"Due to the definitions of ME and CFS, “ME/CFS” does not exist..."

Today, May 12th, is ME/CFS and Fibromyalgia International Awareness Day, a day to designed to "bring awareness to ME/CFS patients, families, caregivers, and researchers." Keep that terminology in mind...

The quote heading the title of this post - "Due to the definitions of ME and CFS, “ME/CFS” does not exist..." - comes from the viewpoint paper published by Frank Twisk [1]. The report covers an important topic in the realms of chronic fatigue syndrome (CFS) also known as myalgic encephalomyelitis (ME) also known as systemic exertion intolerance disease (SEID) in terms of whether it is appropriate to use such terms of defining the illness in a mixed or interchangeable fashion. Indeed, whether the connections between all those 'also known as' words I just used are actually accurately reflective of current diagnostic descriptions...

It's no secret that science and clinical practice is still coming to grips with some of the fundamentals of CFS, ME and SEID (see here and see here for examples) in terms of what to call it, how to define it and how to test for some of the fundamental diagnostic characteristics (see here). It's also still dealing with things like the definition of recovery (see here), which might seem like common sense (a complete and sustained remission of symptoms) but hasn't been particularly straightforward in this area for quite a few reasons.

Twisk takes the reader through some of the history of the terminology used and, how, whilst there is overlap in the way that ME, CFS and SEID are defined (chronic and long-lasting weakness or fatigue is a commonality), there are also some important differences. Take for example the authors description of the Ramsay criteria for ME and specifically onset: "Illness commonly initiated by respiratory and/or gastrointestinal infection, but an insidious or more dramatic onset following neurological, cardiac, or endocrine disability occurs." This contrasts with the onset criteria for CFS and SEID which basically says little about how symptoms start or come about.

Twisk concludes that: "ME is a neuromuscular disease" and should typically not to be viewed as 'equivalent' to CFS. CFS, he argues, tends to rely heavily on a single mandatory 'chronic fatigue' symptom, something that might intersect with ME but does not go far enough to evoke a full diagnosis of ME. As for SEID, well, trumpeted as being the solution to all the diagnostic confusion, SEID has it's own issues according to Twisk. Not least that it can't serve both masters (ME and CFS) in diagnostic terms. Also important: "SEID case criteria are also applicable to subsets of people with other diseases, for example, Multiple Sclerosis (MS) and lupus; and psychological conditions, for example, major depression." There is the propensity for diagnostic confusion.

I do think that Twisk is on to something with his observations. I know quite a few people who don't like the confusion caused by combination terminology like 'ME/CFS'; often seeing it as conflating two (or even more!) quite different conditions. Add in yet another potentially important variable to such an argument - the addition of chronic disabling fatigue (CDF) as "a proxy for clinically diagnosed CFS/ME" as some authors have (see here) - and things get even more muddled. I daresay a lot of this confusion might also intersect with discussions/debates/arguments as to how far something like the biopsychosical (BPS) model should or rather shouldn't be applied to such fatigue related conditions (see here)...

You want to do something for ME/CFS and Fibromyalgia International Awareness Day? Well, first thing you could do is watch 'Unrest', then follow the #millionsmissing hashtag and then push for more research, biological research...

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[1] Twisk FNM. Myalgic Encephalomyelitis, Chronic Fatigue Syndrome, and Systemic Exertion Intolerance Disease: Three Distinct Clinical Entities. Challenges. 2018; 9(1): 19.

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Estimated autism prevalence in Northern Ireland: 2.9% for 2017-2018

Consider this post discussing the publication: "The Prevalence of Autism (including Aspergers Syndrome) in School age Children in Northern Ireland 2018" an extension of other musings on previous figures to come out of Northern Ireland (see here).

On my last blogging occasion on this topic, the report (see here) covered the period 2015/2016 and detailed an estimated prevalence rate of autism spectrum disorder ("including Asperger syndrome") in Northern Ireland of 2.3%. I actually missed a report that covered the period 2016/2017 (see here) that detailed an estimated rate of 2.5%. This latest report covering 2017/2018 sets the rate at 2.9%. You can perhaps see the direction of the trend, mirroring other population estimated data (see here)...

The report(s) are open-access for anyone to see, but I'm going to pick out a few choice snippets of information.

So: the last blogging time I talked about the Northern Ireland (NI) report, I mentioned that the [estimated] prevalence rate for boys was approaching 4% based on those 2015/2016 figures. Well, that's been well and truly surpassed and is now heading towards 5% of boys in NI "identified with autism." How are they identified I hear you ask? Well, school data is the answer, "from the ‘Northern Ireland School Census’" where schools are legally obliged by the Department of Education in NI to provide information about registered pupils. Further: "The data only captures those children identified with autism, at any time there may be additional children who may be progressing through the full assessment process and it is possible that a number of children may be identified as having autism at a later date." That last point is important in the context that NI has a bit of a history of 'long-waiting lists' for autism assessments (see here). Oh, I should also mention that the National Health Service (NHS) functions in Northern Ireland just as it does in other parts of the United Kingdom (UK) meaning that healthcare (including autism assessment and diagnosis) is free at the point of need. This does not mean that things are going to be 'fast or rapid' temporally, but does mean that people don't have to typically pay extra for such clinical services.

Next: the 4:1 male:female ratio for diagnosis seems to be holding true (as it did in the latest CDC report on 'estimated' autism prevalence in the United States). I know that quite a few people talk about this ratio figure being 'inaccurate' in view of how autism may/may not present slightly 'differently' in females (see here for example), but, at the moment at least, that's what the statistics are telling us. One thing I perhaps am slightly cautious about in the latest report with regards to the sex/gender ratio thing is the phrase: "Autism could therefore be considered to be an extreme of the normal male profile." Hmm...

Also: autism prevalence by school year shows some interesting patterns. Take a look at the screen grab I've added observing that 3.4% of children in Year 9 were "identified with autism." Just in case you're not up to speed with what Year 9 translates as in age terms, have a look at this link which covers England. I think things are slightly different in NI (see here) but generally speaking, Year 9 covers somewhere between 12-14 years of age.

Finally, something else potentially quite important: "The Northern Ireland urban population has a statistically significant higher prevalence rate than the rural population." Note those words 'statistically significant', inferring that chance alone, is probably not the driver of such disparity. It's been a while since I've blogged about 'urban vs. rural' in the context of autism (see here) and I'm sure there are 101 different explanations for the mismatch. Combined however with some other observations on a possible influence of deprivation and poverty on the recent figures ("In 2017/18, the rate of autism in the most deprived MDM [Multiple Deprivation Measure] decile was 31% higher than the Northern Ireland average") one could argue that any explanation is going to be multi-factorial.

I look at these most recent figures and cannot help but think that 'increased awareness' and/or other 'artificial' explanations are (yet again) unable to entirely account for the sorts of increase in diagnosed autism being noted (see here). Were schools and other professionals 'so bad at recognising and/or recording autism' just a few years ago? No, they weren't. And to infer they were is bit a slur on the professionalism of many teachers and other associated professionals, many of whom have seen literally generations of schoolchildren pass through their educational doors.

There's also another important question to attend to on the basis of the recent figures: are the resources currently and in the future, in place to cope with the rising demands on things like education, health and social care following the increase in the numbers of children being diagnosed with autism? I say this in the context that if there are already insufficient resources to cope with the numbers requiring assessment for autism (assessments that are typically not inexpensive [1]), how can we hope that there will be sufficient resources in place over a lifetime of potential need?

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[1] Galliver M. et al. Cost of assessing a child for possible autism spectrum disorder? An observational study of current practice in child development centres in the UK. BMJ Paediatr Open. 2017 Nov 30;1(1):e000052.

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"psychiatric diagnoses, psychiatric care and psychotropic medication" in older age adults with autism

Is 55 years old still considered older age?

Well, according to the findings reported by Lena Nylander and colleagues [1] it represents the lower end cut-off point for their study looking at "the pattern of coexistent psychiatric diagnoses and the utilisation of psychiatric care and psychotropic medication among any individuals found to have ASD [autism spectrum disorder] diagnoses." Said individuals were aged between 55 and 96 in 2012 and "had a registered diagnosis of any ASD—defined as an ICD-10 code."

Those individuals 'found to have ASD' were located via some of those very useful Scandinavian population registries, this time based in Sweden. As a function of their registration for 'municipal services' researchers were also able to access other collected records and subsequently mined data on "gender, other psychiatric diagnoses, psychiatric care utilisation and psychotropic medication [use]" for a group of increasing research and clinical importance in the context of autism (see here).

The results proved interesting. Of the 600 people included for study, most had received a diagnosis of childhood autism (~40%), most had not received a concomitant intellectual disability (ID) diagnosis (~60%) and quite a few had received more than one 'type' of autism diagnosis (~15%).

When it came to the receipt of other psychiatric diagnoses such as affective disorder, personality disorder, anxiety or psychotic disorder, several notable observations were made. As a function of the total group, including everyone whether diagnosed with an ID or not, around 50% of participants had received at least one psychiatric diagnosis. A nebulous category defined as "other psychiatric diagnosis" was most frequently mentioned, but when it came to a named class of condition(s), affective disorders led the way in terms of frequency irrespective of the presence of ID or not. Affective disorders covers quite a bit of diagnostic ground but typically includes labels/conditions such as depression and/or bipolar disorder; conditions that are no stranger to autism (see here and see here respectively). Anxiety and psychotic disorders were also mentioned as being present among this cohort too; again not for the first time (see here and see here).

With regards to 'psychiatric care utilisation', it was more typical to see psychiatric care used than not used as nearly two-thirds of the cohort had used some kind of psychiatric care over the period included for study examination. Most were categorised as "general adult psychiatric care" and: "The group with Asperger’s syndrome had the highest number of people who had spent time as psychiatric inpatients" reflected in the odds ratio (OR) generated from the study for this group (OR: 6.87, 95% CI 3.80–12.43).

Finally, on the topic of psychotropic medication use, researchers observed that "63% of patients without registered ID diagnosis and as many as 84% of those with ID in combination with ASD had been prescribed antipsychotic medication." Antipsychotics were the most frequent medication mentioned in records, closely followed by anxiolytics (to manage anxiety) and antidepressants. Around 1 in 5 participants received more than one type of medication (irrespective of the presence of ID or not).

An important picture emerges from the Nylander findings. A picture suggesting that psychiatric diagnoses feature fairly prominently in the clinical profile of many older age adults with autism, and their identification and intervention need to be more clearly recognised. Nylander also pointed out that certain sub-groups within the autism spectrum should perhaps be more closely followed in relation to their achieving and maintaining good mental health. So, without trying to focus too much attention on one label: "It seems that the group with Asperger’s syndrome, or ASD without ID, is especially vulnerable to psychiatric disorders" on the basis that: "Only 15 individuals, or 11%, of the group with Asperger’s syndrome had not been in contact with psychiatric care, and 43% had been psychiatric in-patients, which may be interpreted as a sign of vulnerability in these individuals." That word again  - vulnerability - arises in the context of autism (see here and see here for other examples). And here is yet another example (see here) illustrating that phrases like 'high-functioning' in the context of the autism spectrum, really don't do justice to the lived experience of autism and the effects of it's important add-ons.

And on the topic of ageing and autism, and specifically ageing well, there are the findings reported by Ye In Hwang and colleagues [2] to consider, and specifically: "A very small proportion (3.3%) of autistic adults were found to be aging well."

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[1] Nylander L. et al. Older Adults with Autism Spectrum Disorders in Sweden: A Register Study of Diagnoses, Psychiatric Care Utilization and Psychotropic Medication of 601 Individuals. J Autism Dev Disord. 2018. April 16.

[2] Hwang YI. et al. Aging Well on the Autism Spectrum: An Examination of the Dominant Model of Successful Aging. J Autism Dev Disord. 2018. May 2.

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"To Be Quite Honest, If It Wasn't for Videogames I Wouldn't Have a Social Life at All"

The quote heading this post - "To Be Quite Honest, If It Wasn't for Videogames I Wouldn't Have a Social Life at All" - comes from the findings reported by Erinn Finke and colleagues [1]. They looked at "the perceptions of individuals with autism spectrum disorder (ASD) who play videogames as their primary leisure activity regarding the role of videogames in their lives and their motivations for playing videogames."

I've kinda touched on this topic before on this blog (see here) and some of the ins-and-outs of 'pathological' videogame use in the context of autism. On that blogging occasion, I mentioned how one has to be a little careful about 'demonising' an activity that (a) is enjoyed by millions of people, some of whom are and some aren't diagnosed with an autism spectrum disorder, and (b) could, through the wonders of online gaming, provide a valuable 'social' outlet to a population who either might prefer limited face-to-face contact or are in many ways, discriminated against when it comes to more traditional forms of social inclusion and participation (see here). Apparently this area is also something that has been mentioned at the INSAR conference this year too. The downsides to gaming: well, every moment sat in front of a screen is a moment that could have been used in other more physically active pursuits (something that is quite a big issue when it comes to quite a proportion of the autism spectrum). I might also add that mention of the word 'online' can sometimes mean opening up a whole can of worms...

Finke et al relied on a qualitative study methodology that involved asking young adults about their experiences of videogame playing. Bearing in mind the small number of participants under study, authors observed that "participants perceived playing videogames to have a positive impact on their lives and their development." Aside from the enjoyment angle (an important angle by all accounts), participants mentioned about the 'social' element to their videogame playing. Interesting too was another comment from Finke: "The motivations for playing videogames described are similar to those reported by typically developing populations." Why would they be any different?

The authors see the potential value of videogaming when it comes to "teaching" in the context of autism but I'm slightly reluctant to take up this idea for a few reasons. Foremost is the idea that not every activity where autism is mentioned really needs to be 'medicalised' or indeed, 'interventionised' (if there is such a word). I think back to all the discussions about Lego 'therapy' (see here) for example, and how it would be really easy to turn a pastime that is really enjoyable into something more like schooling, and then onward potentially make it less enjoyable. I would also mention that the types of videogames likely to be enjoyed by those on the autism spectrum are probably the same types of videogames that everyone else likes to play. So developing new 'autism-specific' games with intervention(s) in mind are not likely to go down too well in the context of gaming choice and their applicability to larger audiences. Indeed, it could be seen as discriminatory. And yes, then there is also the notion that 'interventionising' videogames will probably also promote yet more sedentary behaviours, something which we should all be keen to discourage (and I have my views about which physical activities might be considered favourable for many young people both on and off the spectrum).

Music to close and Céline Dion with a special guest... Deadpool? Although, his heart probably will go on given his renowned healing abilities...

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[1] Finke EH. et al. "To Be Quite Honest, If It Wasn't for Videogames I Wouldn't Have a Social Life at All": Motivations of Young Adults With Autism Spectrum Disorder for Playing Videogames as Leisure. Am J Speech Lang Pathol. 2018 Apr 2:1-18.

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What factors potentially predict quality of life in adults with autism?

"The study findings are that autistic people on average have lower QoL [quality of life] than the general population in the UK." Further: "Three main characteristics were predictive of lower QoL in almost all domains: being female, having a current mental health diagnosis and higher severity of autism symptoms." By contrast: "Significant positive predictors of QoL were: being employed (physical QoL), receiving support (social and environment QoL), and being in a relationship (social QoL)."

So said the findings reported by David Mason and colleagues [1] describing the results of a study that examined "quality of life (QoL) of a large sample of autistic adults in the UK and investigates characteristics that may be predictive of QoL." This is a vitally important topic because perceived quality of life IS important and has been discussed before in the peer-reviewed research arena with autism in mind (see here).

This time around the focus was on 'self-reported' quality of life for autistic adults who submitted data as part of a research initiative "into the life experiences of autistic adults, the Adult Autism Spectrum Cohort‐UK." Some 370 people (mostly) formally diagnosed with an autism spectrum disorder (ASD) completed the WHOQoL‐BREF, a quality of life assessment schedule developed by the World Health Organisation, as well as the Social Responsiveness Scale (SRS) and a initiative-specific questionnaire that collected various information including that about "everyday life including relationship status; home life including living alone or with family members (family of origin or spouse/partner); employment including paid employment, volunteering, or retired; education including type of school and qualifications achieved; support including who supports the adult and how often support is needed; mental health/neurological conditions including current diagnoses and type of medication/therapy; physical health conditions; and autism spectrum in other family members." Acquired data was put into the statistical 'measuring' machine and trends were reported.

So, a large proportion of participants were aged between 41 and 60 years old (~40%). The sex/gender ratios were fairly evenly split (males: 54% vs. females: 43% vs. 'prefer not to say' ~3%). The vast majority of respondents reported either a mental health issue as being concurrent to their autism diagnosis/status (~70%) or a physical health issue as being present (70%). I don't think we were actually told all the specific diagnostic categories that were included under 'a mental health issue' or 'a physical health issue' but some clues are provided in the text: "most commonly depression and/or anxiety" and "sleep problems, or hypertension." The WHOQoL‐BREF, by the way, provides information on QoL in various domains: physical, psychological, social and environment. Authors therefore report that: "Reported QoL for autistic adults was lower across all four domains than UK norms."

Then to those potential predictors of 'poorer' or 'better' quality of life, as some further statistical analysis was actioned on the collected data. Quite consistently - in the physical, psychological and environment domains - the same three elements cropped up as potentially predicting poorer quality of life: being female, having a comorbid mental health diagnosis and total scores on the SRS (an instrument that "measures autism characteristics" with a focus on social aspects). Looking at the statistical strength of the various factors observed, I'd have to say that the SRS score (total) - that measure of 'autism characteristics' - was the one that seemed to be most strongly related to QoL. Yes, the implications are that the [social] manifestation of autism itself *could* be an important driver of poorer QoL. Insofar as the factors potentially related to a more positive (better) quality of life, being employed, receiving support and being in a relationship were all mentioned, but certainly not as consistently across all the various WHOQoL‐BREF domains as noted in those negative predictors.

The authors highlight a few positives and negatives in relation to their study: use of a "robust measure of QoL is a strength", pretty large sample size and the collection of some good quality 'complete' results. That being said, they also note that a general QoL questionnaire might not gather all the important information relevant to QoL in the context of autism (I do wonder if all that ICF core sets of autism work might help matters on future research occasions). And then there's the issue of representativeness to consider, when it comes to the applicability of Mason results to the (very) wide autism spectrum (see here)...

Recommendations - 'implications' - aplenty spring from the Mason results. Focus in on better screening and treatment/management of mental health (and physical health) issues when concurrent to an autism diagnosis (see here and see here for examples). Make employment - long-term employment - work better for those on the autism spectrum (see here). Devote greater resources to discovering what factors surrounding female autism might lead to poorer quality of life. All noble sentiments worth pursuing. Alongside, are those results about autism severity also seemingly impacting on QoL. Does this perhaps also imply that moves to 'intervene' on core autistic symptoms might also be a target too? Y'know, on the understanding that 'core autism features' have also been *correlated* to some other, rather extreme endpoints also significantly affecting quality of life (see here)?

Addition: 10 May 2018. Y'know I mentioned that SRS scores - "measures autism characteristics" - might be an important variable when it comes to quality of life? Well, it seems another cohort came to similar conclusions [2] (click here for a larger view of figure b and those self SRS scores)...

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[1] Mason D. et al. Predictors of quality of life for autistic adults.  Autism Res. 2018. May 7.

[2] Oakley B. et al. Why Is Quality of Life Reduced in Individuals with Autism Spectrum Conditions? Investigating the Impact of Core Symptoms and Psychiatric Comorbidities on Quality of Life in the EU-AIMS LEAP Cohort. INSAR 2018.

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A gluten-free diet for schizophrenia "with elevations in antigliadin antibodies (AGA IgG)"

Although published on 1st April, the study abstract published by Deanna Kelly and colleagues [1] as part of the 6th Biennial Schizophrenia International Research Society conference is no joke.

Discussing some preliminary results based on the use of a gluten-free (GF) diet with a small participant group diagnosed with schizophrenia and also registering "elevations in antigliadin antibodies (AGA IgG)", authors reported a few things potentially significant to see in terms of the impact of such dietary intervention on presented symptoms.

Such a research topic might be 'new news' to some people but not to me. I've covered the topic of schizophrenia and gluten a few times on this blog (see here and see here for examples), and how names like Curt Dohan and Kalle Reichelt (rest in peace Tiny) really were pioneers of the idea that cereal based foods *might* have quite a few effects on both body and mind. Quite a bit of this early work on gluten and schizophrenia has also 'drifted' over to other diagnostic labels too (see here) (including stretching more generally to a 'secure ward' population [2]) and continues to make some peer-reviewed science waves (see here).

On this research occasion, Kelly et al  - who are not strangers to this area of scientific research - reported initial results based on the use of a randomised, double-blind trial where 16 participants were all assigned to a gluten-free diet and either given "10 gm of gluten flour or 10 gm of rice flour daily in a protein shake" in an inpatient setting for 5 weeks. Various schedules were used to assess symptoms at baseline and again at 5 weeks. Importantly, authors noted that: "The study was not powered to find a treatment effect, but designed to examine the feasibility of conducting an inpatient gluten removal study and examine trends in treatment."

Although not necessarily looking for a treatment effect at this stage, there were some potentially important changes noted between baseline and endpoint as a function of a GF diet. So: "During the clinical trial, participants receiving the gluten free diet had an improvement in negative symptoms as compared to placebo (treatment difference) with an ES=0.53." ES stands for effect size and negative symptoms form part of the clinical profile of schizophrenia. Likewise, scores on one scale (attention) of a cognitive battery also pointed to something potentially to see as a function of dietary intervention. I'll reiterate that this was a pilot trial and the participant number was low... but these results are interesting. And, perhaps as expected, strict adoption of a GF diet affected those antigliadin antibody levels too: "The AGA IgG levels decreased by 35% in the five weeks in the gluten free diet group relative to a 17% decrease in the gluten containing group."

"The feasibility study provided data to design the now ongoing fully powered confirmatory double-blind trial in people with schizophrenia with negative symptoms using a higher gluten amount (30 grams daily) and with aims to examine associated mechanisms, with targets of inflammation, neuroimaging and gut permeability." I'll be keeping my research eyes open for the results of that one on the basis of the presented Kelly results and some other previous research gems (see here and see here)...

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[1] Kelly D. et al. Randomized double-blind feasibility study of a gluten-free diet in people with schizophrenia and elevated antigliadin antibodies (AGA IgG). Schizophrenia Bulletin. 2018; 44: S190.

[2] Vlissides DN. et al. A double-blind gluten-free/gluten-load controlled trial in a secure ward population. Br J Psychiatry. 1986 Apr;148:447-52.

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