Systems Modeling Reveals Shared Metabolic Dysregulation and Novel Therapeutic Treatments in ME/CFS and Long COVID

Abstract:

Myalgic Encephalomyelitis/Chronic Fatigue Syndrome (ME/CFS) and Long COVID are complex, multisystemic conditions that pose ongoing challenges to healthcare professionals. Emerging research suggests that ME/CFS and Long COVID exhibit overlapping metabolic symptoms, indicating possible shared metabolic dysfunctions. This study aims to systematically explore these shared metabolic disturbances and their potential treatments.

Utilizing our novel metabolic modeling method, GPMM, we identified the key metabolic irregularities in patients with ME/CFS and Long COVID, notably the downregulation of the alanine and aspartate metabolism pathway, and the arginine and proline metabolism pathway.

Genome-wide knockout analyses indicated that supplementation with aspartate (ASP) or asparagine (ASN) could potentially ameliorate these metabolic deficiencies. Further metabolic assessments in Long COVID patients highlighted the significant downregulation of ASP in both blood and muscle, supporting our predictions.

Consequently, we propose that the combination of l-ornithine and l-aspartate (LOLA) offers a promising approach to alleviate metabolic symptoms in both ME/CFS and Long COVID patients. This study not only elucidates the shared metabolic pathways in ME/CFS and Long COVID but also positions LOLA as a viable candidate for future clinical trials.

Source: Gong-Hua LiFeifei HanQing-Peng KongWenzhong Xiao. Systems Modeling Reveals Shared Metabolic Dysregulation and Novel Therapeutic Treatments in ME/CFS and Long COVID.

Why exercise magnifies exhaustion for chronic fatigue syndrome patients

The mechanism that causes high-performance athletes to “feel the burn” turns out to be the culprit in what makes people with chronic fatigue syndrome feel exhausted by the most common daily activities, new University of Florida Health research shows.

Published in the February issue of the journal Pain, the study shows that the neural pathways that transmit feelings of fatigue to the brain might be to blame. In those with chronic fatigue syndrome, the pathways do their job too well.

The findings also provide evidence for the first time that peripheral tissues such as muscles contribute to feelings of fatigue. Determining the origins of fatigue could help researchers develop therapies or identify targets for those therapies.

Researchers focused on the role of muscle metabolites, including lactic acid and adenosine triphosphate, or ATP, in the disease. The study has demonstrated for the first time that these substances, released when a person exercises his or her muscles, seem to activate these neural pathways. Also, UF Health researchers have shown that these pathways seem to be much more sensitive in patients with chronic fatigue syndrome than in patients without the disease, something that hasn’t been studied before.

Chronic fatigue syndrome, which the Institute of Medicine recently renamed systemic exertion intolerance disease, or SEID, is characterized by extreme chronic fatigue. Because its chief symptom — fatigue — is often associated with many other diseases, it can be difficult to diagnose SEID for the more than 1 million people who actually have the disease, according to the Centers for Disease Control and Prevention. The disease has no root medical cause, and researchers don’t know what triggers it. But they are studying aspects of the disease to figure out ways to treat it.

“What we have shown now, that has never been shown before in humans, is that muscle metabolites can induce fatigue in healthy people as well as patients who already have fatigue,” said Dr. Roland Staud, a professor of rheumatology and clinical immunology in the UF College of Medicine and the paper’s lead author.

During exercise, muscles produce metabolites, which are sensed by metaboreceptors that transmit information via fatigue pathways to the brain, according to the researchers. But in patients with SEID, these fatigue pathways have become highly sensitive to metabolites and can trigger excessive feelings of fatigue.

“For most of us, at the end of strenuous exertion we feel exhausted and need to stop — but we will recover rapidly,” Staud said. “However, these individuals tire much more rapidly and sometimes just after moving across a room, they are fully exhausted. This takes a toll on their lives.”

Staud and co-author Michael E. Robinson, a professor in the department of clinical and health psychology in the UF College of Public Health and Health Professions, recruited a group of 39 patients with SEID and 29 participants without the disease. The researchers asked the participants to don a blood pressure cuff just above their elbows on their dominant side, pick up a spring-loaded device and squeeze it to 100 percent of their maximum capacity, which was measured by a dial.

With research assistants encouraging them, the study participants then squeezed the device so that the dial showed they were gripping at 50 percent of their maximum capacity for as long as they could.

At the end of the hand-grip exercise, the blood pressure cuff on the participant’s arm was inflated, almost instantly trapping the metabolites generated by the exercise within the forearm muscles. This allowed the metabolites to collect in the forearm tissue without being cleared by the circulatory system. There, the metabolites continued to activate fatigue pathways, sending messages of fatigue to the brain and allowing researchers to measure how much fatigue and pain may occur because of the trapped metabolites.

With the blood pressure cuff still inflated, the participants rated fatigue and then pain in their forearms every 30 seconds. Both patients with SEID and patients without the disease reported increasing fatigue, but patients with the disease recorded much higher levels of fatigue and pain.

“We found that the fatigued individuals reported more fatigue than the non-fatigued individuals during the exercise, and also found that they had more pain compared to the non-fatigued individuals,” Staud said.

On the Fatigue Visual Analog Scale used to measure participants’ fatigue, patients with SEID rated their fatigue at approximately 5.5 on a scale of 0 to 10 after the hand-grip exercise while wearing the inflated blood pressure cuff, whereas participants without the disease rated their fatigue at approximately 1.5.

After 30 minutes, the participants repeated the exercise, but with the opposite arm and without the cinching blood pressure cuff so the metabolites could be cleared from the arm. Both sets of participants experienced fatigue, but the feeling of fatigue in those with the disease was much lower than when the metabolites were trapped with the blood pressure cuff.

“This suggests that hypersensitive fatigue pathways play an important role for the often pronounced exercise-related fatigue of patients with the disease,” Staud said.

Next, Staud plans to explore treatment interventions and to conduct brain-imaging studies of patients with SEID.

“The take-home message here is, like many of the pain studies we have conducted, there are both peripheral and central nervous system factors at play in these complex syndromes,” said Robinson, who is also the director of the UF Center for Pain Research and Behavioral Health. “Our study seems to highlight the important role of these peripheral tissues.”

Journal Reference: Roland Staud, Meriem Mokthech, Donald D. Price, Michael E. Robinson. Evidence for sensitized fatigue pathways in patients with chronic fatigue syndrome. Pain, 2015; 156 (4): 750 DOI: 10.1097/j.pain.0000000000000110

 

Source: University of Florida. “Why exercise magnifies exhaustion for chronic fatigue syndrome patients.” ScienceDaily. ScienceDaily, 12 March 2015. https://www.sciencedaily.com/releases/2015/03/150312154135.htm

 

Understanding Muscle Dysfunction in Chronic Fatigue Syndrome

Abstract:

Introduction. Chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) is a debilitating disorder of unknown aetiology, characterised by severe disabling fatigue in the absence of alternative diagnosis. Historically, there has been a tendency to draw psychological explanations for the origin of fatigue; however, this model is at odds with findings that fatigue and accompanying symptoms may be explained by central and peripheral pathophysiological mechanisms, including effects of the immune, oxidative, mitochondrial, and neuronal pathways. For example, patient descriptions of their fatigue regularly cite difficulty in maintaining muscle activity due to perceived lack of energy. This narrative review examined the literature for evidence of biochemical dysfunction in CFS/ME at the skeletal muscle level.

Methods. Literature was examined following searches of PUB MED, MEDLINE, and Google Scholar, using key words such as CFS/ME, immune, autoimmune, mitochondria, muscle, and acidosis.

Results. Studies show evidence for skeletal muscle biochemical abnormality in CFS/ME patients, particularly in relation to bioenergetic dysfunction.

Discussion. Bioenergetic muscle dysfunction is evident in CFS/ME, with a tendency towards an overutilisation of the lactate dehydrogenase pathway following low-level exercise, in addition to slowed acid clearance after exercise. Potentially, these abnormalities may lead to the perception of severe fatigue in CFS/ME.

 

Source: Rutherford G, Manning P, Newton JL. Understanding Muscle Dysfunction in Chronic Fatigue Syndrome. J Aging Res. 2016;2016:2497348. doi: 10.1155/2016/2497348. Epub 2016 Feb 22. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4779819/ (Full article)

 

Abnormalities of AMPK activation and glucose uptake in cultured skeletal muscle cells from individuals with chronic fatigue syndrome

Abstract:

BACKGROUND: Post exertional muscle fatigue is a key feature in Chronic Fatigue Syndrome (CFS). Abnormalities of skeletal muscle function have been identified in some but not all patients with CFS. To try to limit potential confounders that might contribute to this clinical heterogeneity, we developed a novel in vitro system that allows comparison of AMP kinase (AMPK) activation and metabolic responses to exercise in cultured skeletal muscle cells from CFS patients and control subjects.

METHODS: Skeletal muscle cell cultures were established from 10 subjects with CFS and 7 age-matched controls, subjected to electrical pulse stimulation (EPS) for up to 24h and examined for changes associated with exercise.

RESULTS: In the basal state, CFS cultures showed increased myogenin expression but decreased IL6 secretion during differentiation compared with control cultures. Control cultures subjected to 16 h EPS showed a significant increase in both AMPK phosphorylation and glucose uptake compared with unstimulated cells. In contrast, CFS cultures showed no increase in AMPK phosphorylation or glucose uptake after 16 h EPS. However, glucose uptake remained responsive to insulin in the CFS cells pointing to an exercise-related defect. IL6 secretion in response to EPS was significantly reduced in CFS compared with control cultures at all time points measured.

CONCLUSION: EPS is an effective model for eliciting muscle contraction and the metabolic changes associated with exercise in cultured skeletal muscle cells. We found four main differences in cultured skeletal muscle cells from subjects with CFS; increased myogenin expression in the basal state, impaired activation of AMPK, impaired stimulation of glucose uptake and diminished release of IL6. The retention of these differences in cultured muscle cells from CFS subjects points to a genetic/epigenetic mechanism, and provides a system to identify novel therapeutic targets.

 

Source: Brown AE, Jones DE, Walker M, Newton JL. Abnormalities of AMPK activation and glucose uptake in cultured skeletal muscle cells from individuals with chronic fatigue syndrome. PLoS One. 2015 Apr 2;10(4):e0122982. doi: 10.1371/journal.pone.0122982. ECollection 2015. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4383615/ (Full article)