Psychometric properties of health-related quality of life instruments in patients undergoing palmar fasciectomy for dupuytren’s disease: a prospective study

Background

The traditional outcome measured following treatment of Dupuytren’s Disease (DD) has been digital range of motion; specifically the gain in digital extension. The outcomes research movement in the last three decades however has been advocating the measurement of outcomes from the patient’s perspective using Health-Related Quality of Life questionnaires (HRQOL). Although several generic and region-specific HRQOL questionnaires exist, there is no guidance as to which one is the most appropriate for this population. The objective of this study is to evaluate the psychometric properties of three self-reported HRQOL outcome measures in patients with DD.

Methods

Patients with DD were enrolled from the practices of three plastic surgeons. Test-retest reliability, concurrent validity and responsiveness of three HRQOL questionnaires were compared in a prospective study design. The HRQOL measures included Health Utilities Index Mark 3 (HUI3), Short Form-36 (SF-36), and the Michigan Hand Questionnaire (MHQ).

Results

All three measures demonstrated good test-retest reliability (ICC = 0.77–0.85). Concurrent validity was found between the HUI3 pain and dexterity attributes and SF-36 physical summary score. The sensitivity of the MHQ to detect changes in the status of the patient was found to be high (effect size = 1.14) whereas that of the SF-36 was trivial.

Conclusions

The HUI3 and the MHQ seem to be reliable and valid tools to assess the HRQOL in patients with Dupuytren’s Disease.     

Language Impairment and Early Social Competence in Preschoolers with Autism Spectrum Disorders: A Comparison of DSM-5 Profiles

Children with autism spectrum disorder (ASD) and structural language impairment (LI) may be at risk of more adverse social-developmental outcomes. We examined trajectories of early social competence (using the Vineland-II) in 330 children aged 2–4 years recently diagnosed with ASD, and compared 3 subgroups classified by: language impairment (ASD/LI); intellectual disability (ASD/ID) and ASD without LI or ID (ASD/alone). Children with ASD/LI were significantly more socially impaired at baseline than the ASD/alone subgroup, and less impaired than those with ASD/ID. Growth in social competence was significantly slower for the ASD/ID group. Many preschool-aged children with ASD/LI at time of diagnosis resembled “late talkers” who appeared to catch up linguistically. Children with ASD/ID were more severely impaired and continued to lag further behind.     

Investigating phenotypic heterogeneity in children with autism spectrum disorder: a factor mixture modeling approach

Background: Autism spectrum disorder (ASD) is characterized by notable phenotypic heterogeneity, which is often viewed as an obstacle to the study of its etiology, diagnosis, treatment, and prognosis. On the basis of empirical evidence, instead of three binary categories, the upcoming edition of the DSM 5 will use two dimensions – social communication deficits (SCD) and fixated interests and repetitive behaviors (FIRB) – for the ASD diagnostic criteria. Building on this proposed DSM 5 model, it would be useful to consider whether empirical data on the SCD and FIRB dimensions can be used within the novel methodological framework of Factor Mixture Modeling (FMM) to stratify children with ASD into more homogeneous subgroups. Methods: The study sample consisted of 391 newly diagnosed children (mean age 38.3 months; 330 males) with ASD. To derive subgroups, data from the Autism Diagnostic Interview‐Revised indexing SCD and FIRB were used in FMM; FMM allows the examination of continuous dimensions and latent classes (i.e., categories) using both factor analysis (FA) and latent class analysis (LCA) as part of a single analytic framework. Results: Competing LCA, FA, and FMM models were fit to the data. On the basis of a set of goodness‐of‐fit criteria, a ‘two‐factor/three‐class’ factor mixture model provided the overall best fit to the data. This model describes ASD using three subgroups/classes (Class 1: 34%, Class 2: 10%, Class 3: 56% of the sample) based on differential severity gradients on the SCD and FIRB symptom dimensions. In addition to having different symptom severity levels, children from these subgroups were diagnosed at different ages and were functioning at different adaptive, language, and cognitive levels. Conclusions: Study findings suggest that the two symptom dimensions of SCD and FIRB proposed for the DSM 5 can be used in FMM to stratify children with ASD empirically into three relatively homogeneous subgroups.     

Circulating tumour markers can define patients with normal colons, benign polyps, and cancers

Background:

Early diagnosis represents the best opportunity for cure of colorectal cancer. Current screening programmes use faecal occult blood testing for screening, which has limited sensitivity and poor specificity.

Methods:

In this study we looked at a series of previously described diagnostic markers utilising circulating free DNA (cfDNA), with a preparation method allowing small DNA fragments to be isolated. The Circulating free DNA was isolated from samples obtained from 85 patients, including 35 patients without endoscopic abnormality, a group of 26 patients with benign colorectal adenomas, and 24 patients with colorectal carcinomas. In each case, polymerase chain reaction (PCR) was performed for Line1 79 bp, Line1 300 bp, Alu 115 bp, Alu 247 bp, and mitochondrial primers. In addition, carcinoembryonic antigen (CEA) was measured by ELISA. Each marker was analysed between normal, polyp, and cancer populations, and the best performing analysed in combination by logistic regression.

Results:

The best model was able to discriminate normal from populations with adenoma or carcinoma using three DNA markers and CEA, showing an area under the receiver operator characteristic (ROC) curve of 0.855 with a positive predictive value of 81.1% for polyps and cancer diagnosis.

Conclusion:

These circulating markers in combination with other markers offer the prospect of a simple blood test as a possible secondary screen for colorectal cancers and polyps in patients with positive faecal occult blood tests.     

Similar developmental trajectories in autism and Asperger syndrome: from early childhood to adolescence

Objective:  The objective of this study was to chart the developmental trajectories of high-functioning children with autism spectrum disorders (ASD) from early childhood to adolescence using the presence and absence of structural language impairment (StrLI) as a way of differentiating autism from Asperger syndrome (AS).
Method:  Sixty-four high-functioning children with ASD were ascertained at 4–6 years of age from several different regional diagnostic and treatment centers. At 6–8 years of age, the ADI-R and the Test of Oral Language Development were used to define an autism group (those with StrLI at 6–8 years of age) and an AS group (those without StrLI). Growth curve analysis was then used to chart the developmental trajectories of these children on measures of autistic symptoms, and adaptive skills in communication, daily living and socialization.
Results:  Differentiating the ASD group in terms of the presence/absence of StrLI provided a better explanation of the variation in growth curves than not differentiating high-functioning ASD children. The two groups had similar developmental trajectories but the group without StrLI (the AS group) was functioning better and had fewer autistic symptoms than the group with StrLI (the autism group) on all measures across time. The differences in outcome could not be explained by non-verbal IQ or change in early language skills.
Conclusion:  Distinguishing between autism and Asperger syndrome based on the presence or absence of StrLI appears to be a clinically useful way of classifying ASD sub-types.     

The Joint Effect of Maternal Marital Status and Type of Household Cooking Fuel on Child Nutritional Status in Sub-Saharan Africa: Analysis of Cross-Sectional Surveys on Children from 31 Countries

The current study sought to investigate the joint effect of maternal marital status and type of household cooking fuel on child nutritional status in sub-Saharan Africa. Data in the children’s files of 31 sub-Saharan African countries were pooled from the Demographic and Health Surveys collected between 2010 and 2019. The outcome variables were three child anthropometrics: stunting (height-for-age z-scores); wasting (weight-for-height z-scores); and underweight (weight-for-age z-scores). The joint effect of maternal marital status and type of household cooking fuel on child nutritional status was examined using multilevel regression models. The results were presented as adjusted odds ratios (aORs) at p < 0.05. The percentages of children who were stunted, wasted and underweight in the 31 countries in sub-Saharan Africa were 31%, 8% and 17%, respectively. On the joint effect of maternal marital status and type of household cooking fuel on stunting, we found that compared to children born to married mothers who used clean household cooking fuel, children born to single mothers who use unclean household cooking fuel, children born to single women who use clean household cooking fuel, and children born to married women who used unclean household cooking were more likely to be stunted. With wasting, children born to single mothers who used unclean household cooking fuel and children born to married women who used unclean household cooking fuel were more likely to be wasted compared to children born to married mothers who used clean household cooking fuel. With underweight, we found that compared to children born to married mothers who used clean household cooking fuel, children born to single mothers who used unclean household cooking fuel, children born to single women who used clean household cooking fuel and children born to married women who used unclean household cooking were more likely to be underweight. It is imperative for the governments of the 31 sub-Saharan African countries to double their efforts to end the use of unclean household cooking fuel. This goal could be achieved by promoting clean household cooking fuel (e.g., electricity, gas, ethanol, solar, etc.) through effective health education, and promotion programmes. The attention of policymakers is drawn to the urgent need for children’s nutritional status policies and programmes (e.g., dietary supplementation, increasing dietary diversity, improving agriculture and food security) to be targeted towards at-risk sub-populations (i.e., single mothered households).     

A comparison of the superficial inferior epigastric artery flap and deep inferior epigastric perforator flap in postmastectomy reconstruction: A cost-effectiveness analysis

PURPOSE:

To perform a cost-effectiveness analysis comparing the superficial inferior epigastric artery (SIEA) and deep inferior epigastric perforator (DIEP) flaps in postmastectomy reconstruction.

METHODS:

A decision analytic model with seven clinically important health outcomes (health states) was used, incorporating the Ontario Ministry of Health’s perspective. Direct medical costs were estimated from a university-based hospital. The utilities of each health state converted into quality-adjusted life years (QALYs) were obtained from previously published data. Health state probabilities were computed from a systematic literature review. Analyses yielded SIEA and DIEP expected costs and QALYs allowing calculation of the incremental cost-utility ratio (ICUR). One-way sensitivity analyses were conducted under five plausible scenarios, assessing result robustness.

RESULTS:

Five SIEA and 27 DIEP studies were identified. The baseline SIEA expected cost was slightly higher than that for the DIEP ($16,107 versus $16,095), with slightly higher QALYs (33.14 years versus 32.98 years), giving an ICUR of $77/QALY. Taking into account conversions from SIEA to DIEP, the ICUR increased to $4,480/QALY. Sensitivity analysis gave ICURs ranging from $2,614/QALY to ‘dominant’, all consistent with the adoption of the SIEA over the DIEP.

CONCLUSION:

The best available evidence suggests the SIEA is a cost-effective procedure. However, given the high SIEA to DIEP conversion rates and small marginal differences in cost and effectiveness, the ICUR may be sensitive to minor changes in costs or QALYs. The ‘truth’ can only be obtained from a randomized, controlled trial comparing both techniques side by side, simultaneously capturing the costs of the competing interventions.     

Differentiating Autism and Asperger Syndrome on the Basis of Language Delay or Impairment

Asperger syndrome (AS) is differentiated from high-functioning autism (HFA) largely on a history of “language delay.” This study examined “specific language impairment” as a predictor of outcome. Language skills of 19 children with AS and 45 with HFA were assessed at 4–6 years of age (Time 1) and 2 years later (Time 2). Children’s symptoms and functional outcome scores were assessed every 2 years (Times 3, 4, and 5) until ages 15–17 years old. Regression analysis revealed that specific language impairment at time 2 more often accounted for the greatest variation in outcome scores in adolescence than the standard diagnosis of AS versus HFA based on history of language delay. Diagnostic implications are discussed.     

Alexithymia in Parents of Children with Autism Spectrum Disorder

Given the recent findings regarding the association between alexithymia and Autism Spectrum Disorder (ASD) and the accumulating evidence for the presence of the Broader Autism Phenotype (BAP) in relatives of individuals with ASD, we further explored the construct of alexithymia in parents of children with ASD as a potential part of the BAP. We hypothesized that (a) parents of children with ASD will demonstrate higher impairment in their emotion processing when compared to controls, and (b) high impairment in emotion processing in parents will be associated with severity of symptoms in children with ASD. Psychometric and diagnostic data were collected on 188 children with a diagnosis of ASD. The Toronto Alexithymia Scale (TAS-20) was completed by 439 parents of children with ASD and a control group of 45 parents of children with Prader Willi syndrome (PW). Results show that ASD parents score higher than controls on the TAS-20 total score. Within the ASD group, children of fathers with high alexithymia score higher on repetitive behaviour symptoms compared to children of fathers with low alexithymia. The alexithymia trait appears to be one of the many building blocks that make up the BAP.