Osteomalacia with Bone Marrow Fibrosis Due to Severe Vitamin D Deficiency After a Gastrointestinal Bypass Operation for Severe Obesity

ObjectiveTo present 5 cases of bone biopsy-proven osteomalacia with marrow fibrosis (in 3 cases) after gastric bypass operation, review the relevant literature, and offer preventive strategies.MethodsWe summarize the clinical presentation, pertinent biochemical and radiologic data, and bone histomorphometric findings in 5 patients, encountered during a period of 17 years, in whom severe vitamin D deficiency developed after a gastrointestinal bypass surgical procedure for morbid obesity.ResultsFive patients (39 to 60 years of age) were seen for evaluation of metabolic bone disease not responding to “usual” therapy after a gastric bypass surgical procedure. All had generalized bone pain and tenderness, muscle weakness, stooping posture, difficulty walking, and waddling gait due to severe proximal muscle weakness for a period of 2 to 5 years. Diagnoses before the referral varied from arthritis and gout to vitamin D deficiency and osteoporosis despite highly suggestive biochemical or radiologic findings (or both) of osteomalacia in each patient, which was confirmed by bone biopsy. After therapy with pharmacologic doses of ergocalciferol (100,000 IU daily) and calcium carbonate (1 to 2.5 g daily), considerable improvements occurred in clinical symptoms and functional status, biochemical indices, bone mineral density, and bone histomorphometric features.ConclusionGastric bypass operations predispose patients to severe vitamin D deficiency and osteomalacia in the absence of pharmacologic doses of vitamin D therapy. In general, the current recommendations are grossly inadequate in this high-risk population, and the clinical presentation is both nonspecific and often misleading. Prospective long-term studies are needed to determine the appropriate vitamin D dose required to prevent osteomalacia in such patients. (Endocr Pract. 2009;15:528-533)     

Pathological femoral fractures due to osteomalacia associated with adefovir dipivoxil treatment for hepatitis B: a case report

ABSTRACT: We present a case of a 62-year-old man who underwent total hip arthroplasty for treatment of pathologic femoral neck fracture associated with adefovir dipivoxil-induced osteomalacia. He had a 13-month history of bone pain involving his shoulders, hips, and knee. He received adefovir dipivoxil for treatment of lamivudine-resistant hepatitis B virus infection for 5 years before the occurrence of femoral neck fracture. Orthopedic surgeons should be aware of osteomalacia and pathological hip fracture caused by drug-induced renal dysfunction, which results in Fanconi's syndrome.Virtual slidesThe virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/1600344696739249.     

Persistent Tumor-Induced Osteomalacia Confirmed by Elevated Postoperative Levels of Serum Fibroblast Growth Factor-23 and 5-Year Follow-up of Bone Density Changes

ObjectiveTo describe a case of persistent tumor-induced osteomalacia, determine whether serum fibroblast growth factor-23 (FGF-23) levels postoperatively indicate incomplete tumor resection, and report lumbar spine and forearm bone mineral density (BMD) changes during 5 years of follow-up.MethodsWe present clinical, radiologic, histologic, and bone densitometry data as well as serum FGF-23 levels (determined with use of a novel C-terminal enzyme-linked immunosorbent assay) from the study patient and discuss these findings in the context of previous literature.ResultsA 52-year-old man, who presented with muscle weakness and multiple fractures, was found to have low values for serum phosphorus, serum 1,25-dihy-droxyvitamin D, and maximal tubular reabsorption of phosphate per glomerular filtration rate, a high level of serum alkaline phosphatase, and a normal serum concentration of parathyroid hormone, characteristic of tumor-induced osteomalacia. Magnetic resonance imaging to evaluate an abnormality of the left foot revealed a soft tissue mass, biopsy of which confirmed the presence of a benign, phosphaturic, mesenchymal tumor. The baseline serum FGF-23 level (2,050 RU/mL) was more than 17 times the upper limit of normal for adults (23 to 118 RU/mL) and decreased substantially within 1 day after partial resection of the tumor but remained above normal postoperatively. BMD changes indicated rapid substantial recovery of vertebral BMD but ongoing loss of forearm bone density.ConclusionThe serum FGF-23 level is high in a substantial proportion of patients with tumor-induced osteomalacia. The postoperative above normal levels of serum FGF-23 correlated with known persistence of tumor in our study patient. In a patient with normal renal function, such as our study patient, levels of serum FGF-23 studied with use of the C-terminal enzyme-linked immunosorbent assay reached their nadir within 24 hours postoperatively. This result suggests that this assay can provide clinicians with rapid prognostic information in patients with known or suspected residual tumor. BMD should be assessed at both appendicular and axial sites in patients with persistent tumor-induced osteomalacia. (Endocr Pract. 2005;11:108-114)     

Phosphatonins: From Discovery to Therapeutics

ObjectivePhosphate is crucial for cell signaling, energy metabolism, nucleotide synthesis, and bone mineralization. The gut-bone-parathyroid-kidney axis is influenced by parathyroid hormone, 1,25-dihydroxyvitamin D, and phosphatonins, especially fibroblast growth factor 23 (FGF23). These hormones facilitate maintenance of phosphate homeostasis. This review summarizes current knowledge regarding the phosphate homeostasis, phosphatonin pathophysiology, and clinical implications of FGF23-related hypophosphatemic disorders, with specific focus on burosumab treatment.MethodA focused literature search of PubMed was conducted.ResultsPhosphatonins including FGF23, secreted frizzled-related protein 4, matrix extracellular phosphoglycoprotein, and fibroblast growth factor 7 play a pathogenic role in several hypophosphatemic disorders. Excess FGF23 inhibits sodium-dependent phosphate cotransporters (NaPi-2a and NaPi-2c), resulting in hyperphosphaturia and hypophosphatemia. Additionally, FGF23 suppresses 1,25-dihydroxyvitamin D synthesis in the proximal renal tubule, and thus, it indirectly inhibits intestinal phosphate absorption. Disorders of FGF23-related hypophosphatemia include X-linked hypophosphatemia (XLH), autosomal dominant hypophosphatemic rickets, autosomal recessive hypophosphatemic rickets, fibrous dysplasia/McCune-Albright syndrome, and tumor-induced osteomalacia (TIO). Complications of conventional therapy with oral phosphate and vitamin D analogs comprise gastrointestinal distress, hypercalcemia, nephrocalcinosis, and secondary/tertiary hyperparathyroidism. In both children and adults with XLH and TIO, the anti-FGF23 antibody burosumab exhibits a favorable safety profile and is associated with healing of rickets in affected children and improvement of osteomalacia in both children and adults.ConclusionThe treatment paradigm for XLH and TIO is changing based on data from recent clinical trials. Research suggest that burosumab is effective and safe for pediatric and adult patients with XLH or TIO.     

Oncogenic osteomalacia associated with mesenchymal tumor in the middle cranial fossa: a case report

ABSTRACT: INTRODUCTION: Tumor-induced osteomalacia is a paraneoplastic syndrome of hypophosphatemia. Osteomalacia causes multiple bone fractures and severe pain. CASE PRESENTATION: We report the case of a 57-year-old Japanese man with tumor-induced osteomalacia associated with a middle cranial fossa bone tumor. The tumor was successfully resected by using a middle fossa epidural approach. His phosphate level recovered to a normal range immediately after the surgery. CONCLUSIONS: It is rare that tumor-induced osteomalacia originates from the middle skull base. This report suggests that, if patients have a clinical and biochemical picture suggestive of tumor-induced osteomalacia, it is crucial to perform a meticulous examination to detect the tumor or the lesion responsible for the tumor. The serum level of fibroblast growth factor 23 is the most reliable marker for evaluating the treatment outcome of tumor-induced osteomalacia.     

A Case of Hypercalcemia Associated with Castleman Disease

ObjectiveTo present a case of hypercalcemia associated with Castleman disease.MethodsThe details of case presentation, evaluation, diagnosis, and treatment are reviewed in a 25-year-old woman with severe hypercalcemia, joint pain, conjunctival injection, and lymphadenopathy.ResultsEndocrinopathies such as primary hyperparathyroidism were ruled out. Infectious and rheumatologic laboratory evaluations revealed normal results. On a whole-body scan, the patient was noted to have diffusely increased osseous uptake in conjunction with increased periarticular uptake, consistent with a metabolic superscan. After extensive evaluation, the patient underwent a right axillary lymph node biopsy and was found to have multicentric Castleman disease. She was treated with high-dose corticosteroids and eventually immunomodulators to help control her disease.ConclusionTo the best of our knowledge, this is the first published case report demonstrating hypercalcemia in association with Castleman disease. The pathologic mechanism is likely bone turnover mediated by cytokines such as interleukin-6. (Endocr Pract. 2010;16:1007-1011)     

FGF-23 transgenic mice demonstrate hypophosphatemic rickets with reduced expression of sodium phosphate cotransporter type IIa

Fibroblast growth factor (FGF)-23 was identified as a causative factor of tumor-induced osteomalacia and also as a responsible gene for autosomal dominant hypophosphatemic rickets. To clarify the pathophysiological roles of FGF-23 in these diseases, we generated its transgenic mice. The transgenic mice expressing human FGF-23 reproduced the common clinical features of these diseases such as hypophosphatemia probably due to increased renal phosphate wasting, inappropriately low serum 1,25-dihydroxyvitamin D level, and rachitic bone. The renal phosphate wasting in the transgenic mice was accompanied by the reduced expression of sodium phosphate cotransporter type IIa in renal proximal tubules. These results reinforce the notion that the excessive action of FGF-23 plays a causative role in the development of several hypophosphatemic rickets/osteomalacia.     

An Interesting Case of Peripheral Vascular Disease,Vascular Reperfusion,and Subsequent Development of Pain Due to Paget's Disease of Bone

ObjectiveTo present a case of Paget’s disease of bone that was unmasked after vascular reperfusion.MethodsIn this case study, we review the presentation, evaluation, diagnosis, and management of a patient with Paget’s disease and peripheral vascular disease.ResultsA 79-year-old-woman with a history of coronary artery heart disease and recent finding of a T5 compression fracture was hospitalized for evaluation of right lower extremity claudication. Angiography demonstrated a focal complete occlusion of the distal right femoral and popliteal arteries. A self-expanding stent was placed in the distal femoral and popliteal arteries. Approximately 48 hours after the procedure, the patient developed severe, right lower leg pain. On endocrine evaluation, the patient was found to have clinical signs suggesting Paget’s disease of bone, which was subsequently confirmed by imaging.ConclusionThis patient’s development of severe pain following reperfusion of distal femoral and popliteal arteries is in keeping with the known and aforementioned hypervascularity of pagetic bone. The finding of increased warmth over an area of skeletal deformation should always raise the possibility of Paget’s disease of bone.(Endocr Pract. 2013;19:e61-e63)     

Idiopathic Fanconi syndrome with progressive renal failure: a case report and discussion

Fanconi syndrome is a complex of renal tubular dysfunctions defined by glycosuria without diabetes, aminoaciduria, phosphaturia, and renal tubular acidosis. It is often associated with hypokalemia, hypophosphatemia, and rickets or osteomalacia. Although it is usually found in the setting of other well-established non-renal diseases, Fanconi syndrome may present without identifiable etiology or association. Very infrequently a patient with idiopathic Fanconi syndrome will progress to chronic renal failure. This case report details the course of such a patient over the 20 years since his diagnosis and discusses the syndrome's genetic background, clinical features, putative pathophysiology, and therapeutic options, including transplantation.     

Une ostéomalacie liée à un diabète phosphoré – rôle de la scintigraphie osseuse couplée à la TEMP-TDM

We report the case of a 59-year-old woman, investigated for disabling pain of the left thigh, unrelated to any traumatic event. Interrogation had found diffuse pain of myalgia-type and arthralgia-type for approximately a year without local inflammatory signs and insufficiency fractures of both calcaneus two years before. The Technetium 99m-labeled hydroxymethylene diphosphonate (^99mTc-HDP) whole-body bone scintigraphy evidenced multiple hot spots on the higher third of left femur, rib cage, sternum, scapulae, pelvis, right hip and both calcaneus. Moreover, a more diffuse and heterogeneous prominent uptake appeared on rib cage, spine and pelvis. These images suggested a diffuse metastatic disease of the skeleton. The single photon emission computerized tomography guided by computerized tomography (SPECT/CT), centered on lumbar spine, pelvis and the upper end of femurs showed that the multiple hot spots were infact bone fractures. These findings pointed diagnosis to a metabolic disease. The clinical context was in favour of an osteomalacia. Further explorations showed an osteomalacia related to phosphate diabetes. A thorough work-up did not reveal any known aetiology. To date, idiopathic phosphate diabetes seems the most likely diagnosis. Nuclear medicine input in osteomalacia is discussed.     

Subungual Exostosis in an Osteoporotic Patient Treated with Teriparatide

ObjectiveTo report an unusual case of subungual exostosis in a patient on teriparatide.MethodsWe describe the presentation and symptoms of the patient and review of the relevant literature.ResultsTeriparatide is used for the treatment of osteoporosis. Rat studies using 3-60x the approved human dose have shown an association between teriparatide and an increased risk of osteosarcoma. Subungual exostosis, to our knowledge, has not been reported. We report the case of a 54-year-old female who presented with a 4-month history of pain and swelling in the medial side of her right thumb with no preceding trauma. The patient had history of severe osteoporosis with multiple fractures and was on teriparatide for 16 months. On examination, the right thumb was swollen and tender with no superficial erythema or signs of an infection. X-ray imaging revealed a trabecular bony overgrowth consistent with subungual exostosis. The patient was treated with subungual excision. Pathology showed endochondral bone formation with reactive atypia, consistent with osteocartilaginous exostosis.ConclusionTo our knowledge, this is the first case hypothesizing an association between teriparatide and subungual exostosis. Subungual exostosis is a benign growth of bone that arises in the distal phalanx, under or adjacent to the nail bed. The pathophysiology is not clearly understood, but the lesion has base of trabecular bone with a proliferating fibrocartilaginous cap. Teriparatide can stimulate the trabecular bone formation. Hence, an association between the use of teriparatide and the development of subungal exostosis cannot be excluded. Further studies delineating this relationship are needed. (Endocr. Pract. 2013;19:e115-e117)     

Ostéomalacie secondaire au cisplatine : un diagnostic difficile

IntroductionCisplatin is an antineoplastic agent which can cause renal magnesium loss.Case reportA 42-year-old female followed for a primary duodenal large B-cell lymphoma treated with 12 cycles of chemotherapy including “CHOP” (cyclophosphamide, adriamycin, vincristine, prednisone) and bleomycin then 3 cures “DHAP” (cisplatin, cytosine-arabinoside, dexamethasone) consulted for bone pain with muscle cramps. Serum calcium and magnesium were low. The radiograph of the pelvis showed an osteolytic lesion in the right sacroiliac joint. The bone scan showed increased uptakes in the left fronto-parietal bone, the right sacroiliac and the mandible. Iliac, sacral and skull biopsies were negative. The parathormone value was 564 ng, l, 25-(OH) vitamin D lower than 7 μg/L and 1, 25-(OH) 2 vitamine D 15 ng/L. Bone densitometry showed osteopenia. The diagnosis was osteomalacia caused by hypomagnesemia secondary to cisplatin.ConclusionCisplatin can cause osteomalacia through hypomagnesemia.     

X-Linked hypophosphatemic rickets: a disease often unknown to affected patients

X-Linked hypophosphatemic rickets (XLH) is an X-linked dominant disorder that is secondary to renal phosphate wasting. Affected individuals frequently present the following characteristics: short stature, lower-extremity deformity, bone pain, dental abscesses, en-thesopathy, rickets, and osteomalacia. Since the disorder is characterized by evident phenotypic abnormalities, we hypothesized that there would be a high degree of knowledge about the disease in affected kindreds. Thus, we constructed a six-page, self-administered questionnaire to determine whether family members are, in fact, aware of their disease and properly diagnosed and treated. We also designed the survey to determine rates of symptoms thought to be associated with rickets/osteomalacia in a population with a lower referral bias than is usually seen in tertiary care centers. We administered the questionnaire to 234 study subjects (57 affected) who were members of one of three large kindreds. Although 62% of affected individuals knew they had some problem with their bones, only 22.6% were told by a physician that they had rickets or osteomalacia. This apparent lack of awareness occurred in spite of 61.1% of affected subjects complaining of bone or joint problems to their personal physician. Indeed, of those patients who had persistent complaints, only 34.5% were told they had rickets or osteomalacia. Only one patient was taking phosphate and vitamin D. The spectrum of symptoms evident in affected subjects compared with normals included: dental abscesses (54.5% vs. 13.0%, P < 0.001), bone pain (45.5% vs. 28.2%, P = 0.027), back pain (51.8% vs. 35.1%, P = 0.036), joint stiffness (48.2% vs. 16.8%, P < 0.001), joint pain (55.4% vs. 31.1%, P = 0.003), weakness (25.0% vs. 10.7%, P = 0.023), and hearing loss (28.6% vs. 9.8%, P = 0.002). Surprisingly, although affected individuals complained of many symptoms due to XLH, they fractured bones less frequently than controls (20% vs. 38.1%., P = 0.018). Our data demonstrate that, despite the presence of disease in family members, few affected subjects knew that they had XLH. Although the presence of symptoms did increase knowledge of disease status, only one-third of symptomatic individuals knew of their diagnosis.     

A cannabinoid 2 receptor agonist attenuates bone cancer-induced pain and bone loss

AimsCannabinoid CB₂ agonists have been shown to alleviate behavioral signs of inflammatory and neuropathic pain in animal models. AM1241, a CB₂ agonist, does not demonstrate central nervous system side effects seen with CB₁ agonists such as hypothermia and catalepsy. Metastatic bone cancer causes severe pain in patients and is treated with analgesics such as opiates. Recent reports suggest that sustained opiates can produce paradoxical hyperalgesic actions and enhance bone destruction in a murine model of bone cancer. In contrast, CB₂ selective agonists have been shown to reduce bone loss associated with a model of osteoporosis. Here we tested whether a CB₂ agonist administered over a 7 day period inhibits bone cancer-induced pain as well as attenuates cancer-induced bone degradation.Main methodsA murine bone cancer model was used in which osteolytic sarcoma cells were injected into the intramedullary space of the distal end of the femur. Behavioral and radiographic image analysis was performed at days 7, 10 and 14 after injection of tumor cells into the femur.Key findingsOsteolytic sarcoma within the femur produced spontaneous and touch evoked behavioral signs of pain within the tumor-bearing limb. The systemic administration of AM1241 acutely or for 7 days significantly attenuated spontaneous and evoked pain in the inoculated limb. Sustained AM1241 significantly reduced bone loss and decreased the incidence of cancer-induced bone fractures.SignificanceThese findings suggest a novel therapy for cancer-induced bone pain, bone loss and bone fracture while lacking many unwanted side effects seen with current treatments for bone cancer pain.     

Oncogenic Osteomalacia Cured by Removal of an Organized Hematoma

ObjectiveTo describe a patient with oncogenic osteomalacia whose symptoms were rapidly resolved after surgical removal of an organized hematoma of the hip.MethodsA case report is presented, including clinical and laboratory findings. The relevant literature is reviewed, and the current understanding of oncogenic osteomalacia is summarized.ResultsIn September 1996, a 44-year-old black woman presented with a 2-year history of bone pain, progressive muscle weakness, depression, osteomalacia, and hypophosphatemia. Her condition did not improve with use of calcitriol and phosphate replacement. During the previous year, her serum phosphorus levels were low, ranging from 1.0 to 2.2 mg/dL, and the levels of serum 1,25-dihydroxyvitamin D [1,25-(OH)₂D] were very low, ranging from < 5 to 19.4 pg/mL (normal, 15 to 60). The serum 25-hydroxyvitamin D levels were low, ranging from 8 to 14 ng/mL (normal, 9 to 52). The higher values were noted after she had received large doses of phosphate, 1,25-(OH)₂D, and vitamin D. During the previous year, her serum alkaline phosphatase levels were high, ranging from 253 to 314 U/L; serum calcium and parathyroid hormone levels were normal. The abnormalities on physical examination were obesity and a 10- by 10-cm firm, poorly demarcated mass superior to the left greater trochanter. A computed tomographic scan of this region showed a water-density fluid collection in the left buttock measuring 7.8 by 7.8 cm, consistent with a chronic hematoma. The mass was resected, and histopathologic examination revealed features of an organized hematoma with areas of myxoid changes and cartilaginous metaplasia. Postoperatively, the patient’s strength improved, and the levels of serum phosphorus and 1,25-(OH)₂D became supranormal.ConclusionThe symptoms and laboratory abnormalities of this patient with oncogenic osteomalacia promptly resolved after resection of an organized hematoma of the left hip. (Endocr Pract. 2005;11:190-193)     

Tumor-induced osteomalacia: a single center experience

ObjectiveTo describe the clinical presentation, localization modalities, and management of patients with tumor-induced osteomalacia (TIO).MethodsWe performed a retrospective analysis of case records of patients diagnosed with TIO between January 1996 and March 2010 at our institution in Mumbai, India.ResultsNine patients (6 female and 3 male) with a mean age of 37.5 ± 17.5 years with biochemical and imaging evidence of TIO were included in the study. Overall, patients presented with proximal muscle weakness and pain. Three patients had neurofibromatosis 1, one had isolated schwannoma, and one had epidermal nevus syndrome. The mean delay in diagnosis was 7.67 years. Biochemical studies revealed normal serum calcium (mean, 9.2 ± 0.8 mg/dL), low serum phosphorus (mean, 1.36 ± 0.54 mg/dL), and low maximal tubular reabsorption of phosphorus factored for glomerular filtration rate (mean, 0.94 ± 0.49 mg/dL). Fibroblast growth factor-23 was increased in 3 of the patients without neurofibromatosis but was normal or near-normal in all the patients with neurofibromas. A fludeoxyglucose F 18 positron emission tomography (FDG PET) scan helped to localize the tumors in 4 of the 5 patients with diagnoses other than neurofibromatosis. Three patients underwent surgical excision and were cured. One patient underwent biopsy and partial excision. Histopathologic findings were suggestive of phosphaturic mesenchymal tumor, benign fibrous histiocytoma, nonossifying fibroma, and sciatic nerve schwannoma.ConclusionThere is a well-known delay in the diagnosis of TIO. FDG PET is a useful modality for localization of tumors. Preoperative localization increases the odds for cure after surgical excision. (Endocr Pract. 2011;17: 177-184)     

Clinical and Bone Density Outcomes of Tumor-Induced Osteomalacia after Treatment

ObjectiveTo report the outcomes of tumor-induced osteomalacia after treatment, particularly related to recovery of bone mass.MethodsWe review the clinical course of a 61-year-old man extremely debilitated from multiple fractures and neuromuscular weakness due to tumor-induced osteomalacia and report the changes in biochemical markers and bone density after removal of the causative neoplasm.ResultsAt the time of diagnosis, the patient’s serum phosphorus and 1,25 dihydroxyvitamin D levels were depressed, and his fibroblast growth factor-23 level was markedly elevated. These values normalized 2 days after surgery and remained within their respective reference ranges 4 and 12 months after resection of a mesenchymal tumor. Lumbar bone density values (T-scores) were 0.445 g/cm² (-5.9) preoperatively, 0.939 g/cm² (–1.4) 4 months after surgery, and 1.152 g/cm² (0.7) 12 months after surgery. Left femoral neck values at the same time points were 0.525 g/cm² (-3.0), 1.035 g/cm² (-0.8), and 1.184 g/cm² (1.9). Ultra-distal radius values at the same time points were 0.128 g/cm² (-7.0), 0.191 g/cm² (-5.9), and 0.259 g/cm² (-4.8). In addition, he recovered neuromuscular function and was able to leave his wheelchair.ConclusionTumor-induced osteomalacia can be an extremely debilitating disease. With successful localization, identification, and resection of the neoplasm, bone mass and physical function can recover. (Endocr Pract. 2007;13:458-462)     

The Risk of Bone Fractures in Post-Poliomyelitis Patients Transitioning to Middle Adulthood

ObjectiveWhile osteoporotic fractures are reported in up to 40% of adults with post-poliomyelitis syndrome (PPS), clinical guidelines regarding bone mineral density (BMD) and indications for treatment are scarce. We investigated the characteristics of PPS patients, focusing on fractures and osteoporosis as the primary outcomes.MethodsA cross-sectional retrospective data analysis from medical records of 204 PPS patients regarding their clinical characteristics and long-term outcome, with emphasis on bone metabolism status.ResultsOur cohort included 53% women; mean age was 65 years at study entry and 1.7 years at the diagnosis of acute poliomyelitis. The lower limb was involved in 97.5% of patients, and the BMD in the affected limb tended to be lower than the unaffected, with a mean T-score of -1.64 vs. -1.19, respectively (P = .06). Recurrent falls were documented in 39.2% of patients, and osteoporosis in 20.6%, being more frequent in women (P = .003) and patients with fractures (P = .002). At least one fracture occurred in 52.2% of patients, and more than one in 40.3%. The median age for the first fracture was 57.5 years (range, 30 to 83 years), and most fractures occurred in the affected limb (73.2%).ConclusionsUnderdiagnosis and delayed treatment of osteoporosis in late-adulthood post-poliomyelitis patients underlie the need for comprehensive clinical guidelines to manage these patients, including recommendations on bone health assessment, medical treatment, and their inclusion as a high-risk group for bone fractures.     

人工髋关节置换术治疗高龄患者股骨粗隆间骨折的临床疗效研究

目的:探究人工髋关节置换术治疗高龄患者股骨粗隆间骨折的临床疗效。方法:选择40例高龄股骨粗隆间骨折患者并将其随机分为两组,观察组给予人工髋关节置换术治疗,对照组经股骨近端髓内钉内固定术治疗。观察比较两组L2-4椎体骨密度、疼痛程度、髋关节功能恢复情况及Harris评分。结果:治疗后,两组L2-4椎体骨密度均显著高于治疗前(P0.05),且观察组显著高于对照组(P0.05);观察组疼痛比例较治疗前明显降低(P0.05),且显著低于对照组(P0.05);观察组髋关节恢复优良率为95.0%,显著高于对照组(70.0%,P0.05);两组Harris评分均显著高于治疗前(P0.05),且观察组显著高于对照组(P0.05)。结论:采用人工髋关节置换术治疗高龄股骨粗隆间骨折的疗效明显优于经股骨近端髓内钉内固定术治疗,其可有效促进髋关节功能恢复,减轻患者疼痛。     

Coexistence of tumor-induced osteomalacia and primary hyperparathyroidism

ObjectiveTo present an unusual case of coexisting tumor-induced osteomalacia (TIO) and primary hyperparathyroidism (PHPT).MethodsWe report the clinical features, imaging studies, and the results of laboratory investigations before and after surgical resection of both a soft-tissue tumor and a parathyroid adenoma.ResultsA 44-year-old woman was referred to the endocrinology department with a diagnosis of PHPT accompanied by unusually severe hypophosphatemia, despite having received treatment with cinacalcet. Debilitating muscle weakness and bone pain, severe phosphaturia and hypophosphatemia, inappropriately normal calcitriol, and elevated fibroblast growth factor-23 and intact parathyroid hormone levels raised the suspicion of coexisting TIO and PHPT. Imaging studies were negative, but histologic characteristics of a palpable subcutaneous mass from the patient’s thigh revealed a phosphaturic mesenchymal tumor. Complete remission after surgical removal of both the soft-tissue tumor and the parathyroid adenoma confirmed the diagnosis.ConclusionThe coexistence of TIO and PHPT has not been described before and can cause life-threatening hypophosphatemia. Diagnosis and localization of the tumor is of paramount importance since surgery is the treatment of choice for both TIO and PHPT. (Endocr Pract. 2011;17:e144-e148)