"Rates of death are higher for autistic individuals compared to the general population"

It's world autism awareness day today (2 April). The theme this year (2019), according to the United Nations, is "Assistive Technologies, Active Participation", something rather timely given the recent realisation that quite a few people diagnosed with autism are not properly represented [1] in various fields. Indeed, it continues a theme on the under-represented and under-studied populations within the autism spectrum being highlighted today of all days (see here)...

To mark this years autism awareness day, I'm turning to a topic that is probably about as important as it gets with regards to autism or any label: premature death. Specifically, highlighting how premature premature death can actually be when autism is diagnosed. I appreciate that this is not great PR for autism. But it's real-life for too many; and needs to be talked about as much as possible so people sit up and (hopefully) do something about it...

So...

The quote heading up this post - "Rates of death are higher for autistic individuals compared to the general population" - taken from the findings reported by Ye In (Jane) Hwang and colleagues [2] is perhaps not altogether accurate. A more precise meaning is required: as a group, those diagnosed with autism or autism spectrum disorder (ASD) are at some elevated risk for a premature (untimely) death than the non-autistic population (see here). Not exactly great dinner party conversation but vitally important nonetheless.

This is a topic that has cropped up time and time again in the peer-reviewed research literature and beyond (see here and see here). The grim trend crosses geographical boundaries and seems to cover the entirety of the autism spectrum. This time around New South Wales in Australia provided the source data with the aim "to report the rates and risk factors for mortality and cause of death in individuals on the autism spectrum (n = 35,929 age range 5-64) with and without concurrent intellectual disability (ID)."

The results: "Mortality rates for those on the autism spectrum were 2.06 times that of the general population." In line with other data (see here), those who also presented with intellectual (learning) disability alongside autism were at a higher risk of death. Epilepsy is also reported as being an important issue too (see here). A few other details are highlighted in the Hwang paper including the observations that "injury and poisoning" were some of the top-ranked causes of death as per other data (see here and see here).

What's more to say on this topic? Another call to action (see here)? Some more big news headlines (see here)? Another 'long-term plan' (see here)? All well-and-good, but what's actually being done to tackle such frightening statistics here and now and actually improve and extend lives in the here and now? What's currently helping autistic people to 'age well' [3]? And just in case you're of the inclination that epilepsy and intellectual (learning) disability are some how not a 'core' part of some autism, you're probably wrong in many cases (see here and see here)...

If the Hwang and other data aren't enough to satisfy you on this topic, another research paper with another (similar) set of grim findings has also recently been published [4]. Faced with such data, discussions about a puppet depicting one face of autism (see here) for example, don't seem all that important do they? Indeed, if half as much time was dedicated to highlighting the mortality stats associated with autism and making sure this makes headlines - over a 20 year period "6.4% of individuals died at an average age of 39 years" - may be more would be done to tackle them, including providing "adequate access to health care for individuals with autism spectrum disorder."

Food for thought, and hopefully important fuel for action...

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[1] Russell G. et al. Selection bias on intellectual ability in autism research: a cross-sectional review and meta-analysis. Molecular Autism. 2019; 10: 9.

[2] Hwang YIJ. et al. Mortality and cause of death of Australians on the autism spectrum. Autism Res. 2019 Feb 25.

[3] 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.

[4] Smith DaWalt L. et al. Mortality in individuals with autism spectrum disorder: Predictors over a 20-year period. Autism. 2019. Feb 28.

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The NHS Long Term Plan and autism

The NHS (National Health Service) here in Blighty has recently released its Long Term Plan [1]. 'Long term' covers a period of 10 years and how, faced with ever-mounting financial and resource pressures, the NHS is continually trying to serve the needs of the population it serves in an ever-changing world.

Needless to say that lots of media outlets have covered the Long Term Plan (see here for example) and the various priorities that it includes. 'Prevention' and 'Early Detection' are words used quite a bit both in the reporting and the document itself, as the focus moves slightly away from hospitals to other front-line services like general practitioners (GPs) and community care agencies.

Mental health also figures in the Long Term Plan, as this area continues on its 'parity of esteem' journey (see here). Being careful not to label autism as a mental health condition, I note that autism and learning (intellectual) disability also gets a mention in the Plan for quite a few reasons...

So, starting on page 52 of the report: "Action will be taken to tackle the causes of morbidity and preventable deaths in people with a learning disability and for autistic people." It's about time that this was 'tackled'. I say that because the statistics on early mortality with autism in mind are truly, truly shocking (see here and see here). It's also of interest that the LeDeR (Learning Disabilities Mortality Review) initiative is mentioned in this context too (see here). How exactly such inequalities will be tackled is however, not precisely detailed in the Long Term Plan.

Then: "The whole NHS will improve its understanding of the needs of people with learning disabilities and autism, and work together to improve their health and wellbeing." Allied to improving uptake of annual health checks that should be available, the document talks about working with partners to "bring hearing, sight and dental checks to children and young people with a learning disability, autism or both in special residential schools." It's a start given what said overlooked issues might be involved with (see here) but what about this who aren't in special residential schools? The Plan also mentions how: "By 2023/24, a ‘digital flag’ in the patient record will ensure staff know a patient has a learning disability or autism." I believe this would also solve a few 'issues' with regards to the (estimated) prevalence of autism and/or learning disability here in Blighty (see here and see here).

Also: "Children and young people with suspected autism wait too long before being provided with a diagnostic assessment." Yes, yes they do (see here). The Plan therefore sets out to "test and implement the most effective ways to reduce waiting times for specialist services." You've got to be kinda careful with the wording here because, as far as I can see, there is no commitment to a timescale of diagnosis unlike the commitment to reducing those with autism being accommodated at inpatient units for example: "By March 2023/24, inpatient provision will have reduced to less than half of 2015 levels (on a like for like basis and taking into account population growth) and, for every one million adults, there will be no more than 30 people with a learning disability and/or autism cared for in an inpatient unit".

I'll leave readers to decide whether this Long Term Plan represents something 'good for autism' or just skirts around some of the bigger issues. Personally I see some positives and some 'missed opportunities'. Positives? Well as I said, anything that can impact on those shameful early mortality figures in the context of autism is a good thing. I do have questions about how issues like suicidality, that contribute quite a bit to the early mortality stats, are for example, going to be addressed, but if lives are going to be saved and hopefully enhanced, I'm all for that. Reducing waiting times for assessments is also a good thing, as is the idea of the 'digital flag' to (hopefully) help enhance the doctor-patient interaction where autism is a feature.

Negatives? Well, there doesn't seem to be a great amount of details mentioned and even less discussed about adult autism and what the Long Term Plan is going to do for the thousands of autistic adults (many of whom are not also described as 'learning disabled'). There are many pressing issues for this group (see here and see here), some of which cross-over with the primary tenets of the Long Term Plan (see here) with autism in mind. It strikes me that there is much more to do in this area.

So I guess we'll just have to see what happens...

Music to close: And given some recent news about Weezer, a sublime blast from the past...

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[1] The NHS Long Term Plan. January 2019.

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Long-term health conditions and autism: a Scottish perspective

There were a few things that raised my eyebrow (Roger Moore style) in the paper published by Ewelina Rydzewska and colleagues [1]. The primary finding that: "Comorbidity is substantially greater in adults with reported autism than in other people" was one of the eyebrow raisers. But also the observation that: "Scotland’s Census is one of the few country censuses that asks every person in the country whether or not they have autism, indeed it may be unique in this regard" was another. There were others too...

Back to the first point and the primary purpose of the Rydzewska study: "to investigate the prevalence and predictors of deafness or partial hearing loss, blindness or partial sight loss, intellectual disabilities, mental health conditions, physical disability and other condition, in a whole country population of adults with reported autism aged 25+ compared with their peers without autism." Said whole country was somewhere not a million miles away from where I write this post - Scotland - and results based on Scotland's Census 2011. The Census did what Census (I don't know what the plural should be!) are normally designed to do: to provide details on the population (N=5 295 403) covering everyone "whether living in communal establishments (such as care homes and student halls of residence) or private households." As mentioned, I wasn't aware that Scotland asked about whether someone has autism or not as part of the Census but I'm impressed that they do. As to the authors' testing the idea that autism is rarely a stand-along diagnosis, well let's just say that this has already been discussed a few times in the peer-reviewed science arena (see here for example) so we kinda had some clues as to what might have been observed.

Results: "There were 6649/3 746 584 (0.2%) adults aged 25+ recorded to have autism as defined here, 4610 (69.3%) of whom were men and 2039 (30.7%) women compared with 1 776 845 (47.5%) men and 1 963 090 (52.5%) women in the adult population without autism." Let me just say that again: in the Scottish 2011 Census, that covered both private households and 'communal establishments' only 0.2% of the respondents above 25 years of age were reported to have a diagnosis of a "developmental disorder (eg, autistic spectrum disorder or Asperger’s syndrome)." Interesting.

Continuing: "The rate of autism was lowest in the oldest age groups (autism may be associated with reduced life expectancy)." 'Autism may be associated with reduced life expectancy' is another important detail mentioned in the Rydzewska study and something that taps into a wealth of other science (see here for example) observing that the diagnosis of autism can, very much, be a life-limiting label for some.

Then to the main event: lots of different diagnoses/labels were seemingly over-represented alongside a diagnosis of autism. Indeed, pretty much all of the ones enquired about by researchers fell into that category. A third of participants for example, reported experiencing 'something' under the heading of "a mental health condition"; a finding also fairly consistently stretching across individual analyses based on different age bandings. When it came also to the category titled 'intellectual disabilities' (also known as learning disability here in Blighty) the figures stood out: "A high rate of intellectual disabilities in children with autism has been described previously; we have now quantified the extent of this—29% (25%–32% depending on age group; 22%–35% for men and 31%–42% for women)—in a much larger study of adults." This [roughly] accords with other independent data too (see here).

What's more to say? Well yet again, it's all about not looking at autism as some sort of 'stand alone' label given the over-representation of some pretty significant categories of health issues, covering both the somatic (physical) and the psychological. I don't think anyone would disagree with such an observation in this day and age as the words 'Autism Plus' are also mentioned in the Rydzewska paper. Once again, this also has implications for preferential screening when a diagnosis of autism is received and also the treatment/management of any issues that are detected (see here and see here for examples).

As for that 0.2% adult (over 25 years old) prevalence figure noted by the authors, I'm not going to say too much more about that for now. Aside that is, from dropping in some discussions about another adult autism prevalence study (see here) which reported that figures were potentially climbing in line with the childhood autism prevalence figures (see here). I'm sure that there are multiple discussions that could be generated around such figures but not here, not now, and not to distract from the important observation that autism rarely appears in a diagnostic vacuum...

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[1] Rydzewska E. et al. Prevalence of long-term health conditions in adults with autism: observational study of a whole country population. BMJ Open. 2018;8:e023945.

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Even more on wandering and autism

I don't know if it's just me being more attentive but I seem to be more regularly seeing media reports of children and adults diagnosed with autism or autism spectrum disorder (ASD) wandering from their family/caregiver home or other place. Some of these media reports end happily insofar as the person being found safe and unharmed to then be reunited with their loved ones; on occasion, using some important previously learned survival skills. Other reports however, don't have such a happy ending...

The paper by Laura McLaughlin and colleagues [1] brings the topic of wandering (elopement) back into research view, observing that in their cohort of nearly 1500 parents talking about their children diagnosed with an ASD: "22.4% of the children wandered from their home or yard and 24.6% from a public place more than monthly." Such research continues a theme down the years illustrating how wandering is an important issue when it comes to autism (see here) and how for some, wandering can lead to a very, very final outcome (see here).

Researchers distributed their anonymous on-line questionnaire about various aspects related to wandering through several autism-related organisations, encouraging parents of children diagnosed with autism/ASD to respond. This wasn't your typical 'does your child wander?' questionnaire, but instead also incorporated several related items such as "the use of electronic tracking devices,... use of restraints and/or seclusion to prevent wandering at school, and receipt of guidance about wandering."

About a quarter of parents said their child wanders (and wanders quite routinely) and the effects of such behaviour seemed to be quite wide-ranging. Not only did wandering reflect a worry for many parents - "48.6% and 58.7% of parents were moderately/very worried about their child wandering from home or yard or a public place" - but almost three-quarters of parents reported that wandering concerns affected "decisions to let their child spend time with friends or family in their absence." With such sentiments being expressed, it's not difficult to see why some parents with children with autism have to announce 'why I can never die' (see here). Perhaps also surprisingly, McLaughlin et al reported that only a third of parents in their cohort "had previously received any counseling about wandering."

I still maintain that wandering represents one of the most important 'issues' linked to autism (see here). Lots more resources need to be dedicated to the hows-and-whys of such behaviour (see here) and what can be done to reduce risks all round. I also think more needs to be done to talk about wandering in the context of autism and provide parents/caregivers with the information and tools ('best available evidence') about wandering. Given also the seemingly important relationship between wandering and water safety in particular (see here), I'm minded to again vocalise the (preferential) need to teach water and swimming skills as and when a child is diagnosed...

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[1] McLaughlin L. et al. Wandering by Children with Autism Spectrum Disorder: Key Clinical Factors and the Role of Schools and Pediatricians. J Dev Behav Pediatr. 2018 Jul 6.

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"Health inequalities in relation to people with learning disabilities": LeDeR reports.

LeDeR mentioned in the title of this post, refers to the Learning Disabilities Mortality Review, an English initiative designed to "make improvements to the lives of people with learning disabilities." Part of that 'make improvements' statement covers the important area of health inequality and some truly shocking early mortality risks that seems to follow a diagnosis of learning (intellectual) disability. To quote: "Today, people with learning disabilities die, on average, 15-20 years sooner than people in the general population, with some of those deaths identified as being potentially amenable to good quality healthcare." Remind me what century we live in again...

I've covered early mortality in the context of learning disability before on this blog (see here and see here) and how various factors seem to 'converge' to place someone diagnosed with a learning disability at some significant disadvantage when it comes to living a healthy life or being faced with an early death. So things like an acknowledgement that various physical conditions seems to be 'over-represented' alongside learning disability and the idea that diagnostic overshadowing is quite prevalent are some key points to consider (see here). Minus any sweeping generalisations, I should also mention that the quality of the care received (or sometimes not received) by some people diagnosed with a learning disability is also sometimes part of the picture too (see here).

The LeDeR report covering 2017 [1] once again paints a pretty grim picture of how we, as a society, treat some of how most vulnerable members. Some media coverage of the publication of the report can be seen here and other bloggers have dissected some of the observations (see here). The report notes from the period 1st July 2016 to 30th November 2017 there were over 1300 deaths reported to the LeDeR programme. Also: "The median age at death of people with learning disabilities (aged four years and over) was 58 years (range 4-97 years). For males it was 59; for females 56." And: "More than a quarter (28%) of deaths were of people aged under 50 years." More than a quarter of deaths were noted in those under 50 years of age. Let that sink in, as it appears that being diagnosed with a learning disability impacts on how long you're likely to live. Perhaps just as important was another observation made: "People with profound or multiple disabilities had a median age at death of 41 years; those with mild or moderate learning disabilities had a median age at death of 63 years." There appears to be a dose-related relationship between mortality and how disabled a person was.

Details on the cause of death also revealed some important patterns. Respiratory illness was mentioned on the death certificate of some 30% of people - the most frequently cited cause - and diseases of the circulatory system in around 15%. In more detail, these categorisations covered specific diagnoses such as pneumonia and sepsis. I should also mention that epilepsy was also mentioned as a cause of death in around 4-6% of cases.

Where next? Well, the report does make some recommendations to try and reduce/eliminate such health inequalities in the context of learning disability. Obviously, not every death recorded could have been avoided, but there are potential ways and means in which care might (should!) be improved to help lessen the risk of early mortality for some. Outside of things like 'strengthening collaboration and information sharing' noted in the report, I was drawn to a few other recommendations including one stating that "all people with learning disabilities with two or more long term conditions (related to either physical or mental health) should have a local, named health care coordinator" and another that: "There should be a national focus on pneumonia and sepsis in people with learning disabilities, to raise awareness about their prevention, identification and early treatment." I can't disagree with such future directions assuming that is, that the money required to set in motion such recommendations, also follows suit.

I'm becoming increasingly aware that there is a real issue present in the care of many different groups of people, specifically where a learning, developmental or psychiatric diagnosis is mentioned. People with a learning disability are at a heightened risk of early mortality. People diagnosed with a developmental disorder such as autism are at a heightened risk of early mortality (see here and see here). People diagnosed with a psychiatric disorder such as schizophrenia are at a heightened risk of early mortality (see here and see here). It strikes me that the time may be right to set up an independent commission covering these (often, very often, overlapping) diagnostic areas, and look at putting into action new ways of ensuring that such diagnoses are not a gateway to an early death.

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[1] The Learning Disabilities Mortality Review (LeDeR) Programme. 2018. Annual Report December 2017.

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Physical conditions accompanying intellectual disabilities

By linking to some media discussing the inquest findings for Richard Handley (see here) I don't want to trivialise his death or use it as some kind of 'I told you so' example. Here was a man who died as a result of "gross failures" in his care as someone with Down's syndrome who also had a history of bowel issues. His death, linked to chronic constipation and "as a result of choking on his own vomit having inhaled gastric contents following complications with surgery", represents failures on many different levels. Not least is a seeming lack of appreciation of how bowel issues can very much be part of the clinical profile of many conditions / labels / diagnoses considered within the spectrum of intellectual or learning disabilities. Said issues require both regular monitoring and appropriate and timely intervention.

It is perhaps notable then that the paper by Deborah Kinnear and colleagues [1] is published these days, highlighting how various physical conditions are over-represented alongside diagnoses characterised by intellectual (learning) disability. The authors reported that: "The five most prevalent were visual impairment, obesity, epilepsy, constipation and ataxic/gait disorders."

Based on the examination of data for "people with intellectual disabilities living within the geographical area of Greater Glasgow Health Board, Scotland" between 2002 and 2004, researchers reviewed case records and "completed a comprehensive semi-structured health interview and targeted physical examination and followed a phlebotomy protocol, with the person with intellectual disabilities and their carer." This was time consuming affair - the "complete assessment process took about 4 hours per participant" - covering over 1000 people.

Alongside the reporting of those 'top five most prevalent conditions', researchers also observed several other important details. Namely that nearly all of their cohort had a least one co-existing health condition. The average number of comorbid conditions was 11. Yes, that's 11. And some people had over 25 comorbid physical conditions. When also taking into account the presence or not of Down's syndrome, not much changed in relation to the detected physical comorbidity. Similarly, when age and sex/gender were examined, the authors concluded that there only minor differences noted in terms of comorbidity profiles for example.

Relevant to the case of Richard Handley are some discussions in the Kinnear paper about constipation and learning disability. So: "Constipation was the fourth most prevalent physical health condition." Further, they note in another study [2]: "Eight people with chronic constipation had serious side effects (rectal prolapse, diverticula of colon, intestinal obstruction, megacolon and haemorrhoids) and four eventually died of intestinal obstruction... Thus, as well as being painful, constipation may remain undetected for a long time and can cause death due to missed clinical symptoms." Constipation can cause death. It shouldn't in this day and age, but it still does...

In relation to the implications of their findings, the authors make some additional comments. So: "Medical education is also focused on assessment and management of single conditions, yet management of multimorbidity is far more complex." This is important. It implies that outside of the view of one [primary] diagnosis being present per patient, clinicians and others need to have a more plural view, particularly when it comes to something like learning disability. The focus also on 'physical' comorbidities is also important insofar as how various developmental / behavioural / psychiatric conditions can very much manifest other non-developmental / behavioural / psychiatric conditions. This kinda mirrors what has been discussed with autism in mind (see here and see here for examples) and, alongside, the barriers that need to be overcome when it comes to appropriate diagnoses being made in such contexts (see here).

Set within other data indicating that premature mortality is very much over-represented when it comes to learning disability (see here), and that much of that early mortality is due to the presence of physical health conditions, the Kinnear findings should serve as a wake-up call to many sectors of the professional and other communities. Screening, diagnosing and implementing timely and appropriate intervention(s) are key recommendations; with a specific focus on the person as a whole rather than just being the sum of individual somethings like the diagnosis of learning disability. Bear in mind also that guidance on the management of something like constipation in the context of learning disability does exist in the peer-reviewed domain [3].

And finally, just in case you think that today's discussions aren't directly relevant to autism, constipation has also been mentioned in the death of another young person (see here)...

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[1] Kinnear D. et al. Prevalence of physical conditions and multimorbidity in a cohort of adults with intellectual disabilities with and without Down syndrome: cross-sectional study. BMJ Open. 2018; 8: e018292.

[2] Evenhuis HM. Medical aspects of ageing in a population with intellectual disability: III. Mobility, internal conditions and cancer. J Intellect Disabil Res. 1997 Feb;41 ( Pt 1):8-18.

[3] Robertson J. et al. Constipation management in people with intellectual disability: A systematic review. J Appl Res Intellect Disabil. 2017 Nov 23.

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Mortality patterns in older adults with learning disability

"Older adults with ID [intellectual disability] in Sweden carry a higher mortality risk compared with the general population, mainly attributable to respiratory, nervous and circulatory diseases."

So said the findings reported by Nawi Ng and colleagues [1] (open-access) looking at the records of some 15,000 individuals diagnosed with an intellectual (learning) disability compared with a similar number of control participants with respect to "patterns and risk of mortality." Researchers observed "a 4-fold higher mortality rate and a shorter median survival time among older adults with ID compared with the general older adult population." Such findings add to a growing body of research literature (see here and see here) suggesting that behavioural, cognitive and/or psychiatric diagnoses seem to be accompanied by some significant health inequalities too.

I could go into the details about the Ng study and the various ins-and-outs of how they arrived at their conclusions but to be honest, I'm just not minded to. I say that on the basis that the figures speak for themselves: "the mortality rate among individuals with ID was three times higher than in the control population" and: "Individuals with DS [Down's syndrome] had an 11-fold higher mortality risk than the control population." Sombre reading, perhaps partially reflecting how modern-day society treats some of its most vulnerable citizens.

The ultimate question should be: is there anything that can be done to improve this situation for those with a learning disability? Yes, is the answer; taking into account how a diagnosis of ID for example, means that some might be "more prone to cardiovascular disease risk factors, morbidity and mortality than the general population." The authors add that: "Cardiovascular disease health promotion and prevention programmes should be tailored for individuals with ID, to prevent or delay cardiovascular diseases and premature mortality in this vulnerable group." Corresponding action in relation to deaths resulting from epilepsy for example, should also (and always) be a priority.

"How a society treats its most vulnerable is always the measure of its humanity" is a phrase attributed to many speakers. On the basis of the research from Ng and other study results, society is seemingly failing many of those diagnosed with a learning disability as it is other vulnerable sections of society (see here).

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[1] Ng N. et al. Mortality patterns and risk among older men and women with intellectual disability: a Swedish national retrospective cohort study. BMC Geriatrics. 2017; 17: 269.

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"Spending constraints" a.k.a austerity and a mortality gap?

I try not to be too political on this blog given the focus on peer-reviewed science and my notable lack of political interest or motivation. It is however difficult to completely separate science and politics from each other given the world that we live in. A world that still commonly uses the term 'austerity' quite a few years after 'the crisis', alongside a realisation that behind headlines on the continued drive(s) towards 'fiscal security' and 'balancing the books' there are inevitably going to be winners and losers.

The paper by Jonathan Watkins and colleagues [1] highlights some of the extreme 'not winners' potentially stemming from the "relative constraints in public expenditure on healthcare (PEH) and social care (PES)" here in Blighty over recent years. They concluded that: "Spending constraints between 2010 and 2014 were associated with an estimated 45 368 (95% CI 34 530 to 56 206) higher than expected number of deaths compared with pre-2010 trends." Further, that if current trends continue in relation to funding "approximately 150 000 additional deaths may arise between 2015 and 2020." Cue the sharp intake of breath as words such as 'economic murder' have been banded around in the popular press discussing this research (see here).

I'm not going to go into all the details of the Watkins findings on this occasion. The data for the study was derived from various public databases in relation to population mortality, spending on health and social care and related variables. I don't doubt that as per other debates on the 'weekend effect' for example, different people with different mindsets might arrive at different conclusions based on such data. 'Lies, damn lies and statistics' is a phrase that springs to mind. With my very rudimentary reading and understanding of the Watkins data, I can't however see any obvious flaws in their methods or logic behind their results; aside that is from remembering that correlation does not always equal causation. Others have voiced similar sentiments (see here).

"By setting, deaths at care homes and at home contributed most to the observed ‘mortality gap’, while hospital mortality was lower than expected." The authors specifically attribute such data to the gap between spending on social care vs. spending on health care; also noting that "the recent drive to move patients with poor prognoses and who have reached their ceiling of care away from the hospital environment to care homes or their own homes may have contributed to this." One interpretation of this is that those who are elderly and/or vulnerable placed outside of the hospital environment are perhaps disproportionately being burdened with the effects of austerity.  And one possible solution? Well: "Our study suggests that the number of NHS-qualified nurses is the strongest tested mediator of the relationships between spending, and care home and home mortality." One solution but not the only solution.

With a UK budget announcement set for later this week, I wonder if the Watkins findings might figure in relation to the suggestion that "a cumulative spending increase of approximately £25.3 billion would be required to close this gap across health and social care by 2020/2021, equating to around £6.3 billion annually." Yes, those are some quite staggering sums of money, but at the end of day what is to be valued more: balancing the books or plugging quite a significant mortality gap? (and even some of our elected officials seem to be interested in this debate).

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[1] Watkins J. et al. Effects of health and social care spending constraints on mortality in England: a time trend analysis. BMJ Open 2017;7:e017722.

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Risk of death by injury and autism

"Individuals with autism appear to be at substantially heightened risk for death from injury."

That was the research bottom line published by Joseph Guan & Guohua Li [1] from Columbia University Medical Center, who trawled the accumulated data included in the US National Vital Statistics System screening death certificates issued between 1999 and 2014. Of the approximate 32 million death certificates issued, researchers found that some 1300 people diagnosed as on the autism spectrum had their death recorded and recorded with mention alongside the word 'autism'.

A few details about those 1367 deaths among those with autism compared with the not-autism population have captured media headlines (see here) not least the finding that: "The mean age at death for individuals with autism was 36.2 years (SD = 20.9 years), compared with 72.0 years (SD = 19.2 years) for the general population." This data tallies with other reports on premature mortality in relation to autism (see here) and also in respect of some of the over-represented comorbidity that accompanies the label such as learning (intellectual) disability (see here). I might also forward the idea that another over-represented comorbidity - attention-deficit hyperactivity disorder (ADHD) - appearing alongside autism (see here) might also have some relevance when it specifically comes to injury risk too (see here). Researchers noted that around a quarter of deaths recorded for those with autism were attributed to injury - quite a bit more than compared to those general population death stats - and that suffocation, asphyxiation and drowning made up a considerable portion of those deaths following injury.

OK a step back is required here. This data does not mean that those diagnosed on the autism spectrum have a life expectancy of 36.2 years (plus or minus 20 years or so). I know it might seem a little obvious to make such a statement but I do want to make that point clear. The data did find that premature mortality due to injury and non-injury does seem to 'hit younger' when it comes to autism (bear in mind also that the majority of recorded deaths where autism was mentioned were not caused as a result of injury).

Appreciating that some people have voiced their concern about these and other aspects of the Guan/Li paper (see here) I have to say that I'm slightly more focused on the pattern of death by injury reported over above whether the data is truly representative of the autistic population at large or not. Death by drowning is sadly something all too familiar when it comes to autism, particularly when taken in the context of the wandering/elopement data (see here). Yes, for some, autism, the presentation of autism and autism+, can be a life-limiting diagnosis. I know that last sentence does not make for great PR but readers should look at it in the context of other important peer-reviewed information too (see here and see here).

So, what can be done to improve life prospects for those on the autism spectrum?  Well, outside of moves to reduce the multitude of health inequalities that seem to coincide with receipt of a diagnosis of autism (see here and see here for examples), there are some practical solutions. Lessons about water safety and learning to swim represent a first course of action [2] and something that may have various other beneficial effects in terms of physical fitness (another issue that pervades the autism research literature). I appreciate that some children (and adults) might be a little nervous around water but with a bit of creative thinking (i.e. making swimming fun and not just about learning to do lengths of the pool) I don't doubt that learning such a life-saving skill can be made much more palatable. In the age where cinemas and theatres have 'autism-friendly' screenings, surely autism-friendly swimming sessions can't be too far behind?

In relation to reducing other potential causes of death such as suffocation and asphyxia, there may be other things to consider. I'm assuming that food and eating patterns may be something to be looked at when it comes to problems such as choking. This means education about eating small morsels of food and the importance of properly chewing food before swallowing so reducing the frequency of food packing for example. This might also have implications as and when medication is taken orally (via the mouth) and little details such as ensuring that a person drinks some water with any tablets that need to be taken. You may again think this is a rather obvious thing to say but sadly, there have been occasions when choking on food [3] has led to rather extreme consequences in the context of the autism spectrum.

In short, death by injury in relation to autism is not something that should ever be accepted. Yes, injury and accidents happen, but that's not to say nothing that can be done to prevent some/many of them...

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[1] Guan J. & Li G. Injury Mortality in Individuals With Autism. American Journal of Public Health. 2017. Mar 21.

[2] Pan CY. Effects of water exercise swimming program on aquatic skills and social behaviors in children with autism spectrum disorders. Autism. 2010 Jan;14(1):9-28.

[3] Hudson A. et al. Packing and Problematic Feeding Behaviors in CHARGE Syndrome: A Qualitative Analysis. Int J Pediatr Otorhinolaryngol. 2016 Mar;82:107-15.

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Guan, J., & Li, G. (2017). Injury Mortality in Individuals With Autism American Journal of Public Health DOI: 10.2105/AJPH.2017.303696

Premature mortality in intellectual disability in Australia (and England)

"Adults with ID [intellectual disability] experience premature mortality and over-representation of potentially avoidable deaths."

The paper by Julian Trollor and colleagues [1] (open-access available here) provides some sombre reading today, as once again the topic of early mortality is raised on this blog. Looking at several measures - the "Age Standardised Mortality Rate (ASMR), Comparative Mortality Figure (CMF), years of productive life lost (YPLL) and proportion of deaths with potentially avoidable causes" - authors paint a depressing picture of a 'mortality gap' between those diagnosed with a learning (intellectual) disability and the wider, general population.

I don't want to trawl through the Trollor paper in great detail given that it is open-access for all to see, but a few points are worthwhile raising. So, based on data from some 20,000 adults (aged 20 or over) registered with an intellectual disability (ID) in New South Wales (NSW) in Oz, there were 732 deaths reported (4%) "equivalent to a crude death rate of 5.9 deaths per 1000 people per year." The median age at death was 54 years and about 60% of deaths were in men. A control cohort consisting of adults from NSW was used as a comparator where "a crude death rate of 9.1 deaths per 1000 person years" was calculated. The median age at death however, for the control group, was 81 years. When looking at death rates between the groups according to age banding (20-44 years, 45-64 years, 65+ years) authors noted that: "People with ID in the 20–44 years age category had four times the death rate of the comparison group."

Looking at the causes of death between the ID and control groups, authors noted some potentially important trends. So: "Cause of death in [the] ID cohort was dominated by respiratory, circulatory, neoplasm and nervous system." This bearing in mind that cause of death was not available for everyone diagnosed with an ID (only 87%). Such causes were not wildly different from those noted in the control population but when it came to 'potentially avoidable deaths' the ID group were placed at some quite notable disadvantage, with 31% of deaths falling into this category (revised up to 38% depending on the 'death classification' used) compared with 17% in the general population. Readers should also note that: "Potentially avoidable deaths are deaths from conditions that are preventable through individualised care and/or treatable through existing primary or hospital care for persons aged under 75 years and which are avoidable in the context of the present health system."

As you can see, there are some quite shocking details noted in the Trollor paper. The emerging picture that some of the most vulnerable people in society (certainly in Australia) are (a) at risk of dying earlier than the general population and (b) at greater risk of suffering a 'potentially avoidable death' is one that no-one should be proud of. And just in case you though the results might not be generalisable to other parts of the world... you're wrong [2] (open-access here) as data from England reveals that: "Mortality rates for people with ID were significantly higher than for those without. Their all-cause standardised mortality ratio was 3.18. Their life expectancy at birth was 19.7 years lower than for people without ID." Truly shocking.

What can society do about such a state of affairs? Well, potentially lots (and it doesn't take monumental shifts to achieve better outcomes either). "Particularly stark is the large proportion of
potentially avoidable deaths due to infections. Such deaths suggest that people with ID experience delays, difficulties or differences in accessing specific and effective interventions for infections. Medical assistance must be sought assertively in individuals who manifest symptoms, but this is made difficult as patients with ID may not readily report symptoms, and some providing direct care
lack skills in early identification of relevant physical signs. Primary care providers should consider careful assessment, proactive treatment and close monitoring of progress if there are infections in this population." Sorry for the large chunk of replication text there but several important themes are laid out by Trollor, some of which overlap with other work in relation to autism for example (see here). Not least is the need for 'proactivity' on the part of clinicians and other professionals, potentially dealing with a group who may not be able to readily communicate their physical state for example and so shifting the responsibility on medical care being inspective and proactive. This means regular health screening and, at the basic level, understanding that a diagnosis of ID (or autism or schizophrenia [3]) does not seemingly provide any protection against the development of life-threatening illness or other conditions becoming evident.

I close with an article discussing another part of the reason why people with ID are being placed at an unacceptably high risk of early death: when those who are supposed to provide care, fail.

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[1] Trollor J. et al. Cause of death and potentially avoidable deaths in Australian adults with intellectual disability using retrospective linked data. BMJ Open. 2017. Feb 7.

[2] Glover G. et al. Mortality in people with intellectual disabilities in England. J Intellect Disabil Res. 2017 Jan;61(1):62-74.

[3] Hjorthøj C. et al. Years of potential life lost and life expectancy in schizophrenia: a systematic review and meta-analysis. Lancet Psychiatry. 2017 Feb 22. pii: S2215-0366(17)30078-0.

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Trollor J, Srasuebkul P, Xu H, & Howlett S (2017). Cause of death and potentially avoidable deaths in Australian adults with intellectual disability using retrospective linked data. BMJ open, 7 (2) PMID: 28179413

Gut barrier integrity meets blood-brain barrier integrity with autism in mind

"In the ASD [autism spectrum disorder] brain, there is an altered expression of genes associated with BBB [blood-brain barrier] integrity coupled with increased neuroinflammation and possibly impaired gut barrier integrity."

Although pretty enthused to see research linking names like Anna Sapone, Tim Buie and Alessio Fasano in the recent paper published by Maria Fiorentino and colleagues [1] (open-access), I was slightly less impressed with the use of the term 'the ASD brain' in their paper potentially joining two concepts that I've been quite interested in down my research years: gut barrier and blood-brain barrier function in the context of autism. Yes, I accept that those most precious of resources, donated brains from the deceased, represented some of the 'material' under scientific scrutiny, but if science has learned anything about autism down the years, it is that sweeping generalisations such as terms like 'the autism brain' don't reflect what the existing research tells us about the heterogeneity under the label. I might just as well use the term 'blogger brain' to denote some of my activities, but such a label tells you nothing about me aside from my pastime.

After that little rant, the paper from Fiorentino is an interesting one in that the goal was to "investigate whether an altered BBB and gut permeability is part of the pathophysiology of ASD." To do this, tissue from both brain and gastrointestinal (GI) tract donated by a small number of deceased and non-deceased participants who were diagnosed with autism, schizophrenia or nothing related (not-autism controls) were analysed "for gene and protein expression profiles." This work was undertaken on the basis of "the interconnectivity of the gut–brain axis, [that] suggests that inappropriate antigen trafficking through an impaired intestinal barrier, followed by passage of antigens or activated immune complexes through a permissive blood–brain barrier (BBB), can be part of the chain of events leading to neuroinflammation and thereby subsequent disease." I might add that the use of the word 'disease' in that sentence is, I think, aiming to describe the physiological effects of 'leaky barriers' not the diagnosis of autism. It is unfortunate however that 'disease' still continues to be banded around in the context of autism [2].

I think it's important to stress that the Fiorentino study was in effect two studies: one that looked at brain samples from one participant group who had died, and one that looked at GI samples from those who were still living (at the time of sample collection) and who presented with "GI symptoms undergoing esophagogastroduodenoscopy (EGD) for clinically indicated reasons." This was not a study where biological samples - brain and gut - came from the same person but rather a mash-up. Keep that in mind for now. The sorts of genes that were focused in on were those "associated with the formation, integrity, and function of the BBB and neuroinflammation" and included the claudins and something called MMP-9 and MMP-2 that have been discussed previously on this blog (see here) with leaky barriers in mind. The key words are 'barrier integrity' when it comes to the list of compounds that were under inspection.

Results: well it was good to see the authors list details of each of the participants from which tissue were used in their study. Brain tissue from the deceased with autism for example, is subject to quite a few factors that can influence the outcome of any results obtained; not least whether specific comorbidity accompanied their autism diagnosis and the nature of their death. Indeed, looking through the various case report numbers, I'm struck by how young many participants, particularly those diagnosed with autism, were at the time of their death. This ties into other discussions and debates (see here).

"Our molecular analysis of the BBB integrity and function shows an altered BBB in the ASD subjects evaluated." This was evidenced by elevations in the gene expression of MMP-9 and its proposed connection to disturbances of BBB integrity. Further: "Of the four claudins (i.e., CLDN-1, -3, -5 and -12) that to date are thought to be incorporated in the BBB... we found that two were significantly more expressed in the ASD brain as compared in HC [healthy controls]." Once again I might suggest the term 'healthy controls' is not an inappropriate one when it comes to determining not-autism or not-schizophrenia.

Then to analysis of those [independent] gut biopsy samples: "results, showing increased expression levels of pore-forming (66% of the ASD samples) and decreased levels of barrier-forming (75% of the ASD samples) TJ [tight junction] components in the duodenal samples, suggest an impaired gut barrier and serve as a proof of concept to support the hypothesis of a gut–brain axis dysfunction in a subgroup of ASD patients." So, those compounds linked to making the gut barrier more 'leaky' were seemingly increased in expression, and those linked to making the gut barrier less 'leaky' were reduced in quite a few of the samples from those diagnosed with autism. Mmm...

There is quite a bit more science included in the Fiorentino study but I think I've gone on long enough in this post. Suffice to say that the whole gut-brain axis thing with autism in mind gets a boost but more work is indicated, not least with larger sample groups and perhaps combining tissues from gut and brain from the same person. I would also like to see a little more done on this topic with some 'interventions' in mind, based on the other autism research that potentially links the authors (see here). Drawing for example, on a paper written by Prof Fasano titled: 'Zonulin, regulation of tight junctions, and autoimmune diseases' [3] suggesting that "gliadin, a storage protein present in wheat and that triggers celiac disease in genetically susceptible individuals, also affect the intestinal barrier function by releasing zonulin" one might see how far from being a set-in-stone state of affairs, dietary changes for some on the autism spectrum, might actually set in motion a host of biological changes pertinent to this area of work. And such changes might not be just confined to accepted gluten-related conditions either...

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[1] Fiorentino M. et al. Blood–brain barrier and intestinal epithelial barrier alterations in autism spectrum disorders. Molecular Autism. 2016; 7:49.

[2] Simms MD. When Autistic Behavior Suggests a Disease Other than Classic Autism. Pediatr Clin North Am. 2017 Feb;64(1):127-138.

[3] Fasano A. Zonulin, regulation of tight junctions, and autoimmune diseases. Annals of the New York Academy of Sciences. 2012;1258(1):25-33.

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Fiorentino, M., Sapone, A., Senger, S., Camhi, S., Kadzielski, S., Buie, T., Kelly, D., Cascella, N., & Fasano, A. (2016). Blood–brain barrier and intestinal epithelial barrier alterations in autism spectrum disorders Molecular Autism, 7 (1) DOI: 10.1186/s13229-016-0110-z

Anxiety disorders and mortality risk: implications for autism?

"Anxiety disorders significantly increased mortality risk. Comorbidity of anxiety disorders and depression played an important part in the increased mortality."

So said the findings reported by Sandra Meier and colleagues [1] looking to assess any relationship between the presence of an anxiety disorder and mortality risk. Based on data from one of those oh-so-useful Scandinavian population registries (Denmark this time), researchers reported that: "The risk of death by natural and unnatural causes was significantly higher among individuals with anxiety disorders... compared with the general population." Death by unnatural causes was also linked in quite a few cases to "comorbid diagnoses of depression."

Although making sombre reading, the data from Meier et al provide further evidence [2] that a psychiatric/behavioural diagnosis might have far-reaching implications when it comes to the risk of early mortality, be that based on natural causes or something rather more unnatural such as death by suicide or an enhanced risk of accidental death or death because of illness. This also follows a trend suggesting that severe mental illness also has social implications such as an increased risk of becoming a victim of crime too (see here). Quality of life, health-related or otherwise, is nearly always affected by such diagnoses.

I introduced the 'implications for autism' bit to this post simply because (a) when it comes to comorbidity surrounding the diagnosis of autism, anxiety and depression (various types) pretty much come top with regards to psychiatric labels applied (see here and see here respectively) and (b) enhanced risk of early mortality is also an unfortunate feature when it comes to autism too (see here). Putting these findings together and well, I'm sure you can understand the need for quite a bit more study in this area and in particular, a reiteration of how utterly disabling anxiety and/or depression can be when it comes to autism.

If and when possible roles for anxiety and/or depression are found to contribute to some of the excess risk of early mortality when it comes to autism, the bright side is that this could have implications for intervention and management and onwards a reduction in mortality risk. I might also introduce the findings reported by Butnoriene and colleagues [3] at this point, who suggested that sex differences might also be relevant to the type of risk factors associated with mortality. Discussions in this area should also probably include discussions on a related topic based on a particularly extreme path being selected by some on the autism spectrum (see here).

Without trying to make connections where none might exist, I'm also inclined to suggest that outside of psychological and pharmacological interventions to tackle anxiety and/or depression comorbid to autism, one might also look to treating certain somatic correlates also potentially exerting an effect (see here). There is potentially lots to examine across such comorbidities as yet again, another very important line of study opens up that intersects with autism.

Finally, dare I also add that other labels such as obsessive-compulsive disorder (OCD) that may also intersect with some autism (see here - yes, this is another Meier paper) might also increase the risk of early mortality when it comes to autism too [4]?

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[1] Meier SM. et al. Increased mortality among people with anxiety disorders: total population study. The British Journal of Psychiatry. 2016; 209: 216-221.

[2] Pratt LA. et al. Excess mortality due to depression and anxiety in the United States: results from a nationally representative survey. Gen Hosp Psychiatry. 2016 Mar-Apr;39:39-45.

[3] Butnoriene J. et al. Metabolic syndrome, major depression, generalized anxiety disorder, and ten-year all-cause and cardiovascular mortality in middle aged and elderly patients. Int J Cardiol. 2015;190:360-6.

[4] Fernández de la Cruz L. et al. Suicide in obsessive-compulsive disorder: a population-based study of 36 788 Swedish patients. Mol Psychiatry. 2016 Jul 19.

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Meier SM, Mattheisen M, Mors O, Mortensen PB, Laursen TM, & Penninx BW (2016). Increased mortality among people with anxiety disorders: total population study. The British journal of psychiatry : the journal of mental science PMID: 27388572

Risk of premature death and autism: some reflections

"People with autism 'die younger', warns charity" went the very stark BBC headline recently.

Today I'd like to bring your attention to the recent report published by Autistica titled: 'Personal tragedies, public crisis' making the headlines, highlighting how people with autism face a considerably enhanced risk of early mortality compared with the general population [1] (see here for my take).

Although making quite sober reading and rightly using some very emotive language, I think most people would welcome this report in highlighting an issue that for too long has seemingly been 'brushed under the carpet'. I've talked about early mortality and autism a few times on this blog (see here) and how it potentially intersects with other issues such as wandering/elopement (see here) and access to appropriate medical care that takes into consideration the manifestation(s) of autism (see here). The bottom line being that for some people, autism and/or the 'effects' of autism for whatever reason, can be life-limiting as well as life-changing. I know that last sentence might not make a great autism awareness message but lives are being needlessly lost. Indeed, I wonder whether with World Autism Awareness Day approaching, that should be a primary message this year and every year...

I appreciate the calls for further research on the issue of premature mortality and autism highlighted in the Autistica report and how for example, we do need to know more about the prevalence of early mortality in relation to autism here in the UK. I'm fearful however that potentially spending years (and precious monetary resources) waiting for such prevalence data to be forthcoming does little for those at risk here and now. Indeed, the more important issue of how to reduce (eradicate) the early mortality risk in relation to autism, is something that I would particularly champion and what this might mean for the way we think about autism in terms of the provision of screening and intervention and providing greater social and health support for those on the spectrum (and their families and loved ones).

Epilepsy and suicide are discussed as important causes of death when it comes to autism in the published report but are not then only ones. Accepting that suicide - ideation or completed - is a complex act with many potential roads bringing a person to such a final decision (see here), one of the primary opportunities for reducing suicide risk has to be to screen for comorbid symptoms/diagnoses that might enhance such risk. Y'know, things like depression (see here) in light of where that can potentially lead; taking into account that the label depression covers quite a bit of diagnostic ground and might not always appear as expected in relation to autism (see here). Treating and/or managing symptoms of depression should then be indicated, accepting that talking- and the traditional pharmaco-therapies might not be the only tools in the intervention arsenal (see here). I might also advance the idea that a person's social environment can also influence risk of suicide; such that this should likewise be assessed and acted upon accordingly. Social attitudes and policy have to change.

Epilepsy has a long history of association with autism (see here). Again, it's all about appropriate (and perhaps preferential) screening and monitoring as and when autism is diagnosed to keep an eye on symptoms pertinent to the development of epilepsy or seizure disorder. Medication can be life-saving when it comes to epilepsy but this should perhaps also come with some good medicines management including screening other parameters (see here). That quite a few 'types' of autism might actually come with epilepsy as part of the diagnostic package (see here) is worth noting in terms of receipt of a diagnosis of autism being a springboard to further screening and assessment. The idea that some of the more complementary interventions indicated for autism might also have an important impact on comorbid seizure issues outside of the label (see here) should be pointed out (with no medical or clinical advice given or intended).

As uncomfortable as it might be, the idea that a diagnosis of autism elevates the risk of premature mortality provokes the questions: should science be doing more to help lessen the risk(s) of someone developing autism in the first place and/or should we be focusing greater attention on ways to 'alleviate' the more disabling - life-threatening - symptoms of the label? I've already aired some of my views on this in previous posts on the equally emotive topic of euthanasia / assisted suicide in respect to behavioural / psychiatric labels where autism has been mentioned (see here). Alongside various research suggesting that childhood behavioural issues elevate the risk of adult psychopathology and onwards poorer life outcomes (see here), childhood neurodevelopmental issues such as autism have similarly been tied to later outcomes adversely affecting quality of life (see here). In short, what happens in childhood affects what happens in adulthood. Armed with that knowledge, should we not be doing all we can in childhood as well as adulthood?

Moves to increase awareness of autism - including the idea of heterogeneity - and importantly, creating a more welcoming society for people on the autism spectrum should of course remain a priority. I dare say that if more meaningful and 'sustainable' opportunities were afforded to those with autism, the risk of suicide might, for example, be lessened for quite a few who consider turning to such an extreme option. But alongside all the talk about 'celebrating autism' (to coin a term - see here) the report from Autistica highlights some very real and very raw implications attached to the diagnosis. How potentially moving autism from the (generalised) description of a 'life-long condition' to that of a potentially 'life-limiting condition' should be a call to action to ensure that a reduction of between 16 and 30 YEARS of life is no longer tolerated as and when autism is diagnosed. I'd say that represents a crisis indeed.

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[1] Hirvikoski T. et al. Premature mortality in autism spectrum disorder. Br J Psychiatry. 2015 Nov 5.

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Hirvikoski T, Mittendorfer-Rutz E, Boman M, Larsson H, Lichtenstein P, & Bölte S (2016). Premature mortality in autism spectrum disorder. The British journal of psychiatry : the journal of mental science, 208 (3), 232-8 PMID: 26541693

On the question of suicide risk and chronic fatigue syndrome

The paper by Emmert Roberts and colleagues [1] (open-access) forms the basis of today's post and the finding that: "There was no significant difference in age-standardised and sex-standardised mortality ratios (SMRs) for all-cause mortality... or cancer-specific mortality in patients with chronic fatigue syndrome when compared with the general population in England and Wales." This is good news indeed bearing in mind how much a diagnosis of chronic fatigue syndrome/myalgic encephalomyelitis (CFS/ME) can affect so many aspects of a person's life (see here).

The news about the diagnosis of CFS/ME not increasing 'all-cause mortality' is however tempered by the finding that author's results did show "a substantial increase in mortality from suicide"; something that has been picked up by some quarters of the media (see here). I might also direct readers to an accompanying commentary attached to the Roberts paper (see here).

The Roberts paper is open-access but a few points are worth highlighting.

Based on a participant group of over 2000 people diagnosed with CFS identified "using data from the South London and Maudsley NHS Foundation Trust Biomedical Research Centre (SLaM BRC) Clinical Record Interactive Search (CRIS) register", authors set about calculating mortality rates over a 7-year observation period (2007-2013). As per previous 'issues' on how a diagnosis of CFS is arrived at (see here) we are told that "the most inclusive criteria, and thus included all patients with a clinical diagnosis of chronic fatigue syndrome" were used.

Mortality details were cross-referenced with the participant group data initially "from the “Service User Death Report” of “the Spine”, maintained by NHS Care Records Service." Some further checking on the basis of the death certificate details and cross-referencing with ICD-10 codes "for cause of death" provided further details. Various other variables were also analysed including ethnicity, socio-economic status (SES) and the: "Presence of a lifetime diagnosis of depression was defined as having had a recorded depressive episode (ICD-10 code: F32.x) or recurrent depressive disorder (F33.x)."

Results: after some examination of the data authors "identified 2147 cases of chronic fatigue syndrome in CRIS with 17 deaths." Just over 1500 women were included for study and 11 of them had died (11/1533). Of 614 men included for study, 6 had died (6/614). Most deaths (n=8) were "from malignant neoplasm" but 5 deaths were recorded as suicide. Further: "When stratified by lifetime diagnosis of depression, 216 patients had a recorded lifetime diagnosis of F32.x or F33.x. Four (26%) of 17 patients who died had a lifetime diagnosis of depression, for two of whom the cause of death was suicide." The authors conclude that theirs is "the first study to show a specific increased risk of suicide in a population of patients with chronic fatigue syndrome compared with the general population." But also: "if there had been two fewer deaths by suicide, this risk would not be significantly increased."

Without belittling the tragedy that is suicide and the devastating impact it can have on a person's family and friends (as well as themselves), I do think it is worth reiterating a few cold, objective points. First, yes the suicide rate was heightened in this group over this period in comparison to population figures, but the overall number of cases was still low. Second, without making too many sweeping generalisations about depression, two of those five cases of suicide had a history of depression. Given the quite widely accepted greater vulnerability to suicide (attempted or actual) following a diagnosis of depression (see here), one could argue that a diagnosis of CFS/ME is potentially only coincidental to any such association. Other research has asked similar questions [2]. That is also if you assume that depression as a condition should not be included under the banner of ME/CFS [3]? Indeed reiterating that statement about 'if there were two fewer deaths by suicide, this risk would not be significantly increased' one *could* argue that the suicide rate in relation to CFS/ME is actually not increased with the depression findings taken into account. Finally, I'd also suggest that given the extent of the debilitating symptoms often associated with CFS/ME and the 'cruel' way that some/many people with CFS/ME have been treated down the years, the suicide rate reported by Roberts is perhaps lower than I think many people would have expected. Although not directly comparable with the Roberts data, suicide has been discussed on other occasions in the peer-reviewed domain with CFS/ME in mind [4].

Continuing: "This study highlights the importance of adequate assessment of mood and other psychiatric symptoms in patients with chronic fatigue syndrome, because lifetime diagnosis of depression is an independent risk factor for increased risk of completed suicide in this population. Although completed suicide was a rare event, the findings strengthen the case for robust psychiatric assessment by mental health professionals when managing individuals with chronic fatigue syndrome." I don't think anyone would quibble with the idea that a comprehensive assessment be provided to patients with ME/CFS given what is known about how psychology can be affected by the presentation of the condition (see here) and also that depression is not something to be taken lightly. I am however cautious of the idea that a 'robust psychiatric assessment' is the only thing offered to patients - "when managing" -  particularly when there is a wealth of peer-reviewed evidence on the more 'biological side' of the condition too (see here) with potentially important implications for treatment for example. I am, for example, going to be talking about the case report from Galán and colleagues [5] soon on how the "initial symptoms of mitochondrial disease in adults can easily be mistaken with CFS." Indeed, going back to that paper by Zdunke and colleagues [6] on how "there may be important differences in illness characteristics across individuals with CFS in the US and the UK, and this has implications for the comparability of research findings across these two countries" one might appreciate how much further the UK experience of CFS/ME needs to go in order to put biology and genetics on a research/clinical practice par with the influence of psychology and psychiatry in relation to the label.

And just in case you might not believe that CFS/ME can be a life-limiting condition, due respect should be paid to those who've paid the ultimate price as a result of their diagnosis.

For those who might need it, details of someone who will listen...

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[1] Roberts E. et al. Mortality of people with chronic fatigue syndrome: a retrospective cohort study in England and Wales from the South London and Maudsley NHS Foundation Trust Biomedical Research Centre (SLaM BRC) Clinical Record Interactive Search (CRIS) Register. Lancet. 2016. Feb 9.

[2] Fuller-Thomson E. & Nimigon J. Factors associated with depression among individuals with chronic fatigue syndrome: findings from a nationally representative survey. Fam Pract. 2008 Dec;25(6):414-22.

[3] Jason LA. et al. Unintended Consequences of not Specifying Exclusionary Illnesses for Systemic Exertion Intolerance Disease. Diagnostics (Basel). 2015 Jun 23;5(2):272-86.

[4] Jason LA. et al. Causes of death among patients with chronic fatigue syndrome. Health Care Women Int. 2006 Aug;27(7):615-26.

[5] Galán F. et al. Mitochondrial Myopathy in Follow-up of a Patient With Chronic Fatigue Syndrome. J Investig Med High Impact Case Rep. 2015 Sep 24;3(3):2324709615607908.

[6] Zdunek M. et al. A Cross Cultural Comparison of Disability and Symptomatology Associated with CFS. Int J Psychol Behav Sci. 2015;5(2):98-107.

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Roberts, E., Wessely, S., Chalder, T., Chang, C., & Hotopf, M. (2016). Mortality of people with chronic fatigue syndrome: a retrospective cohort study in England and Wales from the South London and Maudsley NHS Foundation Trust Biomedical Research Centre (SLaM BRC) Clinical Record Interactive Search (CRIS) Register The Lancet DOI: 10.1016/S0140-6736(15)01223-4