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Pain and quality of life of children and adolescents with osteogenesis imperfecta over a bisphosphonate treatment cycle

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Abstract

The objective was to describe the pain and quality of life among children and adolescents with any osteogenesis imperfecta (OI) type over one intravenous bisphosphonate treatment cycle from a child and parental perspective. A prospective, observational study was conducted, where children and adolescents evaluated their pain intensity, location, and quality, as well as quality of life before, 1 week after treatment, and 6 months later. Quality of life was also evaluated from the parental perspective at the same three time points. Thirty-three child/parent dyads participated. The results showed that pain intensity on the 0–10 self-report scale after the Zoledronate infusion (median = 0, range = 0–6) was not different from pre (median = 2, range = 0–10) and 6-months post-scores (median = 2, range = 0–8) (p = 0.170). Children and adolescents with OI reported experiencing pain mainly in the ankles and the anterior and posterior shoulders. They selected evaluative pain descriptors such as uncomfortable (n = 16, 48%) and annoying (n = 13, 39%). Children and adolescents’ functioning and quality of life did not change significantly across the bisphosphonate treatment cycle (p = 0.326), parents perceived an improvement immediately after the treatment compared to before (p = 0.016).

Conclusion: Children and adolescents with OI experience mild, yet complex pain localized across several body areas. There is little fluctuation in the pain intensity and functioning of children with OI undergoing bisphosphonate treatment.

What is Known:

• Acute and chronic musculoskeletal pain remains a major issue in OI.

• Pain has a negative impact on quality of life.

What is New:

• New and unpublished methods and findings describing the pain and quality of life of children and adolescents with OI over one intravenous bisphosphonate treatment cycle from a child- and parental-proxy perspective.

• Children and adolescents with OI experience pain intensity that is mild, yet complex in quality and localized across several body areas.

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Abbreviations

APPT:

Adolescent Pediatric Pain Tool

FPS-R:

Faces Pain Scale–Revised

IASP:

International Association for the Study of Pain

IV:

Intravenous

OI:

Osteogenesis imperfecta

OTC:

Over the counter

PedsQL4:

Pediatric Quality of Life 4.0

QoL:

Quality of life

RCTs:

Randomized controlled trials

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Funding

Argerie Tsimicalis received research grants from Quebec Nursing Intervention Research Network (RRISIQ), Newton Foundation, Scotiabank®, and Tunis Temple.

Author information

Authors and Affiliations

  1. Ingram School of Nursing, McGill University, Montreal, Quebec, Canada

    Argerie Tsimicalis, Madalina Boitor, Jaimie Isabel Carrier & Tracy Ngheim

  2. McGill University Health Centre, Montreal, Quebec, Canada

    Argerie Tsimicalis & Catherine E. Ferland

  3. Clinical Research, Shriners Hospitals for Children®-Canada, 1003 Decarie Boulevard, Montreal, Quebec, Canada

    Argerie Tsimicalis, Catherine E. Ferland, Frank Rauch, Jaimie Isabel Carrier, Tracy Ngheim & Claudette Bilodeau

  4. Department of Anesthesia, McGill University, Montreal, Quebec, Canada

    Catherine E. Ferland

  5. Department of Pediatrics, McGill University, Montreal, Quebec, Canada

    Frank Rauch

  6. Faculty of Nursing, University of Montreal, Montreal, Quebec, Canada

    Sylvie Le May

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  1. Argerie Tsimicalis
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  2. Madalina Boitor
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  3. Catherine E. Ferland
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  4. Frank Rauch
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  7. Tracy Ngheim
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  8. Claudette Bilodeau
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Contributions

Madalina Boitor, a doctoral trainee, analyzed the data, interpreted the findings, and wrote the manuscript. Catherine Ferland designed the study as part of her postdoctoral training, reviewed the findings, and edited and reviewed the manuscript. Frank Rauch designed the study, was responsible for ethics, oversaw recruitment and data collection, reviewed the findings, and reviewed the manuscript. Sylvie Le May reviewed the findings and manuscript; offering her expertise in pain research with children. Jaimie Isabel Carrier, an undergraduate research trainee, recruited and collected data, participated in data analysis, reviewed the manuscript. Tracy Nghiem, a graduate research trainee, reviewed the findings, edited and reviewed the manuscript; offering her training expertise in pain in OI. Claudette Bilodeau recruited and collected data, supervised undergraduate trainee, participated in data analysis, reviewed the manuscript. Argerie Tsimicalis edited the study design, was responsible for ethics and budget, oversaw recruitment, data collection, and analysis, supervised staff and trainees, participated in data analysis and interpretation, and wrote, edited, and reviewed the manuscript.

Corresponding author

Correspondence to Argerie Tsimicalis.

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The authors declare that they have no conflict of interest.

Informed consent

All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation (institutional and national) and with the Helsinki Declaration of 1975, as revised in 2008 (5). Informed consent was obtained from all patients for being included in the study.

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Communicated by Peter de Winter

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Tsimicalis, A., Boitor, M., Ferland, C.E. et al. Pain and quality of life of children and adolescents with osteogenesis imperfecta over a bisphosphonate treatment cycle. Eur J Pediatr 177, 891–902 (2018). https://doi.org/10.1007/s00431-018-3127-9

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