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Brain SciencesBrain Sciences
  • Review
  • Open Access

28 August 2021

19 Pages

Developmental Dyslexia, Reading Acquisition, and Statistical Learning: A Sceptic’s Guide

,
and
1
Department of Child and Adolescent Psychiatry, Psychosomatics and Psychotherapy, University Hospital, Ludwig Maximilian University of Munich, 80336 Munich, Germany
2
Department of Psychology and Cognitive Sciences, University of Trento, 38068 Rovereto, Italy
*
Author to whom correspondence should be addressed.
†
Current address: Pettenkoferstr. 8a, 80336 Munich, Germany.
This article belongs to the Section Neuropsychology

Abstract

Many theories have been put forward that propose that developmental dyslexia is caused by low-level neural, cognitive, or perceptual deficits. For example, statistical learning is a cognitive mechanism that allows the learner to detect a probabilistic pattern in a stream of stimuli and to generalise the knowledge of this pattern to similar stimuli. The link between statistical learning and reading ability is indirect, with intermediate skills, such as knowledge of frequently co-occurring letters, likely being causally dependent on statistical learning skills and, in turn, causing individual variation in reading ability. We discuss theoretical issues regarding what a link between statistical learning and reading ability actually means and review the evidence for such a deficit. We then describe and simulate the “noisy chain hypothesis”, where each intermediary link between a proposed cause and the end-state of reading ability reduces the correlation coefficient between the low-level deficit and the end-state outcome of reading. We draw the following conclusions: (1) Empirically, there is evidence for a correlation between statistical learning ability and reading ability, but there is no evidence to suggest that this relationship is causal, (2) theoretically, focussing on a complete causal chain between a distal cause and developmental dyslexia, rather than the two endpoints of the distal cause and reading ability only, is necessary for understanding the underlying processes, (3) statistically, the indirect nature of the link between statistical learning and reading ability means that the magnitude of the correlation is diluted by other influencing variables, yielding most studies to date underpowered, and (4) practically, it is unclear what can be gained from invoking the concept of statistical learning in teaching children to read.

1. Introduction

In order to learn to read, a child needs to learn statistical regularities that are ingrained in their orthography: any orthography contains regularities, such as the same visual symbols mapping to the same sounds across words. It has been proposed that learning such orthographic regularities (as well as other types of statistical regularities) relies on a domain-general cognitive mechanism, called statistical learning. Statistical learning enables individuals to detect a probabilistic pattern from an input stream and to be able to apply this knowledge when they encounter a similar stimulus in the future [1].
As reading, almost by definition, requires sensitivity to regularities such as print-to-speech-sound correspondences, research has investigated the role of statistical learning as a predictor of reading ability. The idea is that a statistical learning deficit may be causally related to developmental dyslexia (hereafter: dyslexia; e.g., the work of [2]). While some researchers argue that statistical learning is causally related to reading [3,4], others remain sceptical [5,6,7,8].
The statistical learning deficit theory is by far not the only causal theory of dyslexia that is controversial: other studies have proposed lower-level perceptual, neural, or cognitive deficits as causes of dyslexia (e.g., auditory temporal sampling framework; pro: [9]; sceptical: [10]; visual magnocellular deficit hypothesis; pro: [11]; sceptical: [12]). Such proposed deficits are distal causes of dyslexia [13,14]. In the distinction between distal versus proximal deficits as causes of dyslexia, a proximal deficit refers to a deficit that is located in the cognitive reading system, and thus specific to reading: for example, if a child has poor knowledge of grapheme-phoneme correspondences, this is a proximal cause of the resulting poor reading ability. Distal deficits are causes of proximal deficits or causes of other distal deficits. In the case of statistical learning (as outlined in detail below), a statistical learning deficit (distal) could result in reduced ability to learn which letters can or cannot occur in one’s orthography (distal), which might prevent the build-up of an orthographic lexicon (proximal). Thus, in a metaphorical chain between a distal cause and dyslexia, each link acts as a distal cause except for the link(s) closest to the end-state of reading ability. As another example of a distal cause theory, the temporal sampling framework [9] proposes that a cause of dyslexia is a problem with neural synchronisation to speech sounds at the temporal frequency of syllables (distal). This leads to over-reliance on phonetic information (distal), which, in turn, reduces children’s ability to perceive phonemic categories, such as hearing the /p/ in “pin” ([p]) and “spin” ([ph]) as the same phoneme (distal). The failure to perceive phonemic categories affects the learning of grapheme-phoneme correspondences (proximal), as this requires children to understand that the phonemic categories map onto graphemes (in alphabetic orthographies).
We examine the statistical learning deficit theory as a case study of a distal deficit hypothesis. First, we critically evaluate the evidence for a statistical learning deficit as a cause of dyslexia. Then, we turn to the more general question of what issues need to be resolved that are common to distal cause theories. Overall, this will provide directions for future research aiming to provide a comprehensive theory of the causes of dyslexia.

4. Issues in Establishing Distal Causes: A Noisy Chain Hypothesis

The above section describes two possible causal chains between statistical learning and reading ability or dyslexia. In both of these, statistical learning would act as a distal cause of reading ability and a distal deficit associated with dyslexia. As noted in the Introduction, other distal cause theories have been put forward, such as a procedural motor learning deficit [44], an auditory temporal sampling deficit [9], and a visual magnocellular deficit [11]. All of these theories remain controversial, and the evidence is mixed. In contrast, theories that focus on proximal causes are less controversial: for example, it is well established that children with dyslexia have problems with phonological processing (e.g., the work of [30,78]; though note that even here, it is controversial whether a phonological deficit is causal to dyslexia; [79]). Furthermore, in treatment studies of children with dyslexia, training children on cognitive mechanisms that have a proximal connection with reading ability is generally more effective than treating distal deficits [14,80].
While the above section has focussed on methodological and empirical issues specific to statistical learning, here we describe a theoretical-statistical issue that is relevant to all distal cause theories. The acknowledgement of this issue can help shed light on why the distal cause theories tend to be controversial. In a metaphorical causal chain between a perceptual (e.g., auditory), neural (e.g., neural synchronisation to speech), or low-level cognitive (e.g., statistical learning) process and reading, we expect the connection between each link, representing a cognitive skill, will not be deterministic, in the sense that a deficit in a link will not necessarily lead to dyslexia. For example, a child’s statistical learning ability is unlikely to be the only determinant of their graphotactic sensitivity; one obvious additional determinant is the amount of exposure to printed materials. This additional determinant dilutes the correlation between statistical learning ability and graphotactic sensitivity: even if we develop measures of both constructs that are completely free of measurement error, we will not obtain a correlation of 1 between them. The same holds true of the relationship between sensitivity to graphotactic regularities and spelling ability: even if we assume that this relationship is causal, other factors, such as the child’s phonological decoding ability, will additionally contribute to determining their spelling ability. Thus, as we travel further down the chain, the relationship between our first distal cause (e.g., statistical learning or the perceptual impairment following a magnocellular deficit) and reading ability or dyslexia will be diluted, which will be manifested as a reduced correlation between increasingly distal skills.
Theoretically, it should be possible to identify all contributors of a single skill or link, but we are still far from such a comprehensive understanding of the predictors of reading ability and dyslexia. If we treat all unknown contributors of a skill as random noise, it becomes possible to model the correlation between a distal skill and the end-state outcome of reading under a range of assumptions. We expect that such modelling will show two dependencies: (1) The greater the number of links between a distal cause and the end-state of reading, the smaller the correlation between the most distal cause and reading ability, and (2) the smaller the relative contribution of Link L-1 to Link L, the smaller the correlation between the most distal cause and reading ability.
The extent to which Link L contributes to Link L+1, relative to the additional predictors, is likely to vary. Some links may be very tight, such as knowledge of grapheme-phoneme correspondences as a predictor of pseudoword reading ability: though other skills (such as the ability to blend phonemes after successful conversion) are likely to affect pseudoword reading ability, this error term will be relatively small. For other links, the causal relationship may be relatively weak: if we assume that knowledge of grapheme-phoneme correspondences is affected by statistical learning ability, it will also be affected, for example, by the amount of exposure, explicit instruction of basic or even more complex grapheme-phoneme correspondences. We can model this by multiplying the contribution of Link L-1 and the noise term by xL and yL, where xL + yL = 1. In the case of the link between knowledge of grapheme-phoneme correspondences and reading ability, the contribution of grapheme-phoneme correspondence knowledge (xL) might be 0.8, with the remaining 20% (yL = 0.2) reflecting the influence of all other predictors. In the case of statistical learning to the knowledge of grapheme-phoneme correspondences, this contribution will be much smaller (e.g., xL = 0.2), with the remaining 80% reflecting the influence of all other predictors (yL = 0.8)
We will model each Skill X as a standard normal distribution (Skill X ~ N(0,1)). Skill A, denoting the most distal cause, is thus modelled by drawing numbers from a standard normal distribution (μ = 0, σ = 1). Skill B is partly determined by Skill A and partly by noise (ε), resulting from additional, unknown predictors, again, modelled as a standard normal distribution (εn ~ N(0, 1)). Taken together, then, the model, for n amount of links, including the most distal cause (Skill A), will look as follows:
Skill A ~ N(0, 1)
Skill B = x1 ∗ Skill A + y1 ∗ ε1, ε1 ~ N(0, 1)
Skill C = x2 ∗ Skill B + y2 ∗ ε2, ε2 ~ N(0, 1)
…
Reading ability = xn ∗ Skill n + yn ∗ εn, εn ~ N(0, 1)
We implemented this model in R (version 4.0.2; [81]); the code can be found on the Open Science Framework: https://osf.io/epu8m/ (accessed on 1 June 2021). We varied both the number of links (n) and the relative contributions of Link L versus the unknown predictors (x and y). The number of links varied between a direction relationship (Skill A → reading) with no intermediate links, and 1 (Skill A → Skill B → reading), 2, 3, or 4 intermediate links.
For values of x, we arbitrarily chose different values for scenarios where the contribution of Link L to Link L+1 (xn) may be low (0.2), medium (0.5), or high (0.8). This value is likely to vary for each link within a chain; however, to keep the number of simulations with different parameters manageable, we kept it constant within each chain (i.e., for all links within a chain, xn is either 0.2, 0.5, or 0.8). For each scenario, we simulated 1000 participants’ scores on Skills A to Skill n, as well as reading ability, and calculated the correlation between Skill A (the most distal cause) and reading ability. We standardised each skill to retain the interpretability of the scores as z-scores. To derive a confidence interval in addition to a point estimate for the correlation, we repeated this process, for each scenario, 10,000 times (the number of simulated participants and simulations were chosen within a trade-off between getting reliable output values and computational restraints). This allowed us to provide a point estimate for the correlation (average correlation across 10,000 simulations) and an upper and lower bound of a 95% CI (bounds of the part distribution that contain 95% of the simulated data). These are depicted in Table 1. From the correlation coefficients, it is trivial to provide a power calculation: how many participants are required to detect the correlation with p < 0.05, 80% of the time, for the given correlation? These calculations were conducted with the R package pwr (version 1.3-0; [82]).
Table 1. Results of simulations, showing the estimated correlation coefficient for different scenarios of relationship strength and number of intermediates.
We note that the scenarios with a strong relationship between Link L and Link L+1 are likely to be overly optimistic: in most real-life cases, additional skills would account for more than 20% of performance on Skill L+1. Furthermore, the current simulations ignore the role of measurement error for the sake of simplicity: measurement error further reduces the correlations between any set of skills. The causal chains for statistical learning that we have described above contain, as a minimum, two intermediate links: for example, statistical learning (Skill A) → sensitivity to complex grapheme-phoneme correspondences (Skill B) → ability to read unfamiliar words (Skill C) → reading ability. In this case, assuming a medium-strength relationship (which seems to us like the most optimistic but still realistic case scenario), one would need 170 participants to detect a correlation between statistical learning and reading ability. To name some recent examples, correlational studies that have assessed this relationship tested 38 children and 37 adults [4], 84 adults [6], 65 children [83], 72 adults and children [84].
In dyslexia research, rather than calculating correlations, researchers select a sample of dyslexia and compare their performance on Skill A to a matched control group. We simulated this scenario to derive an effect size estimate: instead of calculating a correlation between Skill A and reading, we divided the simulated participants into dyslexic participants (standardised reading score z < −2) and controls (standardised reading score z ≥ −2) and derived a standardised effect size (Cohen’s d) by subtracting the mean simulated performance on Skill A of the control group from the mean simulated performance on Skill A of the group with dyslexia, and dividing the difference score by the overall simulated population standard deviation (d = (Skill A mean dyslexia − Skill A mean control)/Skill A standard deviation all). To increase the number of participants in the dyslexia group for reliable statistics, we changed the number of simulated participants to 10,000. Otherwise, the simulation and parameters were identical to the previous simulations. The results of these simulations are presented in Table 2.
Table 2. Results of simulations, showing the estimated standardised effect size estimate for different scenarios of relationship strength and number of intermediates.
An optimistic-realistic-case scenario (medium-strength relationship, 2 intermediate links) shows that, in order to detect a significant group difference between participants with and without dyslexia in the most distal deficit, we would need 45 participants (approximately 23 participants per group). Again, most studies have a smaller sample size: in a systematic review of artificial grammar learning studies in participants with and without dyslexia, we have found a median sample size of 32 participants (16 per group), with a minimum of 30 and a maximum of 223 in both groups [5].

5. Directions for Future Research

So far, we have identified both theoretical and statistical issues pertaining to the statistical learning theory of dyslexia. Some of these can be applied to other distal theories of dyslexia. From a statistical perspective, the noisy chain hypothesis and our simulations thereof clearly show that most studies, to date, are underpowered to detect a correlation between a distal cause and the end-state outcome of reading ability, or a group difference in measures of the distal cause between participants with and without dyslexia. This is an issue that is common to all distal theories of dyslexia. Across simulations, we showed a wide range of correlations and effect sizes, resulting in vast differences in sample size calculations for achieving adequate power. Future research could aim to narrow these down by establishing plausible parameter values. These are likely to depend on the particular research question.
For a researcher proposing a distal theory, the implication of this finding is that the number of links in the causal chain should be taken into account when determining sample size. This will improve the research on the causes of dyslexia in two ways. First, this will force the researcher to specify an exact causal pathway. Second, it is likely that the sample size required to find a link between the most distal cause and reading ability will be much larger than what is generally used in this kind of research. The specification of the causal pathway will have a theoretical benefit, as it will avoid ambiguity, which results in unclear predictions, thus making it difficult to either support or falsify a given theory.
Improving sample size, overall, will improve our ability to draw conclusions from mixed sets of findings. Low statistical power is problematic in most areas of psychology, with no apparent improvement, on average, since the phenomenon was first described in the 1960s [85,86]. A commonality between distal causal theories of dyslexia is the mixed empirical results, with failures to replicate an initial, often positive finding of a group deficit. By definition, we would expect that many studies would yield non-significant results when we have low statistical power. Thus, the false-negative rate is likely to be high. However, other common issues in social and behavioural sciences increase the false positive rate: for example, publication bias [87] and questionable research practices [88]. This leaves us in a situation where it is very difficult to interpret a mixed set of findings, as both a true effect with false negatives due to low power and the absence of a true effect with some false positives are plausible scenarios.
An obvious solution to the problem of low statistical power is to run larger studies. Depending on the scenario, however, this may require sample sizes in the thousands or even up to 200,000,000 participants. Due to the practical hurdles of testing such a big number of participants, this might not always be feasible. To circumvent this issue, researchers may take a more experimental approach when possible. In artificial orthography learning studies, we can minimise the effect of existing print-related knowledge, which, in itself, is likely to be caused by a plethora of known and unknown, measured, and unmeasurable external variables. While such experiments lack external validity, they can lay the groundwork for a follow-up study [89]: if a researcher can show a model of the causes of dyslexia and support the links of their causal chain with experimental evidence, they can use this knowledge to apply for an amount of funding that will allow them to perform an adequately powered study.
Another recommendation is to focus on individual links in the causal chain instead of focussing, from the very beginning of a new line of research, on the relationship between the most distal skill and reading ability. The correlation between two adjacent links is likely to be strongest. From a theoretical perspective, preliminary studies focussing on only a part of a causal chain can be used to provide evidence for or against the proposed theory before a large-scale observational study is conducted. The findings of the study can also be used to estimate the parameters in the simulation above: the strength of the relationship between individual links and the number of linking skills. This will allow for a more accurate effect size estimate. Furthermore, the methods of preliminary studies can be tweaked to minimise measurement error, leading to stronger correlations and a smaller number of participants, which will be required for a large-scale observational study.

5.1. Establishing Causality for the Statistical Learning Deficit Theory

In the section on making links in a causal chain for the statistical learning deficit theories of dyslexia, we have shown correlational evidence but concluded that evidence for causality is missing. Again, this is an issue that is relevant not only for the statistical learning deficit theory but also for other distal theories, which are often correlational.
In the case of statistical learning and dyslexia, previous studies have already provided important puzzle pieces towards identifying the direction of causality between sensitivity to orthography-related regularities and reading ability. Experimental studies with artificial orthographies are able, in principle, to establish causality, but they are limited in their ecological validity: it remains unclear to what extent adults learning an artificial script use similar cognitive mechanisms as children learning to read. Correlational studies can be conducted with children learning to read in a natural setting but cannot be used to establish causality. The missing link to establish causality would be either a longitudinal study or a training study. Such studies would need to be carefully designed to truly understand the direction of causality: children who are inherently good at reading tend to read more [90], which means that better readers have more exposure to texts and the regularities that they contain [91].
A longitudinal study would be able to shed further light on the relationship between statistical learning, knowledge of print-related regularities, and reading. Such a study should start with preschool children, akin to Zhang and Treiman [37]. The first question is whether individual differences in the ability to rely on graphotactic regularities in spelling continue to predict reading acquisition after the onset of formal instruction. However, even here, we cannot exclude the reverse direction of causality: even if children have not started reading acquisition yet, their knowledge of graphotactic regularities must come from having been exposed to print. Such a study would therefore not provide an unequivocal link between statistical learning ability and reading but would at best show that knowledge of statistical regularities aids reading acquisition. A second question is whether preschool children’s ability to extract regularities from an artificial orthography would predict their later reading abilities in their native language. If a statistical learning deficit causes dyslexia, one would predict that all children who are impaired in extracting regularities will develop dyslexia after the onset of reading instructions. This combination of experimental and observational research would help to establish a causal link between the ability to learn regularities and reading acquisition.
What would be the implications of further research on statistical learning and dyslexia, and what are the implications of the findings so far? The studies to date have provided important theoretical insights into learning mechanisms that potentially underlie not only reading but also other visual learning tasks [92]. Understanding the direction of causality would shed further light on the cognitive mechanisms underlying reading acquisition. This would help tighten the link between linguistics and educational psychology, as it would open directions for linguistic research on relevant regularities that are present in orthographies and spoken languages.

5.2. Practical Implications of the Statistical Learning Deficit Hypothesis

The practical implications of statistical learning and dyslexia research are likely to be more limited. In general, identifying a causal deficit is associated with the promise of early identification and treatment. To date, we lack any indication that general statistical learning ability is causally related to reading ability. If such evidence is found in future studies, it will be a challenge to devise statistical learning tasks for children with psychometric properties that would allow for them to be used as a diagnostic test [46]. The link that does have empirical support is between sensitivity to print-related regularities and reading ability. Sensitivity to print-related regularities cannot develop without already having had some exposure to print; thus, it is unclear whether anything is gained by taking into account the sensitivity to regularities beyond simply assessing children’s reading ability.
In terms of treatment, there are three levels on which one could try to improve children’s reading ability. The first would be to train a domain-general statistical learning ability. To date, it is unclear whether this is possible or whether there would be any benefits even if a reliable training method is found. Second, one could increase children’s sensitivity to print-related statistical regularities by exposing them to more reading material. This would seem to be a sensible strategy in any case, as the number of books in a child’s house has been shown to be a predictor of reading ability, over and above genetic influences [93].
Third, one could explicitly teach children about regularities in their orthography. For example, if a child spells a word with an implausible letter sequence, such as ckwick for quick, one could explain that, even though ck is, indeed, a plausible spelling for the sound /k/, in English, words never start with ck. Similarly, one can teach a child to consider morphology in their spellings: if they spell, for example, magician as majishun, one can draw their attention to the relationship of this word to the word magic and other words containing the morpheme ian, such as mathematician. The focus is no longer on a cognitive statistical learning process but rather on explicitly teaching children print-related regularities. This raises the question of what is gained by invoking the concept of statistical learning in research on print-related regularities. From a theoretical perspective, it is relevant why a child has not been able to learn a particular regularity. From a practical perspective, however, it is likely that the treatment will be identical, regardless of the reasons why a child has not learned a given regularity: be it due to a statistical learning deficit, lack of exposure to reading materials, or a lack of explicit instructions relating to these regularities.

5.3. Further Theoretical Implications of Statistical Learning

As discussed in the Introduction, print consists mostly of orthographic regularities, and learning the statistical properties of these regularities is, by definition, necessary for reading acquisition. Thus, statistical learning is definitely critical for reading acquisition, but as this describes the very nature of an orthography, referring to reading as “statistical learning” arguably serves as a one-word explanation, which has a less well-defined meaning than the concept that it is proposed to describe, and shifts the focus away from the underlying cognitive mechanisms [94]. From a sceptic’s perspective, therefore, we propose to reserve the term “statistical learning” for the domain-general cognitive learning mechanism that is likely to play a role in many, if not all, learning tasks.

6. Conclusions

In the current review paper, we discuss issues related to the statistical learning deficit theory of dyslexia, some of which apply to distal deficit theories of dyslexia in general. First, we reviewed the evidence for a statistical learning deficit in dyslexia. While we find convincing evidence for a correlation, we argue that none of the existing studies establish causality: this is a concern for theories of dyslexia in general, which are often based on correlational or quasi-experimental evidence (i.e., differences in a given skill between participants with and without dyslexia). This issue can be addressed, at least in part, by future experimental and longitudinal work.
We then describe and model the noisy chain hypothesis, which could help in driving future studies. The hypothesis builds on the assumption that the strength of the relationship between a distal cause and reading ability or dyslexia is diluted with each intermediate linking skill. Our simulations show that this leads to a decrease in correlation between the most distal cause and reading ability with each additional intermediate link. Applied to (statistically) predicting whether a given child will show symptoms of dyslexia rather than predicting reading ability as a continuous variable, this should result in smaller group differences between participants with and without dyslexia for distal causes. The dilution of effect sizes is due to additional variables that influence each of the linking skills. This means that a larger number of participants is required to reach adequate statistical power compared to studies that test a proximal cause, which is closely linked to the end-state outcome of reading ability. For theories of distal causes of dyslexia, this means that much of the current research is likely to be underpowered. To circumvent issues with low statistical power, we propose (1) well-defined theories, which specify the number and nature of the intermediate links, (2) experimental research, where the influence of additional variables is minimised, and (3) studies focussing on adjacent links, which can be used to minimise measurement noise and to provide a proper effect size estimate to ultimately test the effect of the distal cause on reading ability or dyslexia.

Author Contributions

Conceptualization, X.S., B.T. and C.M.; Methodology, X.S., B.T. and C.M.; Formal analysis: X.S.; Writing—original draft preparation, X.S.; Writing—review and editing, X.S., B.T. and C.M.; Project administration, X.S.; Funding acquisition, B.T. & X.S. All authors have read and agreed to the published version of the manuscript.

Funding

X.S. was supported by a grant from the Deutsche Forschungsgemeinschaft (SCHM3450/1-1) and by the Programme “Fellows Freies Wissen” from the Stifterverband, Wikimedia, and Volkswagenstiftung (H190 5909 5096 32135). Parts of the manuscript were prepared while XS was a visiting researcher at the University of Trento. The APC was funded by DIPSCO - University of Trento.

Data Availability Statement

The code to the model can be found at https://osf.io/epu8m/, accessed on 27 June 2021.

Conflicts of Interest

The authors declare no conflict of interest.

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