Stress in Parents of Children With Genetically Determined Leukoencephalopathies: A Pilot Study.

Genetically determined leukoencephalopathies comprise a group of rare inherited white matter disorders. The majority are progressive diseases resulting in early death. We performed a cross-sectional pilot study including 55 parents from 36 families to assess the level of stress experienced by parents of patients with genetically determined leukoencephalopathies, aged 1 month to 12 years. Thirty-four mothers and 21 fathers completed the Parenting Stress Index-4th Edition. One demographic questionnaire was completed per family. Detailed clinical data was gathered on all patients. Statistical analysis was performed with total stress percentile score as the primary outcome. Mothers and fathers had significantly higher stress levels compared with the normative sample; 20% of parents had high levels of stress whereas 11% had clinically significant levels of stress. Mothers and fathers had comparable total stress percentile scores. We identified pediatric behavioral difficulties and gross motor function to be factors influencing stress in mothers. Our study is the first to examine parental stress in this population and highlights the need for parental support early in the disease course. In this pilot study, we demonstrated that using the Parenting Stress Index-4th Edition to assess stress levels in parents of patients with genetically determined leukoencephalopathies is feasible, leads to valuable and actionable results, and should be used in larger, prospective studies.

Silicon carbide pneumoconiosis: a radiographic assessment.

Interstitial lung disease and airflow limitation have been reported in silicon carbide workers but it is uncertain whether these recent observations reflect past working conditions alone (30 years ago) or both past and present conditions even if the latter have improved. To investigate this, we analyzed pairs of chest radiographs obtained in 1977 and 1984 in 128 silicon carbide plant workers. Three indices, related to density of small opacities, profusion of opacities, and presence of pleural plaques, were obtained. Student t-tests on results obtained in each time period showed the mean density (1.02 in 1977 and 0.56 in 1984) and profusion (4.27 in 1977 and 2.13 in 1984) indices to be significantly different. However, the pleural plaque index was not different in 1984 (0.34) as compared with 1977 (0.32). There was no deterioration of the radiographic image even in groups with density ratios equal or greater than 1/1. We concluded that the actual environmental working conditions did not influence the integrity of the lung radiographic image in these workers within a time span of 6 or 7 years.