scholarly journals Influence of taurine on phenotype expression of muscular dystrophy in Drosophila melanogaster

2020 ◽  
Vol 27 ◽  
pp. 202-207
Author(s):  
T. K. Orlov ◽  
S. M. Gorbulinska ◽  
H. M. Klepach ◽  
M. A. Kryzhanovska ◽  
N. Ia. Holub

Aim. To investigate the effect of 0.1% taurine on the manifestation of muscular dystrophy in Drosophila melanogaster mutants under both larval and adult feeding. Methods. Larval and adult feeding of 0.1 % taurine, construction of survival curves and study of moving activity, making thorax muscles histological preparations. Results. It has been shown the increasing of life span indexes (on 46-75%). moving activity (on 50-75%), decreasing of fatigue rates, and reducing of muscle damages (on 6-20%) in Drosophila dystrophy mutants under the influence of 0.1% taurine. Conclusions. The results indicate a positive effect of 0.1% taurine as a stimulant, antioxidant, myoprotector, blocker of fatigue processes in dystrophin mutants. However, following further studies on vertebrates, taurine may be recommended as a therapeutic agent in the treatment of Duchenne muscular dystrophy. Keywords: Drosophila, dystrophin, miodystrophy, taurine.

2021 ◽  
Vol 2 (1) ◽  
pp. 38-50
Author(s):  
Tatiana A. Gremiakova ◽  
Vasiliy M. Souslov ◽  
Gulzhan E. Sakbaeva ◽  
Andrey A. Stepanov

Duchenne muscular dystrophy (DMD) is an X-linked recessive degenerative neuromuscular disorder due to a deficiency of dystrophin protein. This protein is most common in skeletal and cardiac muscles, to a lesser extent in smooth muscles and the brain. With DMD, progressive damage and muscle degeneration, a delay in motor development, and respiratory cardiac disorders are progressing. Patients with DMD have an increased risk of developing osteoporosis, fractures of the tubular bones and vertebrae, and neurocognitive impairment. Vitamin D is recommended prophylactically for DMD since many studies have shown its deficiency. The purpose of this work is to consolidate the literature data on the vitamin D deficiency in DMD patients and its effects on the development of concurrent comorbid conditions of the musculoskeletal, endocrine, and nervous systems. The authors discuss data concerning the appropriate level of vitamin D throughout the life span of DMD has a positive effect on the course of the disease patients’ quality of life ends. Primary clinical outcomes of vitamin D normalization include prevention of the development of osteoporosis (especially after the start of steroid therapy), fractures of the tubular bones and vertebrae, prolonged ability to walk, more effective treatment with bisphosphonates, including a decrease in the number of complications during initial use and lower jaw necrosis, positive effect on the expression of autistic spectrum symptoms. For patients with long-term steroid therapy, metabolic and liver disorders, calcidiol could be used, allowing quick deficiency compensation instead of standard vitamin D preparations.


2017 ◽  
Vol 27 ◽  
pp. S191
Author(s):  
P. Piñol ◽  
E. Fernández-Simón ◽  
X. Suárez ◽  
N. de Luna ◽  
A. Molins ◽  
...  

2010 ◽  
Vol 18 (12) ◽  
pp. 2155-2163 ◽  
Author(s):  
Sébastien Goudenege ◽  
Yann Lamarre ◽  
Nicolas Dumont ◽  
Joël Rousseau ◽  
Jérôme Frenette ◽  
...  

2020 ◽  
Vol 14 (4) ◽  
pp. 349-352
Author(s):  
Claudia Weiß ◽  
Corinna Stoltenburg ◽  
Dilan Bayram ◽  
Julia Funk ◽  
Susanne Lebek

Purpose In the 1980s the first results of an early multilevel contracture release (MLCR) in patients suffering from progressive Duchenne muscular dystrophy (DMD) showed a positive effect on ambulation. Despite the demonstrated positive effects of prolongation of walking this treatment is not part of current guidelines. The aim of our study was to evaluate the effect of MLCR as well as its combination with glucocorticoid (GC) treatment on ambulation. Methods Data of all boys (n = 86) with DMD treated in our outpatient department were analyzed regarding the treatment and loss of independent ambulation. In all, 23 were treated with GC only, ten were operated on, 21 received GC and underwent MLCR and 32 received neither of the two treatments. Results The analysis of the loss of independent ambulation in our cohort showed a comparable extension of the ambulatory period between the GC-treated and MLCR-treated boys (p = 0.008 and p = 0.005, respectively). Furthermore, an additive effect of both therapies was found; patients with DMD who had both treatments were able to walk two years longer than those with only one of the two treatment options (p<0.001). Conclusion Standard GC treatment and early MLCR in lower limbs have an independent positive effect on prolongation of ambulation in patients with DMD. In our cohort, the combination of both therapies is significantly more effective than each therapy alone. We suggest both should be offered to all DMD patients eligible. Level of evidence: III


2019 ◽  
Vol 25 ◽  
pp. 32-37
Author(s):  
O. V. Gorenskaya ◽  
V. V. Navrotskaya

Aim. To analyze life span in mutant Drosophila stocks with impaired tryptophan-kynurenine metabolism. Methods. Wild type stocks Canton-S and Oregon, stocks with mutations of the locus white: white, whiteapricot, whitesatsuma, and stocks with the mutation vermilion have been used. The average life span of imago has been determined, survival curves have been analyzed. Results. It has been shown that the average life span of Drosophila females with mutant alleles of the white gene does not differ from the wild-type stock; in males of the w(C-S) and wa(C-S) stocks the index is increased. The presence of the mutantion vermilion in the genotype also increases the average life span of imago of both sexes, but in males the extension is more pronounced. Conclusions. The results suggest that aging is associated with the regulation of tryptophan-kynurenine metabolism. Keywords: Drosophila melanogaster, life span, kynurenine pathway of tryptophan metabolism.


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