Bone health in childhood cancer: review of the literature and recommendations for the management of bone health in childhood cancer survivors

Marcucci, Gemma; Beltrami, Giovanni; Tamburini, Angela; Body, Jean‐Jacques; Confavreux, C; Hadji, Peyman; Holzer, Gerold; Kendler, David L.; Napoli, Nicola; Pierroz, Dominique D.; Rizzoli, René; Brandi, Maria Luisa

doi:10.1093/annonc/mdz120

Cited by 49 publications

(65 citation statements)

References 139 publications

(204 reference statements)

Supporting

Mentioning

Contrasting

Order By: Relevance

“…The program is based on a Dutch population study (26) and calculates pubertal SDS for breast development, pubic hair stage and testicular volume. While the calculation is based on the Tanner stages for breast development and pubic hair stage, the application allows only discrete measures for testicular volume (i.e., 2,3,4,8,12,16,20, and 25 ml). Therefore, volume measurements that fell in between these values, which were determined using an orchidometer, were rounded to the closest possible measurement (e.g., a measurement of 6 ml was rounded to 8 ml for this study).…”

Section: Clinical Parameters and Questionnairementioning

confidence: 99%

See 1 more Smart Citation

Prevalence of Osteopathologies in Children and Adolescents After Diagnosis of Acute Lymphoblastic Leukemia

et al. 2020

View full text Add to dashboard Cite

Background: Impaired bone health is a late effect of childhood malignancies which can be difficult to detect in juvenile survivors. It may, however, lead to compromised quality of life, or even permanent disability later in life due to osteoporosis, pain or fractures if left untreated. Acute lymphoblastic leukemia (ALL) is the most frequent childhood malignancy with an over 85% five-year survival. ALL and its treatment cause bone alterations in adults, but little information on the bone health status in juvenile survivors is available. Objective: To report data on skeletal late effects in juvenile survivors of childhood ALL based on a comprehensive assessment of bone health and to assess the influence of a vitamin D deficiency on bone health in this cohort. Methods: In a single center cross sectional study 128 pediatric patients (11.9 ± 4.76 years) with a mean follow up of 5.88 ± 3.75 years after diagnosis of ALL were recruited. The bone health status of the survivors was assessed based on clinical examination, review of medical records, biochemical and radiographic analyses, by clinical experts. A score which utilized 8 different parameters was formed and used to assess the effect of a vitamin D deficiency on bone health. Results: In this cohort, 18% of survivors displayed overt osteopathologies as defined by clinical expert assessment. Impaired bone health, defined by at least one pathological screening parameter, was detected in 77%. Despite recommendations for adequate vitamin D supplementation, 15% displayed a vitamin D deficiency associated with hyperparathyroidism. The applied score identified survivors with osteopathologies with high sensitivity and specificity. The median score did not differ between patients without and with severe vitamin D deficiency. Conclusion: Our findings suggest that impaired bone health and osteopathologies are common skeletal late effects following treatment of childhood ALL. Major contributing factors are BMT, irradiation and older age at diagnosis. Vitamin D deficiency likely Schündeln et al. Osteopathologies After Acute Lymphoblastic Leukemia accounts for hyperparathyroidism in some patients but does not seem to further affect bone health in this cohort. Survivors of ALL need thorough surveillance to investigate bone health, since bone morbidity is common and still poorly understood. Early detection and appropriate intervention may improve bone health.

show abstract

Section: Clinical Parameters and Questionnairementioning

confidence: 99%

“…Endocrine late effects in general play a major role in survivorship morbidity (4)(5)(6)(7). Bone health has been shown to be impaired in a significant number of children and adults surviving pediatric malignancies (8). Mostoufi-Moab and Halton review the subject for patients with childhood ALL (9).…”

Section: Introductionmentioning

confidence: 99%

Prevalence of Osteopathologies in Children and Adolescents After Diagnosis of Acute Lymphoblastic Leukemia

et al. 2020

View full text Add to dashboard Cite

show abstract

“…Patients with ALL are extensively treated with chemotherapy, including high-dose methotrexate, in addition to high cumulative doses of corticosteroids. Patients with Ewing's sarcoma and osteosarcoma are treated with intense chemotherapy and surgery, with or without radiotherapy [25].…”

Section: Discussionmentioning

confidence: 99%

Low bone mineral density is associated with hypogonadism and cranial irradiation in male childhood cancer survivors

et al. 2020

View full text Add to dashboard Cite

We investigated if bone mineral density was related to testosterone deficiency and/or previous cancer treatment in men who were childhood cancer survivors. Men with untreated testosterone deficiency or previous treatment with cranial irradiation were at increased risk of impaired bone health. Prevention of osteoporosis should be considered in their follow-up. Introduction Childhood cancer survivors (CCS) are at increased risk of hypogonadism. Reduced bone mineral density (BMD) has been reported in CCS but it is unclear whether this is due to hypogonadism or a direct effect of cancer therapy. This study investigated BMD in CCS, and association with hypogonadism, previous treatment and cancer type. Methods Investigation of 125 CCS (median age 33.7 at inclusion; 9.6 at diagnosis) and 125 age-matched population controls. Serum testosterone and luteinizing hormone were assayed and BMD at total hip and lumbar spine L1-L4 measured. The mean difference in BMD (g/cm 2 ; 95% CI) between CCS and controls was analysed. Odds ratios (OR; 95% CI) for low BMD were also calculated. Results Overall, BMD in the CCS cohort did not significantly differ from controls. However, compared with eugonadal CCS, the CCS with untreated hypogonadism had lower BMD at the hip (mean difference − 0.139 (− 0.210; − 0.067); p < 0.001) and spine (− 0.102 (− 0.174; − 0.030); p = 0.006). They also had a higher risk of low hip BMD (OR 4.1 (1.3; 14); p = 0.018). CCS treated with cranial irradiation also had lower BMD (hip − 0.076 (− 0.133; − 0.019); p = 0.009; spine − 0.071 (− 0.124; − 0.018); p = 0.009) compared with controls. The latter associations remained statistically significant after adjustment for hypogonadism. Conclusions CCS with hypogonadism or previously treated with cranial irradiation are at increased risk of impaired bone health. Prevention of osteoporosis should be considered as an important part in future follow-up of these men.

show abstract

“…9 Hospital Infantil Universitario Niño Jesús, Madrid, Spain. 10 16 Biohealth Research Institute in Granada (ibs.GRANADA), E-18012 Granada, Spain. 17 Department of Physiotherapy, University of Granada, E-18016 Granada, Spain.…”

Section: Acknowledgmentsmentioning

confidence: 99%

“…Paediatric cancer is a life-threatening condition that also occurs during the period of bone development and strengthening. Gonadal failure following to pelvic radiation or gonadotoxic chemotherapy and hypothalamic pituitary dysfunction by means of cranial radiation can adversely affect areal bone mineral density (aBMD), increasing osteoporosis risk later in life [9,10]. In addition, direct radiation to bone not only causes hypovascularity but has a direct cytotoxic effect on the epiphyseal chondrocytes [11].…”

Section: Introductionmentioning

confidence: 99%

The effect of an online exercise programme on bone health in paediatric cancer survivors (iBoneFIT): study protocol of a multi-centre randomized controlled trial

et al. 2020

View full text Add to dashboard Cite

Background New approaches on paediatric cancer treatment aim to maintain long-term health. As a result of radiotherapy, chemotherapy or surgery, paediatric cancer survivors tend to suffer from any chronic health condition. Endocrine dysfunction represents one of the most common issues and affects bone health. Exercise is key for bone mass accrual during growth, specifically plyometric jump training. The iBoneFIT study will investigate the effect of a 9-month online exercise programme on bone health in paediatric cancer survivors. This study will also examine the effect of the intervention on body composition, physical fitness, physical activity, calcium intake, vitamin D, blood samples quality of life and mental health. Methods A minimum of 116 participants aged 6 to 18 years will be randomized into an intervention (n = 58) or control group (n = 58). The intervention group will receive an online exercise programme and diet counselling on calcium and vitamin D. In addition, five behaviour change techniques and a gamification design will be implemented in order to increase the interest of this non-game programme. The control group will only receive diet counselling. Participants will be assessed on 3 occasions: 1) at baseline; 2) after the 9 months of the intervention; 3) 4 months following the intervention. The primary outcome will be determined by dual energy X-ray absorptiometry (DXA) and the hip structural analysis, trabecular bone score and 3D-DXA softwares. Secondary outcomes will include anthropometry, body composition, physical fitness, physical activity, calcium and vitamin D intake, blood samples, quality of life and mental health. Discussion Whether a simple, feasible and short in duration exercise programme can improve bone health has not been examined in paediatric cancer survivors. This article describes the design, rationale and methods of a study intended to test the effect of a rigorous online exercise programme on bone health in paediatric cancer survivors. If successful, the iBoneFIT study will contribute to decrease chronic health conditions in this population and will have a positive impact in the society. Trial registration Prospectively registered in isrctn.com: isrctn61195625. Registered 2 April 2020.

show abstract

Bone health in childhood cancer: review of the literature and recommendations for the management of bone health in childhood cancer survivors

Cited by 49 publications

References 139 publications

Prevalence of Osteopathologies in Children and Adolescents After Diagnosis of Acute Lymphoblastic Leukemia

Prevalence of Osteopathologies in Children and Adolescents After Diagnosis of Acute Lymphoblastic Leukemia

Low bone mineral density is associated with hypogonadism and cranial irradiation in male childhood cancer survivors

The effect of an online exercise programme on bone health in paediatric cancer survivors (iBoneFIT): study protocol of a multi-centre randomized controlled trial

Contact Info

Product

Resources

About