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Despite well-defined criteria for radiographic diagnosis of atypical femur fractures (AFFs), missed and delayed diagnosis is common. An AFF diagnostic software could provide timely AFF detection to prevent progression of incomplete or development of contralateral AFFs. In this study, we investigated the ability for an artificial intelligence (AI)-based application, using deep learning models (DLMs), particularly convolutional neural networks (CNNs), to detect AFFs from femoral radiographs. A labelled Australian dataset of pre-operative complete AFF (cAFF), incomplete AFF (iAFF), typical femoral shaft fracture (TFF), and non-fractured femoral (NFF) X-ray images in anterior-posterior view were used for training (N = 213, 49, 394, 1359, respectively). An AFFnet model was developed using a pretrained (ImageNet dataset) ResNet-50 backbone, and a novel Box Attention Guide (BAG) module to guide the model's scanning patterns to enhance its learning. All images were used to train and internally test the model using a 5-fold cross validation approach, and further validated by an external dataset. External validation of the model's performance was conducted on a Sweden dataset comprising 733 TFF and 290 AFF images. Precision, sensitivity, specificity, F1-score and AUC were measured and compared between AFFnet and a global approach with ResNet-50. Excellent diagnostic performance was recorded in both models (all AUC >0.97), however AFFnet recorded lower number of prediction errors, and improved sensitivity, F1-score and precision compared to ResNet-50 in both internal and external testing. Sensitivity in the detection of iAFF was higher for AFFnet than ResNet-50 (82 % vs 56 %). In conclusion, AFFnet achieved excellent diagnostic performance on internal and external validation, which was superior to a pre-existing model. Accurate AI-based AFF diagnostic software has the potential to improve AFF diagnosis, reduce radiologist error, and allow urgent intervention, thus improving pa

OBJECTIVE: To formulate clinical consensus recommendations on the presentation, assessment, and management of primary hyperparathyroidism (PHPT) in adults. METHODS: Representatives from relevant Australian and New Zealand Societies used a systematic approach for adaptation of guidelines (ADAPTE) to derive an evidence-informed position statement addressing nine key questions. RESULTS: PHPT is a biochemical diagnosis. Serum calcium should be measured in patients with suggestive symptoms, reduced bone mineral density or minimal trauma fractures, and in those with renal stones. Other indications are detailed in the manuscript. In patients with hypercalcaemia, intact parathyroid hormone, 25-hydroxy vitamin D, phosphate, and renal function should be measured. In established PHPT, assessment of bone mineral density, vertebral fractures, urinary tract calculi/nephrocalcinosis and quantification of urinary calcium excretion is warranted. Parathyroidectomy is the only definitive treatment and is warranted for all symptomatic patients and should be considered for asymptomatic patients without contraindications to surgery and with >10 years life expectancy. In patients who do not undergo surgery, we recommend annual evaluation for disease progression. Where the diagnosis is not clear or the risk-benefit ratio is not obvious, multidisciplinary discussion and formulation of a consensus management plan is appropriate. Genetic testing for familial hyperparathyroidism is recommended in selected patients. CONCLUSIONS: These clinical consensus recommendations were developed to provide clinicians with contemporary guidance on the assessment and management of PHPT in adults. It is anticipated that improved health outcomes for individuals and the population will be achieved at a decreased cost to the community.

BACKGROUND: Low bone density (BMD) and fractures commonly affect women with premature ovarian insufficiency (POI). However, bone microarchitecture and body composition data are lacking. OBJECTIVE: To assess and characterise musculoskeletal phenotype and effects of oestrogen replacement therapy (ERT) in women with POI. METHOD: Cross-sectional and longitudinal studies of 60 normal karyotype women with POI, aged 20-40 years, from 2005-2018. Dual x-ray absorptiometry (DXA)-derived spinal (LS) and femoral neck (FN) BMD, trabecular bone score (TBS), appendicular lean mass (ALM), total fat mass (TFM), and fracture prevalence were compared with 60 age-, and BMI-matched population-based controls. Longitudinal changes in bone and body composition variables and ERT effects were analysed using linear mixed models over a median duration of 6 years. RESULTS: Women with POI were subdivided into spontaneous (s)-POI (n=25) and iatrogenic (i)-POI (n=35). Median(range) age of POI diagnosis was 34 (10-40) years with baseline DXA performed at median 1(0-13) year post-diagnosis. ERT was used by 82% women (similar for both POI groups). FN-BMD were lowest in s-POI (p<0.002). Low TBS was more common in s-POI [(44%), p=0.03], versus other groups. LS-BMD and ALM were lower in both s-POI and i-POI groups than controls (p<0.05). Fracture prevalence was not significantly different: 20% (s-POI), 17% (i-POI), and 8% (controls) (p=0.26). Longitudinal analysis of 23 POI women showed regular ERT was associated with ALM increment of 127.05 g/year (p<0.001) and protected against bone loss. However, ERT interruption was associated with annual reductions in FN BMD and TBS of 0.020g/cm2 and 0.0070 (p<0.05), respectively. CONCLUSION: Deficits in BMD, trabecular microarchitecture, and lean mass were present in women with POI. However, regular ERT protected against declines in bone variables, with an increase in ALM. Assessment of skeletal and muscle health, and advocating ERT adherence, is essential in POI

Objective: Glucocorticoids constitute a considerable risk for developing osteoporosis in both younger and older adults. However, currently available bone imaging modalities and fracture-risk assessment tools do not adequately capture the dramatic changes in bone microarchitecture, heterogeneity of glucocorticoid exposure, the impact of chronic disease and other osteoporosis risk factors on the assessment of osteoporosis in these individuals. Design(s): A narrative review is presented, following a systematic search of the literature from 2000 to 2021. Result(s): Our current appreciation of glucocorticoid-induced osteoporosis (GIO) is focused on older populations, with limited evidence to guide the investigation, risk assessment and treatment in premenopausal women and men less than 50 years. The impact of the underlying chronic disease on secondary osteoporosis in these younger adults is also poorly understood. Conclusion(s): Through this narrative review, we provide a comprehensive overview of and recommendations for optimising the management of this common cause of secondary osteoporosis younger and older adults.Copyright © 2021 John Wiley & Sons Ltd.

Premature ovarian insufficiency (POI), defined as a loss of ovarian function before the age of 40 years, is a life-changing diagnosis that has numerous long-term consequences. Musculoskeletal complications, including osteoporosis and fractures, are a key concern for women with POI. The risk of bone loss is influenced by the underlying etiology of POI, and the degree and duration of estrogen deficiency. A decline in muscle mass as a result of estrogen and androgen deficiency may contribute to skeletal fragility, but has not been examined in women with POI. This article aims to review musculoskeletal health in POI; summarize the traditional and novel modalities available to screen for skeletal fragility and muscle dysfunction; and provide updated evidence for available management strategies.Copyright © 2021 International Menopause Society.

Accelerated bone loss and muscle decline coexist in women with premature ovarian insufficiency (POI), but there are significant gaps in our understanding of musculoskeletal health in POI. This article is the first of a two-part review which describes estrogen signaling in muscle and its role in musculoskeletal health and disease. Current evidence regarding the utility of available diagnostic tests and therapeutic options is also discussed. A literature review from January 2000 to March 2020 was conducted to identify relevant studies. Women with POI experience significant deterioration in musculoskeletal health due to the loss of protective effects of estrogen. In addition to bone loss, muscle decay and dysfunction is now increasingly recognized. Nevertheless, there is a paucity of validated tools to assess muscle parameters. There is a growing need to acknowledge bone-muscle codependence to design new therapies which target both muscle and bone, resulting in improved physical performance and reduced morbidity and mortality. More high-quality research and international collaborations are needed to address the deficiencies in our understanding and management of musculoskeletal health in women with POI.Copyright © 2021 EDP Sciences. All rights reserved.

Accelerated bone loss and muscle decline coexist in women with premature ovarian insufficiency (POI), but there are significant gaps in our understanding of musculoskeletal health in POI. This article is the first of a two-part review which describes estrogen signaling in muscle and its role in musculoskeletal health and disease. Current evidence regarding the utility of available diagnostic tests and therapeutic options is also discussed. A literature review from January 2000 to March 2020 was conducted to identify relevant studies. Women with POI experience significant deterioration in musculoskeletal health due to the loss of protective effects of estrogen. In addition to bone loss, muscle decay and dysfunction is now increasingly recognized. Nevertheless, there is a paucity of validated tools to assess muscle parameters. There is a growing need to acknowledge bone-muscle codependence to design new therapies which target both muscle and bone, resulting in improved physical performance and reduced morbidity and mortality. More high-quality research and international collaborations are needed to address the deficiencies in our understanding and management of musculoskeletal health in women with POI.Copyright © 2021 Georg Thieme Verlag. All rights reserved.

Summary: This study evaluated mediators of fracture risk in postmenopausal women with type 1 (T1D) and type 2 diabetes (T2D), over a 15-year follow-up period. This study provides evidence that the increased fracture risk in women with T1D or T2D is partially explained by falls. Furthermore, a shorter reproductive lifespan in women with T1D contributes modestly to fracture risk in this cohort. Purpose(s): Skeletal fragility is associated with diabetes mellitus, while limited estrogen exposure during the reproductive years also predisposes to lower bone mass and higher fracture risk. We aimed to determine osteoporosis diagnosis, fall and fracture rates in women with type 1 (T1D) and type 2 (T2D) diabetes mellitus, and explore mediators of the diabetes-fracture relationship. Method(s): Prospective observational data drawn from the Australian Longitudinal Study in Women's Health (ALSWH) from 1996 to 2010. Women were randomly selected from the national health insurance database. Standardized data collection occurred at six survey time points, with main outcome measures being self-reported osteoporosis, incident fracture, falls, and reproductive lifespan. Mediation analyses were performed to elucidate relevant intermediaries in the diabetes-fracture relationship. Result(s): Exactly 11,313 women were included at baseline (T1D, n = 107; T2D, n = 333; controls, n = 10,873). A total of 885 new cases of osteoporosis and 1099 incident fractures were reported over 15 years. Women with T1D or T2D reported more falls and fall-related injuries; additionally, women with T1D had a shorter reproductive lifespan. While fracture risk was increased in women with diabetes (T1D: OR 2.28, 95% CI 1.53-3.40; T2D: OR 2.40, 95% CI 1.90-3.03), compared with controls, adjustment for falls attenuated the risk of fracture by 10% and 6% in T1D and T2D, respectively. In women with T1D, reproductive lifespan modestly attenuated fracture risk by 4%. Conclusion(s): Women with T1D and T2D have an increas

Introduction: Bone mineral density, measured by dual X-ray absorptiometry (DXA), is the gold standard for diagnosis of osteoporosis. The utility of DXA relies on the accuracy of scan acquisition, interpretation of data, and the adequacy of reports. The International Society for Clinical Densitometry (ISCD) has published guidelines regarding minimum reporting guidelines. This study assessed whether DXA reports for patients receiving care at an academic teaching hospital adhere to these reporting standards, and determine whether differences exist depending on patient factors and the imaging service. Method(s): Patients aged >=18 years, receiving care at specialist outpatient clinics between January 1, 2018 and December 31, 2019, with a DXA report available, were eligible for inclusion. DXA reports were manually reviewed for adherence to ISCD guidelines, with each criterion scored as one point, giving a total score of 14 for baseline DXA scans and 18 for repeat DXA scans. The score was then converted to a percentage. Result(s): Of 459 DXA scans included, 214 were performed internally at our hospital and 245 performed at 23 external imaging services. Mean (SD) patient age was 60 (16.3) years, and 75.8% were female. The overall median (IQR) report score was 57.1% (42.9, 82.4). ISCD criteria with the lowest scores were recommendation and timing of future DXA scans (included in 1.1% of reports) and investigation for secondary causes of osteoporosis (included in 1.2% of reports). Reports performed internally had significantly higher scores than those performed externally, after adjusting for age, sex, indication, and type of scan (incidence rate ratio 1.83, 95% confidence interval 1.77, 1.89). Baseline DXA reports had slightly higher scores than repeat DXA scans, and, among external imaging services, rural services had higher scores than metropolitan services. Conclusion(s): This study, the largest comprehensive evaluation of DXA reports, highlights significant deficiencies

BACKGROUND: Patients with end-stage kidney disease (ESKD) have higher fracture rates and post-fracture mortality than the general population, but bone mineral density by dual-energy X-ray absorptiometry (DXA) is less predictive of fracture in this patient group. Bone biopsy and high-resolution imaging indicate that cortical thickness (CT) is reduced and cortical porosity is increased in ESKD. The aim of this study was to assess cortical parameters using DXA in patients with ESKD. It was hypothesized that these parameters would show deterioration and be associated with fracture. METHOD(S): Using advanced hip analysis, normal age-related ranges were determined from 752 female and 861 male femur scans and were compared with scans of 226 patients with ESKD at the time of transplantation. RESULT(S): Compared with controls, female patients had lower mean+/-SD CT (mms) at the femoral neck (FN) (2.59+/-1.42 versus 5.23+/-1.85), calcar (3.46+/-1.07 versus 5.09+/-1.30) and shaft (4.42+/-1.21 versus 7.44+/-2.07; P<0.001 for each), and buckling ratios were higher (8.21+/-4.6 versus 3.63+/-1.42; P<0.001), indicating greater FN instability. All findings were similar for men. Prevalent fracture was documented in 28.8% of patients; 12.4% vertebral only, 8.4% non-vertebral only and 8% vertebral plus non-vertebral. In adjusted models, each 1 SD reduction in FN CT and increase in the buckling ratio was associated with a respective 1.73 (1.22-2.46)- and 1.82 (1.49-2.86)-fold increase in the risk of prevalent vertebral fracture. CONCLUSION(S): In patients with ESKD, DXA-derived cortical parameters are markedly abnormal compared with age- and sex-matched controls. These parameters should be assessed for incident fracture prediction and targeting treatment.Copyright © The Author(s) 2019. Published by Oxford University Press on behalf of ERA-EDTA. All rights reserved.

Although hypercalciuria, osteoporosis, fractures and nephrolithiasis are well described in thalassemia, there is limited literature to guide clinical surveillance of the renal-bone disease in this group. Bone mineral density as assessed by dual-energy x-ray absorptiometry (DXA) is cannot adequately capture the microarchitectural changes seen in thalassaemia bone disease. The trabecular bone score (TBS), derived from the lumbar spine using DXA provides a surrogate measures of bone microarchitecture. There is limited information available on the utility of TBS for fracture risk assessment in the thalassemia cohort. In this cross-sectional study of 71 subjects with transfusion-dependent thalassemia, the relationship between TBS, BMD, fractures, nephrolithiasis and biochemical parameters were investigated. TBS was significantly associated with non-vertebral fractures (OR 1.99 [95% CI 1.28 - 3.09]) and nephrolithiasis (OR 1.98 [1.33, 2.95]). Nephrolithiasis was highly prevalent (40.6%) and common in men (60%). Conventional DXA parameters were not significantly associated with nephrolithiasis but were associated with all types of fracture. This study demonstrates the potential use of TBS in predicting non-vertebral fractures and nephrolithiasis in transfusion-dependent thalassemia. The association between TBS and nephrolithiasis deserves further study.

Objective: To formulate clinical consensus recommendations on the presentation, assessment, and management of primary hyperparathyroidism (PHPT) in adults. Method(s): Representatives from relevant Australian and New Zealand Societies used a systematic approach for adaptation of guidelines (ADAPTE) to derive an evidence-informed position statement addressing nine key questions. Result(s): PHPT is a biochemical diagnosis. Serum calcium should be measured in patients with suggestive symptoms, reduced bone mineral density or minimal trauma fractures, and in those with renal stones. Other indications are detailed in the manuscript. In patients with hypercalcaemia, intact parathyroid hormone, 25-hydroxy vitamin D, phosphate, and renal function should be measured. In established PHPT, assessment of bone mineral density, vertebral fractures, urinary tract calculi/nephrocalcinosis and quantification of urinary calcium excretion is warranted. Parathyroidectomy is the only definitive treatment and is warranted for all symptomatic patients and should be considered for asymptomatic patients without contraindications to surgery and with >10 years life expectancy. In patients who do not undergo surgery, we recommend annual evaluation for disease progression. Where the diagnosis is not clear or the risk-benefit ratio is not obvious, multidisciplinary discussion and formulation of a consensus management plan is appropriate. Genetic testing for familial hyperparathyroidism is recommended in selected patients. Conclusion(s): These clinical consensus recommendations were developed to provide clinicians with contemporary guidance on the assessment and management of PHPT in adults. It is anticipated that improved health outcomes for individuals and the population will be achieved at a decreased cost to the community.Copyright © 2021 John Wiley & Sons Ltd.

Objective: To develop evidence-based recommendations to guide the surgical management and postoperative follow-up of adults with primary hyperparathyroidism. Method(s): Representatives from relevant Australian and New Zealand Societies used a systematic approach for adaptation of guidelines (ADAPTE) to derive an evidence-informed position statement addressing eight key questions. Result(s): Diagnostic imaging does not determine suitability for surgery but can guide the planning of surgery in suitable candidates. First-line imaging includes ultrasound and either parathyroid 4DCT or scintigraphy, depending on local availability and expertise. Minimally invasive parathyroidectomy is appropriate in most patients with concordant imaging. Bilateral neck exploration should be considered in those with discordant/negative imaging findings, multi-gland disease and genetic/familial risk factors. Parathyroid surgery, especially re-operative surgery, has better outcomes in the hands of higher volume surgeons. Neuromonitoring is generally not required for initial surgery but should be considered for re-operative surgery. Following parathyroidectomy, calcium and parathyroid hormone levels should be re-checked in the first 24 h and repeated early if there are risk factors for hypocalcaemia. Eucalcaemia at 6 months is consistent with surgical cure; parathyroid hormone levels do not need to be re-checked in the absence of other clinical indications. Longer-term surveillance of skeletal health is recommended. Conclusion(s): This position statement provides up-to-date guidance on evidence-based best practice surgical and postoperative management of adults with primary hyperparathyroidism.Copyright © 2021 John Wiley & Sons Ltd.

Introduction: Fracture prevention in young adults with chronic disease and glucocorticoid (GC) exposure is complex and limited by insufficient data. Current guidelines(1) recommend monitoring of bone mineral density (BMD) with dual x-ray absorptiometry (DXA); oral bisphosphonates are recommended over anabolic therapy for those at moderate-high fracture risk.Clinical Case: A 47-year-old woman with a 30-year history of severe rheumatoid arthritis presented for review of osteoporosis management. She had a 20-year fracture history including vertebral, metatarsal and periprosthetic fractures of the left ulna and a more recent non-healing left patella and tibia-fibula fracture. Risk factors for fracture included over 20 years of GC exposure, premature ovarian insufficiency (POI) secondary to cyclophosphamide and pancreatitis secondary to sulfasalazine. She had received oral bisphosphonates for over five years followed by four doses of zoledronic acid. Two years after her final dose of zoledronic acid, she developed osteonecrosis of the jaw (ONJ) after a dental extraction.The patient had normal vitamin D (25-OH vitamin D 102nmol/L; 50-250), creatinine 63umol/L (45 - 85) and corrected calcium 2.44mmol/L (2.15-2.55) levels. Bone turnover markers were unremarkable: CTx 220ng/L (150-800), P1NP 50ug/L (15-70) and ALP 59U/L (20-105). BMD was normal on DXA: L1-L4 (LS) 1.254g/cm2, Z-score -0.3, left neck of femur (NOF) 0.921g/cm2, Z score -1.1 in 2010, increasing to 1.353g/cm2 LS and 1.142g/cm2 NOF by 2018. However, high resolution peripheral quantitative computed tomography (HR-pQCT) identified reduced trabecular and cortical bone volume and a 7.8x6.5x6.7mm area of focal bone loss (Figure 1). While her ONJ improved, trabecular volumetric BMD (Tb.vBMD: 157mgHA/cm3 at radius and 144.3 mgHA/cm3 at tibia) and cortical volumetric BMD (Ct.vBMD; 845.3 mgHA/cm3 radius and 889.5 mgHA/cm3 tibia) did not, (Tbv.BMD 119.0 mgHA/cm3 radius and 114.3 mgHA/cm3 tibia, Ct.vBMD 848.4 mgHA/cm3 radius

Osteoporosis is a global public health problem, with fractures contributing to significant morbidity and mortality. Although post-menopausal osteoporosis is most common, up to 30% of post-menopausal women, >50% of premenopausal women, and between 50-80% of men have secondary osteoporosis. Exclusion of secondary causes is important as treatment of such patients often commences by treating the underlying condition. These are varied but often neglected, ranging from endocrine to chronic inflammatory and genetic conditions. General screening is recommended for all patients with osteoporosis with advanced investigations reserved for premenopausal women and men aged <50 years, for older patients in whom classical risk factors for osteoporosis are absent, and for all patients with the lowest bone mass (Z-score <=-2). The response of secondary osteoporosis to conventional anti-osteoporosis therapy may be inadequate if the underlying condition is unrecognized and untreated. Bone densitometry, using dual energy x-ray absorptiometry (DXA), may underestimate fracture risk in some chronic diseases including glucocorticoid-induced osteoporosis, type 2 diabetes and obesity, and may overestimate fracture risk in others (e.g. Turner syndrome). FRAX and TBS may provide additional information regarding fracture risk in secondary osteoporosis, but their use is limited to adults aged >=40 years and >=50 years, respectively. In additional FRAX requires adjustment in some chronic conditions e.g. glucocorticoid dose, type 2 diabetes and HIV. In most conditions, evidence for antiresorptive or anabolic therapy is limited to increases in bone mass. Current osteoporosis management guidelines also neglect secondary osteoporosis and these existing evidence gaps are discussed.Copyright © The Author(s) 2021. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.

Background: Type 1 diabetes (T1D) is associated with reproductive dysfunction, particularly in the setting of poor metabolic control. Improvements in contemporary management ameliorate these problems, albeit at the cost of increased exogenous insulin and rising obesity, with emerging reproductive implications. Objective(s): To evaluate changes in body mass index (BMI) and the relationship between obesity, menstrual irregularity and polycystic ovary syndrome (PCOS) in young women with T1D, compared with controls. Method(s): Longitudinal observational study using data from the Australian Longitudinal Study in Women's Health of the cohort born in 1989-95, from 2013 to 2015. Three questionnaires administered at baseline and yearly intervals were used to evaluate self-reported menstrual irregularity, PCOS and BMI. Result(s): Overall, 15 926 women were included at baseline (T1D, n = 115; controls, n = 15 811). 61 women with T1D and 8332 controls remained at Year 2. Median BMI was higher in women with type 1 diabetes (25.5 vs 22.9 kg/m2, P <.001), where over half were overweight or obese (54.4% vs 32.9%, P <.001). Median BMI increased by 1.11 and 0.45 kg/m2, in the T1D and control groups, respectively. T1D was independently associated with an increased risk of menstrual irregularity (RR 1.22, 95% CI 1.02-1.46) and PCOS (RR 2.41, 95% CI 1.70-3.42). Obesity conferred a 4-fold increased risk of PCOS, compared to those with normal BMI (RR 3.93, 95% CI 3.51-4.42). Conclusion(s): Obesity is prevalent amongst women with T1D and may be a key contributor to the higher risk of menstrual irregularity and PCOS in this cohort, representing an important opportunity for prevention and intervention.Copyright © 2020 John Wiley & Sons Ltd

Accelerated bone loss and muscle loss coexist in women with premature ovarian insufficiency (POI), but there are significant gaps in our understanding of musculoskeletal health in POI. This review describes estrogen signaling in bone and its role in skeletal health and disease. Possible mechanisms contributing to bone loss in different forms of POI and current evidence regarding the utility of available diagnostic tests and therapeutic options are also discussed. A literature review from January 2000 to March 2020 was conducted to identify relevant studies. Women with POI experience significant deterioration in musculoskeletal health due to the loss of protective effects of estrogen. In bone, loss of bone mineral density (BMD) and compromised bone quality result in increased fracture risk; however, tools to assess bone quality such as trabecular bone score (TBS) need to be validated in this population. Timely initiation of HRT is recommended to minimize the deleterious effects of estrogen deficiency on bone in the absence of contraindications; however, the ideal estrogen replacement regimen remains unknown. POI is associated with compromised bone health, regardless of the etiology. Ongoing research is warranted to refine our management strategies to preserve bone health in women with POI.Copyright © 2021 Georg Thieme Verlag. All rights reserved.

OBJECTIVE: To develop evidence-based recommendations to guide the surgical management and postoperative follow-up of adults with primary hyperparathyroidism. METHODS: Representatives from relevant Australian and New Zealand Societies used a systematic approach for adaptation of guidelines (ADAPTE) to derive an evidence-informed position statement addressing eight key questions. RESULTS: Diagnostic imaging does not determine suitability for surgery but can guide the planning of surgery in suitable candidates. First-line imaging includes ultrasound and either parathyroid 4DCT or scintigraphy, depending on local availability and expertise. Minimally invasive parathyroidectomy is appropriate in most patients with concordant imaging. Bilateral neck exploration should be considered in those with discordant/negative imaging findings, multi-gland disease and genetic/familial risk factors. Parathyroid surgery, especially re-operative surgery, has better outcomes in the hands of higher volume surgeons. Neuromonitoring is generally not required for initial surgery but should be considered for re-operative surgery. Following parathyroidectomy, calcium and parathyroid hormone levels should be re-checked in the first 24 h and repeated early if there are risk factors for hypocalcaemia. Eucalcaemia at 6 months is consistent with surgical cure; parathyroid hormone levels do not need to be re-checked in the absence of other clinical indications. Longer-term surveillance of skeletal health is recommended. CONCLUSIONS: This position statement provides up-to-date guidance on evidence-based best practice surgical and postoperative management of adults with primary hyperparathyroidism.

Purpose: To determine whether geographic variation exists in osteoporosis knowledge, management, and barriers to care in the setting of premature ovarian insufficiency (POI), among general practitioners (GPs) and women with POI. Method(s): Australian GPs completed an online questionnaire regarding osteoporosis knowledge, barriers to care and educational preferences for managing osteoporosis in POI. Women with POI/early menopause (EM) completed an online questionnaire regarding osteoporosis knowledge, risk factors and health beliefs. Clinicians and consumers in metropolitan areas were compared to those in rural areas. Result(s): Of 688 GP respondents, 62.2% practised in major capital cities, 13.1% in major regional cities, 7.8% in regional centres, 8.7% in rural areas and 8.1% in remote areas. Mean +/- SD osteoporosis knowledge score was 9.1 +/- 1.5/13, with no difference by location. Forty-one percent of GPs reported barriers to care which varied by location. Of 316 women with POI/EM, 61.1% lived in metropolitan, 22.5% in regional, 11.7% in rural and 4.4% in remote locations. The mean osteoporosis knowledge score was 8.2 +/- 3.1/20, with lower scores in women living in rural and remote versus metropolitan locations (difference - 1.3; 95% CI - 2.3, - 0.25; p = 0.02). Women in rural areas were less likely to use vitamin D supplements and more likely to have a family history of osteoporosis (both p < 0.05). Conclusion(s): GP knowledge gaps and specific, location-dependent care barriers for osteoporosis in POI were identified. Geographic differences in osteoporosis knowledge and risk factors exist in women with POI/EM. These factors require consideration when designing programs to improve bone health in POI.Copyright © 2020, International Osteoporosis Foundation and National Osteoporosis Foundation.

Familial hypocalciuric hypercalcemia (FHH) is a group of autosomal dominant disorders caused by dysfunction of the calcium sensing receptor (CaSR) and its downstream signaling proteins, leading to generally asymptomatic hypercalcemia. During pregnancy, distinguishing FHH from primary hyperparathyroidism (PHPT) is important, as the latter is associated with adverse outcomes and can be treated surgically during pregnancy, whereas the former is benign. This case report highlights the difficulties in diagnosing FHH during pregnancy. A 32-year-old woman was found to have asymptomatic hypercalcemia at 14-weeks' gestation. Investigations showed a corrected calcium (cCa) of 2.61 mmol/L (2.10 to 2.60), ionized Ca (iCa) of 1.40 mmol/L (1.15 to 1.28), 25OHD of 33 nmol/L (75 to 250), and PTH of 9.5 pmol/L (1.5 to 7.0). The patient was treated with 2000 IU cholecalciferol daily with normalization of 25OHD. The urine calcium / creatinine clearance ratio (CCCR) was 0.0071, and neck US did not visualize a parathyroid adenoma. Upon a retrospective review of the patient's biochemistry from 2 years prior, hypercalcemia was found that was not investigated. The patient was monitored with serial iCa levels and obstetric US. She delivered a healthy boy at 38-weeks' gestation. Postnatal iCa was 1.48 mmol/L and remained elevated. Her son had elevated iCa at birth of 1.46 mmol/L (1.15 to 1.33), which rose to 1.81 mmol/L by 2 weeks. He was otherwise well. Given the familial hypercalcemia, a likely diagnosis of FHH was made. Genetic testing of the son revealed a missense mutation, NM_000388.3(CASR):c.2446A > G, in exon 7 of the CaSR, consistent with FHH type 1. To our knowledge, there are only three existing reports of FHH in pregnancy. When differentiating between FHH and PHPT in pregnancy, interpretation of biochemistry requires an understanding of changes in Ca physiology, and urine CCCR may be unreliable. If the decision is made to observe, clinical symptoms, calcium levels, and fetal US s

Asian race, younger age, higher body mass index (BMI) and antiresorptive drugs have all been associated with atypical femur fractures (AFFs). This increased risk of AFF in Asians is important as by 2050, >50% of hip fractures globally will occur in Asia, with an increased demand for antiresorptive drugs being likely. It is also currently unclear whether AFF risk is increased in all Asian subgroups. We therefore aimed to identify the incidence of AFFs in an Australian tertiary hospital, the contribution of ethnic origin to AFF risk, and determine other clinical risk factors for AFF. From January 1, 2009 to December 31, 2017, 97 AFFs (82 complete and 15 incomplete) occurred in 71 individuals in the overall study population of 204,358. Patients with AFF were more likely to be female (88.7% vs 69.1%, p < 0.001) and younger [median (IQR): 74(52-92) years vs 83(75-88) years, p < 0.001] than the "typical" femur fracture group (n = 3330). The cumulative incidence rate of AFF was 4.2 per 100,000 person-years, far lower than for any ICD-10 AM coded "typical" femur fracture (202.9 per 100,000 person-years). Asians were 3.4 (95%CI, 2.1-5.6) times more likely to sustain an AFF than non-Asians, the highest incidence being in those from South East Asian countries (16.6 per 100,000 person years), suggesting differences in risk between Asian countries. In the nested case-control study, bisphosphonate use was an independent association with AFF development. We conclude Asian ethnicity is an important association with AFF in this large Australian cohort.Copyright © 2020 Elsevier Inc.

Patients with end-stage kidney disease (ESKD) have higher fracture rates and post-fracture mortality than the general population, but bone mineral density by dual energy X-ray absorptiometry (DXA) is less predictive of fracture in this population. Data from bone biopsy and high-resolution imaging indicate that patients on dialysis have reduced cortical thickness and increased cortical porosity, which contribute to fracture risk. The aim of this study was to assess cortical parameters using DXA in patients with ESKD. Using advanced hip analysis, normal age-related ranges were determined from 752 female and 861 male femur scans, and were compared to scans of 226 dialysis patients at time of transplantation. Female dialysis patients had lower cortical thickness (mm) at the femoral neck (2.59 +/- 1.42 vs. 5.23 +/- 1.85), calcar (3.46 +/- 1.07 vs. 5.09 +/- 1.30) and femoral shaft (4.42 +/- 1.21 vs. 7.44 +/- 2.07); p= 0.001 for each site. Buckling ratios (BR), higher values indicating greater femoral neck instability, were also higher for these women (8.21 +/- 4.6 vs. 3.63 +/- 1.42, P = 0.001). All findings were similar for men. Prevalent fracture was documented in 29% of dialysis patients, and in adjusted models, lower femoral neck cortical thickness and a higher BR were associated respectively with a 1.73 (1.22-2.46) and 1.82 (1.49-2.86) fold increased risk of prevalent vertebral fracture per standard deviation change. Cortical parameters measured by DXA are markedly abnormal in dialysis patients and are associated with prevalent vertebral fracture. These parameters should be assessed prospectively in patients with ESKD for utility in fracture prediction and targeting treatment.

Background: Osteoporosis is a key concern of women with premature ovarian insufficiency (POI) but there are gaps in clinicians' knowledge of bone health. Objective(s): 1) To systematically evaluate the quality of clinical practice guidelines (CPGs) related to POI and bone health; 2) to formulate a management algorithm. Method(s): Systematic search for English-language clinical practice guidelines (CPGs) from August 2012 to August 2017 (PROSPERO registration number CRD42017075143). Four reviewers independently evaluated the methodological quality of included CPGs using the Appraisal of Guidelines for Research and Evaluation II (AGREE II) instrument (comprising 23 items across 6 domains) using the My AGREE PLUS platform. Inter-rater reliability was assessed using the intraclass correlation coefficient (ICC). Individual domain and total percentage scores were calculated for each CPG. Data from high-scoring CPGs were extracted and summarised to develop the algorithm, with subsequent refinement via expert and end-user clinician feedback. Result(s): The systematic search yielded 16 CPGs for appraisal. ICC values were 0.71 (good) to 0.95 (very good). The quality of the CPGs was appraised as "high" in 4 cases, "average" in 8 and "low" in 4. High-quality CPGs had mean total scores of 82-96%. Recommendations from high-quality CPGs were summarised into 6 categories: screening; risk factors; initial assessment; diagnosis; subsequent assessment; and management. Only "management" had recommendations (moderate-quality to low-quality evidence) from all four high-quality CPGs. Limitations are reflected in the algorithm. Conclusion(s): Most CPGs regarding bone health and POI are of average to poor quality. High-quality CPGs have evidence limitations and recommendation gaps indicating the need for further research.Copyright © 2019 Elsevier B.V.

Objective: Osteoporosis associated with premature ovarian insufficiency (POI) and early menopause (EM) is a major concern for women. We aimed to (a) identify information and knowledge gaps and behaviours regarding bone health in women with POI/EM and (b) co-design an osteoporosis fact sheet. Design(s): Mixed-methods study: survey of women and online resource appraisals to develop and refine, using semi-structured interviews, an osteoporosis fact sheet. Patient(s): Women with POI/EM (menopause before ages 40 and 45 years respectively). Measurements: Demographics, comorbidities, information needs, calcium intake, exercise, osteoporosis knowledge (OKAT), beliefs and self-efficacy, DISCERN appraisal (validated scales). Analysis: descriptive statistics, logistic regression and thematic analysis of interviews. Result(s): Median age of survey respondents (n = 316) was 54(IQR47-63) years, median age of menopause was 40(IQR38-43) years, and osteoporosis diagnosis was reported in 19%. Most reported inadequate dietary calcium intake (99%) and exercise (65%). Median OKAT score 8 [IQR6-10]/19 indicated knowledge gaps regarding risk factors and treatment options. Adjusting for age and education, OKAT predicted calcium intake (OR 1.126 [CI 1.035-1.225]; P = 0.006) and screening (OR 1.186 [CI 1.077-1.305]; P = 0.001); beliefs predicted screening (OR 1.027 [CI 1.004-1.050]; P = 0.019); and self-efficacy predicted calcium intake (OR1.040 (CI 1.013-1.069); P = 0.003] and exercise (OR 1.117 [CI 1.077-1.160]; P < 0.001). Current online resources have deficiencies. Five themes identified from two interview rounds (n = 10/ round) were as follows: content, emotional response, design, perceived usefulness and clinical considerations. The final fact sheet was considered acceptable and useful in addressing knowledge gaps, promoting information-seeking, impacting behaviours and facilitating healthcare discussions. Conclusion(s): A co-designed fact sheet is acceptable and addresses identified o

Introduction: Representatives appointed by relevant Australian medical societies used a systematic approach for adaptation of guidelines (ADAPTE) to formulate clinical consensus recommendations on assessment and management of bone health in women with oestrogen receptor-positive early breast cancer receiving endocrine therapy. The current evidence suggests that women receiving adjuvant aromatase inhibitors and pre-menopausal woman treated with tamoxifen have accelerated bone loss and that women receiving adjuvant aromatase inhibitors have increased fracture risk. Both bisphosphonates and denosumab prevent bone loss; additionally, denosumab has proven anti-fracture benefit in post-menopausal women receiving aromatase inhibitors for hormone receptor-positive breast cancer. Main recommendations: Women considering endocrine therapy need fracture risk assessment, including clinical risk factors, biochemistry and bone mineral density measurement, with monitoring based on risk factors. Weight-bearing exercise and vitamin D and calcium sufficiency are recommended routinely. Anti-resorptive treatment is indicated in women with prevalent or incident clinical or morphometric fragility fractures, and should be considered in women with a T score (or Z score in women aged < 50 years) of < - 2.0 at any site, or if annual bone loss is >= 5%, considering baseline bone mineral density and other fracture risk factors. Duration of anti-resorptive treatment can be individualised based on absolute fracture risk. Relative to their skeletal benefits, risks of adverse events with anti-resorptive treatments are low. Changes in management as result of the position statement: Skeletal health should be considered in the decision-making process regarding choice and duration of endocrine therapy. Before and during endocrine therapy, skeletal health should be assessed regularly, optimised by non-pharmacological intervention and, where indicated, anti-resorptive treatment, in an individualised, mul

Context: Cerebral palsy (CP) is a motor disorder affecting movement, muscle tone and posture due to damage to the foetal or infant brain. The subsequent lack of ambulation, nutritional deficiencies, anticonvulsant use and hormonal deficiencies have been implicated in the low bone mass associated with this condition. Objective(s): To assess changes in areal bone mineral density (aBMD) during adolescence and young adulthood in individuals with CP. The effect of ambulation, nutrition, hypogonadism on longitudinal changes in aBMD is also examined. Design(s): Retrospective longitudinal study. Setting and participants: Forty-five subjects with CP who had longitudinal dual-energy X-ray absorptiometry (DXA) scans at a single tertiary hospital between 2006 and 2018. Result(s): Mean age at first DXA was 19.4 years (range: 10-36 years), 57.8% were male and 80% were nonambulatory. The mean Z-scores at baseline were <-2.0 at all sites - lumbar spine (LS), femoral neck (FN), total hip (TH) and total body (TB). The median change in aBMD was +1.2%-1.9% per year in all subjects but in those <20 years of age, the median change was 4%-8% per year. Z-scores across all sites remained stable over time. Reduced functional state as measured by the gross motor functional classification scale (GMFCS) had a small negative effect on aBMD over time. Conclusion(s): In adolescents with CP, low bone mass was evident from the baseline DXA. However, significant bone accrual occurred during the second decade, followed by bone maintenance in young adulthood. Future studies should focus on optimizing bone health from early childhood.Copyright © 2019 John Wiley & Sons Ltd

CONTEXT: Cerebral palsy (CP) is a motor disorder affecting movement, muscle tone and posture due to damage to the foetal or infant brain. The subsequent lack of ambulation, nutritional deficiencies, anticonvulsant use and hormonal deficiencies have been implicated in the low bone mass associated with this condition. OBJECTIVE: To assess changes in areal bone mineral density (aBMD) during adolescence and young adulthood in individuals with CP. The effect of ambulation, nutrition, hypogonadism on longitudinal changes in aBMD is also examined. DESIGN: Retrospective longitudinal study. SETTING AND PARTICIPANTS: Forty-five subjects with CP who had longitudinal dual-energy X-ray absorptiometry (DXA) scans at a single tertiary hospital between 2006 and 2018. RESULTS: Mean age at first DXA was 19.4 years (range: 10-36 years), 57.8% were male and 80% were nonambulatory. The mean Z-scores at baseline were <-2.0 at all sites - lumbar spine (LS), femoral neck (FN), total hip (TH) and total body (TB). The median change in aBMD was +1.2%-1.9% per year in all subjects but in those <20 years of age, the median change was 4%-8% per year. Z-scores across all sites remained stable over time. Reduced functional state as measured by the gross motor functional classification scale (GMFCS) had a small negative effect on aBMD over time. CONCLUSION: In adolescents with CP, low bone mass was evident from the baseline DXA. However, significant bone accrual occurred during the second decade, followed by bone maintenance in young adulthood. Future studies should focus on optimizing bone health from early childhood.

INTRODUCTION: Representatives appointed by relevant Australian medical societies used a systematic approach for adaptation of guidelines (ADAPTE) to formulate clinical consensus recommendations on assessment and management of bone health in women with oestrogen receptor-positive early breast cancer receiving endocrine therapy. The current evidence suggests that women receiving adjuvant aromatase inhibitors and pre-menopausal woman treated with tamoxifen have accelerated bone loss and that women receiving adjuvant aromatase inhibitors have increased fracture risk. Both bisphosphonates and denosumab prevent bone loss; additionally, denosumab has proven anti-fracture benefit in post-menopausal women receiving aromatase inhibitors for hormone receptor-positive breast cancer. MAIN RECOMMENDATIONS: Women considering endocrine therapy need fracture risk assessment, including clinical risk factors, biochemistry and bone mineral density measurement, with monitoring based on risk factors. Weight-bearing exercise and vitamin D and calcium sufficiency are recommended routinely. Anti-resorptive treatment is indicated in women with prevalent or incident clinical or morphometric fragility fractures, and should be considered in women with a T score (or Z score in women aged < 50 years) of < - 2.0 at any site, or if annual bone loss is ≥ 5%, considering baseline bone mineral density and other fracture risk factors. Duration of anti-resorptive treatment can be individualised based on absolute fracture risk. Relative to their skeletal benefits, risks of adverse events with anti-resorptive treatments are low. CHANGES IN MANAGEMENT AS RESULT OF THE POSITION STATEMENT: Skeletal health should be considered in the decision-making process regarding choice and duration of endocrine therapy. Before and during endocrine therapy, skeletal health should be assessed regularly, optimised by non-pharmacological intervention and, where indicated, anti-resorptive treatment, in an individualised, mult

Objectives: The detrimental effects of coeliac disease (CD) on bone health are well established. Individuals with CD have an increased risk of osteoporosis and fractures, but few studies have evaluated bone microarchitecture in CD. Our aim was to evaluate trabecular bone score (TBS) derived from DXA images, as an indirect assessment of bone microarchitecture in individuals with CD compared with controls. Method(s): TBS was obtained from 29 female participants who were diagnosed with CD at age <=55 years, 5 years after diagnosis(1), and from29 female controls without CD. There was no history of prior fracture in either group. Multiple linear regression was performed to evaluate associations between age, BMI, CD and menopausal status, and TBS. Result(s):Women in the CD group were five years younger (47.1+/-10.0 vs. 52.2+/-7.4 years, p=0.03). There were no significant differences in BMI (25.7+/-3.2 vs. 25.2+/-4.9 kg/m2, p=0.64), lumbar spine BMD (1.16+/-0.21 vs. 1.15+/-0.18 g/cm2, p=0.83), femoral neck BMD (0.96+/-0.12 vs. 0.92 +/-0.10 g/cm2, p=0.21) or menopausal status (54.8% vs. 75.9% postmenopausal, p=0.11) between groups. Individuals with CD had significantly lower TBS compared with controls (1.36+/-0.11 vs. 1.41+/-0.06, p=0.03), despite an 89.7% mucosal remission rate on duodenal biopsy, 5 years after CD diagnosis. 27.5% of the CD group demonstrated seropositivity to either tissue transglutaminase or deamidated gliadin peptide antibodies. Using a cutoff of 1.35, 45.2% of the CD group had TBS in the degraded range, compared with 13.8% of controls (p=0.01). TBS was negatively associated with both CD and postmenopausal status, but positively associated with lumbar spine BMD and BMI, suggesting neither BMD, nor menopausal status were responsible for the lower TBS in CD. Conclusion(s): We show, for the first time, women with CD had significantly lower TBS compared with older controls matched by BMI and BMD. Degraded TBS secondary to CD was present in half of CD patien

Context: Turner syndrome (TS) is associated with short stature, gonadal failure, and fractures. Spinal trabecular bone score (TBS) is a novel bone imaging modality that has not been evaluated in TS. Objective(s): To evaluate TBS in TS and its association with bone mineral density (BMD), prevalent fracture, and risk factors. Design and Setting: Longitudinal study of TS from a single tertiary hospital between 2006 and 2017. Patients or Other Participants: Fifty-eight subjects with TS aged 20 to 49 years who underwent dualenergy X-ray absorptiometry (DXA). Main Outcome Measure(s): TBS, DXA parameters, and prevalent fractures were investigated. Result(s): Normal, partially degraded, and degraded TBSs were observed in 39 (67%), 15 (26%), and four (7%) subjects, respectively. High rates of prescribed estrogen replacement therapy (ERT) with stable TBS and BMD were observed during follow-up. TBS was positively correlated with spine and femoral neck (FN) BMD and Z-scores (all P<0.05) and negatively correlated with age (20.004 per year; P = 0.014) and delay in ERT initiation inwomenwith primary amenorrhea (20.010 per year; P<0.001). Fractureswere present in 17 (31%) subjects. LowTBS had a significantly higher area under the receiver operator curve for predicting prevalent fracture than low bone mass at either the spine or FN (P < 0.05). Subjects with no history of fracture were more likely to have a normal TBS (P = 0.023). Conclusion(s): BMD and TBS can be preserved with early initiation and continued use of ERT. TBS may provide additional fracture risk prediction to standard DXA parameters in TS and needs to be validated in larger prospective studies.Copyright © 2018 Oxford University Press. All rights reserved.

Introduction: Progressive chronic kidney disease (CKD) confers a marked increase in risk for vascular calcification, cardiovascular disease, fracture and mortality, with likely contributing factors including dysregulated bone metabolism and mineral homeostasis. In general population studies, increased vascular calcification is directly related to mortality and inversely related to bone mineral density (BMD) measured by dual-energy X-ray absorptiometry (DXA). In patients with CKD, complex bone histomorphometry reduces the ability of DXA to predict fracture. The trabecular bone score (TBS) obtained from lumbar spine DXA images, provides a surrogate measure of microarchitectural integrity not captured by BMD. This study aimed to examine the association of the TBS to prevalent abdominal aortic calcification (AAC) in patients with CKD receiving dialysis. Method(s): We performed a cross-sectional study of dialysis patients awaiting transplantation. All patients underwent laboratory testing, lateral spinal radiographs including the abdominal aorta, DXA imaging and TBS assessment. AAC scores were determined using the Kauppila method. Correlations and linear regression models were used to determine predictors of AAC scores. Result(s): 146 patients (60% male, mean age 48+/-13 years) were included, of whom 49% had prevalent calcification with an AAC score >=1. Of those with calcification, the mean AAC score was 7 +/- 5.5 and 42 patients had scores >=6, considered to indicate severe AAC. TBS values corresponding to intermediate or high risk for fracture (<1.31) were present in 35% of patients. TBS values correlated inversely to AAC scores (beta= -0.206, p=0.013) and remained significant in multivariable linear regression, adjusting for age, BMI and time on dialysis (-0.160, p=0.031). There was no significant correlation of AAC scores to any BMD parameter. Conclusion(s): There is a high prevalence of AAC in relatively young dialysis patients awaiting transplantation and their AA

Introduction: Progressive chronic kidney disease (CKD) confers a marked increase in risk for vascular calcification, cardiovascular disease, fracture and mortality, with likely contributing factors including dysregulated bone metabolism and mineral homeostasis. In general population studies, increased vascular calcification is directly related to mortality and inversely related to bone mineral density (BMD) measured by dual-energy X-ray absorptiometry (DXA). In patients with CKD, abnormalities in turnover, mineralization and bone volume reduce the ability of DXA to predict fracture. The trabecular bone score (TBS) obtained from lumbar spine DXA images, provides a surrogate measure of microarchitectural integrity not captured by BMD. This study aimed to examine the association of the TBS to prevalent abdominal aortic calcification (AAC) in patients with CKD receiving dialysis. Method(s): We performed a cross-sectional study of dialysis patients awaiting transplantation. All patients underwent laboratory testing, lateral spinal radiographs including the abdominal aorta, DXA imaging and TBS assessment. AAC scores were determined using the Kauppila method. Correlations and linear regression models were used to determine predictors of AAC scores. Result(s): 146 patients (60% male, mean age 48 +/- 13 years) were included, of whom 49% had prevalent calcification with an AAC score >= 1. Of those with calcification, the mean AAC score was 7 +/- 5.5 and 42 patients had scores >= 6, considered to indicate severe AAC. TBS values corresponding to intermediate or high risk for fracture (<1.31) were present in 35% of patients. TBS values correlated inversely to AAC scores (beta = -0.206, p = 0.013) and remained significant in multivariable linear regression, adjusting for age, BMI and time on dialysis (-0.160, p = 0.031). There was no significant correlation of AAC scores to any BMD parameter. Conclusion(s): There is a high prevalence of AAC in relatively young dialysis patients a

To date, clinical research has focused on postmenopausal osteoporosis, while the aetiology and management of osteoporosis in young adults remain poorly understood. Individuals with transfusion-dependent haemoglobinopathies, chronic neurological conditions, renal disease, premature menopause and young hip fractures are particularly underserved by current literature. This lecture will explore recent developments in the management of the bone disease in several of these groups. Improvements in transfusion medicine have significantly improved life expectancy for patients with thalassemia major. However, osteoporosis and fracture are one of the main causes of morbidity. Multiple factors are implicated in bone disease including marrow expansion, iron toxicity, endocrinopathies and more recently renal tubular dysfunction (1). Our discovery of accelerated bone loss (2), renal calculi (3) and deferasirox-associated hypercalciuria (4) in haemoglobinopathies provides a new pathogenic mechanism underlying bone loss in this cohort. Current work explores methods to minimise and manage hypercalciuria, examining bone and renal outcomes. Cerebral palsy (CP) is the most common motor disorder in children, with increased fracture risk through diminished ambulation, nutritional deficiencies and anticonvulsant use. Improvements in medical care have seen increases in life-expectancy, but studies examining bone health beyond childhood are limited. Our recent work in young adults suggests that fragility fractures are common with predominant ankle, vertebral and rib fractures (5). In addition, hypogonadism was present in 20% of young adults with CP, prompting further work to optimise management in this group.