Development of Atypical Ulnar Fracture is Associated with Bisphosphonate Therapy and Severe Spinal Deformity: A Case Report.

Atypical ulna fracture (AUF) is relatively rare and is known to be associated with prolonged bisphosphonate (BP) use. The developmental mechanism remains unclear. We report a patient with an AUF associated with BP and severe spinal deformity. The patient was an 85-year-old woman receiving oral alendronate for 8 years without vitamin D supplementation. During regular kitchen work, she needed left upper limb support. She presented with atraumatic pain over the ulna. Radiographs revealed a transverse fracture in the proximal ulna and ulna bowing deformity. Whole-spine standing radiographs showed severe degenerative kyphoscoliosis. The skin induration with pigmentation on her left elbow that suggested prolonged overload and during standing work, coincided exactly with fracture location. This report suggests that 'direct stress', with persistent local overload on the proximal ulna, is one of the developmental mechanisms of AUF, in addition to persistent suppression of bone remodelling by prolonged BP use and vitamin D deficiency. Level of Evidence: Level V (Therapeutic).

Diagnostic Criteria and Treatment of Atypical Ulnar Fractures Associated With Prolonged Bisphosphonate Therapy: Multicenter Case Analysis.

PURPOSE: Atypical ulnar fracture (AUF) related to prolonged bisphosphonate therapy is a rare complication. We propose diagnostic criteria of AUFs and present a treatment algorithm. METHODS: Twelve AUFs in 10 patients were studied. The diagnosis of AUF was based on the case definition of atypical femoral fracture (AFF). We investigated clinical and radiographic characteristics of AUFs according to major and minor features of AFFs, and modified the case definition of an AFF to fit the characteristics of AUFs. All AUFs were treated surgically. The radiographic union of fractures was investigated, and delayed fracture healing was defined as a delay of 6 months or more. RESULTS: The average point at which AUFs occurred was at a point 35.1% along the proximal diaphysis of the total ulnar length. All major features of AFFs were identified in the 12 AUFs. Among the minor features, generalized cortical thickening was observed in 6 AUFs, prodromal symptoms in 2 AUFs, bilateral involvement in 2 patients, and delayed fracture healing in 10 AUFs (5 delayed union, 5 nonunion). Initially, 11 of 12 AUFs were treated with plating, and 1 was treated with intramedullary nailing. Two nonunions were revised with sclerotic bone resections, bone grafts, and plate fixation. Finally, union was achieved in 9 AUFs. CONCLUSIONS: The case definition of AFFs can be used for the diagnosis of AUFs, although some modifications must be included in the case definition. Plating is useful in managing AUFs, although sclerotic bone resections and bone grafts may be required. Atypical ulnar fractures occurred in patients who took bisphosphonates longer than AFFs or those whose bisphosphonates were discontinued a few years earlier. Therefore, physicians should be aware of AUFs in those patients and, if necessary, perform a screening test to look for atypical fractures in other bones. TYPE OF STUDY/LEVEL OF EVIDENCE: Diagnostic V.

Case Report: Prophylactic Plate Fixation for Incomplete Atypical Ulnar Fractures Resulting From the Use of Denosumab for Bone Metastases.

Patients with bone metastases are treated with long-term bone resorption inhibitors such as bisphosphonates and denosumab. However, resorption inhibitors have been known to cause fractures, such as atypical femoral fractures (AFFs). In recent years, there have been an increasing number of reports of atypical ulna fractures (AUFs) caused by bone resorption inhibitor usage. Treatment of AUFs is complicated, especially when they occur in patients with bone metastases, because it is difficult to discontinue bone resorption inhibitor treatment without the risk of aggravating metastatic lesions. Prophylactic surgery is recommended in AFFs when fractures are predicted, but there are few reports of prophylactic surgery for AUFs. Here, we report a case of incomplete AUF in a 74-year-old woman which was surgically treated with prophylactic plate fixation. The patient had been using denosumab for 6 years to treat bone metastases due to thyroid cancer. After surgery, no fractures were observed for 2 years without discontinuing denosumab, and her forearm function was adequate. AUFs are rare and difficult to treat, so oncologists who treat bone metastases need to pay special attention to diagnose this incomplete AUF before the fracture worsens. We believe that detection of a possible fracture and prophylactic surgery can improve prognosis.

Bilateral atypical ulnar fractures occurring after long-term treatment with bisphosphonate for 7 years and with teriparatide for 2 years: a case report.

Most atypical fractures associated with the long-term treatment with bisphosphonates (BP) commonly develop in the femoral shaft or subtrochanteric region. We report a rare case of bilateral atypical ulnar fractures in an 86-year-old woman with osteoporosis who finished the treatment with teriparatide for 2 years after long-term treatment with BP. She slid down from an approximately 30-cm-tall seat and slightly contused her left elbow. Plain radiography revealed that both ulnae had a noncomminuted short oblique fracture with cortical thickening and sclerosis at the fracture site. Based on the clinical and radiological findings, she was diagnosed with bilateral atypical ulnar fractures. The fracture of the left ulna was completely displaced and treated surgically. On the other hand, since the right ulna was an incomplete fracture, it was treated conservatively. During surgery, drilling with Kirschner wire and curettage were performed in the osteosclerotic lesion, and an autologous cancellous bone graft was inserted from the ipsilateral olecranon. Bone union was achieved in both fractures at 1 year after surgery. There have been no reports regarding the development of atypical ulnar fractures occurring after the long-term treatment with BP and 2-year use of teriparatide, and the treatment strategies of such fractures have not been established. If teriparatide cannot be used after occurring atypical fractures, the use of low-intensity pulsed ultrasound (LIPUS) and subsequent treatment for osteoporosis are recommended for the bone union. In addition, the treatment of the osteosclerotic lesion and rigid internal fixation are required in surgery.

Atypical Fracture of the Proximal Shaft of the Ulna Associated with Prolonged Bisphosphonate Therapy.

Atypical fractures have been reported as a complication of long-standing bisphosphonate therapy and occur commonly in the subtrochanteric region or shaft of the femur showing typical radiographic features. We encountered a case of atypical fracture of the proximal one-third of the shaft of the ulna. Radiographic findings of this case differed from previously reported cases of ulnar fracture in terms of showing a transverse fracture line with cortical thickening and an oblique fracture line with anterior cortical spike. On the other hand, these findings were similar to radiographic features of atypical femoral fractures. The present case was managed surgically and union of fracture was achieved after 6 months. As there are possibilities of occurrence of atypical fractures in sites other than the femur, the physicians should cautiously examine the patients' history and radiographic findings.

Multiple pathological fractures and delayed union associated with lead exposure in a German Shepherd Dog.

CASE REPORT: An 8-year-old 40.8-kg intact male German Shepherd Dog was evaluated for bilateral antebrachial fractures. Radiographs revealed osteopenia and comminuted proximal diaphyseal fractures of the left radius and ulna, and proximal articular fracture of the right ulna. A dual energy X-ray absorptiometry scan confirmed decreased bone mineral density. Bone mineral analysis collected at the time of definitive surgical repair demonstrated high lead concentration. Analysis further demonstrated normal bone calcium and phosphorus concentrations. Serum lead concentration was normal. The left radial and ulnar fractures were surgically stabilised with an external fixator. The right ulnar fracture was splinted. The left antebrachial fractures were palpably unstable at 12 weeks after surgery. Moderate callus formation and incomplete bone union were present at 17 weeks postoperatively. The dog was re-presented 15 months later for right metacarpal and left metatarsal fractures, which were managed conservatively. Complete bone union of the right radial and ulnar fractures was not present at that time. CONCLUSION: Excessive lead accumulation in bone should be considered as a differential diagnosis for increased susceptibility to pathologic fracture and delayed fracture healing in dogs.

Ulnar fractures with bisphosphonate therapy: a systematic review of published case reports.

SUMMARY: A systematic review of eight ulnar fractures in seven patients with bisphosphonate therapy was performed to describe the characteristics and predisposing factors. The proximal ulna is likely to be fractured, especially in the dominant limb of elderly female patients using walking aids after 7 to 15 years of bisphosphonate use. INTRODUCTION: Long-term bisphosphonate use has been suggested to result in decreased bone remodelling and increased risk of atypical fractures. While the relationship between bisphosphonate use and atypical femoral fractures has been extensively studied, there is relative rarity and unawareness of these fractures in the forearm. We conducted a systematic review of existing case reports to better describe the characteristics and predisposing factors for fractures occurring in patients with bisphosphonate therapy. METHODS: The systematic review was conducted according to PRISMA guidelines. All studies with ulnar fractures in individuals with history of bisphosphonate use were included, with data extracted and analysed in totality. RESULTS: Seven patients with eight fractures are included. Predisposing factors include elderly females requiring walking aids. There is a propensity for the proximal ulna to be fractured, especially in the dominant limb used for ambulation or transfer. All patients were on bisphosphonate for 7 to 15 years. All fractures were atraumatic, non-comminuted, transverse in configuration, had localised periosteal or endosteal thickening at the fracture site and generalised cortical thickening of the diaphysis. CONCLUSION: Ulnar fractures in patients with bisphosphonate therapy demonstrate features similar to those described for atypical femoral fractures, suggesting that these fractures could also possibly be due to bisphosphonate use. However, the ulna appears to be able to tolerate longer periods of alendronate use prior to fracture development. The mechanism and characteristics of these fractures additionally suggest the presence of repetitive stress that accumulates over time due to suppressed bone remodelling in patients on bisphosphonates, eventually resulting in these fractures.

Atypical forearm fractures associated with long-term use of bisphosphonate.

Recent reports on atypical femoral fracture have raised concerns about the long-term use of bisphosphonate. More recent case series focus specifically on the subtrochanteric fractures. But, there is relatively rarity and unawareness of atypical fracture in upper extremity. We report forearm fracture in two women receiving long-term bisphosphonate therapy. First woman presented with pain in the forearm and both thighs and radiographs showed incomplete fractures in a proximal ulnar shaft and bilateral femoral shaft. The other woman had a fracture in the radial shaft. This report suggests atypical fractures associated long-term use of bisphosphonate could occur in bones other than femur. More study is required to identify the magnitude of clinical features of this emerging concern.

Bilateral ulna fractures associated with bisphosphonate therapy.

We report a case of bilateral ulna stress fractures following bilateral femoral fractures associated with long-term bisphosphonate use. The patient is an 84-year-old woman receiving 15 years of bisphosphonate therapy. She did not have any preexisting medical conditions which are known secondary causes of bone loss. She was mostly housebound and used a walking frame for ambulation. She presented with atraumatic right ulna pain and subsequent atraumatic left ulna pain a month later. She was treated conservatively in backslabs and her bisphosphonate was stopped. Investigations did not reveal any secondary causes of osteoporosis or metabolic bone disorders.

Alendronate-associated ulnar and tibial fractures: a case report.

Bisphosphonates are widely used to prevent osteoporotic fractures. Long-term use has resulted in unexpected subtrochanteric fractures in some elderly patients. We report a patient on long-term alendronate therapy who sustained fractures of the ulna and tibia sequentially over a 7-month period, without any trauma or fall.

Spontaneous fractures of uninvolved bones in patients with Paget's disease during unduly prolonged treatment with disodium etidronate (EHDP).

Three patients with Paget's disease of bone were treated by disodium etidronate (EHDP) without interruption during periods of 18 to 30 months. In one case the daily dose was also unduly high (approximately equal to 18 mg/kg/day). A moderate to conspicuous diminution of the renal function was observed in all cases. The three patients developed skeletal pain in a gradual but progressively severe pattern. Seven nontraumatic fractures in nonpagetic bones were encountered. EHDP produces blockage of bone mineralization and excessive suppression of bone remodeling, therefore increasing the risks of fracture. EHDP affects not only pagetic bones but normal skeleton. A baseline evaluation of the renal function might help to identify those patients with greater risk to develop skeletal side effects during EHDP treatment.