Latent metabolic bone disease, skeletal dysplasia and other conditions related to low bone formation among 38 patients with subtrochanteric femoral fractures: a retrospective observational study.

Subtrochanteric femoral fracture is rare and intractable due to the possible association with low bone formation. Retrospective analysis of 38 patients with subtrochanteric femoral fractures revealed that four patients suffered from disorders related to low bone formation and there were specific treatments for two of them. PURPOSE: The main aim of this study was to detect latent metabolic bone diseases and skeletal dysplasia associated with low bone formation among patients with morphologic atypical femoral fracture (AFF). A second aim was to evaluate the frequency of recognized risk factors, such as antiresorptive agents, glucocorticoids, and age. METHODS: Clinical information was retrospectively analyzed among 38 Japanese patients who were admitted to the Department of Orthopedic Surgery and Spinal Surgery and the Division of Emergency and Critical Care Medicine at the University of Tokyo Hospital with diagnoses of subtrochanteric fractures between February 2012 and March 2022. RESULTS: Among 38 patients (including 30 females), 21 patients were aged 75 and over. Ten patients had past oral glucocorticoid use, and 18 had past antiresorptive agent use. Two patients were diagnosed with hypophosphatemic osteomalacia after the development of fractures. One patient was suspected to be a carrier of a loss-of-function variant of alkaline phosphatase, biomineralization associated (ALPL), and one other patient had previously been genetically diagnosed with pycnodysostosis. Among four patients with a diagnosis or suspicion of these metabolic bone diseases and skeletal dysplasia, four had past clinical fractures, two had past subtrochanteric femoral fractures, and two had subtrochanteric femoral fractures on both sides. CONCLUSION: If clinicians encounter patients with morphologic AFF, latent diseases related to low bone formation should be carefully differentiated because appropriate treatment may prevent delayed union and recurrent fractures. Additionally, it may be desirable to exclude these bone diseases in advance before initiating long-term use of antiresorptive agents in osteoporotic patients by screening with serum alkaline phosphatase levels to reduce the risk of morphologic AFF.

Teriparatide Does not Have Beneficial Effects on Bone Healing in Complete Atypical Femur Fractures.

Teriparatide is an anabolic drug sometimes administered to patients who have atypical femoral fracture (AFF). However, whether teriparatide has beneficial effects on bone healing remains uncertain. The present study aimed to analyze the association between teriparatide and bone healing in complete AFF. A total of 59 consecutive cases (58 patients) who underwent intramedullary nailing for complete AFF were categorized based on postoperative use of teriparatide into the non-teriparatide (non-TPTD, n = 34) and teriparatide groups (TPTD, n = 25). Time-to-bone union was evaluated and compared between the two groups. Additionally, multiple regression analysis was performed to evaluate factors affecting time-to-bone union. All participants were women, with a mean age of 77.6 years (range: 62-92). No significant difference in time-to-bone union was found between the non-TPTD and TPTD groups (5.5 months vs. 5.8 months, p = 0.359). Two patients in the non-TPTD group underwent reoperation (p = 0.503) due to failure caused by inadequate fixation, and both achieved bone healing after additional fixation with blocking screws. Multiple regression analysis revealed that the anterior gap of the fracture site postoperatively was a factor affecting time-to-bone union (p = 0.014). The beneficial effect of teriparatide on bone healing in complete AFF could not be confirmed. Additional randomized controlled trials are required. Nonetheless, appropriate techniques, including efforts to reduce the gap on the tensile side during the surgery, are important for reliable bone healing.

The effect of teriparatide on patients with atypical femur fractures: a systematic review and meta-analysis.

INTRODUCTION: Bisphosphonates (BPs) are one of the most often used drugs to lower fracture risk in osteoporosis patients; nonetheless, BPs have been linked to atypical femoral fracture (AFF). Teriparatide (TPTD) is a parathyroid hormone analogue and anabolic drug that may accelerate fracture repair. TPTD has been considered as a possible treatment for AFF, particularly those caused by BP use. We evaluate the effect of TPTD on AFF in this systematic review and meta-analysis. MATERIALS AND METHODS: A thorough search of: Web of Science, Scopus, PubMed, and Cochrane was conducted on August 2, 2023. Trials evaluating the effect of TPTD on the incidence of: complete bone healing, non-union, early and delayed bone union, progression of incomplete AFF to complete AFF, and time to bone union were included. Using Review Manager (RevMan) version 5.4, the risk ratio (RR) and mean difference (MD) with the corresponding 95% confidence interval (CI) were estimated for dichotomous and continuous outcomes, respectively. The Newcastle-Ottawa Scale was used to assess the quality of studies. RESULTS: Eight studies met the eligibility criteria and were included in our analysis. TPTD significantly increased the incidence of early bone union (RR = 1.45, 95% CI [1.13, 1.87], P = 0.004) and time to bone union (MD = -1.56, 95% CI [-2.86, -0.26], P = 0.02) compared to the control group. No significant differences were observed in terms of complete bone healing (RR = 1.09, 95% CI [0.99, 1.13], P = 0.12), non-union (RR = 0.48, 95% CI [0.22, 1.04], P = 0.06), and progression of incomplete AFF to complete AFF (RR = 0.27, 95% CI [0.04, 1.97], P = 0.19). CONCLUSIONS: TPTD is an effective therapy for enhancing and hastening healing following AFF, particularly in postoperative settings. Future large randomized clinical trials are needed to confirm or dispute the results.

Factors Affecting the Second Complete Atypical Femoral Fracture after the First Atypical Fracture.

OBJECTIVES: Atypical femoral fracture (AFF) is an atypical low-energy subtrochanteric and diaphyseal femoral fracture. Even if bone fusion is achieved in patients with AFF, the risk of AFF in the contralateral femur must be considered. This study aimed to investigate the factors affecting complete AFF in the contralateral femur and conservatively treated incomplete AFF. SUBJECT AND METHODS: Radiographs of 111 femurs in 104 AFF cases were examined, and the femurs were classified as follows: 85 contralateral femurs with complete AFF; 18 contralateral femurs with incomplete AFF; 8 femurs with incomplete AFF without surgical treatment. Various patients' clinical data were collected, and we investigated the factors affecting the second complete AFF. RESULTS: Complete fractures occurred in 10 (9.7%) of 103 femurs without incomplete AFF at the first visit and in 3 (37.5%) of 8 femurs with incomplete AFF. The Kaplan-Meier curve revealed that lateral cortical bone thickening and thigh pain were associated with significantly poorer prognoses (p = 0.026 and p = 0.013, respectively). Multivariate analyses revealed that eldecalcitol usage after AFF onset (p = 0.0094) and previous use of bisphosphonate or denosumab (p = 0.0126) were protective factors for second complete AFF and that the presence of thigh pain (p = 0.0134) was a risk factor for second complete AFF. CONCLUSIONS: Eldecalcitol administration after bone union of first AFF may prevent AFF recurrence. In addition, painful incomplete AFF has a high risk of developing a complete fracture.

The effect of teriparatide on fracture healing after atypical femoral fracture: A systematic review and meta-analysis.

This meta-analysis demonstrated that a greater prevalence of delayed union and nonunion and a longer time to fracture healing in the group that did not receive TPTD treatment after AFFs than in the group that received TPTD treatment. PURPOSE: To date, there is no hard evidence for medical management after atypical femoral fracture (AFF), even though weak data indicate faster healing with teriparatide (TPTD). Herein, we aimed to investigate the effect of postfracture TPTD treatment on AFF healing using a pairwise meta-analysis focusing on delayed union, nonunion, and fracture healing time. METHODS: A systematic search of the MEDLINE (PUBMED), Embase, and Cochrane Library databases was performed for studies investigating the effect of TPTD after AFF up to October 11, 2022. We compared the incidence of delayed union and nonunion and the time of fracture healing between the TPTD ( +) and TPTD (-) groups. RESULTS: The 6 studies analyzed a total of 214 AFF patients, including 93 who received TPTD therapy after AFF and 121 who did not. The pooled analysis showed a significantly higher rate of delayed union in the TPTD (-) group than in the TPTD ( +) group (OR, 0.24; 95% CI, 0.11-0.52; P < 0.01; I2 = 0%), and a higher nonunion rate was observed in the TPTD (-) group than in the TPTD ( +) group with low heterogeneity (OR, 0.21; 95% CI, 0.06-0.78; P = 0.02; I2 = 0%). The TPTD (-) group required 1.69 months longer to achieve fracture union than the TPTD ( +) group, with statistical significance (MD = - 1.69, 95% CI: - 2.44 to - 0.95, P < 0.01; I2 = 13%). Subgroup analysis for patients with complete AFF showed that the TPTD (-) group had a higher rate of delayed union with low heterogeneity (OR, 0.22; 95% CI, 0.10-0.51; P < 0.01; I2 = 0%), but there was no significant difference in the nonunion rate between TPTD ( +) and TPTD (-) groups (OR, 0.35; 95% CI, 0.06-2.21; P = 0.25; I2 = 0%). Fracture healing took significantly longer in the TPTD (-) group (MD = - 1.81, 95% CI: - 2.55 to - 1.08; P < 0.01; I2 = 48%). The reoperation rate showed no significant difference between the two groups (OR, 0.29; 95% CI, 0.07-1.20; P = 0.09; I2 = 0%). CONCLUSIONS: The current meta-analysis supported the hypothesis that TPTD treatment following AFF might benefit fracture healing, lowering the rate of delayed union and nonunion and shortening the fracture healing time.

Dual Energy X-Ray Absorptiometry (DXA) Extended Femur Scans to Support Opportunistic Screening for Incomplete Atypical Femoral Fractures: A Short Term in-vivo Precision Study.

BACKGROUND: Atypical femoral fracture (AFF) is documented as a known but rare complication of bisphosphonate use for the treatment of osteoporosis. These present in an incomplete form prior to failure, which results in a complete fracture requiring surgical intervention. Dual energy x-ray absorptiometry (DXA) is the gold standard for the diagnosis of Osteoporosis and for monitoring the response to therapeutic interventions. This provides an opportunity to use routine DXA scans to identify incomplete atypical fractures, which can subsequently be monitored for progression and pre-fracture intramedullary nailing undertaken where necessary. DXA manufacturers have developed extended femur scans to assess and measure the femoral cortex for incipient atypical femoral fractures. The aim of this study was to evaluate the precision errors related to the cortical measurements and for hip bone mineral density using the extended femur setting. METHODOLOGY: A single operator performed duplicate same day in-vivo measurements of the femur in 30 consenting participants, with repositioning between scans, during their visit for routine DXA scanning. The study was performed on a single GE Lunar Prodigy scanner (GE Lunar, Bedford, UK). Root mean squared standard deviation (RMS SD) and coefficient of variation (RMS CV%) were calculated for the cortex measurements known as beaking index (BI) and hip bone mineral density (BMD) measurements. RESULTS: The use of the extended femur scan software yielded an RMS SD (RMS CV%) of 0.011 (1.43%) for the total hip and 0.015 (2.05%) for the femoral neck. The BI measurement RMS SD (RMS CV%) was 0.473 (38.10%) Visual assessment of the femoral cortex discounted all positive BI anomalies as software generated in this dataset. CONCLUSIONS: The use of extended femur scan software did not affect the precision errors of the BMD measurements at the hip when compared to the literature on focused hip scans, however this study is unique with nothing similar being found in the published literature. The BI precision errors were much greater than those seen at the hip and therefore unreliable unless accompanied by visual assessment which is recommended to avoid unnecessary investigation in around one fifth of the scan population.

Risks of femoral localized periosteal thickening in patients with autoimmune inflammatory rheumatic diseases.

OBJECTIVES: The incidence of femoral localized periosteal thickening (LPT), which can precede atypical femoral fracture (AFF), is not low (1-10%) in Japanese patients with autoimmune inflammatory rheumatic diseases (AIRDs). We explored the associations between underlying AIRDs and the prevalence of LPT. METHODS: We conducted post hoc analyses of two cohorts that included a total of 280 Japanese women, 105 of whom had AIRDs and had been taking bisphosphonate (BP) and prednisolone (PSL) and 175 of whom had rheumatoid arthritis (RA). RESULTS: LPT was detected in a total of 18 patients (6.4%) and 3 (1.1%) developed AFFs. RA was negatively correlated with LPT. A disease other than RA requiring glucocorticoid treatment, BP use ≥5 years, PSL use ≥7 years, and a PSL dose ≥5.5 mg/day were positively correlated with LPT. After adjusting for age, diabetes mellitus, and BP duration or daily PSL dose, RA was no longer associated with LPT. CONCLUSIONS: LPT in Japanese patients with AIRDs was associated with BP and glucocorticoid treatment rather than underlying AIRDs. When PSL dose ≥5.5 mg/day is required long-term [typically combined with long-term BP treatment (≥5 years)], clinicians need to pay particular attention in cases LPT and AFF as well as glucocorticoid-induced osteoporosis.

Risk Factors Associated with Intraoperative Iatrogenic Fracture in Patients Undergoing Intramedullary Nailing for Atypical Femoral Fractures with Marked Anterior and Lateral Bowing.

Background and objectives: Iatrogenic fractures are potential complications during intramedullary (IM) nailing for atypical femoral fractures (AFFs). The risk factors associated with iatrogenic fractures remain unclear, although excessive femoral bowing and osteoporosis are hypothesized to be contributing factors. The present study aimed to determine the risk factors for the occurrence of iatrogenic fractures during IM nailing in patients with AFFs. Materials and Methods: This retrospective cross-sectional study evaluated 95 patients with AFF (all female; age range: 49-87 years) who underwent IM nailing between June 2008 and December 2017. The patients were divided into two groups: Group I (with iatrogenic fracture: n = 20) and Group II (without iatrogenic fracture: n = 75). Background characteristics were retrieved from medical records and radiographic measurements were obtained. Univariate and multivariate logistic regression analyses were performed to identify risk factors for the occurrence of intraoperative iatrogenic fractures. Receiver operating curve (ROC) analysis was conducted to determine a cut-off value for the prediction of iatrogenic fracture occurrence. Results: Iatrogenic fractures occurred in 20 (21.1%) patients. The two groups exhibited no significant differences regarding age and other background characteristics. Group I exhibited significantly lower mean femoral bone mineral density (BMD) and significantly greater mean lateral and anterior femoral bowing angles than Group II (all p < 0.05). There were no significant differences in AFF location, nonunion, and IM nail diameter, length, or nail entry point between the two groups. In the univariate analysis, femoral BMD and lateral bowing of the femur differed significantly between the two groups. On multivariate analysis, only lateral bowing of the femur remained significantly associated with iatrogenic fracture occurrence. The ROC analysis determined a cut-off value of 9.3° in lateral bowing of the femur for prediction of iatrogenic fracture occurrence during IM nailing for AFF treatment. Conclusions: The lateral bowing angle of the femur is an important predictive factor for intraoperative iatrogenic fracture occurrence in patients undergoing IM nailing for AFF treatment.

Atypical femur fractures associated with bisphosphonate therapy: post-operative outcomes.

PURPOSE: Bisphosphonates are commonly used medication for the treatment of osteoporosis, and a well-established complication of this medication is bisphosphonate-associated atypical femur fractures (BAAFFs). The aim of this study was to assess the post-operative functional and radiographic outcomes of surgically treated BAAFFs. METHODS: An analysis of patients treated at a university-affiliated institution was performed. Patients who had undergone surgical fixation for a subtrochanteric or shaft of femur fracture which had been classified as atypical and treated with bisphosphonate therapy at the time of fracture were included. The outcome measures assessed included post-operative complications, length of stay, discharge destination, post-operative function, independence in activities of daily living (ADLs) and fracture union. RESULTS: Twenty patients were included in this study with a mean age of 75.2 years. The cohort was predominantly female. The average duration of bisphosphonate therapy was 7.35 years. Nine patients had contralateral bisphosphonate-related stress reactions at the time of their initial fracture and underwent prophylactic surgical intervention. The majority of patients received cephalomedullary nail fixation (95%) with more than half (65.0%) of the cohort experiencing one or more post-operative complications. The median length of stay was 6.5 days and 50% of the cohort required inpatient rehabilitation. At final review, independent mobilisation was recorded in only 10.0% of patients, and 70% of patients were dependent with their ADLs at their latest follow-up. Average follow-up was 8 months, and only six cases demonstrated union at the six-month review, with delayed union between 9 and 12 months being common. CONCLUSION: Patients with BAAFFs experience high rates of complications, record poor post-operative functional outcomes, and demonstrate a delayed time to union. Nearly half of our cohort had radiographic evidence of bilateral pathology.

Atypical periprosthetic femoral fracture with stem breakage: a case report.

Although the American Society for Bone and Mineral Research definition of atypical femoral fracture excludes periprosthetic fractures, fractures around the prosthesis with clinical features of atypical femoral fractures have been reported in the literature. All fractures reported thus far have been distal to the prosthetic segment; however, we encountered a case of a stress fracture in the middle of the femoral component segment. An 86-year-old woman with a history of bisphosphonate osteoporosis treatment and revisional total hip arthroplasty visited our outpatient clinic complaining of pain in the left thigh and groin. We diagnosed an incomplete atypical femoral fracture around the hip prosthesis; medical treatment was implemented. Two months later, the patient visited the emergency department with a complete subtrochanteric fracture with stem breakage. Without revision of the broken stem, two plates were applied after reduction. In this case, we recognized the possibility of a stress fracture but overlooked the possibility of stem breakage in an atypical femoral fracture. Even if it is not evident on the radiograph before complete fracture, clinicians should be alert to the signs of stress fracture in the middle of the femoral component segment, as they may be clues to atypical periprosthetic femoral fracture with stem failure. Isolated medical treatment plans are not recommended for incomplete subtrochanteric atypical periprosthetic femoral fracture. Instead, concomitant prophylactic plate fixation is recommended.

Long-term safety of eldecalcitol in Japanese patients with osteoporosis: a retrospective, large-scale database study.

INTRODUCTION: This real-world study evaluated whether long-term use of eldecalcitol (ELD) increases the risk of adverse events (AEs), namely, hypercalcemia, acute kidney injury (AKI), and urolithiasis, and analyzed the ELD-induced risk of rare AEs such as osteonecrosis of the jaw (ONJ) and atypical femoral fracture (AFF). MATERIALS AND METHODS: Patient records were retrieved from Medical Data Vision (MDV) and Japan Medical Data Center (JMDC) databases. The ELD-treated osteoporosis patient cohort (ELD cohort) was analyzed to determine the incidence rate of the aforementioned AEs. The patient cohort that was prescribed active vitamin D3 other than ELD (AVD cohort) was analyzed as the reference. RESULTS: Incidence rates of hypercalcemia, AKI, and urolithiasis in the ELD cohort were 0.942, 0.517, 2.465 events per 100 person-years, respectively, in the MDV dataset, and 0.687, 0.155, 3.785, respectively, in the JMDC dataset. The incidence rates of these AEs in the ELD cohort remained relatively constant throughout ELD treatment. A small number of patients experienced ONJ or AFF during ELD or AVD treatment. The number of ONJ and AFF cases in the both cohorts decreased over time. The two cohorts showed no difference in the concomitant use of anti-bone resorptive agents such as bisphosphonates and denosumab. CONCLUSION: The risk of hypercalcemia and AKI associated with ELD use observed in this retrospective analysis is similar to that reported previously in the Japanese post-marketing surveillance of ELD. Furthermore, ELD, similar to AVD, may not increase the risk of ONJ and AFF.

Mortality Risk After Atypical Femoral Fracture: A Systematic Review and Meta-analysis.

OBJECTIVE: To summarize all available data using systematic review and meta-analysis to estimate the 1-year mortality risk after atypical femoral fracture (AFF) and risk ratio of mortality after AFF versus typical femoral fracture (TFF). METHODS: Potentially eligible studies were identified from MEDLINE and Embase databases from inception to February 2022 using a search strategy that comprised the terms for "atypical femoral fracture" and "mortality." An eligible study must consist of a cohort of patients with AFF. Then, the study must report the 1-year mortality rate after the AFF or report effect estimates with 95% confidence intervals, comparing the incident mortality between patients with AFF and TFF. Point estimates with standard errors were retrieved from each study and combined using the generic inverse variance method. RESULTS: A total of 8967 articles were identified. After 2 rounds of independent review by 3 investigators, we identified 7 studies reporting the 1-year mortality rate of AFFs and 3 studies comparing the mortality rate of AFF with that of TFF. Pooled analysis revealed a pooled 1-year mortality rate after an AFF of 0.10 (95% confidence interval, 0.05-0.16; I2 = 93.3%). Two studies compared the mortality risks of AFF with those of TFF and revealed conflicting results. CONCLUSION: The 1-year mortality rate after an AFF was approximately 10%. However, evidence is insufficient to conclude whether there was a difference in mortality risk between AFF and TFF.

Atypical femoral fracture mimicking lumbar radiculopathy in two patients taking bisphosphonate long-term: A case report.

BACKGROUND: Atypical femoral fracture (AFF) is a rare complication in patients with osteoporosis undergoing long-term bisphosphonate therapy. The most common symptom of incomplete AFF is nonspecific thigh pain, which is often difficult to distinguish from other causes. CASE PRESENTATION: We present two cases of AFF misdiagnosed as lumbar radiculopathy. Both patients visited our hospital for thigh pain, and in both cases the findings of lumbar spine magnetic resonance imaging showed substantial nerve compression. These patients had been treated for lumbar radiculopathy, but localized symptoms became conspicuous and femoral radiographs revealed complete AFF. The initial radiographs were reviewed retrospectively and revealed slight lateral cortical thickening in the affected femur, denoting a missed incomplete AFF. Internal fixation with intramedullary nails was performed. CONCLUSIONS: AFF may mimic lumbar radiculopathy. Therefore, clinicians should consider AFF as a differential diagnosis in older patients with lumbar canal stenosis who are undergoing long-term bisphosphonate therapy and present with thigh pain.

Hip Fractures after Intramedullary Nailing Fixation for Atypical Femoral Fractures: Three Cases.

Secondary hip fractures (SHFs) rarely occur after intramedullary nailing (IMN) fixation without femoral neck fixation for atypical femoral fractures (AFFs). We report three cases of older Japanese women who sustained SHFs presumably caused by osteoporosis and peri-implant stress concentration around the femoral neck after undergoing IMN without femoral neck fixation for AFF. All cases were fixed with malalignment. In AFF patients, postoperative changes due to postoperative femoral bone malalignment may affect the peri-implant mechanical environment around the femoral neck, which can result in insufficiency fractures. At the first AFF surgery, we recommend femoral neck fixation after adequate reduction is achieved.

Successful Treatment of Atypical Femoral Fracture with Bowed Femur Using Contralateral Intramedullary Nail Combined with Early Daily Teriparatide.

We report a case of atypical femoral fracture achieving early fracture union with combination therapy comprising contralateral nail and immediate teriparatide injection. Fracture union of atypical fractures is often delayed due to bowing deformity and bone metabolic disorders. Combination treatment that takes both problems into consideration represents a useful treatment option for atypical femoral fracture.

The importance of reconstruction nailing for diaphyseal atypical femoral fractures: a comparative study with standard nailing.

INTRODUCTION: Subtrochanteric atypical femoral fractures (AFFs) usually require long cephalomedullary or reconstruction nailing to achieve satisfactory healing. Recently, this type of nailing has also been recommended to fix diaphyseal AFFs and prevent fragility fractures around the proximal femur, although standard antegrade nailing seems sufficient from a biomechanical perspective. This study aimed to compare treatment outcomes and complications between reconstruction and standard nailing for diaphyseal AFFs. MATERIALS AND METHODS: A retrospective comparative study of 48 female patients with diaphyseal AFFs who underwent surgical treatment with either reconstruction or standard nailing was conducted. The reconstruction nailing group comprised 23 patients with a mean age of 74.4 years and included 16 complete and 7 incomplete AFFs. The mean follow-up period was 30 months. The standard nailing group comprised 25 patients with a mean age of 71.1 years and included 16 complete and 9 incomplete AFFs. The mean follow-up period was 57.8 months. The healing rate, time to union, and complications were analyzed and compared between the two groups. RESULTS: Bony union was achieved in all 23 cases (100%) of the reconstruction nailing group by a mean of 19.3 weeks postoperative. In the standard nailing group, 23 of 25 cases healed (92%) by a mean of 16 weeks postoperative. The standard nailing group included two delayed fragility fractures of the femoral neck (5 and 10 years after the initial surgery), four cases of newly developed subtrochanteric AFFs at the distal screw of the proximal interlocking screws of the nail (one complete and three incomplete fractures), and two cases of nonunion. Complications only occurred in the standard nailing group (p = 0.01, Fisher's exact test). CONCLUSIONS: In diaphyseal AFFs, reconstruction nailing may achieve a satisfactory outcome and can also decrease delayed peri-implant fragility fractures of the hip, newly developed subtrochanteric AFFs, and nonunion.

Subtrochanteric atypical femoral fracture induced solely by glucocorticoid without bisphosphonate treatment: a case report.

An AFF is a form of stress fracture induced by excessive physiologic repetitive stress over the bone remodeling capacity. Although glucocorticoid administration is a known risk factor for AFF, no case of AFF with glucocorticoid administration as the only risk factor has been previously reported. In this report, we aimed to highlight the risk of AFF associated with long-term administration of glucocorticoids, and the importance of surveillance and correction of risk factors in patients undergoing long-term glucocorticoid therapy. A 58-year-old male patient was diagnosed with subtrochanteric AFF. He had no medical history of any condition that might disrupt bone metabolism and no known risk factors for AFF, except for long-term administration of glucocorticoid. After fixation of the fracture, the glucocorticoid was replaced with an alternative medication. Although AFF is notorious for delayed union or nonunion, complete union of the fracture was obtained at 14 months postoperatively. This case brought to our attention the possibility that glucocorticoids alone may be responsible for inducing AFF and highlighted the importance of regular assessments in case of necessity of glucocorticoid administration. Additionally, correction of risk factors might expedite the union of AFF.

Cumulative incidence of femoral localized periosteal thickening (beaking) preceding atypical femoral fractures in patients with rheumatoid arthritis.

The incidence of localized periosteal thickening (LPT, also termed beaking) of the lateral cortex that often precedes an atypical femoral fracture (AFF) was not high in patients with rheumatoid arthritis (RA) but incomplete AFFs developed in two patients. Higher-dose prednisolone was a significant risk factor for LPT in patients with RA. INTRODUCTION: Atypical femoral fractures (AFFs) are stress fractures; bisphosphonate (BP) use is a major risk factor for the development of such fractures. Localized periosteal thickening (LPT, also termed beaking) of the lateral cortex often precedes a complete or incomplete AFF. We evaluated the incidence of latent LPT in patients with rheumatoid arthritis (RA), to evaluate LPT progression, and to define LPT risk factors. METHODS: A total of 254 patients with RA were included; all underwent annual X-ray evaluation, dual-energy X-ray absorptiometry, and analyses of serum and bone metabolic markers for 2-3 years. LPT of the lateral cortex was sought in femoral X-rays. RESULTS: The incidence of LPT was 2.4% (6/254). Among patients on both BP and prednisolone (PSL) at enrollment, the incidence was 2.3% (3/131). Two femurs of two patients with LPT developed incomplete AFFs; LPT was extensive and associated with endosteal thickening. One patient had been on BP and PSL and microscopic polyangiitis was comorbidity. The other was on a selective estrogen receptor modulator and PSL. A daily PSL dose >5 mg (OR 11.4; 95%CI 2.15-60.2; p = 0.004) and higher-dose methotrexate (OR 1.22; 95%CI 1.01-1.49; p = 0.043) were significant risk factors for LPT. CONCLUSIONS: The incidence of latent LPT was not high (2.4%) but incomplete AFFs developed in two RA patients. Higher-dose PSL because of a comorbid disease requiring glucocorticoid treatment other than RA or refractory RA were risk factors for LPT; X-ray screening for latent LPT would usefully prevent complete AFFs.

Differences in subtrochanteric and diaphyseal atypical femoral fractures in a super-aging prefectural area: YamaCAFe Study.

INTRODUCTION: Atypical femoral fractures (AFFs) have been correlated with long-term use of bisphosphonates (BPs), glucocorticoids (GCs), and femoral geometry. We investigated the incidence and characteristics of subtrochanteric (ST) and diaphyseal (DP) AFFs in all institutes in a super-aging prefectural area. MATERIALS AND METHODS: We performed a blinded analysis of radiographic data in 87 patients with 98 AFFs in all institutes in Yamagata prefectural area from 2009 to 2014. Among the 98 AFFs, 57 AFFs comprising 11 ST fractures in 9 patients and 46 DP fractures in 41 patients with adequate medical records and X-rays were surveyed for time to bone healing and geometry. RESULTS: Of the 87 patients, 67 received BPs/denosumab (77%) and 10 received GCs (11%). Surgery was performed in 94 AFFs. Among 4 AFFs with conservative therapy, 3 required additional surgery. In univariate regression analyses for ST group versus DP group, male-to-female ratio was 2/7 versus 1/40, mean age at fracture was 58.2 (37-75) versus 78 (60-89) years, rheumatic diseases affected 55.5% (5/9) versus 4.9% (2/41), femoral lateral bowing angle was 1.7 (0-6) versus 11.8 (0.8-24)°, GC usage was 67% (6/9) versus 4.9% (2/41), and bone healing time was 12.1 (6-20) versus 8.1 (3-38) months (p < 0.05). In multivariate analyses, higher male-to-female ratio, younger age, greater proportion affected by rheumatic diseases, and higher GC usage remained significant (p < 0.05). CONCLUSIONS: The incidence of AFFs in our prefectural area was 1.43 cases/100,000 persons/year. This study suggests that the onset of ST AFFs have greater correlation with the worse bone quality, vice versa, the onset of DP AFFs correlated with the bone geometry. The developmental mechanisms of AFFs may differ significantly between ST and DP fractures.

Lower trabecular bone score is associated with an increased incidence of localized femoral periosteal thickening.

INTRODUCTION: Femoral localized periosteal thickening (LPT, also termed "beaking") of the lateral cortex often precedes an atypical femoral fracture (AFF). Bisphosphonate (BP) use, glucocorticoid use, and Asian race are major risk factors for developing such fractures. The aim of this study was to determine whether the trabecular bone score (TBS) reflecting the lumbar trabecular microarchitecture was related to LPT in glucocorticoid-treated Japanese patients with autoimmune diseases. MATERIALS AND METHODS: We retrospectively investigated 111 women with autoimmune diseases treated with prednisolone (PSL) who had undergone both femoral X-ray and dual-energy X-ray absorptiometry of the L1 - L4 lumbar vertebrae and for whom TBS could be evaluated for two or more of these. RESULTS: Femoral LPT was evident in the X-rays of 18 of 111 patients (16.2%). Higher body mass index (BMI), longer duration of PSL use and longer duration of BP use were significant in patients with LPT compared to those without. The TBS was significantly lower in patients with LPT than in those without (1.314 ± 0.092 vs. 1.365 ± 0.100, p = 0.044); however, the lumbar bone mineral density did not differ significantly (0.892 ± 0.141 vs. 0.897 ± 0.154 g/cm2, p = 0.897). TBS was significantly associated with LPT (odds ratio, 0.004; 95% CI, 0 - 0.96; p = 0.048), but not in the multivariate analysis including BMI, duration of PSL use and duration of BP use. CONCLUSIONS: The TBS was lower in glucocorticoid-treated Japanese women with autoimmune diseases with LPT than in those without LPT, and deteriorated trabecular microarchitecture influenced by longer use of BP and glucocorticoid might be associated with the development of LPT.