Mitochondrial myopathy – differential diagnosis for ME

Posted on 09/30/2015 by wames

Research abstract:

Introduction: Symptoms of mitochondrial diseases and chronic fatigue syndrome (CFS) frequently overlap and can easily be mistaken.

Methods: We report the case of a patient diagnosed with CFS and during follow-up was finally diagnosed with mitochondrial myopathy by histochemical study of muscle biopsy, spectrophotometric analysis of the complexes of the mitochondrial respiratory chain, and genetic studies.

Results: The results revealed 3% fiber-ragged blue and a severe deficiency of complexes I and IV and several mtDNA variants. Mother, sisters, and nephews showed similar symptoms, which strongly suggests a possible maternal inheritance. The patient and his family responded to treatment with high doses of riboflavin and thiamine with a remarkable and sustained fatigue and muscle symptoms improvement.

Conclusions: This case illustrates that initial symptoms of mitochondrial disease in adults can easily be mistaken with CFS, and in these patients a regular reassessment and monitoring of symptoms is recommended to reconfirm or change the diagnosis.

Mitochondrial Myopathy in Follow-up of a Patient With Chronic Fatigue Syndrome by Fernando Galán, AOPT, Isabel de Lavera, PhD, David Cotán, PhD, José A.
Sánchez-Alcázar, AOPT in Journal of Investigative Medicine High Impact Case Reports July-September 2015 vol. 3 no. 3 [Published September 27, 2015]

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ACT (Acceptance and commitment therapy) for CFS

Posted on 09/30/2015 by wames

Research abstract:

Acceptance is understood to be an important element in coping and  living with chronic illnesses such as diabetes and chronic pain, and increasing acceptance has been related to a number of positive  outcomes such as reduced symptoms as well as greater reported quality  of life.

Chronic Fatigue Syndrome (CFS) is a disabling disorder that is associated with poor reported quality of life even in comparison to others chronic conditions.

Given that ACT (Acceptance and commitment therapy) is a psychotherapeutic model aimed at increasing psychological flexibility and values pursuance, the approach holds potential for coping and living with CFS, with interesting implications for service provision and client care.

We utilized a six-week guided self-help ACT intervention with six detailed case studies to assess the utility of an ACT self-help intervention for this under-researched population, and whether any therapeutic and physical change is driven by predicted ACT mechanisms.

These results are discussed in relation to ACT processes and implications for working with individuals with a diagnosis of CFS.

ACT for chronic fatigue syndrome: a case series approach, by Lauren Roche, Dave Dawson, Nima Moghaddam, Mark Gresswell. Poster presentation at Association for Contextual Behavioral Science World Conference, 14 – 19 July 2015, Berlin, Germany [Published University of Lincoln Repository, September 28, 2015

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Patients’ opinions of activity pacing

Posted on 09/30/2015 by wames

Research abstract:

Objective:

Despite the frequent recommendation of activity pacing as a coping strategy for patients with chronic pain and/or fatigue, pacing is interpreted in different ways and there is an absence of a widely accepted pacing scale. We have developed a new Activity Pacing Questionnaire (APQ). The aims of this study were to explore patients’ views and beliefs about the concept of pacing, together with the acceptability of the APQ.

Design:

Qualitative pragmatic study using semi-structured telephone interviews. Data were analysed using Framework analysis.

Participants:

16 adult patients attending secondary care physiotherapy out-patient departments were recruited via purposive sampling. Diagnoses included chronic low back pain, chronic widespread pain, fibromyalgia and chronic fatigue syndrome/myalgic encephalomyelitis.

Findings:

Pacing emerged as a multifaceted concept from participants’ descriptions. The implementation of pacing was influenced by participants’ age, the presence of co-morbidities and participants’ emotions. The APQ was found to be generally acceptable in comparison to two existing pacing subscales. Participants undertook activities using quota/symptom-contingent approaches. Four behavioural typologies emerged: Task avoidance, Task persistence, Task fluctuation (boom-bust) and Task modification (activity pacing).

Conclusions:

The APQ appears to be easy to complete, and acceptable to patients who are attending physiotherapy for the management of long-term conditions. It emerged that individual patients implemented different pacing facets to varying degrees, and that different behavioural typologies were apparent. The relationships between behavioural typologies and facets of pacing warrant further investigation to facilitate the development of effective tailored pacing interventions.

Exploring patients’ opinions of activity pacing and a new activity pacing questionnaire for chronic pain and/or fatigue: a qualitative study by Deborah Antcliff, Philip Keeley, Malcolm Campbell, Steve Woby, Linda McGowan in Physiotherapy. 2015 Aug

 

 

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Patients’ ME research priorities

Posted on 09/30/2015 by wames

Research abstract:

Background:
The aim of this work was to involve patients in setting future priorities for myalgic encephalomyelitis (M.E.) research.

Methods:
A national on-line survey was developed collecting structured and unstructured data. Respondents were asked what they considered Action for M.E.’s first, second and third research priorities to be from a list of 13 pre-defined options. Individuals were also invited to provide any additional free text comments about Action for M.E.’s research priorities in general.

A total of 1144 individuals completed the on-line survey. Respondents were asked to indicate if:

  • they had M.E. (n=3D822; 90.4%);
  • were a supporting a member of Action for M.E (n=3D94; 10.3%);
  • carer for someone with M.E. (n=3D66; 7.3%),
  • professional with an interest in M.E. (n=3D26; 2.9%); or had a family member or colleague with M.E. (n=3D136; 15%).

Individuals were able to select more than one category as applicable.

Results:
The top five research priorities identified by the respondents were:

  • disease processes to achieve a better understanding of the underlying
    pathology of M.E.;
  • more effective treatments; faster and more accurate diagnosis;
  • clinical course of M.E.;
  • outcomes and prognosis;
  • severely affected patients.

The lower research priorities identified were: sleep; economic research towards identifying the cost of ME for individuals and society; and psychological aspects.

Much of the unstructured data provided by respondents emphasised the importance of funding biomedical research into disease processes to achieve a better understanding of the underlying pathology of M.E. Three themes were identified in relation to this topic: accurate diagnosis and awareness; risk factors and causes; drug development and curative
therapies.

Conclusions:
Individuals affected by M.E. have clear views regarding the most important priorities for research investment. These tended to focus on disease processes to achieve a better understanding of the underlying pathology of M.E. and have been used to inform Action for M.E.’s ongoing research strategy.

Consulting patients in setting priorities in Myalgic Encephalomyelitis
(M.E.) research: findings from a national on-line survey, by Nicola Childs, Lisa Robinson, Sonya Chowdhury, Clare Ogden and Julia L. Newton in Research Involvement and Engagement Vol 1, 11, September 28, 2015

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Effect on siblings of children with CFS/ME

Posted on 09/29/2015 by wames

Research abstract:

Chronic fatigue syndrome or myalgic encephalomyelitis (CFS/ME) is a disabling condition known to have a negative impact on all aspects of a child’s life. However, little is understood about the impact of CFS/ME on siblings.

A total of 34 siblings completed questionnaires measuring depression (Hospital Anxiety and Depression Scale (HADS)), anxiety (HADS and Spence Children’s Anxiety Scale (SCAS)) and European Quality-of-life-Youth (EQ-5D-Y). These scores were compared with scores from normative samples.

Siblings had higher levels of anxiety on the SCAS than adolescents of the same age recruited from a normative sample; however, depression and quality-of-life were similar.

Interviews were undertaken with nine siblings of children with CFS/ME who returned questionnaires. Interview data were analysed using a framework approach to thematic analysis. Siblings identified restrictions on family life, ‘not knowing’ and lack of communication as negative impacts on their family, and change of role/focus, emotional reactions and social stigma as negative impacts on themselves. They also described positive communication, social support and extra activities as protective factors.

Paediatric services should be aware of the impact of CFS/ME on the siblings of children with CFS/ME, understand the importance of assessing paediatric CFS/ME patients within the context of their family and consider providing information for siblings about CFS/ME.

Psychological wellbeing and quality-of-life among siblings of paediatric CFS/ME patients: A mixed-methods study by S  Velleman, SM Collin, L Beasant, E Crawley  in Clin Child Psychol Psychiatry 2015 Sep 22. pii: 1359104515602373. [Epub ahead of print]

 

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Characteristics of CFS/ME sub-group who respond to Rintatolimod (Ampligen)

Posted on 09/29/2015 by wames

Research abstract:

Background: Chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) is a debilitating disease of unknown pathogenesis consisting of  a variety of flu-like symptoms including severe fatigue.

Initial analysis of the use of rintatolimod (Poly I: Poly C12U), a selective TLR3 agonist, in a
Phase III, double-blind, randomized, placebo-controlled trial of CFS/ME demonstrated statistical significance (p<0.05) in the reduction of fatigue  as measured by exercise tolerance (ET) as the primary endpoint using a modified Bruce protocol with reduced physical exertion in patients with  severe CFS/ME as defined by a Karnofsky performance score (KPS) of 40-60.

Methods and Findings: In order to better identify responders to rintatolimod, primary and secondary endpoints have been reexamined post hoc as a function of a pre-specified study baseline ET duration >9 minutes. Analysis of improvement in exercise performance at the ≥ 25% and  ≥ 50% levels using ET at 40 weeks compared to baseline was performed for the intent-to-treat (ITT) population (n=208) using the pre-specified baseline exercise stratum (baseline ET duration >9 minutes). For this subset of patients (n=126), 33% (n=20), and 12% (n=8) of rintatolimod vs. placebo patients, respectively, improved ET duration by ≥ 25% (p=0.004) while 23% (n=14) compared to 4.5% (n=3) of rintatolimod vs.  placebo patients, respectively improved ET duration by ≥ 50% (p=0.003).

This corresponds to increases of ≥ 186 and ≥ 373 seconds for patients receiving rintatolimod, respectively, at ≥ 25% and ≥ 50% improvement responses. A frequency distribution analysis of ≥ 25% improvement, <25% change, and ≥ 25% deterioration in ET from baseline at 40 weeks for the baseline >9 minutes cohort showed net improvement to be 18.3% for the rintatolimod cohort vs. 4.6% deterioration for placebo (p=0.015).

A continuous responder analysis using 5% increments from ≥ 25% to ≥ 50% provided a robust clinical enhancement in ET effect in the rintatolimod cohorts as compared to placebo. The KPS and Vitality (SF-36 subscale) quality of life secondary endpoints demonstrated similar clinically significant improvements for the rintatolimod cohort as a function of the same ET dichotomization. Rintatolimod was generally well-tolerated in this CFS/ME population.

Conclusions: Using a modified Bruce ET protocol with reduced physical exertion allowed clear identification of patient responders to rintatolimod with severe CFS/ME syndrome. Rintatolimod produced significant enhancement in ET and quality of life indicators in patients able to complete >9 minutes in a modified Bruce ET test.

Rintatolimod also reduced deterioration in ET compared to placebo in patients with the poorest initial ET. Exercise endurance >9 minutes in a Bruce protocol modified for patients with CFS/ME provides a method to identify patients most likely
to respond to rintatolimod.

Chronic Fatigue Syndrome/Myalgic Encephalomyelitis (CFS/ME): Characteristics of Responders to Rintatolimod,  by David R Strayer, Bruce C Stouch, Staci R Stevens, Lucinda Bateman, Charles W Lapp, Daniel L Peterson, William A Carter, and William M Mitchell in J Drug Res Dev 1(1). 2015 Aug 8

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UK rituximab trial fundraising total almost reached

Posted on 09/28/2015 by wames

Invest in ME has announced the latest fundraising total for the rituximab/B-cell research, which is a great boost as the research team at UCL are about to visit and discuss the subject with Dr Fluge and Professor Mella in Haukeland University Hospital, Bergen, Norway.

In order to build a trials protocol and identify likely responders a preliminary B-cell study has been underway at University  College London. This will lead to the clinical trial.

In order to continue B-cell study research in ME, and as part of the overall research portfolio that Invest in ME has with UCL, they are committing themselves to fund a new PhD studentship at UCL beginning next year. This will augment the proposed Invest in ME rituximab clinical trial, will secure the longer term research into ME and will continue the relationship which IiME has built up with UCL.

New goal:   £520,000

Achieved:   £458,000

More info and how to donate

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Elevated autoantibodies found in CFS subgroup

Posted on 09/28/2015 by wames

Research highlights:

  • β adrenergic and muscarinic acetylcholine receptor autoantibodies are elevated in a subset of patients with Chronic Fatigue Syndrome (CFS).
  • Elevated autoantibodies in CFS correlate with elevated IgG1-3 subclass levels, thyreoperoxidase and ANA antibodies and T cell activation.
  • In CFS patients responding to rituximab treatment, elevated antibody levels detected pre-treatment normalized in the majority of clinical responders post-treatment.

Research abstract:

Infection-triggered disease onset, chronic immune activation and autonomic dysregulation in CFS point to an autoimmune disease directed against neurotransmitter receptors.

Autoantibodies against G-protein coupled receptors were shown to play a pathogenic role in several autoimmune diseases. Here, serum samples from a patient cohort from Berlin (n= 268) and from Bergen with pre- and post-treatment samples from 25 patients treated within the KTS-2 rituximab trial were analysed for IgG against human α and ß adrenergic, muscarinic (M) 1-5 acetylcholine, dopamine, serotonin, angiotensin, and endothelin receptors by ELISA and compared to a healthy control cohort (n=108).

Antibodies against ß2, M3 and M4 receptors were significantly elevated in CFS patients compared to controls. In contrast, levels of antibodies against α adrenergic, dopamine, serotonin, angiotensin, and endothelin receptors were not different between patients and controls. A high correlation was found between levels of autoantibodies and elevated IgG1-3 subclasses, but not with IgG4. Further patients with high ß2 antibodies had significantly more frequently activated HLA-DR+ T cells and more frequently thyreoperoxidase and anti-nuclear antibodies.

In patients receiving rituximab maintenance treatment achieving prolonged B-cell depletion, elevated ß2 and M4 receptor autoantibodies significantly declined in clinical responder, but not in non-responder. We provide evidence that 29.5% of patients with CFS had elevated antibodies against one or more M acetylcholine and ß adrenergic receptors which are potential biomarkers for response to B-cell depleting therapy.

The association of autoantibodies with immune markers suggests that they activate B and T cells expressing ß adrenergic and M acetylcholine receptors. Dysregulation of acetylcholine and adrenergic signalling could also explain various clinical symptoms of CFS.

Antibodies to ß adrenergic and muscarinic cholinergic receptors in patients with Chronic Fatigue Syndrome, by M Loebel, P Grabowski, H Heidecke, S Bauer, LG Hanitsch, K Wittke, C Meisel, P Reinke, HD Volk HD, Ø Fluge, O Mella, C Scheibenbogen, in Brain Behav Immun. 2015 Sep 20. pii: S0889-1591(15)30020-9. [Epub ahead of print]

Discussion on Phoenix rising forum

New study sheds more light on why some people with ME/CFS may respond to rituximab, in ME Association News blog, 26 September 2015

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Poor self-esteem found in adolescents with CFS

Posted on 09/25/2015 by wames

Research abstract:

Background: The self and self-experience are often assumed to play an important role in adolescent patients presenting with severe somatic symptoms and bodily distress. Nonetheless, most empirical work on this subject is confined to studies of personality and patients’ experience of negative emotionality. This study aims to move beyond mere descriptions of symptoms, traits and distress, and consequently adopts a narrative approach to self-experience in adolescent chronic fatigue syndrome (CFS).

Sampling and Methods: The self-confrontation method (SCM) is a well-validated instrument to systematically analyze narrative self-experience. The SCM was used to study 42 adolescents with CFS, compared to 36 adolescents with juvenile idiopathic arthritis (JIA) and 25 matched healthy controls.

The Child Health Questionnaire (CHQ-CF87) was used to assess mental health, self-esteem, and physical and psychosocial functioning.

Results: Both patient groups reported significantly less positive self-experience of autonomy and success compared to healthy controls. Furthermore, patients with CFS described significantly more negative self-experience of powerlessness, isolation and unfulfilled longing. In the CHQ-CF87, both patient groups scored significantly lower on physical functioning than controls. Adolescents with CFS also scored significantly lower on mental health and self-esteem.

Conclusions: Adolescent CFS entails a serious threat to the self, which might be inherent to the condition. Not only are patients more impaired in mental health, self-esteem, and physical and psychosocial functioning than patients with JIA, they also suffer from a distinct combination of high negative and low positive self-experience. These findings stress the need for strategies that empower patients towards a ‘management of the self’.

The self beyond somatic symptoms: a narrative approach to self-experience in adolescent chronic fatigue syndrome by SM van Geelen SM, et al. in Psychopathology, 2015 Sep 12

 

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Severity of POT fluctuates in ME

Posted on 09/25/2015 by wames

Research abstract:

Central nervous system dysfunction with myalgic encephalomyelitis (ME) has been suggested as the main cause of chronic fatigue syndrome. Fluctuation of the symptom severity and hierarchy is a characteristic feature in ME patients. The characteristics of the sympathetic activation may differ between the “good days” and “bad days” in them.

Twenty-four ME patients with orthostatic intolerance underwent a conventional 10-min active standing test and echocardiography both on a “good day” and a “bad day”, defined according to the severity of their symptoms. The mean heart rate at rest was significantly higher on the “bad days” than on the “good days”. During the standing test on a “bad day”, 5 patients (21 %) failed to maintain an upright posture for 10 min, whereas on a “good day” all the 24 patients maintained it.

Postural orthostatic tachycardia (POT) (increase in heart rate ≥30 beats/min) or severe POT (heart rate ≥120 beats/min) was observed on the “bad days” in 10 patients (43 %) who did not suffer from the severe tachycardia on the “good days”, suggesting the exaggerated sympathetic nervous activation.

In contrast, POT did not occur or severe POT was attenuated on the “bad days” in 5 patients (21 %) who developed POT or severe POT on the “good days”, suggesting the impaired sympathetic activation. Echocardiography revealed significantly lower mean values of both the left ventricular end-diastolic diameter and stroke volume index on the “bad days” compared with the “good days”.

In conclusion, in ME patients with orthostatic intolerance, the exaggerated activation of the sympathetic nervous system while standing appears to switch to the impaired sympathetic activation after the system is loaded with the additional accentuated stimuli associated with the preload reduction.

Variability of postural orthostatic tachycardia in patients with myalgic encephalomyelitis and orthostatic intolerance by Miwa K in Heart Vessels, 2015 Sep 15 [Epub ahead of print]

 

 

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