Selection bias on intellectual ability in autism research: a cross-sectional review and meta-analysis

This is the final version. Available from BMC via the DOI in this record.Background Current global estimates suggest the proportion of the population with autism spectrum disorder (ASD) who have intellectual disability (ID) is approximately 50%. Our objective was to ascertain the existence of selection bias due to under-inclusion of populations with ID across all fields of autism research. A sub-goal was to evaluate inconsistencies in reporting of findings. Methods This review covers all original research published in 2016 in autism-specific journals with an impact factor greater than 3. Across 301 included studies, 100,245 participants had ASD. A random effects meta-analysis was used to estimate the proportion of participants without ID. Selection bias was defined as where more than 75% of participants did not have ID. Results Meta-analysis estimated 94% of all participants identified as being on the autism spectrum in the studies reviewed did not have ID (95% CI 0.91–0.97). Eight out of ten studies demonstrated selection bias against participants with ID. The reporting of participant characteristics was generally poor: information about participants’ intellectual ability was absent in 38% of studies (n = 114). Where there was selection bias on ID, only 31% of studies mentioned lack of generalisability as a limitation. Conclusions We found selection bias against ID throughout all fields of autism research. We recommend transparent reporting about ID and strategies for inclusion for this much marginalised group.Wellcome Trus

Primordial formation of major silicates in a protoplanetary disc with homogeneous 26Al/27Al

Understanding the spatial variability of initial 26Al/27Al in the solar system, i.e., (26Al/27Al)0, is of prime importance to meteorite chronology, planetary heat production, and protoplanetary disc mixing dynamics. The (26Al/27Al)0 of calcium-aluminum–rich inclusions (CAIs) in primitive meteorites (~5 × 10−5) is frequently assumed to reflect the (26Al/27Al)0 of the entire protoplanetary disc, and predicts its initial 26Mg/24Mg to be ~35 parts per million (ppm) less radiogenic than modern Earth (i.e., Δ′26Mg0 = −35 ppm). Others argue for spatially heterogeneous (26Al/27Al)0, where the source reservoirs of most primitive meteorite components have lower (26Al/27Al)0 at ~2.7 × 10−5 and Δ′26Mg0 of −16 ppm. We measured the magnesium isotope compositions of primitive meteoritic olivine, which originated outside of the CAI-forming reservoir(s), and report five grains whose Δ′26Mg0 are within uncertainty of −35 ppm. Our data thus affirm a model of a largely homogeneous protoplanetary disc with (26Al/27Al)0 of ~5 × 10−5, supporting the accuracy of the 26Al→26Mg chronometer

Mapping the autistic advantage from the accounts of adults diagnosed with autism: A qualitative study

This is the final version. Available on open access from Mary Ann Liebert via the DOI in this recordBackground: Autism has been associated with specific cognitive strengths. Strengths and weaknesses have traditionally been conceptualized as dichotomous. Methods: We conducted 28 semi-structured interviews with autistic adults. Maximum variation sampling was used to ensure diversity in relation to support needs. We asked which personal traits adults attributed to their autism, and how these have helped in the workplace, in relationships, and beyond. Data were collected in two stages. Responses were analyzed using content and thematic techniques. Results: The ability to hyperfocus, attention to detail, good memory, and creativity were the most frequently described traits. Participants also described specific qualities relating to social interaction, such as honesty, loyalty, and empathy for animals or for other autistic people. In thematic analysis we found that traits associated with autism could be experienced either as advantageous or disadvantageous dependent on moderating influences. Moderating influences included the social context in which behaviors occurred, the ability to control behaviors, and the extent to which traits were expressed. Conclusions: Separating autistic strengths from weaknesses may be a false dichotomy if traits cannot be isolated as separate constructs of strengths or deficits. If attempts to isolate problematic traits from advantageous traits are ill conceived, there may be implications for interventions that have reduction in autistic traits as a primary outcome measure.Wellcome Trus

People should be allowed to do what they like’: Autistic adults’ views and experiences of stimming

This is the final version. Available from the publisher via the DOI in this record.Data from participants who consented will be deposited in the UK Data Service, in 2019.‘Stereotyped or repetitive motor movements’ are characterised as core features in the diagnosis of autism, yet many autistic adults (and the neurodiversity movement) have reclaimed them as ‘stimming’. Supported by a growing body of scientific research, autistic adults argue that these behaviours may serve as useful coping mechanisms, yet little research has examined stimming from the perspective of autistic adults. Through interviews and focus groups, we asked 32 autistic adults to share their perceptions and experiences of stimming, including the reasons they stim, any value doing so may hold for them and their perceptions of others’ reactions to stimming. Using thematic analysis, we identified two themes: stimming as (1) a self-regulatory mechanism and (2) lacking in social acceptance, but can become accepted through understanding. Autistic adults highlighted the importance of stimming as an adaptive mechanism that helps them to soothe or communicate intense emotions or thoughts and thus objected to treatment that aims to eliminate the behaviour.Wellcome TrustLeverhulme Trus

Perceived importance of components of asynchronous music in circuit training

This study examined regular exercisers’ perceptions of specific components of music during circuit training. Twenty-four men (38.8 years, s = 11.8 years) and 31 women (32.4 years, s = 9.6 years) completed two questionnaires immediately after a circuit training class. Participants rated the importance of 13 components of music (rhythm, melody, etc.) in relation to exercise enjoyment, and each completed the Affect Intensity Measure (Larsen, 1984) to measure emotional reactivity. Independent t tests were used to evaluate gender differences in perceptions of musical importance. Pearson correlations were computed to evaluate the relationships between affect intensity, age and importance of musical components. Consistent with previous research and theoretical predictions, rhythm response components (rhythm, tempo, beat) were rated as most important. Women rated the importance of melody significantly higher than did men, while men gave more importance to music associated with sport. Affect intensity was found to be positively and significantly related to the perceived importance of melody, lyrical content, musical style, personal associations and emotional content. Results suggest that exercise leaders need to be sensitive to personal factors when choosing music to accompany exercise. Qualitative research that focuses on the personal meaning of music is encouraged

A patient-derived orthotopic xenograft (PDOX) mouse model of a cisplatinum-resistant osteosarcoma lung metastasis that was sensitive to temozolomide and trabectedin: implications for precision oncology.

In the present study, we evaluated the efficacy of trabectedin (TRAB) and temozolomide (TEM) compared to cisplatinum (CDDP) on a patient-derived orthotopic xenogrraft (PDOX) of a lung-metastasis from an osteosarcoma of a patient who failed CDDP therapy. Osteosarcoma resected from the patient was implanted orthotopically in the distal femur of mice to establish PDOX models which were randomized into the following groups when tumor volume reached approximately 100 mm3: G1, control without treatment; G2, CDDP (6 mg/kg, intraperitoneal injection, weekly, for 2 weeks); G3, TRAB (0.15 mg/kg, intravenous injection, weekly, for 2 weeks); G4, TEM (25 mg/kg, oral, daily, for 14 days). Tumor sizes and body weight were measured with calipers and a digital balance twice a week. On day 14 after initiation of treatment, TEM and TRAB, but not CDDP, significantly inhibited tumor volume compared to untreated control: control (G1): 814.5±258.8 mm3; CDDP (G2): 608.6±126.9 mm3, TRAB (G3): 286.6±133.0 mm3; TEM (G4): 182.9±69.1 mm3. CDDP vs. control, p=0.07; TRAB vs. control, p=0.0004; TEM vs. control p =0.0002; TRAB vs. CDDP, p =0.0002; TEM vs. CDDP, p =0.00003. The results of the present study show that a PDOX model of an osteosarcoma lung-metastasis that recurred after adjuvant CDDP-treatment has identified potentially, highly-effective drugs for this recalcitrant disease, while precisely maintaining the CDDP resistance of the tumor in the patient, thereby demonstrating the potential of the osteosarcoma PDOX model for precision oncology

Trabectedin arrests a doxorubicin-resistant PDGFRA-activated liposarcoma patient-derived orthotopic xenograft (PDOX) nude mouse model.

BACKGROUND:Pleomorphic liposarcoma (PLPS) is a rare, heterogeneous and an aggressive variant of liposarcoma. Therefore, individualized therapy is urgently needed. Our recent reports suggest that trabectedin (TRAB) is effective against several patient-derived orthotopic xenograft (PDOX) mouse models. Here, we compared the efficacy of first-line therapy, doxorubicin (DOX), and TRAB in a platelet-derived growth factor receptor-α (PDGFRA)-amplified PLPS. METHODS:We used a fresh sample of PLPS tumor derived from a 68-year-old male patient diagnosed with a recurrent PLPS. Subcutaneous implantation of tumor tissue was performed in a nude mouse. After three weeks of implantation, tumor tissues were isolated and cut into small pieces. To match the patient a PDGFRA-amplified PLPS PDOX was created in the biceps femoris of nude mice. Mice were randomized into three groups: Group 1 (G1), control (untreated); Group 2 (G2), DOX-treated; Group 3 (G3), TRAB-treated. Measurement was done twice a week for tumor width, length, and mouse body weight. RESULTS:The PLPS PDOX showed resistance towards DOX. However, TRAB could arrest the PLPS (p < 0.05 compared to control; p < 0.05 compared to DOX) without any significant changes in body-weight. CONCLUSIONS:The data presented here suggest that for the individual patient the PLPS PDOX model could specifically distinguish both effective and ineffective drugs. This is especially crucial for PLPS because effective first-line therapy is harder to establish if it is not individualized

Selection bias on intellectual ability in autism research: a cross-sectional review and meta-analysis

Background: Current global estimates suggest the proportion of the population with autism spectrum disorder (ASD) who have intellectual disability (ID) is approximately 50%. Our objective was to ascertain the existence of selection bias due to under-inclusion of populations with ID across all fields of autism research. A sub-goal was to evaluate inconsistencies in reporting of findings. Methods: This review covers all original research published in 2016 in autism-specific journals with an impact factor greater than 3. Across 301 included studies, 100,245 participants had ASD. A random effects meta-analysis was used to estimate the proportion of participants without ID. Selection bias was defined as where more than 75% of participants did not have ID. Results: Meta-analysis estimated 94% of all participants identified as being on the autism spectrum in the studies reviewed did not have ID (95% CI 0.91–0.97). Eight out of ten studies demonstrated selection bias against participants with ID. The reporting of participant characteristics was generally poor: information about participants’ intellectual ability was absent in 38% of studies (n = 114). Where there was selection bias on ID, only 31% of studies mentioned lack of generalisability as a limitation. Conclusions: We found selection bias against ID throughout all fields of autism research. We recommend transparent reporting about ID and strategies for inclusion for this much marginalised grou

Harbour seals avoid tidal turbine noise: implications for collision risk

1. Tidal stream energy converters (turbines) are currently being installed in tidally energetic coastal sites. However, there is currently a high level of uncertainty surrounding the potential environmental impacts on marine mammals. This is a key consenting risk to commercial introduction of tidal energy technology. Concerns derive primarily from the potential for injury to marine mammals through collisions with moving components of turbines. To understand the nature of this risk, information on how animals respond to tidal turbines is urgently required. 2. We measured the behaviour of harbour seals in response to acoustic playbacks of simulated tidal turbine sound within a narrow coastal channel subject to strong, tidally induced currents. This was carried out using data from animal-borne GPS tags and shore-based observations, which were analysed to quantify behavioural responses to the turbine sound. 3. Results showed that the playback state (silent control or turbine signal) was not a significant predictor of the overall number of seals sighted within the channel. 4. However, there was a localised impact of the turbine signal; tagged harbour seals exhibited significant spatial avoidance of the sound which resulted in a reduction in the usage by seals of between 11 and 41% at the playback location. The significant decline in usage extended to 500 m from the playback location at which usage decreased by between 1 and 9% during playback. Synthesis and applications: This study provides important information for policy makers looking to assess the potential impacts of tidal turbines and advise on development of the tidal energy industry. Results showing that seals avoid tidal turbine sound suggest that a proportion of seals encountering tidal turbines will exhibit behavioural responses resulting in avoidance of physical injury; in practice, the empirical changes in usage can be used directly as avoidance rates when using collision risk models to predict the effects of tidal turbines on seals. There is now a clear need to measure how marine mammals behave in response to actual operating tidal turbines in the long term to learn whether marine mammals and tidal turbines can co-exist safely at the scales currently envisaged for the industry