Saturday, 26 February 2011

Academic publishing: why isn't psychology like physics?


Copyright: www.CartoonStock.com

My job is to conduct research and publish the results, so I’ve become interested in discussions on the internet about models of publishing. There are three tensions in the field: (1) who pays to publish research? (2) who decides what gets published? and (3) who takes any profits?
In the traditional model, based on publishing on paper, the answers have been (1) readers pay, via journal subscriptions, (2) editors decide, advised by reviewers, (3) publishers take the profits.
But publishing on paper is in decline. Most of us read papers on the internet taken from electronic versions of journals; either directly on computer, or printed from a pdf. This has led to some re-evaluation of how publishing is done. Most publishers, with an eye to their profits, have attempted to just continue with the old system, charging subscriptions for electronic journals much as they have done for paper journals. These are largely paid for by institutional libraries rather than individuals. If your institution doesn’t subscribe, or you don’t belong to an institution, there is an option to pay for individual articles, but the charges are typically prohibitively high.
Psychologist Stevan Harnad was among the first to point out that the internet changes the publishing game completely. Way back in 1994, he suggested that academics could leave publishers out of the research communication cycle altogether. For instance, when I write an article, I could post it on my website, and anyone who wanted to read it could download it. Instead of institutions paying thousands of pounds in journal subscriptions, and individual readers being clobbered for $30 for a five-page article, academic exchange would be virtually free. What would be wrong with that? This appealing idea has over the past sixteen years fostered a huge amount of debate. What is surprising to me, though, is that there appears to be a massive discrepancy between disciplines in how far Harnad's ideas have been taken up. And Harnad's own discipline, psychology, is one that has been very slow to respond, compared, say, to physics - see http://arxiv.org/. Because many of my readers are psychologists, and many psychologists are wedded to the traditional printed journal, I'll briefly rehearse some of the arguments that have been made, before commenting on the possibilities opened up by e-publishing.
There are two obvious snags to bypassing journals. The first just concerns presentation. Traditionally, publishers have ensured that journal articles look nice: properly formatted, intelligible figures, grammatical writing, and so on. Their role in this aspect of publishing has, however, dwindled over the years. This does not mean that any academic can produce a nicely-formatted article: some skill is required. And many academics don’t write very well and benefit from the services of professional editors. But increasingly, publishers are cutting down on professional editors and graphic designers - indeed, increasingly they hold authors responsible for getting their figures formatted for direct web publication (see my post).
The second point has to do with quality control. Traditionally, journals have acted as gatekeepers. If an article appears in a peer-reviewed journal, it can usually be assumed that it doesn’t have serious flaws: the higher the quality of the journal, the safer this assumption should be. And for the top-notch journals with the highest impacts, publication of paper generally gives some guarantee that it is not only methodologically sound but also particularly important in terms of advancing the field.
Nevertheless, some have queried whether we need this traditional function of publishers, pointing out flaws in the peer review system and idiosyncratic behaviour by editors. Indeed, gatekeeping may not just be imperfect: In some fields it may damage science, by inducing a publication bias in favour of ‘significant’ (both statistically and theoretically) findings, and biasing against publication of replications (or non-replications).It's even been argued that publishing in high-impact journals may be unethical, because such journals are particularly likely to publish papers that over-state their conclusions. And unless there is a massive media stink, they resist accepting papers that fail to replicate exciting findings that they've previously published.
Such considerations have led some to argue that we should ditch not just publishers, but the whole editorial and reviewing process as well. In effect, everyone would just publish their own stuff, and it would be judged by the academic community. Although this solution appeals to the more anarchic side of my nature, I can see objections to it. The main is that we’d all be swamped by a tidal wave of information. The good stuff would be buried within a heap of rubbish. And because methodological flaws are often hard to detect unless you have done research in the field yourself, problematic but apparently important studies might have a large impact.
As Harnad noted, however, taking publishers out of the loop need not entail abandoning peer review. With the exception of a few top-notch journals, most academic publishers don’t employ editors. Instead, editors do the work for free, or for a small honorarium. It is very unusual for reviewers to be paid anything. So this gate-keeping role of publishers doesn’t need to reside with publishers. Any group of academics could decide to set up a journal, appoint editors and operate a reviewing process. Provided the journal was electronic, with no paper copies, this should be achievable at relatively modest cost.
But has it worked? Well, sadly, not for psychology. I've brooded about this issue for some years, but was stimulated to revisit it by my recent discovery of Kindle direct publishing. It’s different from an Open Access model, because the reader pays for content, but the sums involved are trivially small compared to current journal charges. The Kindle operation is designed for publishing books. Harnad has always distinguished commercial book publishing from what he terms ‘esoteric’ publishing, i.e. publication of academic articles of interest only to a small group of experts. But my Kindle experience made me wonder whether the distinction between the two types of publishing need be as sharp.
I came across Kindle e-publishing when I’d had my first novel turned down by two agents, both of whom spoke of a ‘saturated’ market for crime novels, the genre I’d written in. I’m not a typical author: though I’d be very pleased if people enjoyed reading my book as much as I enjoyed writing it, I don’t have any high expectations of a successful second career as a novelist. I write just for fun, and, being an academic, I’m used to not getting paid for what I write. And being an academic also makes me used to rejection, while at the same time recognising that I shouldn’t let it discourage me if I have a belief my work is good.
After my experience with open access journals, I’d expected that any business that offered a self-publishing option would charge authors, especially for ‘esoteric’ material. As a first novel by an unknown author and with neither agent nor publisher, my book was definitely in that category. So I was pleasantly surprised to find that not only did I not have to pay anything, but I could set my own price and get a 70 per cent royalty. For a book with low-volume sales, this is going to be negligible, especially as a great chunk of tax is taken off any US sales. (This can be avoided if you fill in a lot of forms, send away your passport to the US, and so on, but since I was not expecting more than about two US sales, this clearly was not a sensible option). Anyhow, you are walked through the process with easy instructions, which include downloading a couple of bits of software to convert your manuscript into the right format, and then you press a button and upload your book. The whole process took about the same amount of time than it typically takes to submit a journal article through an electronic portal. And  you don't need a Kindle, though the reading experience is better than on a PC or iPhone. I keep wondering if there’s a catch; no doubt someone will tell me if there is. In common with some others, I am a bit dubious about accuracy of the sales figures provided (there's a rather surreal exchange about this on the Kindle forum). But when I go to Kindle store, there it is: the Case of the Fremantle Fingers by Deevy Bishop at $2.99. This gives me a small glow of satisfaction.
So I started to think, couldn’t research papers adopt this model, but with even lower charges? I’m not saying the articles need be in Kindle format: I don’t think it’s ideal for scientific papers. But rather that the method of author download to a central site, from which others download at a minimal cost, would be an interesting one for psychology (and other discipline) journals to adopt. The difference from current practice is that the material would be affordable; instead of costing 20 to 30 dollars, individual articles would be priced at 20 to 30 cents. The Kindle store does sell some books, often out-of-print classics, for ridiculously low prices, (or even free), and the charging mechanism doesn’t seem to be a problem. Authors of academic articles don’t expect royalties, so these could be waived.
But what about the dangers of the free-for-all and lack of peer review to identify quality? I suspect that even a notional charge per download might be a help here, by identifying material that people would be willing to pay for. And the Amazon system also suggests a more explicit way in which refereeing be adapted to such a system, by using a star rating with optional comment. This is especially useful in the academic context if the reviewers themselves also have ratings. For instance, a 5 star reviewer would be one who’d published significant papers in the same area, a 3-star reviewer would be someone familiar with this or a related area but without significant publications in that area, and those with no expertise would be 1-star reviewers. It shouldn’t then be difficult for readers to see which papers had high ratings from knowledgeable people.
But would people make ratings? There has been disappointing take-up of Comments options for journals that offer this, such as the PLOS series. I don’t like anonymity, but  to encourage frank comments on papers,  there would have to be some way of registering reviewers that made it possible to check their credentials and assign them an anonymous ID. Otherwise, junior people might be scared to make adverse comments on senior figures.
The downside? For me, omitting the usual pre-publication reviewing stage would have disadvantages. As I’ve noted before, I find peer review painful but often helpful in improving papers. Sometimes it has saved me from revealing an embarrassing amount of ignorance. I’d be happier with a system that allowed an option for pre-publication review, and it would be interesting then to see whether papers that were identified as having undergone such review would attract better post-publication ratings than other papers. Learned societies, for instance, which currently produce paper journals, might instead use their existing reviewing systems for papers which are then deposited on the electronic store, with a quality kitemark.
Another drawback is that for some people, particularly those in low-income countries, even a nominal charge per download would be a deterrent. But it should be feasible to devise a system that subsidised those from resource-poor countries. Or  the charge could be waived once a certain number of downloads had been achieved, so that the most popular material becomes automatically freely available.
Since Harnad’s ground-breaking formulation of the Subversive Proposal much has been written about models of science publishing. Many changes have occurred, with increasing pressure for open access, and growing criticism of the peer review system. But there remains a massive difference between disciplines: physics embraced self-archiving rapidly, whereas scientists in other disciplines often don't even realise it's an option. Maybe this has to do with the pace of change and degree of competition in a field: physicists don't want to wait months before their work is published because it could be scooped. Psychology experiments are seldom so time-sensitive. Nevertheless, I think the pace of research in our field could be improved enormously if we broke free from the stranglehold of the traditional commercial publishers. 

btw, Tom Webb has drawn attention to a Commons Select Committee that is seeking evidence on the peer review process. This strikes many people, including me, as a bit strange, in that it would seem to fall outside the remit of government. If you want to comment, you need to do so by Thurs 10th March, 2011.


Saturday, 29 January 2011

Orwellian prize for journalistic misrepresentation: an update


I've been re-reading George Orwell's Nineteen Eighty-Four to get me in the mood for looking at nominations for the Orwellian prize of journalistic misrepresentation. Bizarrely, it cheered me up. I last read it in 1968, and it made me nervous. But here we are in Airstrip One in 2010: we may be heading for mass unemployment, dismantlement of the NHS, the BBC and the Universities, we may be getting increasingly uncomfortable with the state's attitude to controlling dissent, but compared to what Winston Smith went through, this is paradise. New technologies are being used to oppress people the world over, but the internet has also emerged as a tool for fighting oppression. I can expostulate on my blog about things that concern me without the thought police carrying me off.

But, back to the prize. Why, I've been asked, did I choose that name? In Orwell's dystopian world, the press is used to achieve control, such that "nominally free news media are required to present 'balanced' coverage, in which every 'truth' is immediately neutered by an equal and opposite one. Every day public opinion is the target of rewritten history, official amnesia and outright lying, all of which is benevolently termed 'spin', as if it were no more harmful than a ride on a merry-go-round." This captured what I felt when I read the coverage of certain scientific discoveries in the press. I knew that there was an Orwell Prize to celebrate the best of British political journalism; I saw the Orwellian Prize as the inverse – a way of noting the worst of science journalism.

I wasn't so concerned by the fact that journalists sometimes make mistakes. That's inevitable when writing under time pressure on subjects where one does not have expertise. I accept too that journalists, and their editors, are not required to be neutral, and are entitled to promulgate their opinions. I'm concerned, however, when mistakes have implications for people who are vulnerable, who might be misled into adopting an ineffective treatment, or shunning an effective one, by a prominent newspaper article. Or, in the case of major environmental issues, a piece on climate change or the impact of industrial practices might sway public opinion in a direction opposite to that held by informed scientists, and risk making our planet less habitable for future generations.  The aspects of scientific writing that particularly disturb me are when science is misrepresented, and that misrepresentation is done thoughtlessly or even knowingly: sometimes for political ends, but more often to get a 'good story' that will have an eye-catching headline and so sell more newspapers.  The MMR story is the most vivid illustration I'm aware of. It's bad when hard-pressed readers are induced to spend money on ineffective treatments; it's worse when children risk disease or even death as a consequence of irresponsible journalism.

In a brilliant post last September, Martin Robbins lampooned the formulaic treatment of science stories that is so often seen in the media. His was a generic account, but in specific areas of science, one can provide even more detail. Thus, for every new advance in neuroscience or genetics, there seems to be one of two possible conclusions: either (a) if confirmed, the discovery will make it possible treat dementia/dyslexia/autism/Parkinson's disease/depression in future, or more commonly (b), the discovery will lead to better diagnosis of dementia/dyslexia/autism/Parkinson's disease/depression.  Yet most of the research in this area is a long way from translation into clinical practice. We are still learning to interpret tools such as brain imaging and genetic analysis, and the more we do, the more complex it becomes. Researchers become rightly excited when they find a neural or genetic correlate of a disease, which could help our understanding of underlying causal mechanisms. This is a vital step in the direction toward effective clinical procedures, but it's usually unrealistic to imagine these will be available soon. Yet in their desire to appeal to human interest, journalists will exert pressure to make more of a story than is justified, by focusing more on hypothetical rather than actual findings. The impetus behind the Orwellian prize is described in my earlier post, where I dissected an article describing a positive impact of fish oil on concentration in children with attention deficit hyperactivity disorder (ADHD). The study behind the story did not use fish oil, did not include children with ADHD, and did not find any behavioural benefits in the children who were given fatty acids. This article epitomised everything that made me angry about reporting of science in my area, with errors so numerous it was hard to believe they were accidental. I therefore threw down a challenge to others to see if they could find equally worrying examples. As it happens, that article was subsequently the focus of a scathing attack by Ben Goldacre, and was taken off the Observer's website.

Now, one of the things I love about blogging is its interactive nature. A number of commentators made points that made me think more. None of them were apologists for poor reporting, but they noted that journalists are not always to blame for an overhyped article. For a start, they have no control over headlines, which are  typically written by a sub-editor whose job it is to attract the reader's attention. They are also fed information by institutional press officers who may put a spin on a story in the hope that the media would pick it up. And researchers themselves are not immune from wanting their day of glory and being willing to 'accentuate the positive' rather than adopting the cautious, balanced approach that characterises a good scientist.  I have to say that, although I knew this could happen, until I started looking at candidates for the Orwellian, I had always thought that it was only a handful of maverick scientists with personality disorders who would behave in this way. I'd just assumed that for most scientists their reputation with colleagues would be far more important than a brief burst of media attention. I'd also reckoned that press offices would jealously guard the reputation of their institution for impeccably accurate science. But this proved to be naïve. Fame is seductive, and many will compromise their standards for their spot in the limelight.

Since throwing down my challenge, I've had two nominations for the Orwellian. The first was from Cambridge neuroscientist Jon Simons, who true to the spirit of my original post, scored up his submission according to the system I'd proposed, which involved giving points for each statement that was inaccurate when checked against the original source article. The nominated newspaper report was by a Washington Post staff writer and appeared on 9th September under the headline "Scientists can scan brains for maturity, potentially gauging child development". Simons's computations gave it a total of 16 points, putting it on level pegging with the ADHD article. One needs only to look at Figure 1 from the original article, reproduced below, to see one reason why he was exasperated. The prediction from the "functional connectivity Maturation Index" (fcMI), while statistically significant, is far from precise, because of the variation around the average level for each age. Consequently, there is a fair amount of overlap in the range of scores seen for adults and children. If a normal adult can get a brain maturity index of a 8 year old, and a normal 8-year-old can get a maturity index equivalent to an adult, it is questionable just how useful the index would be at detecting abnormality. Also, the regression equation was computed on the combined child and adult data, and nowhere in the paper are data presented on the accuracy of prediction of chronological age from brain measures just within the group of children. As an aside, our research group has tried similar things with the more low-tech methodology of event-related potentials, and we find it is relatively easy to discriminate child brains from adult brains, but not easy to distinguish between a 6-year-old and a 10-year-old (Bishop et al, 2007).
Figure 1 from Dosenbach et al, 2010

I would not be happy, however, giving the Orwellian award to the brain maturity piece, because the journalist does not seem to be at fault. Much of the newspaper article appears to be based on a press release from Oregon Health and Science University.  And furthermore, the Science article reporting the findings is entitled Prediction of individual brain maturity using fMRI and concludes that the method "could one day provide useful information to aid in the screening, diagnosis, and prognosis of individuals with disordered brain function."  I could not find any source for the claim in the newspaper article that the method might tell us "whether teenageers are grown-up enough to be treated as adults", though I did find a Neurolaw blogpost that could be the source of this idea: it stated "in the courtroom, this work could prove useful in determining whether children or adults should be culpable for their actions based on the maturation of the brains" (!).  Overall, I felt that the majority of mistakes in this piece were either misinterpretations that were encouraged by the journal article or press release, or involved extrapolations beyond the data that originated with the authors. And, unlike some other articles focused on developmental disorders, I did not feel this one had much potential for harm.


Another contender for the prize came from Australia, where conservation ecologist Corey Bradshaw was exercised at the way his work on frogs had been misrepresented. In a paper in Conservation Biology entitled Eating frogs to extinction, he and his colleagues called for certification of the trade in frogs' legs, which has the potential to have significant impact on frog populations. I won't attempt to summarise his arguments, which are eloquently stated in his own blogpost. All science journalists should read this – not just because it is witty, but also because it gives some insight into just how angry scientists get when they try hard to explain the science to a journalist who then gets it wrong . But this article, too, didn't seem to merit the prize. Quite apart from the fact it's really too early for the prize, dating back to 2009, there's also a sense of breaking a butterfly on a wheel. The article in question was not in a major newspaper; it was rather on a blog called SlashFood. Admittedly, it's an award-winning blog, described by Time magazine as "a site for people who are serious about what they put in their bodies". But this was clearly a journalist coming at the frogs' legs story from the culinary and not the scientific angle. And as Bradshaw himself says of the journalist, " Now, in all fairness, I think she was trying to do well…" Also, it's hard to see that the article did much harm, beyond raising the blood pressure of a distinguished conservation ecologist to dangerous levels.  Replete with inaccuracies as it was, the overall impact would have been to make environmentally conscious people think twice about eating frogs' legs. Incidentally, journalists at the Guardian will be pleased to hear that Jon Henley's coverage of the same story got a ringing endorsement from Bradshaw.

These were the only nominations I received for the prize, and so I have to announce that it will not be awarded this year, although the two nominators will receive an alcoholic token of my appreciation for their efforts. Although I could give the award to the original Observer article, this does not seem justified given that the newspaper withdrew the piece when it became aware of the scientific criticism.

I will accept  nominations for 2011, and am interested in receiving them even if they don't meet all criteria: I am fascinated by interactions between the media and scientists, in finding out more about what scientists do to irritate journalists, and vice versa. If we are going to improve science reporting, we need to understand one another. I hope that the lack of a serious contender for 2010 is telling us something about improving standards in science journalism - but maybe readers know better.

Wednesday, 12 January 2011

What works for women: some useful links

This is a work in progress! Feel free to suggest additions

Resources from Virginia Valian, including details of her book, "Why So Slow", a webcast and other resources

 
Athene Donald’s blog

Jenny Rohn's blog

Blog on "becoming a domestic and laboratory goddess"

Demonstrations of schemata (see Valian for context)
 

STRIDE Faculty recruitment workshop readings
STRIDE faculty recruitment, other resources



Saturday, 8 January 2011

Should we ration research grant applications?

Researchers can never have enough funds.  Talented people with bright ideas frequently fail to get funded, leading to low morale in academia.  In the current financial climate, it is easy to put all the blame for this on government.  But a recent consultation document by the Economic and Social Research Council (ESRC) shows that’s not the full story.  Between 2007 and 2009, the number of responsive mode grant applications rose by 39%.  A similar picture is seen for other UK research councils.  If more people apply for the same pot of money, it is inevitable that a smaller proportion of applications will be funded: In the case of ESRC, success rates have gone from 30% to 16%.  Reductions of research council budgets in real terms means things can only get worse. ESRC reckons that the status quo is not an option: once success rates fall this low, it ceases to be worthwhile for researchers to submit grant proposals. As well as the costs to applicants in time and effort, there are also financial costs to higher-education institutions in administering grant applications, and for ESRC administrative staff.  The peer review process also starts to break down: on the one hand, it becomes difficult to find enough reviewers to handle the mounting tide of proposals, and on the other, reviewers become reluctant to say anything negative at all about a proposal, as only those with a uniform set of glowing reviews stand any chance.

One of the UK Research Councils, the Engineering and Physical Sciences Research Council (EPSRC) has put new measures in place to attempt to reduce the flood of applications. The other research councils are discussing how to do this, and the ESRC is to be congratulated for taking soundings from the academic community about possible ways forward.

Their document, however, creates an odd mental state in academic readers. We’ve all been told for years that getting grant funding is a Good Thing.  Individuals who can bring in the funds will be rewarded with promotion, tenure, and glittering prizes.  Universities pride themselves on their grant income, which is major factor in higher education rankings. Now, however, we are told that applying for grants is a Bad Thing, which the academic community needs to work with ESRC to cut back, with statements such as:

•    (ESRC has) an ambitious target of halving the number of applications submitted through its standard grants scheme by 2014
•    greater self regulation has the potential to significantly reduce the volume of applications submitted by institutions
•    (self-regulation will) probably not go far enough achieve the 50% reduction that the Council is seeking to deliver

The five options outlined by ESRC (copied verbatim below) are:
•    Researcher Sanctions: This involves limiting the number of proposals from individual researchers who consistently fail to submit applications that reach an agreed quality threshold;
•    Institutional Sanctions:  This involves introducing sanctions for HEIs whose applications fail to meet a certain success rate and/or quality threshold;
•    Institutional Quotas for ‘managed mode’ schemes.  This involves the introduction  of institutional quotas for certain schemes (e.g. early career researcher schemes, Large Grants/Centres, Professorial Fellowships).;
•    Institutional quotas for all schemes: This involves responsive as well as managed mode schemes; 
•    Charging for applications. Levying an agreed fee for institutions submitting applications, with the option that this levy is redeemable if the application is successful.

The pros and cons of each of these is discussed, and readers are asked to comment.

My response to ESRC is that they are looking for solutions in the wrong place. To fix the problem, they need to change the basic structure of university funding so that institutions and individuals are no longer assessed on amount of research funding, but rather on an output/input function, i.e. how much bang do you get for your buck.

I have argued in a previous blog that it makes no sense to reward people simply for securing large amounts of funding. Currently, a person who secures a £500K grant which leads to two publications in lower-ranking journals will be given more credit than one who generates five high-ranking publications over the same period with a £50K grant. Clearly, some sorts of research are much more expensive than others; the problem is that the current system discourages people from undertaking inexpensive research.  In my own field of psychology, there are cases of people who have published an impressive body of work based largely on student projects: they do not, however, get much appreciation from their institutions. Meanwhile, it has become almost mandatory for psychology  grant applications to include an expensive brain scanning component, even if this adds nothing to the scientific value of the research.  The introduction of Full Economic Costing (FEC) has added to the problem, by introducing incentives for researchers to add collaborators to their proposals, as this will bump up the cost of the grant.  In short, the combination of the RAE and FEC does the opposite of encouraging cost-effectiveness in grant applications  - it makes people focus solely on cost, the higher the better.

The same incentives have not only encouraged an anti-thrift mentality in higher education institutions, they have also changed expectations about the numbers of grants that academics should hold.  Doing research takes time, and applicants are typically asked to quantify this in grant applications in terms of hours per week spent on the research. As far as I know, nobody ever adds up the estimated hours of research time for a person  holding grants by different bodies. I suspect that there are cases where, if one were to total up estimated time across all a researcher's grants, it would exceed the number of hours in a week. This is particularly true for grant-holders in lectureships, who presumably are expected to spend some time on teaching activities.   The system as it stands will encourage an academic to apply for 5 grants,  spending 1 hr per week on each of them, than to apply for a single grant, on which they propose to spend 5 hr per week. Yet I’d bet that the quality of research would be better in the latter case, because high quality research takes time and thought.  Over-commitment is encouraged by the current system, yet causes stress and waste. Many research-active academics are overwhelmed by backlogs of research data, because they feel compelled to submit more grant applications rather than writing up what they have done.

I have three suggested solutions to the current crisis. The first involves such a radical change to funding structures of Universities that it is unlikely to be implemented. The other two are both feasible:

1.    For Government: Ditch the current methods for allocating funding to Universities so that cost-effectiveness and thrifty use of research funds are rewarded, rather than punished.
2.    For research councils: When evaluating research quality, be more focused on track record of outputs relative to income, so that funding is steered toward those who have demonstrated good cost-effectiveness. Given differences in costs and time-scale of outputs across disciplines, a generic metric would be unworkable, but just by asking grants panels to look at this would be a step forward. Obviously this would not apply to new investigators.
3.    For Universities:  Scrutinise all the research grants held by individuals to ensure that amount of time specified for research activities is realistic, taking into account other job demands.  A person would be debarred from putting in a grant proposal if they were over a limit in terms of hr per week already allocated to research.

If all three could be implemented, we could achieve a situation where the pressure on research councils is relieved, academics would be able to do good research with less stress on continually applying for funds, and research quality would be enhanced.

P.S. 11th January 2011
My congratulating ESRC for undertaking consultation with academics may have been premature. It seems very few academics knew about this, at least in my discipline of psychology. One who made enquiries at his institution discovered that the consultation document had gone to members of the Association of Research Managers and Administrators (whose website is the only place with a link to the document). In Oxford University, the recipient of the document circulated it to academics for their views, but I suspect in many places this did not happen. So, if rumour is to be believed, those of us who actually write the grants don't get asked about a major change in funding policy - just those who administer grant applications. Also noteworthy that the document was released on 20th December, just before Universities close for a long break.

Thursday, 23 December 2010

A day working from home

copyright: www.CartoonStock.com
8.00 a.m. Resolve that today is the day I will seriously engage with referee comments on a paper. Best to go in to office at work, where there are no distractions right now. Get up and don tights, vests, jumper, warm trousers, boots, cardigan, gloves and quilted Canadian coat with hood. Look more like Michelin Man than usual.
8.30 a.m. Set off to walk to work. Wenceslas-like situation in front garden,and difficulty opening gate against foot-high accumulation of snow.
8.35 a.m. See neighbour slide on ice and tumble on back. Fortunately no bones broken. Persevere, optimistic that the main thoroughfares will be cleared of snow.
8.40 a.m. They aren’t. Snow appears to have melted briefly and then refrozen. See another person slide on ice and land on back.
8.45 a.m. Return home.
9.00 a.m. Turn on gas fire and clear desk in preparation for serious academic activity. Get sidetracked by gas bill and unfiled bank statements. Once all the surface debris cleared, decide desk has too many crumbs on it to be compatible with serious work. Engage in desk-cleaning.
9.15 a.m. Open email. Granting agency has sent me material for a proposal I’d agreed to review consisting of six pdfs. Departmental administrator has sent stern message that we all have to come into work or have our pay docked, unless we have explicit agreement to work from home.
9.20 a.m Email the few postdocs who haven’t already gone on holiday to tell them to ignore message from administrator.
9.30 a.m Have a quick look at Twitter. Everyone tweeting about snow or science funding.
9.35 a.m Bite the bullet and open the referee comments. Save as a new file called ‘response to referees’.
9.40 a.m Make a cup of coffee to steel myself
9.45 a.m Read the referee comments. Aargh, aargh, aargh.
9.50 a.m Remember my Australian collaborator has already sent some new analyses and suggestions for responding to referees. Download these.
9.55 a.m Re-read referee comments. Aargh, aargh, aargh, aargh.
10.00 a.m Print out referee comments. Printer not working. Demanding toner. It has insatiable appetite for toner. Take out toner and shake it about and put it back in. Printer sneers: You’ve tried that before and I’m not playing.  Go into cellar (brrr) where I have providently kept spare toner.
10.05 a.m Unable to get into toner box. Into kitchen for a knife. Side-tracked by sight of coffee jar. Make another cup of coffee.  Open toner box. Find piece of paper with instructions for toner replacement in 12 languages. Go through ritual of shaking toner, removing yellow bit, sliding blue bit up and down, replacing toner in printer. 
10.10 a.m Discover I can save children’s lives by sending old printer cartridge back to special address. Want to save children’s lives, so repackage old toner cartridge in box. Go on hunt for sellotape. Find sellotape. Seal up box. Discover label is inside box. Refind knife. Open box. Extract label. Reseal box. Stick label on box.
10.15 a.m Resend printer command. Nothing happens. Take toner cartridge out again, shake it about, and put back in.
10.20 a.m Belatedly realise I have sent document to be printed on default printer, which is at work. Cancel printer command and resend to home printer.
10.25 a.m Get printout of reviewer comments and re-read.  Aargh, aargh, aargh, aargh, aargh. Reviewer 1 argues there six major flaws with the paper and data need total reanalysis.
10.30 a.m  Distract myself from grief with a quick check of email. Two messages from Microban international warning me about bacteria in my kitchen at Christmas (how do they know about me and my kitchen?), one announcement that I have won 1.5 million Euros in a lottery I didn’t enter (could come in handy), and a request to review a manuscript.
10.35 a.m Twitter proves more interesting: everyone has either got a broken-down heating system, or is stuck in an airport somewhere. Feel inappropriately smug. Schadenfreude is a real phenomenon.
10.40 a.m Glance out of window and notice sadly that birds have failed to find piece of stale bread that I had hung up on tree, or nuts stuck in a bush. Deliberate on whether I should do more to encourage birds. Decide this is no good, and must return my attention to the comments.
10.45 a.m Download relevant-looking manuscript that I hope will provide some salvation. Read it and make notes.
11.15 a.m Well, that was quite interesting but no relevance at all for current paper.
11.20 a.m OK, ready to start thinking about reply to reviewer 1. He's one of those people who accuses you of saying something you haven’t and then reprimands you for it.  Grrr. Quick look at BBC News, to distract myself, and counteract irritaiton with more airport grief.
11.25 a.m Print out the original manuscript so I can see exactly what we did say. Do first pass at responses to reviewers.
11.50 a.m. Postman rings with parcel that won’t fit in letterbox. Need to negotiate snowy path to front gate. Find wellies. Struggle into wellies. Collect parcel from remarkably cheery postman. Return and spend 5 mins getting out of wellies.
12.00 p.m. Heavy snow has started falling! Husband has started bustling in kitchen to make seafood risotto for lunch. Yum!
12.05 p.m. Husband suggests that if I want risotto to be amazing rather than just fabulous, I should go and buy white wine. And that some red wine for mulling this evening would also be a good idea. Since he is (a) in tracksuit & slippers and (b) busy with his culinary art, and I am fully dressed, there is justice in this. Muse on advantages of living with licensed 9-to-9 store a stone’s throw away.
12.10 p.m. Don duvet-like coat and boots again and venture into snow, returning with 2 bottles wine and other sundry essentials, on assumption we may be snowed in for days. The street looks magical, like something out of Dickens.
12.15 p.m. Sad blackbird lands on window ledge and looks at me through window. Rummage in fridge for blackbird food and some water.  Only bird-friendly food I can find is olive bread and couscous. Very North Oxford.
12.25 p.m Check email. Boring, boring, boring, but takes 20 mins!
12.45 p.m. Risotto. Yum yum yum! Radio 4 full of tales of airport woe.
13.15 p.m. Quick twitter-scan and tweet to plug my latest blog.
13.25 p.m. Email check. Administrator now telling people to go home early as the Oxford buses are stopping early and they could get stranded. Have visions of a Heathrow-style psychology dept with people bedding down in the coffee area. Now! back to work.
13.30 p.m. Well, I’ve dealt with two comments from ref 2, both of which involve adding the reference number in two places where I’d left it out. Mild sense of achievement. I like ref 2.
13.40 p.m  One of our analyses involves computing intraclass correlation between an individual’s waveform and that of the group average. Referee wants us to leave out the individual when computing grand average.  I know that with sample size of 40+ this makes no difference, but I can’t find out where I checked this out, and so am now going to need to redo the analysis to make this point. But now I need to find the xls formula for the intraclass correlation, which has to be re-entered every time you want to use it in a workbook, and I can’t really remember how to do that either, though I’ve done it loads of times before. I even think I somewhere stored instructions of how to do this, but I can’t find them. So now I am having to run a Search. All to demonstrate to a reviewer something I know is the case and which is not going to make any difference whatsoever to the results.
14.25 p.m. OK, done that. Found formula, worked out how to reinstate it, tried it out on demonstration data and got satisfactory result. Whew.
Looked at email: Message to say my email account is being migrated from one account to another. Had been told this would happen but had forgotten. Scary. But good incentive to stop looking at email for a bit
14.45 p.m. Coffee break .Reading newspapers
15.00 p.m  Must read various articles sent by collaborator to contest arguments being made by reviewer. OK, brain in gear and articles printed out.
15.30 p.m. Husband decides more ingredients needed for culinary art, and is off to Sainsbury’s. Returns 1 min later saying car is entombed in snow and he needs bucket of hot water.
15.35 p.m. Two articles done, and two to go.  Husband returns saying he has removed snowy carapace from car, but now can’t get into it, as door are frozen shut. He sneers at idea we should check for advice on internet re this situation, and stomps off with more buckets of hot water. Internet advice is to use windscreen wiper fluid. Small problem. Our windscreen wiper fluid is in the car, which we can’t get into.
16.45 p.m Have read 4 articles, two of which were mostly incomprehensible and of dubious relevance. Still uncertain if I really need to do more analysis. Send email to collaborator in Australia. However, email system is in process of migrating and it seems uncertain as to whether it is working or not. Time for a small break and a twitter session.
17.00 p.m Need to read more so I can write explanatory section requested by reviewers. Three important articles to download and summarise.
18.00 p.m. Read two of them. V. good and helpful. Now on to inspect whether my email migrated OK.
18.30 p.m. Think it did, as I have messages telling me I have been bequeathed large sums of money, as well as two requests to review manuscripts. Time for some mulled wine.
Tomorrow is another day. Resolve, no more tweeting, email, shopping, bird tending, until paper is revised. But maybe just a little blog about my day.....

Wednesday, 22 December 2010

“Neuroprognosis” in dyslexia

Every week seems to bring a new finding about brains of people with various neurodevelopmental disorders. The problem is that the methods get ever more technical, and so the scientific papers get harder and harder to follow, even if, like me, you have some background in neuropsychology.  I don’t normally blog about specific papers, but various people have asked what I think of an article that was published this week by Hoeft et al in Proceedings of National Academy of Science, and so I thought I’d have a shot at (a) summarising what they found in understandable language, and (b) giving my personal evaluation of the study. The bottom line is that the paper seems methodologically sound, but I’m nevertheless sceptical because (a) I’m always sceptical, and (b) there are some aspects of the results that just seem a bit hard to make sense of.

What question did the researchers ask?

Fig 1: location of IFG
The team, headed by researchers from Stanford University of Medicine, took as their starting point two prior observations.
First, previous studies have found that children with dyslexia look different from normal readers when they do reading tasks in a brain scanner. As well as showing under-activation of regions normally involved in reading, dyslexics often show over-activation in the frontal lobes, specifically in the inferior frontal gyri (IFG) (Figure 1).
The researchers were interested in the idea that this IFG activation could be a sign that dyslexics were using different brain regions to compensate for their dyslexia. They reasoned that, if so, then the amount of IFG activity observed on one occasion might predict the amount of reading improvement at a later occasion.
So the specific question was: Does greater involvement of the IFG in reading predict future long-term gains in reading for children with dyslexia?

Who did they study?
The main focus was on 25 teenagers with dyslexia, whose average age was 14 years at the start of the study. The standard deviation was 1.96 years, indicating that most would have been 12 to 16 years old.  There were 12 boys and 13 girls. They were followed up 2.5 years later, at which point they were subdivided, according to how much progress they’d made on one of the reading measures, into a group of 12 with ‘no-gain’ and 13 with ‘reading-gain’.

The criteria for dyslexia were that, at time 1, (a) performance was in the bottom 25% for age on a composite measure based on two subtests of the Test of Word Reading Efficiency (TOWRE), and (b) performance on a nonverbal IQ subtest (WASI Matrices) was in normal limits (within 1 SD of average).  The TOWRE is a speeded reading test with two parts: reading of real words, and reading of made-up words – the latter subtest picks up difficulties with converting letters into sounds. The average scores are given in Supporting Materials, and confirm that these children had a mean nonverbal ability score of 103 and a mean TOWRE score of 80. This is well in line with how dyslexia is often defined, and confirms they were children of normal ability with significant reading difficulties.

In addition, a ‘control’ group of normal readers was recruited, but they don’t feature much in the paper. The aim of including them was to see whether the same brain measures that predicted reading improvement in dyslexics would also predict reading improvement in normal readers.  However, the control children were rather above average in reading to start with, and, perhaps not surprisingly, they did not show any improvement over time beyond that which you’d expect for their age.

How was reading measured?

Children were given a fairly large battery of tests of reading, spelling and related skills, in addition to the TOWRE, which had been used to diagnose dyslexia. These included measures of reading accuracy (how many words are read correctly from a word list), reading speed, and reading comprehension (how far the child understands what is read). The key measure used to evaluate reading improvement over time was the the Word Identification Subtest from the Woodcock Reading Mastery Test (WID).

It is important to realise that all test scores are shown as age-scaled scores. This allows us to ignore the child’s age, as the score just indicates how good or bad the child is relative to others of the same age. For most measures, the scaling is set so that 100 indicates an average score, with standard deviation (SD) of 15 . You can tell how abnormal a score is by seeing how many SDs it is from the mean; around 16% of children get a score of 85 or less (1 SD below average), but only 3% score 70 or less (2 SD below average).  At Time 1, the average scores of the dyslexics were mostly in the high 70s to mid 80s, confirming that these children are doing poorly for their age.

When using age-scaled scores, the expectation is that, if a child doesn’t get better or worse relative to other children over time, then the score will stay the same. So a static score does not mean the child has learned nothing: rather they have just not changed their position relative to other children in terms reading ability.

Another small point: scaled scores can be transformed so that, for instance, instead of being based on a population average of 100 and SD of 15, the average is specified at 10 and SD as 3. The measures used in this study varied in the scaling they used, but I transformed them so they are all on the same scale: average 100, SD 15. This makes it easier to compare effect sizes across different measures (see below).

How was brain activity measured?
Functional magnetic resonance imaging (fMRI) was used to measure brain activity while the child did a reading task in a brain scanner at Time 1. The Wikipedia account of fMRI gives a pretty good introduction for the non-specialist, though my readers may be able to recommend better sources.  The reading task involved judging whether two written words rhymed, e.g. bait-gate (YES) or price-miss (NO). Brain activity was also measured during rest periods, when no task was presented, and this was subtracted from the activity during the rhyme task. This is a standard procedure in fMRI that allows one to see what activation is specifically associated with task performance. Activity is measured across the whole brain, which is subdivided into cubes measuring 2 x 2 x 2 mm (voxels). For each voxel, a measure indicates the amount of activation in that region. There are thousands of voxels, and so a huge amount of data is generated for each person.

The researchers also did another kind of brain imaging measurement, diffusion tensor imaging (DTI). This measures connectivity between different brain regions, and reflects aspects of underlying brain structure. The DTI results are of some interest, but not a critical part of the paper and I won’t say more about them here.

No brain imaging was done at Time 2 (or if it was it was not reported). This was because the goal of the study was to see whether imaging at one point in time could predict outcome later on.


How were the data analysed?

The dyslexics were subdivided into two groups, using a median split based on improvement on the WID test. In other words, those showing the least improvement formed one group (with 12 children) and those with most improvement formed the other (13 children).

The aim, then, was to see how far (a) behavioural measures, such as initial reading test scores, or (b) fMRI results were able to predict which group children came from.

Readers  may have come across a method that is often used to do this kind of classification, known as discriminant function analysis. The basic logic is that you take a bunch of measures, and allocate a weighting to each measure according to how well it distinguishes the two groups. So if the measure had the same average score for both groups, the weighting would be zero, but if it was excellent at distinguishing them, the weighting might be 1.0. You then add together all the measures, multiplied by their weightings, with the aim of getting a total score that will do the best possible job at distinguishing groups.  You can then use this total score to predict, for each person, which group they belong to. This way you can tell how good the prediction is, e.g. what percentage of people are accurately classified.

The extension of this kind of logic to brain imaging is known as multivariate pattern analysis (MVPA). It is nicely explained, with diagrams, on Neuroskeptic’s blog. .  For a more formal tutorial, see www.princeton.edu/~fpereira/Papers/tutorial.pdf.

It has long been recognised that there’s a potential problem with this approach, as it can give you spuriously good predictions, because the method will capitalise on chance fluctuations in the data that are not really meaningful. This is known as ‘over-fitting’. One way of getting around this is to use the leave-one-out method.  You repeatedly run the analysis, leaving out data from one participant, and then see if you could predict that person’s group status from the function derived from all the other participants. This is what was done in this study, and it is an accepted method for protecting against spurious findings.

Another way of checking that the results aren’t invalid is to directly estimate how likely it would be to get this result if you just had random data. To do this, you assign all your participants a new group code that is entirely arbitrary, using random numbers. So every person in the study has a 50% chance of being in group A or group B. You then re-run the analysis and see whether you can predict whether a person is an A or B on the basis of the same brain data. If you can, this would indicate you are in trouble, as the groups you have put in to the analysis are arbitrary. Typically, one re-runs this kind of arbitrary analysis many times, in what is called a permutation analysis; if you do it enough times, occasionally you will get a good classification result by chance, but that does not matter, so long as the likelihood of it occurring is very rare, say less than 1 in 1000 runs.  For readers with statistical training, we can say that the permutation analysis is a nice way of getting a direct estimate of the p-value associated with the analysis done with the original groups.

So what did they find?



View My Stats
Fig 2: discriminant function (y-axis) vs reading gain


The classification accuracy of the method using the whole-brain fMRI data was reported as an impressive 92%, which was well above chance.  Also, the score on the function used to separate groups was correlated .73 with the amount of reading improvement. The brain regions that contributed most to the classification included the right IFG, and left prefrontal cortex, where greater improvement was associated with higher activation. Also the left parietotemporal region showed the opposite pattern, with greater improvement in those who showed less activation.

So could the researchers have saved themselves a lot of time and got the same result if they’d just used the time 1 behavioural data as predictors? They argue not. The prediction from the behavioural measures was highly significant, but not as strong, with accuracy reported (figure S1 of Supporting Materials) as less than 60%.  Also, once the brain measures had been entered into the equation, adding behavioural measures did not improve the prediction.

And what conclusions did they draw?

  • Variation in brain function predicts reading improvement in children with dyslexia. In particular, activation of the right IFG during a reading task predicts improvement. However, taking a single brain region alone does not give as good a prediction as combining information across the whole brain.
  • Brain measures are better than behavioural measures at predicting future gains in reading.
  • This suggests that children with dyslexia can use the right IFG to compensate for their reading difficulties.
  • Dyslexics learn to read by using different neural mechanisms than those used by normal readers.

Did they note any limitations of the study?
  • It’s possible that different behavioural measures might have done a better job in predicting outcomes.
  • It’s also possible that a different kind of brain activation task could have given different results.
  • Some children had received remediation during the course of the study: but this didn’t affect their outcomes. (Bad news for those doing the remediation!).
  • Children varied in IQ, age, etc, but this didn’t differentiate those who improved and those who didn’t.

Questions I have about the study

Just how good is the prediction from the brain classifier?

Figure 2 (above) shows on the y-axis the discriminant function (the hyperplane), which is the weighted sum of voxels that does the best job of distinguishing groups. The x-axis shows the reading gain. As you can see clearly, there are two individuals who fall in the lower right quadrant, i.e. they have low hyperplane scores, and so would be predicted to be no-gain cases, but actually they make positive gains. The figure of 92% appears to come by treating these as cases where prediction failed, i.e. accurate prediction for the remainder gives 23/25 = 92% correct.
Fig 3: Vertical line dividing groups moved

However, this is not quite what the authors said they did.  They divided the sample into two equal-sized groups (or as equal as you can get with an odd number) in order to do the analysis, which means that the ‘no improvement’ group contains four additional cases, and that the dividing line for quadrants needs to be moved to the right, as shown in Figure 3.  Once again, accurate prediction occurs for those who fall in the top right quadrant, or the bottom left. Prediction is now rather less good, with 4 cases misclassified (three in the top left quadrant, one in the bottom right, i.e. 84% correct).However, it must be accepted that this is still good prediction.


 
Why do the reading gain group improve on only some measures?
One odd feature of the data is the rather selective nature of the reading improvement seen in the reading-gain group. Table 1 shows the data, after standardising all measures to a mean of 100, SD 15. The analysis used the WRMT-WID test, which is shown in pink. On this test, and on the other WMRT tests, the reading-gain group do make impressively bigger gains than the no-gain group. But the two groups look very similar on the TOWRE measures, which were used to diagnose dyslexia, and also on the Gray Oral Reading Test (GORT).  Of course, it’s possible that there is something critical about the content of the different tests – the GORT involves passage-reading, and the TOWRE involves speeded reading of lists of items.  But I’d have been a bit more convinced of the generalisability of the findings, if the reading improvement in the reading-gain group had been evident across a wider range of measures.(Note that we also should not assume all gain is meaningful: see my earlier blog for explanation of why).


Why do the control group show similar levels of right IFG activation to dyslexics?
The authors conclude that the involvement of certain brain regions, notably the right IFG, is indicative of an alternative reading strategy to that adopted by typical readers. Yet the control group appear to show as wide a range of activation of this area as the dyslexics, as shown in Figure 4. The authors don’t present statistics on this, but eyeballing the data doesn’t suggest much group difference.
Figure 4: activation in dyslexics (red) and controls (blue)


If involvement of the right IFG improves reading, why don’t dyslexic groups differ at time 1?

This is more of a logical issue than anything else, but it goes like this. Children who improve in reading by time 2 showed a different pattern of brain activation at time 1. The authors argue that right IFG activation predicts better reading. But at time 1, the groups did not differ on the reading measures – or indeed on their performance of the reading task in the scanner. This would be compatible with some kind of ‘sleeper’ effect, whereby the benefits of using the right IFG take time to trickle through. But what makes me uneasy is that this implies the researchers had been lucky enough to just catch the children at the point where they’d started to use the right IFG, but before this had had any beneficial effect.  So I find myself asking what would have happened if they’d started with younger children? 

Overall evaluation
This is an interesting attempt to use neuroimaging to throw light on mechanisms behind compensatory changes in brains of people with dyslexia.  The methodology appears very sound and clearly described (albeit highly technical in places). The idea that the IFG is involved in compensation fits with some other studies in the field.

There are, however, a few features of the data that I find a bit difficult to make sense of, and that makes me wonder about generalisability of this result.

Having said that, this kind of study is challenging. It is not easy to do scanning with children, and just collecting and assessing a well-documented sample can take many months. One then has to wait to follow them up more than two years later. The analyses are highly demanding.  I think we should see this as an important step in the direction of understanding brain mechanisms in dyslexia, but it’s far from being conclusive.

Hoeft F, McCandliss BD, Black JM, Gantman A, Zakerani N, Hulme C, Lyytinen H, Whitfield-Gabrieli S, Glover GH, Reiss AL, & Gabrieli JD (2011). Neural systems predicting long-term outcome in dyslexia. Proceedings of the National Academy of Sciences of the United States of America, 108 (1), 361-6 PMID: 21173250

Saturday, 18 December 2010

What's in a name?


In a recent blog post in the Guardian, Maxine Frances Roper discussed how her dyspraxia made it hard for her to get a job. She had major problems with maths and poor physical co-ordination and was concerned that employers were reluctant to make accommodations for these. The comments that followed the blog fell mostly in one of two categories: a) people who described their own (or their child’s) similar experiences; b) people who thought of dyspraxia as an invented disorder with no validity.

Although the article was about dyspraxia, it could equally well have been about developmental dyslexia, dyscalculia or dysphasia. These neurological labels are applied to children whose development is uneven, with selective deficits in the domains of literacy, mathematical skills, and oral language development respectively.  They are often described as neurodevelopmental disorders, a category which can be extended to encompass attention deficit hyperactivity disorder (ADHD), and autistic disorder. Unlike conditions such as Down syndrome or Fragile X syndrome, these are all behaviourally defined conditions that can seldom be pinned down to a single cause.  They are subject to frequent challenges as to their validity. ADHD, for instance, is sometimes described as a medical label for naughty children , and dyslexia as a middle-class excuse for a child’s stupidity.   Autism is a particularly interesting case, where the challenges are most commonly made by individuals with autism themselves, who argue they are different rather than disordered.

So, what does the science say? Are these valid disorders?  I shall argue that these medical-sounding labels are in many respects misleading, but they nevertheless have served a purpose because they get  developmental difficulties taken seriously. I’ll then discuss alternatives to medical labels and end with suggestions for a way forward.


Disadvantages of medical labels

1. Medical labels don't correspond to syndromes

Parents often have a sense of relief at being told their child is dyslexic, as they feel it provides an explanation for the reading difficulties. Most people assume that dyslexia is a clearcut syndrome with a known medical cause, and that affected individuals can be clearly differentiated from other poor readers whose problems are due to poor teaching or low intelligence.

In fact, that is not the case.  Dyslexia, and the other conditions listed above, are all diagnosed on the basis of behavioural rather than neurological criteria. A typical definition of developmental dyslexia specifies that there is a mismatch between reading ability and other aspects of cognitive development, which can’t be explained by any physical cause (e.g. bad eyesight) or poor teaching.  It follows that if you have a diagnosis of dyslexia, this is not an explanation for poor reading; rather it is a way of stating in summary form that your reading difficulties have no obvious explanation. 

But medicine progresses by first recognising clusters of symptoms and then identifying underlying causes for individuals with common patterns of deficits. So even if we don’t yet understand what the causes are, could there could be value in singling out individuals who meet criteria for dyslexia, and distinguishing them from other poor readers? To date, this approach has not been very effective. Forty years ago, an epidemiological study was conducted on the Isle of Wight: children were screened on an extensive battery of psychological and neurological measures.  The researchers were particularly interested in whether poor readers who had a large discrepancy between IQ and reading ability had a distinctive clinical profile.  Overall, there was no support for dyslexia as a distinct syndrome, and in 1976, Bill Yule concluded: “The era of applying the label 'dyslexic' is rapidly drawing to a close. The label has served its function in drawing attention to children who have great difficulty in mastering the arts of reading, writing and spelling, but its continued use invokes emotions which often prevent rational discussion and scientific investigation".(p 166).  Subsequent research has focused on specifying what it is about reading that is so difficult for children who struggle with literacy, and it’s been shown that for most of them, a stumbling block is in the process of breaking words into sounds, so-called phonological awareness.   However, poor phonological awareness is seen in poor readers of low IQ as well as in those with a mismatch between IQ and reading skill.

2. Medical labels don’t identify conditions with distinct causes

What about if we look at underlying causes? It's an exciting period for research as new methods make it possible to study the neurological and genetic bases of these conditions.  Many researchers in this field anticipated that once we could look at brain structure using magnetic resonance imaging, we would be able to identify ‘neural signatures’ for the different neurodevelopmental disorders. Despite frequent over-hyped reports of findings of ‘a brain scan to diagnose autism’ and so on, the reality is complicated.

I'm not attacking researchers who look for brain correlates of these conditions: we know far more now than we did 20 years ago about how typical and atypical brains develop, and basic neuroscience may help us understand the underlying processes involved, which in turn could lead to better diagnosis and intervention. But before concluding that a brain scan can be a feasible diagnostic test, we need studies that go beyond showing that an impaired group differs from an unimpaired group.  In a recent review of pediatric neuroimaging and neurodevelopmental disorders,  Giedd and Rapoport concluded: “The high variability and substantial overlap of most measures for most groups being compared has profound implications for the diagnostic utility of psychiatric neuroimaging” (p. 731) (my italics)

Similar arguments apply in the domain of genetics. If you are interested in the details, I have a blog explaining in more detail, but in brief, there are very few instances where a single genetic mutation can explain dyslexia, ADHD, autism and the rest. Genes play a role, and often an important one, in determining who is at risk for disorder, but it seems increasingly likely that the risk is determined by many genes acting together, each of which has a small effect in nudging the risk up or down. Furthermore, the effect of a given gene will depend on environmental factors, and the same gene may be implicated in more than one disorder. What this means is that research showing genetic influences on neurodevelopmental disorders does not translate into nice simple diagnostic genetic tests. 

3. No clear boundaries between individuals with different diagnostic labels

To most people, medical labels imply distinct disorders with clear boundaries, but in practice, many individuals have multiple difficulties.  Maxine Frances Roper’s  blogpost on dyspraxia illustrates this well: dyspraxia affects motor co-ordination, yet she described major problems with maths, which would indicate dyscalculia. Some of her commentators described cases where a diagnosis of dyspraxia was accompanied by a diagnosis of Asperger syndrome, a subtype of  autistic disorder. In a textbook chapter on neurodevelopmental disorders, Michael Rutter and I argued that pure disorders, where just one domain of functioning is affected, are the exception rather than the rule. This is problematic for a diagnostic system that has distinct categories, because people will end up with multiple diagnoses. Even worse, the diagnosis may depend on which professional they see. I know of cases where the same child has been diagnosed as having dyslexia, dyspraxia, ADHD, and “autistic spectrum disorder” (a milder form of autism), depending on whether their child is seen by a psychologist, an occupational therapist, a paediatrician or a child psychiatrist.

4. No clearcut distinction between normality and abnormality

There has been much debate as to whether the causes of severe difficulties are different from causes of normal variation. The jury is still out, but we can say that if there are qualitative differences between children with these neurodevelopmental disorders and typically developing children, we have yet to find them.  Twenty years ago, many of us expected that we might find single genes that caused SLI or autism, for instance, but although this sometimes occurs, it is quite exceptional.  As noted above, we are usually instead dealing with complex causation from a mixture of multiple genetic and environmental causes.  Robert Plomin and colleagues have argued, on the basis of such evidence, that ‘the abnormal is normal’ and that there are no disorders.

Consequences of abandoning medical labels 

Many people worry that if we say that a label like dyslexia is invalid, then we are denying that their child has real difficulties. This was brought home to me vividly when I was an editor of Journal of Child Psychology and Psychiatry. Keith Stanovich wrote a short piece for the journal putting forward arguments to the effect that there were no qualitative differences between poor readers of average or below average IQ, and therefore the construct of ‘dyslexia’ was invalid. This attracted a barrage of criticism from people who wrote in to complain that dyslexia was real, they worked with dyslexic children, and it was disgraceful for anyone to suggest that these children’s difficulties were fictional.  Of course, that was not what Stanovich had said. Indeed, he was very explicit: “Whether or not there is such a thing as 'dyslexia', there most certainly are children who read markedly below their peers on appropriately comprehensive and standardized tests. In this most prosaic sense, poor readers obviously exist.” (p. 580). He was questioning whether we should distinguish dyslexic children from other poor readers, but not denying that there are children for whom reading is a major struggle.  Exactly the same cycle of events followed a Channel 4 TV documentary, the Dyslexia Myth, which raised similar questions about the validity of singling out one subset of poor readers, the dyslexics, and giving them extra help and attention, when other poor readers, with very similar problems but lower IQs, were ignored. A huge amount of debate was generated, some of which featured in the Psychologist. Here again, those who had tried to make this case were attacked vehemently by people who thought they were denying the reality of children’s reading difficulties. 

Among those taking part in such debates are affected adults, many of whom will say ”People said I was stupid, but in reality I had undiagnosed dyslexia”. This is illuminating, as it stresses how the label has a big effect on people’s self-esteem. It seems that a label such as dyslexia is not viewed by most people as just a redescription of a person’s problems. It is seen as making them more real, emphasises that affected people are not unintelligent, and leads the condition to be taken more seriously than if we just say they have reading difficulties.


Should we abandon medical labels?

So what would the consequences be if we rejected medical labels? Here, it is fascinating to chart what has happened for different conditions, because different solutions have been adopted and we can compare and contrast the impact this has had. Let’s start with dyslexia. On the basis of the Isle of Wight study, Bill Yule and colleagues argued that we should abandon the term ‘developmental dyslexia’ and use instead the less loaded and more descriptive term ‘specific reading retardation’. Because of the negative connotations of ‘retardation’ their proposal did not take off, but the term ‘specific reading disability’ was adopted in some quarters. But, actually, neither term has really caught on.  When I did a bibliometric survey of studies on neurodevelopmental disorders, I tried to include all possible diagnostic labels as search terms. I've just looked  the frequency with which different terms were used to describe studies on developmental reading difficulties. Dyslexia won by a long margin, with over 97% of articles using this term.

Quite the opposite happened, though, with ‘developmental dysphasia’, which was used in the 1960s to refer to difficulties in producing and understanding spoken language in a child of otherwise normal ability.  This term was already going out of fashion in the UK and the USA in the 1970s, when I was doing my doctoral studies, and in my thesis I used ‘specific developmental language disorder’. Subsequently, ‘specific language impairment’ (SLI) became popular in the US research literature, but there is current concern that it implies that language is the only area of difficulty, when children often have additional problems.  Among practitioners, there is even less agreement, largely because of an explicit rejection of a ‘medical model’ by the profession of speech and language therapy (speech-language pathology in the US and Australia). So instead of diagnostic labels practitioners use a variety of descriptive terminology, including ‘language difficulties’, ‘communication problems’, and, most recently in the UK ‘speech, language and communication needs’ (SLCN). [If you've never heard of any of these and want to see how they affect children's lives, see http://www.afasicengland.org.uk].

There do seem to be important negative consequences, however. As Gina Conti-Ramsden has argued , specific language impairment (or whatever else you want to call it) is a Cinderella subject.  The amount of research funding directed to it is well below what you’d expect, given its frequency and severity, and it would seem that most members of the public have no idea what it is. Furthermore, if you say a child has ‘developmental dysphasia’, that sounds more serious and real than if you say they have ‘specific language impairment’. And to say they have language ‘difficulties’ or ‘needs’ implies to many people that those difficulties are fairly trivial.  Interestingly, there also seems to be an implicit assumption that, if you don’t have a medical label, then biological factors are unimportant, and you are dealing with problems with purely social origins, such as poor parenting or teaching.

An article by Alan Kamhi had a novel take on this issue. He argued that a good label had to have the properties of a meme. The concept of a meme was introduced by Richard Dawkins in the Selfish Gene,  and subsequently developed by Susan Blackmore in her book The Meme Machine. A meme is an element of culture that is transmitted from person to person, and a successful meme has to be easy to understand, remember and communicate to others. Importantly, it does not necessarily have to be accurate or useful.  Kamhi asked “Why is it more desirable to have dyslexia than to have a reading disability? Why does no one other than speech-language pathologists and related professionals seem to know what a language disorder is? Why is Asperger’s syndrome, a relatively new disorder, already familiar to many people?” (p. 105).  Kamhi’s answer is that terms with ‘language’ in them are problematic because everyone thinks they know what language is, but their interpretations differ from those of the professionals. I think there is some truth in this, but there is more to it than that. In general, I’d argue, the medical-sounding terms are more successful memes than the descriptive terms because they convey a spurious sense of explanation, with foreign and medical-sounding labels lending some gravity to the situation.

What to do?
We are stuck between the proverbial rock and hard place.  It seems that if we stick with medical-sounding labels for neurodevelopmental disorders, they are treated seriously and gain public recognition and research funding. Furthermore, they seem to be generally preferred by those who are affected by these conditions. However, we know these labels are misleading in implying that we are dealing with clearcut syndromes with a single known cause.

So here’s a proposal that attempts to steer a course through this morass. We should use the term ‘neurodevelopmental disability’ as a generic term, and then add a descriptor to indicate the areas of major difficulty. Let me explain why each part of the term is useful. “Neurodevelopmental” indicates that the child’s difficulties have a constitutional basis.  This is not the same as saying they can’t be changed, but it does move us away from the idea that these are some kind of social constructs with no biological basis. The evidence for a biological contributory causes is considerable for those conditions where there have been significant neurological and genetic investigations: dyslexia, SLI, autism and ADHD.

I suggest ‘disability’ rather than ‘disorder’ in the hope this may be more acceptable to those who dislike dividing humanity into the disordered and normal. Disability has a specific meaning in the World Health Organization classification, which focuses on the functional consequences of an impairment for everyday life. People who are the focus of our interest are having difficulties functioning at home, work or school, and so ‘disability’ seems a reasonable term to use.

It follows from what I’ve said above, that the boundary between disability and no disability is bound to be fuzzy: most problems fall on a scale of severity, and where you put the cutoff is arbitrary. But in this regard, neurodevelopmental disability is no different from many medical conditions. For instance, if we take a condition such as high blood pressure: there are some people whose blood pressure is so high that it is causing them major symptoms, and everyone would agree they have a disease. But other people may have elevated blood pressure and doctors will be concerned that this is putting health at risk, but where you actually draw the line and decide that treatment is needed is a difficult judgement, and may depend on presence of other risk factors. It’s common to define conditions such as dyslexia or SLI in terms of statistical cutoffs: the child is identified as having the condition if a score on a reading or language test is in the bottom 16% for their age. This is essentially arbitrary, but it is at least an objective and measurable criterion. However, test scores are just one component of diagnosis: a key factor is whether or not the individual is having difficulty in coping at home, work or school.

‘Neurodevelopmental disability’ alone could be used to indicate that the person has real difficulties that merit attention and support, but it lumps together a wide range of difficulties. That is no bad thing, however, given that many individuals have problems in several domains. The term would actively discourage the compartmentalised view of these different conditions, which leads to an unsatisfactory situation where, for instance, researchers in the US have difficulty doing research on the relationship between reading and language disabilities because these are seen as falling under the remit of different funding streams (NICHD and NIDCD respectively), or where a researcher who is studying language difficulties in autism will have much greater chance of obtaining funding (from NIMH) than one who is studying language difficulties in non-autistic children (which are far more common).

Having defined our generic category, we need to add descriptors that specify weaknesses and strengths. Identification of areas of weakness is crucial both for ensuring access to appropriate services, and to make it possible to do research on individuals with common characteristics. Table 1 shows how traditional medical categories would map on to this system, with a downward arrow denoting a problem area, and = denoting no impairment. But this is just to illustrate how the system corresponds to what we already have: my radical proposal is that we could do away with the labels in the top row.

Table 1: Traditional categories (top row) vs new system
A major advantage of this approach is that it would not force us to slot a person into one diagnostic category; rather it will encourage us to consider the whole gamut of developmental difficulties and document which apply in a given case. We know that many people with reading difficulties also have impairments in maths, oral language and/or attention: rather than giving the person a dyslexia label, which focuses on the reading difficulties, the full range of problem areas could be listed.  Intelligence does not feature in the diagnostic definition of autism, yet it makes a big difference to a person’s functioning if intelligence is in the normal range, or above average. Further some people with autism have major problems with literacy, motor skills or attention, others do not. This framework would allow us to specify areas of weakness explicitly, rather than implying that everyone with a common diagnostic label is the same. Further, it would make it easier to document change in functioning over time, as different areas of difficulty emerge or resolve with age.

In addition, a key feature of my proposed approach would be that assessment should also aim to discover any areas that parents or children themselves identify as areas of strength (up arrows), as fostering these can be as important as attempting to remediate areas of difficulty. If we take Maxine Frances Roper as an example, she evidently has good language and intelligence, so her profile would indicate this, together with weaknesses in maths and motor skills.

In the past, the only area of strength that anyone seemed interested in was IQ test performance.  Although this can be an important predictor of outcome, it is not all that matters, and to my mind should be treated just like the other domains of functioning: i.e., we note whether it is a weakness or strength, but do not rely on it to determine whether a child with a difficulty gains access to services.

When we consider people’s strengths, these may not be in cognitive or academic skills. Consider, for example, Temple Grandin. She is a woman with autism who has become a highly respected consultant in animal husbandry because of her unusual ability to put herself in the mind of the animals she works with. Obviously, not every person will have an amazing talent, but most will have some activities that they enjoy and can succeed in. We should try and find out what these are, and ensure they are fostered.

Will it happen?

Although I see this approach as logical and able to overcome many of the problems associated with our current diagnostic systems, I’d be frankly amazed if it were adopted.

For a start, it is complex and has resource implications. Few practitioners or researchers would have the time to do a comprehensive assessment of all the areas of functioning shown in Table 1. Nevertheless, many people would complain that this list is not long enough! What about memory, speech, spelling, executive function, or visuospatial skills, which are currently not represented but are studied by those interested in specific learning disabilities? The potential list of strengths is even more open-ended, and could encompass areas such as sports, music, craft and cookery activities, drama, ability to work with animals, mechanical aptitude and so on.  I’d suggest, though, that the approach would be tractable if we think about this as a two-stage procedure. Initial screening would rely on parent and/or teacher and/or self report to identify areas of concern. Suitable well-validated screening instruments are already available in the domains of language, attention, and social impairment, and this approach could be extended. Areas identified as specific weaknesses could then be the focus of more detailed assessment by a relevant professional.

The main reason I doubt my system would work is that too many people are attached to the existing labels. I’m sure many will feel that terms such as autism, ADHD, and dyslexia have served us well and there’s no need to abandon them.  Professional groups may indeed be threatened by the idea of removing barriers between different developmental disorders. And could we lose more than we gain by ditching terminology that has served us well, as least for some disorders?


Please add your comments

I certainly don’t have all the answers, but I am hoping that by raising this issue, I’ll stimulate some debate. Various academics in the US and UK have been talking about the particularly dire situation of terminology surrounding speech and language disorders, but the issues are broader than this, and we need to hear the voices of those affected by different kinds of neurodevelopmental disabilities, as well as practitioners and researchers.

With thanks to Courtenay Frazier Norbury and Gina Conti-Ramsden for comments on a draft of this post.


PS. 27th December 2010
A couple of relevant links:

More on failure of speech-language pathologists to agree on terminology for developmental language disorders.

Kamhi, A. G. (2007). Thoughts and reflections on developmental language disorders. In A. G. Kamhi, J. J. Masterson & K. Apel (Eds.), Clinical Decision Making in Developmental Language Disorders: Brookes.

A recent Ofsted report, concluding that many children with 'special educational needs' are just poorly taught. 

PPS. 19th June 2011
Problems with the term 'speech, language and communication needs':
Lindsay, G. (2011). The collection and analysis of data on children with speech, language and communication needs: The challenge to education and health services. Child Language Teaching & Therapy, 27(2), 135-150.


This article (Figshare version) can be cited as:
Bishop, Dorothy V M (2014): What's in a name?. figshare.
http://dx.doi.org/10.6084/m9.figshare.1022866


Tuesday, 14 December 2010

When ethics regulations have unethical consequences

I've been blogging away from home lately. On 1st December, Guardian Science published a guest blog from me relating to a recent PLOS One article, in which I examined the amount of research, and research funding, for different neurodevelopmental disorders. There are some worrying disparities between disorders, which need explanation.  I've already had lots of interesting emails and comments on the post, but I'm planning to revisit this topic shortly, and would welcome more input, so please feel free to add comments here if you wish.

My latest blog is a guest post for Science 3.0, on ethical (and unethical) issues in data-sharing.
How (some) researchers view ethics committees (IRBs)

Disclaimer: the author is vice-chair of the Medical Sciences Interdiscliplinary Research Ethics Committee at the University of Oxford, and notes that most committee members are on the side of the angels.