Contribution
The largest survey of chronic locked-in patients’ own ratings of their lives at the time: a postal questionnaire to the membership of ALIS, the French LIS association, measuring overall subjective well-being with a biographically anchored scale, reintegration into normal life, and views on resuscitation and euthanasia. It establishes that a majority of surviving, association-member LIS patients rate their lives as good, and names the correlates of the minority who do not.
Key insight
Long survival in LIS is compatible with a life rated as good by the person living it, and the rating improves with time in the condition, but the sample that shows it is survivors who have, for the most part, recovered some action: over half speak some words, most move some limb. The paper is evidence about lucid evaluation over years in LIS, not about lucidity with action reduced to an eyelid.
Assumptions
- Population. Members of ALIS, invited by letter in January 2008; answers “aided by the patient’s proxy”. All included patients had chronic LIS (> 1 year) “following a brainstem vascular accident”.
- Definition. LIS “is defined by quadriplegia (or quadriparesis) and aphonia (or severe hypophonia) with a primary mode of communication by eye movements or blinking”, citing the 1995 American Congress of Rehabilitation Medicine nomenclature.
- The forms. “‘classic’ LIS is characterised by quadriplegia and aphonia with coded communication by vertical eye movement or blinking; ‘incomplete’ LIS patients have remnants of voluntary motion other than vertical eye movement; and ‘total’ LIS is defined by complete immobility, including all eye movements”, citing Bauer, Gerstenbrand and Rumpl (1979).
- Measure. The Anamnestic Comparative Self-Assessment (ACSA), whose +5 and −5 anchors are “the patients’ memories of the best period in their life before LIS and their worst period ever”. Happy: ACSA 0 to +5; unhappy: −1 to −5. Reintegration by the RNLI on a 4-point scale.
- Analysis. Univariate tests, then backward stepwise logistic regression with casewise deletion; only questions answered by > 70% analysed.
Key results
- Response. 168 invited; 77 (46%) did not reply; of 91 replies, 26 excluded for missing RNLI or ACSA data (5 lacked RNLI, 21 lacked ACSA); 65 analysed. Excluded respondents were less educated (p = 0.009) and had more pain (p = 0.009).
- Sample. Mean age 49 ± 11; median 8 years in LIS (range 1–28); 64% at home; 64% with a partner. Recovery of speech production: none 26/58 (45%), words 11/58 (19%), sentences 21/58 (36%). Some limb mobility 39/56 (70%).
- Well-being. 47 happy (72%, median ACSA +3), 18 unhappy (28%, median −4). RNLI: 51% severe and 49% mild-to-moderate restrictions in community reintegration; 82% comfortable with personal relationships; 21% occupied most of the day in activities important to them.
- Correlates of unhappiness (univariate). Dissatisfaction with mobility in the community (p = 0.042), with recreational activities (p = 0.028) and with the capacity to deal with life events (p = 0.022); shorter time in LIS (median 7 vs 9 years, p = 0.005); non-recovery of speech (p = 0.049); anxiety (p = 0.015); depression (28% vs 7%, p = 0.040).
- Multivariate (Table 3). Duration in LIS OR 1.5 per year (95% CI 1.1–2.0, p = 0.007); speech production OR 20.47 (1.89–221.45, p = 0.013); anxiety OR 0.19 (0.04–0.87, p = 0.032). The variables “explained only 38% of the variance of overall SWB”.
- End of life. 58% (32/55) did not wish to be resuscitated in case of cardiac arrest, 86% of the unhappy and 49% of the happy; euthanasia envisaged by 53% (31/59); a current wish for euthanasia by 4 of 59 (7%). Suicidal thoughts often: 5/59 (8%).
Claims
| id | claim | strength | support |
|---|---|---|---|
| C1 | Most chronic LIS survivors in this association rate their present life as good relative to their own past | moderate | 47/65; 54% response, 26 further excluded; biases “likely to result in overestimations” |
| C2 | Longer time in LIS goes with happiness | moderate | OR 1.5/yr, p = 0.007; authors offer adaptation or attrition as explanations |
| C3 | Recovery of speech and absence of anxiety go with happiness | moderate | ORs 20.5 and 0.19, wide CIs |
| C4 | Recently affected patients should be told they have a high chance of regaining a happy life, and a moratorium on end-of-life requests should be proposed | weak | clinical recommendation from cross-sectional data; causality not shown |
| C5 | LIS patients are “cognitively intact” and so competent to decide | weak | asserted in the Discussion with one citation (Schnakers et al. 2008); not measured here |
Concepts
- Disability paradox. People with severe persistent disability tend to report good subjective well-being.
- ACSA. A self-anchored scale whose ends are the respondent’s own best and worst periods; chosen to reduce comparison with peers and “under-the-circumstances” responses.
- Response shift / relativity bias. Rating relative to peers or circumstances, which the ACSA is meant to limit; its own risk is a “nostalgia effect” that would depress current ratings.
Connections
It extends a 15-patient quality-of-life study and Doble et al.’s long-term cohort, and follows Cummins’s homeostatic theory of subjective well-being in interpreting the result. The forms of LIS come from Bauer et al. (1979), and its cognitive claim from Schnakers et al. (2008). It points to brain–computer interfaces (Kübler and Neumann 2005, in the same Progress in Brain Research volume as Laureys et al. 2005, LIT-1198) as expected progress. Laureys is senior author here too.
Bearing on the record
- CASE-107.
- The definition. The CASE’s quotation from the abstract is verbatim. The paper’s own definition, in the Introduction, says LIS is quadriplegia “or quadriparesis” and aphonia “or severe hypophonia”, with eye-coded communication “primary”, and its three forms (classic, incomplete, total) are attributed to Bauer et al. (1979), not to Laureys et al. (2005). The CASE’s “The literature distinguishes classic, incomplete and total forms (Laureys et al. 2005)” can be supported from this paper with the original source named.
- “Lucid over years”. Correct, with the qualifier the CASE does not state: this cohort is largely not on one eyelid. 55% had recovered some speech (36% sentences) and 70% some limb mobility, the Introduction notes that “many classic LIS patients may evolve to incomplete LIS”, and answers were given with a proxy’s help. So it shows lucid, evaluative life over years in people whose world-directed action was severely reduced but, in most, partly restored. The survey does not report how many were still in classic LIS.
- “Answered through the spared channels”. The paper says only that the questionnaire was filled in “aided by the patient’s proxy” and adapted “Given the specific constraint of eye-coded communication”; it does not report channels per patient.
- CLAIM-341. The paper bears on the claim’s “What it does not say” (the loop that keeps a built model in touch) only weakly. A biographically anchored self-rating is a judgement made against the person’s own remembered past, maintained over a median of 8 years in a condition where action is reduced, and it moves with time (OR 1.5 per year). That is consistent with judgement keeping a model working with little action. It is not evidence for the thesis against active inference, because most respondents retained or recovered action, and speech recovery is the strongest correlate of happiness (OR 20.5): the data are equally read as action mattering to how well the life goes. Neither reading concerns whether the model stays in touch.
- CLAIM-339, QUESTION-121. No direct bearing. The authors’ explanation of the minority, those “who cannot shed the needs and values from their previous life”, is a judgement-side account of adaptation (recalibrating and reprioritising), not an action-side one, but it is offered as a hypothesis.
- The LIT. Its summary is right in its numbers. “Communication is classically by vertical eye movement or blinking” is the definition, not a description of this sample; the summary should say that most had recovered some speech or limb movement and that answers were given with a proxy. The published date can be the e-publication date, 23 February 2011 (PMC).
- For the anthology. No instruction for machine-learning practice.
Limitations
- 54% response, then 29% of responses excluded; association members, “indicating a stable condition and possibly a degree of social integration”; caregiver-assisted answers open to social desirability. The authors judge “most biases were likely to result in overestimations of SWB”.
- Cross-sectional: the time-in-LIS association may be adaptation or “selection by attrition”.
- Only vascular brainstem LIS; nothing on ALS or CLIS.
- Cognitive status was not measured; competence is asserted from the literature.
Open questions
- Longitudinal: does well-being rise within patients over time, or do the unhappy die sooner? The authors call for this.
- For the record: how does well-being compare between those still in classic LIS and those who recovered speech or limb movement? The paper reports speech as a predictor but no split by LIS form.