Do Bisphosphonates Alleviate Pain in Children? A Systematic Review

Celin, Mercedes Rodríguez; Simon, Jackeline C.; Krzak, Joseph J.; Fial, Alissa; Kruger, Karen M.; Smith, Peter A.; Harris, Gerald F.

doi:10.1007/s11914-020-00621-3

Cited by 14 publications

(4 citation statements)

References 117 publications

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“…Existing reviews of OI literature do not provide a systematic overview of the scope of published records, but rather follow a narrative design [ 61 – 63 ], or aim to answer specific research questions [ 9 , 11 , 18 – 21 , 64 – 72 ]. Therefore, the aim of this review was to determine the breadth of literature available and provide a snapshot of the evidence it covers.…”

Section: Discussionmentioning

confidence: 99%

The patient clinical journey and socioeconomic impact of osteogenesis imperfecta: a systematic scoping review

Rapoport¹,

Bober

Raggio

et al. 2023

Orphanet J Rare Dis

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Background Osteogenesis imperfecta (OI) is a rare heritable connective tissue disorder primarily characterised by skeletal deformity and fragility, and an array of secondary features. The purpose of this review was to capture and quantify the published evidence relating specifically to the clinical, humanistic, and economic impact of OI on individuals, their families, and wider society. Methods A systematic scoping review of 11 databases (MEDLINE, MEDLINE in-progress, EMBASE, CENTRAL, PsycINFO, NHS EED, CEA Registry, PEDE, ScHARRHUd, Orphanet and Google Scholar), supplemented by hand searches of grey literature, was conducted to identify OI literature published 1st January 1995–18th December 2021. Searches were restricted to English language but without geographical limitations. The quality of included records was assessed using the AGREE II checklist and an adapted version of the JBI cross-sectional study checklist. Results Of the identified 7,850 records, 271 records of 245 unique studies met the inclusion criteria; overall, 168 included records examined clinical aspects of OI, 67 provided humanistic data, 6 reported on the economic impact of OI, and 30 provided data on mixed outcomes. Bone conditions, anthropometric measurements, oral conditions, diagnostic techniques, use of pharmacotherapy, and physical functioning of adults and children with OI were well described. However, few records included current care practice, diagnosis and monitoring, interactions with the healthcare system, or transition of care across life stages. Limited data on wider health concerns beyond bone health, how these concerns may impact health-related quality of life, in particular that of adult men and other family members, were identified. Few records described fatigue in children or adults. Markedly few records provided data on the socioeconomic impact of OI on patients and their caregivers, and associated costs to healthcare systems, and wider society. Most included records had qualitative limitations. Conclusion Despite the rarity of OI, the volume of recently published literature highlights the breadth of interest in the OI field from the research community. However, significant data gaps describing the experience of OI for individuals, their families, and wider society warrant further research to capture and quantify the full impact of OI.

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Section: Discussionmentioning

confidence: 99%

The patient clinical journey and socioeconomic impact of osteogenesis imperfecta: a systematic scoping review

Rapoport¹,

Bober

Raggio

et al. 2023

Orphanet J Rare Dis

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“…There is still no cure for OI but treatment with intravenous bisphosphonate has been beneficial [ 3 ]. Bisphosphonates have been shown to have several positive effects, and one of them is to alleviate pain [ 4 ]. In addition, children may receive orthopaedic surgery with rodding, and physiotherapy aiming to support motor development and physical activity, increase opportunities for participation in everyday life, improve muscle strength, reduce fractures and chronic pain [ 5 ].…”

Section: Introductionmentioning

confidence: 99%

Exploring pain interference and self-perceived health status in children with osteogenesis imperfecta - a cross-sectional study

Provenzano

Albrecht

Löwing

2022

BMC Musculoskelet Disord

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Background Chronic pain may affect and interfere in children’s everyday life and can be present in children with Osteogenesis Imperfecta (OI). However, the knowledge is still sparse to what extent pain is present, how pain interfere in children’s everyday life and affect their self-perceived health status. The purpose of the study was therefore to explore presence of chronic pain, pain interference in daily life, and self-perceived health status in children with OI. Methods Children with OI, aged 6–18 years, were recruited consecutively to this cross-sectional study. Participants answered a standardised interview including five pre-structured questions, and the Numeric Pain Rating Scale (NPRS), the Pain Interference Index, and a questionnaire concerning self-perceived health status the Patient Reported Outcomes Measurement Information System Pediatric-25 Profile v1.1 (PROMIS-25). Results Twenty-eight children (median: 11 years, IQR 6) with OI type I, III, or IV participated. Pain was present in 27 of 28 children and interfered in their everyday life regardless of OI-type, sex, and age. The median NPRS for average pain intensity was 4 (IQR 2), the median for pain frequency was 2–3 times/week, and the median frequency of school absence due to pain was 2–3 times per month. The most common pain locations were back and feet. Pain in the feet was more frequently reported in children with type I (p = 0.032), and pain in the hip was more often reported in children ≥13 years (p = 0.011). The children were asked what they thought to be the cause of pain and the most frequent response was “walking long distances”. Self-perceived health status for mobility was lower than the general population, and lowest for children with type III (p = 0.016). Pain interference was associated with children’s self-perceived health status (rs = 0.84, p < 0.001). Conclusion Almost all children experienced pain, which interfered in children’s everyday lives, affected participation in various activities and was associated with reduced self-perceived health status. If children avoid physical activities because of pain, it might cause a vicious circle of inactivity, which further decreases bone density and increase the risk of fractures. The results emphasize the importance to offer adequate pain reducing interventions.

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“…This prompt clinical improvement has been docu-mented in some case reports, including a 45-year-old woman who had rapid disappearance of local pain after pamidronate monotherapy [24] and a child who described immediate improvement of local pain after intravenous neridronate added to the previously prescribed alpha-2b interferon [2]. A systematic review analyzing the effect of bisphosphonates in treating bone pain related to various pathologies documented that 20 of 24 studies reported a positive effect of bisphosphonates in relieving pain [32]. However, the mechanism by which bisphosphonates reduce pain remains unclear.…”

Section: Discussionmentioning

confidence: 88%

Case Report and Literature Review: Bisphosphonate, Sirolimus, and Atenolol Treatment in a 4-Year-Old Child Diagnosed with Gorham–Stout Disease

Park,

Yoo,

Ahn

2023

Pharmaceuticals

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We report a 4-year-old with Gorham–Stout disease (GSD) who was treated with a combination of bisphosphonate, sirolimus, and atenolol. A previously healthy 4-year-old girl presented with back pain after falling on her back 2 months prior. Thoracolumbar spine X-ray revealed diffuse compression spinal fractures in T9-L2. Magnetic resonance imaging (MRI) confirmed multiple compression fractures at T9-L5 and revealed a paraspinal mass along the T1-L1 level. Based on clinical, radiological, and histopathological findings, Gorham–Stout disease was diagnosed. Treatment with sirolimus (0.5 mg twice daily, 1.6 mg/m2) was initiated and intravenous bisphosphonate (pamidronate, 1 mg/kg for 3 days, total 3 mg/kg every 4 months) was added for back pain; she had immediate improvement in back pain. After 9 months with this treatment, she had a mild increase in paraspinal lymphangiomatosis and aggravation in T9-L5 compression fractures; atenolol was administered. The patient underwent 11 months of combination treatment with bisphosphonate, sirolimus, and atenolol, and MRI showed mild degree of reduction in the paraspinal lesions at L1-L5. The patient is currently in stable condition with no back pain or side effects. The triple combination treatment with bisphosphonate, sirolimus, and atenolol may be helpful in stabilizing the disease course of GSD.

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Do Bisphosphonates Alleviate Pain in Children? A Systematic Review

Cited by 14 publications

References 117 publications

The patient clinical journey and socioeconomic impact of osteogenesis imperfecta: a systematic scoping review

The patient clinical journey and socioeconomic impact of osteogenesis imperfecta: a systematic scoping review

Exploring pain interference and self-perceived health status in children with osteogenesis imperfecta - a cross-sectional study

Case Report and Literature Review: Bisphosphonate, Sirolimus, and Atenolol Treatment in a 4-Year-Old Child Diagnosed with Gorham–Stout Disease

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