Email updates

Keep up to date with the latest news and content from BMC Pediatrics and BioMed Central.

Open Access Study protocol

Designing and implementing a longitudinal study of children with neurological, genetic or metabolic conditions: charting the territory

Harold Siden13*, Rose Steele2, Rollin Brant3, Susan Cadell4, Betty Davies5, Lynn Straatman1, Kimberley Widger6 and Gail S Andrews3

  • * Corresponding author: Harold Siden hsiden@cw.bc.ca

  • † Equal contributors

Author Affiliations

1 Faculty of Medicine, University of British Columbia, Vancouver, BC, Canada

2 School of Nursing, York University, Toronto, ON, Canada

3 Child & Family Research Institute, University of British Columbia, Vancouver, BC, Canada

4 Faculty of Social Work, Wilfrid Laurier University, Kitchener, ON, Canada

5 School of Nursing, University of Victoria, Victoria, BC, Canada

6 Faculty of Nursing, University of Toronto, Toronto, ON, Canada

For all author emails, please log on.

BMC Pediatrics 2010, 10:67  doi:10.1186/1471-2431-10-67

Published: 20 September 2010

Abstract

Background

Children with progressive metabolic, neurological, or chromosomal conditions and their families anticipate an unknown lifespan, endure unstable and often painful symptoms, and cope with erratic emotional and spiritual crises as the condition progresses along an uncertain trajectory towards death. Much is known about the genetics and pathophysiology of these diseases, but very little has been documented about the trajectory of symptoms for children with these conditions or the associated experience of their families. A longitudinal study design will help to close this gap in knowledge.

Methods/Design

Charting the Territory is a longitudinal descriptive, correlational study currently underway with children 0-19 years who are diagnosed with progressive neurological, metabolic, or chromosomal conditions and their families. The purpose of the study is to determine and document the clinical progression of the condition and the associated bio-psychosocial-spiritual experiences of the parents and siblings age 7-18 years. Approximately 300 families, both newly diagnosed children and those with established conditions, are being recruited in six Canadian cities. Children and their families are being followed for a minimum of 18 months, depending on when they enroll in the study. Family data collection will continue after the child's death if the child dies during the study period. Data collection includes monthly parental assessment of the child's symptoms; an annual functional assessment of the child; and completion of established instruments every 6 months by parents to assess family functioning, marital satisfaction, health status, anxiety, depression, stress, burden, grief, spirituality, and growth, and by siblings to assess coping and health. Impact of participation on parents is assessed after 1 year and at the end of the study. Chart reviews are conducted at enrollment and at the conclusion of the study or at the time of the child's death.

Discussion

Knowledge developed from this study will provide some of the first-ever detailed descriptions of the clinical symptom trajectory of these non-curable progressive conditions and the bio-psychosocial-spiritual aspects for families, from diagnosis through bereavement. Information about developing and implementing this study may be useful to other researchers who are interested in designing a longitudinal study.