria, such as heteroplasmy and independent molecular transport mechanisms, pose distinct challenges for the clinical implementation of mitochondrial gene therapy strategies. Accordingly, this review elaborates on the current limitations of each approach. Finally, it highlights potential optimization directions to address these challenges, emphasizing that understanding heteroplasmy dynamics and their corresponding phenotypes, ensuring the safe delivery and tissue-specific expression of therapeutic elements, and maintaining long-term therapeutic specificity and efficiency are essential for the clinical translation of mitochondrial gene therapy.
    Keywords:  Allotopic expression; Base editing; Mitochondrial DNA; Mitochondrial disorders; Nuclease
    DOI:  https://doi.org/10.1186/s12967-025-07420-3
  3. Mol Genet Genomic Med. 2025 Dec;13(12): e70172
       INTRODUCTION: Rare diseases sometimes present with deceptively common symptoms, complicating diagnosis and decisions about genetic testing. While testing for rare disease offers important benefits, it also carries risks that warrant careful consideration.
    METHODS: We review illustrative cases of rare diseases, along with current screening and diagnostic practices, to reexamine guiding principles for genetic testing. The analysis focuses on balancing clinical utility, patient-centered care, and broader policy implications.
    RESULTS: We propose a number of recommendations to guide testing, including ruling out common causes before proceeding, ensuring the presentation is atypical for other common conditions, confirming consistency with a specific, treatable rare disease entity or group, assessing patient or family capacity for informed decision-making, and matching test invasiveness to expected diagnostic utility. Comparison with newborn screening and diagnostic testing highlights discrepancies between these principles and common practices, highlighting the difficulty of achieving consistency.
    CONCLUSIONS: Establishing uniform guidelines for genetic testing remains challenging, particularly given the limited knowledge surrounding rare disorders. Coordinated efforts are needed to protect patient interests, assess the utility of diagnoses across varied contexts, and ensure that both clinical practice and policy development maximize benefits while minimizing harms.
    Keywords:  diagnosis; genetic testing; rare and undiagnosed diseases; zebra
    DOI:  https://doi.org/10.1002/mgg3.70172
  4. Int J Neonatal Screen. 2025 Nov 28. pii: 109. [Epub ahead of print]11(4):
      Following Newborn Screening (NBS), parents receiving positive results experience various psychosocial effects upon learning their child's genetic information or unexpected findings. These factors warrant careful consideration. The Japanese Medical Association's Guidelines for Genetic Testing and Diagnosis in Medical Care highlight the importance of genetic counseling (GC) in NBS; however, its current implementation status remains unclear. This study aimed to determine current approaches to GC following positive NBS results in Japan. A questionnaire was conducted with pediatric metabolic specialists responsible for treating individuals who screen positive through NBS results to evaluate GC implementation and their views on its provision. GC was provided at most referral centers for NBS (although not routinely at approximately half of the facilities). In over 70% of cases, GC was performed by a metabolic specialist, regardless of clinical geneticist certification. Furthermore, some metabolic specialists may be reluctant to provide GC due to limited understanding or time constraints. Raising awareness that all parents are eligible for GC, regardless of their child's diagnosis or health status, is essential. In addition, a GC system incorporating multidisciplinary and multidepartmental collaboration is important for the multifaceted support of patients and families.
    Keywords:  clinical geneticist; genetic counseling; genetic counselor; multidisciplinary care; neonatal screening; newborn screening; psychosocial
    DOI:  https://doi.org/10.3390/ijns11040109