scholarly journals A Case of Femoral Neck Insufficiency Fracture due to Tumor-Induced Osteomalacia

2021 ◽  
Vol 2021 ◽  
pp. 1-5
Author(s):  
Yu Inoue ◽  
Tomoaki Fukui ◽  
Keisuke Oe ◽  
Shinya Hayashi ◽  
Teruya Kawamoto ◽  
...  
Keyword(s):  
Femoral Neck ◽  
Fibroblast Growth Factor 23 ◽  
Insufficiency Fracture ◽  
Tumor Resection ◽  
Rotational Stability ◽  
Insufficiency Fractures ◽  
Fracture Union ◽  
Delayed Fracture ◽  
Calcium And Phosphorus ◽  
Fgf 23

Tumor-induced osteomalacia (TIO) is a rare skeletal disease caused by hypersecretion of fibroblast growth factor 23 (FGF-23) from neoplasms of mesenchymal origin; patients with TIO present with insufficiency fractures, progressive bone pain, and delayed fracture unions. Herein, we report the case of a 48-year-old man with an insufficiency fracture in his left femoral neck associated with TIO. The causative tumor located in the patient’s maxillary sinus had been resected; however, complete resection was impossible due to the location of the tumor. Therefore, the patient’s osteomalacia persisted, and he experienced a left femoral neck fracture in the absence of severe trauma. Because delayed fracture union was anticipated in this patient, we performed an internal fixation using an implant with a lateral plate for angular stability and multiple screws for rotational stability. Although fracture union took 15 months, the patient’s postoperative course was uneventful, and he could walk without any symptoms or assistance at his most recent follow-up 30 months after surgery. In TIO, hypersecretion of FGF-23 leads to increased renal excretion of phosphorus, increased bone resorption of calcium and phosphorus, decreased osteoblastic bone mineralization, and decreased gastrointestinal absorption of calcium and phosphorus, leading to insufficiency fractures and delayed fracture unions. Diagnosis of TIO is often delayed due to its rarity and vague symptoms. Total resection of the causative tumor is the optimal treatment; however, in cases wherein complete tumor resection is impossible, drug therapy may be insufficient, and the underlying TIO pathology, including bone fragility, may persist. Early diagnosis of TIO is important for preventing insufficiency fractures; however, when fractures are unavoidable, the surgical treatment of femoral neck fractures in patients with osteomalacia should account for a longer time frame for complete fracture union and therefore utilize implants with sufficient stability.

Insufficiency Fractures vs. Low-Energy Pelvic Ring Fractures – Epidemiological, Diagnostic and Therapeutic Characteristics of Fragility Fractures of the Pelvic Ring

2021 ◽  
Author(s):  
Manuel Sterneder ◽  
Patricia Lang ◽  
Hans-Joachim Riesner ◽  
Carsten Hackenbroch ◽  
Benedikt Friemert ◽  
...  
Keyword(s):  
Pelvic Ring ◽  
Diagnostic Testing ◽  
Insufficiency Fracture ◽  
Fragility Fractures ◽  
Relative Increase ◽  
Trauma Centre ◽  
Low Energy ◽  
Insufficiency Fractures ◽  
Type Iv ◽  
Pelvic Ring Fractures

Abstract Background Fragility fractures of the pelvis (FFP) encompass two fracture entities: fracture after low-energy trauma and insufficiency fracture without trauma. It is unclear whether the two subgroups differ in terms of diagnosis and therapy. The aim of this retrospective study was to evaluate insufficiency fractures with regard to defined parameters and to compare specific parameters with the fractures after low-energy trauma. Patients and Methods In the period from 2008 to 2017, 203 patients with FFP were recorded at our clinic (Level 1 Trauma Centre DGU, SAV approval). Of these, 25 had an insufficiency fracture and 178 had a pelvic ring fracture after low-energy trauma. Epidemiological, diagnostic and therapeutic parameters were examined. Results There was a relative increase in the insufficiency fracture within the FFP (2008 – 2009: 5.0% vs. 2015 – 2017: 17.8%). In these patients, osteoporosis tended to be more pronounced than in patients after low-energy trauma (t-value: − 3.66 vs. − 3.13). The diagnosis of insufficiency fractures showed increased use of MRI and DECT (60.9% vs. 26.0%) and a high proportion of type IV fractures after FFP (40.0% vs. 7.9%). In terms of therapy, surgical treatment of the insufficiency fracture was sought more often (68,2% vs. 52,1%), with a tendency towards increased use of combined osteosynthesis procedures (14.3% vs. 7.6%). Conclusion We were able to show that as the number of cases increases, the insufficiency fracture becomes more important within FFP. If these patients tend to have more pronounced osteoporosis, particular attention should be paid to the diagnosis and adequate therapy of the osteoporosis, especially in the case of an insufficiency fracture. In addition to the increased diagnostic testing using MRI and DECT to detect oedema and the increased surgical therapy for this type of fracture, it is also noteworthy that the insufficiency fracture can cause higher-grade fractures after FFP.

scholarly journals THU0421 TUMOR-INDUCED OSTEOMALACIA: DATA FROM A MONOCENTRIC EXPERIENCE ON 16 PATIENTS

2020 ◽  
Vol 79 (Suppl 1) ◽  
pp. 448.2-449
Author(s):  
C. Crotti ◽  
F. Bartoli ◽  
M. Manara ◽  
P. A. Daolio ◽  
F. Zucchi ◽  
...  
Keyword(s):  
Functional Imaging ◽  
Serum Alkaline Phosphatase ◽  
Fibroblast Growth Factor 23 ◽  
Diagnostic Delay ◽  
The Body ◽  
Definitive Treatment ◽  
Surgical Removal ◽  
Tumor Biopsy ◽  
Pet Ct ◽  
Fgf 23

Background:Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome due to a phosphaturic tumor, which overproduces fibroblast growth factor-23 (FGF-23), causing hyperphosphaturia, hypophosphoremia, low 1,25(OH)2VitD3and osteomalacia. Locating the tumor is critical, because lesions are typically small, benign mesenchymal tumors, anywhere in the body; the delay between onset of symptoms and diagnosis ranges from 2.5–28 years. Surgical removal is the only effective therapeutic approach.Objectives:To retrospectively evaluate patients affected by TIO, investigating clinical management and disease outcome.Methods:We retrospectively collected data of patients affected by TIO referred to a tertiary Rheumatology Center between Sep 2000 and Jan 2020.Results:We included 16 patients with a definite diagnosis of TIO, mean age±standard deviation 62.4±14.6 yrs, 56.2% females, mean age at symptoms onset 48.0±14.3 yrs (53.8±13.1 at diagnosis). Mean diagnostic delay between symptoms onset and tumor detection was 6.8±6.4 yrs. All patients complained bone pain, muscle weakness, and fractures before diagnosis of TIO. Biochemical findings were: mean serum Phosphorus (PS) 1.4±0.4 mg/dL (reference range (RR) 2.5-4.6), mean serum Calcium 9.4±0.7 mg/dL (RR 8.4-10.2), mean serum 1,25(OH)2VitD330.5±23.4 ng/L (RR 25-86). Intact-FGF-23 was dosed in 9 patients, always resulting elevated: mean 396.6±707.3 pg/mL (RR 25-45). PTH was increased in 30% of cases, while serum alkaline phosphatase was increased in 87.5%. 24h-Urine Phosphorus (PU) was increased in only 13% of patients, but, when renal phosphate wasting by tubular reabsorption of phosphate (TRP) was calculated, PU resulted increased in all.Tumor was localized in all cases (Fig.1) and were localized in bone and soft tissue, by using functional imaging, followed by anatomical techniques. Before the introduction in routinely practice of68Ga-DOTATATE-PET-CT in 2013, Octreoscan-SPECT/CT and18F FDG-PET were used as imaging modalities. Since 2013, diagnostic delay consistently reduced, from 8.6±8.3 yrs (7 patients) to 4.5±2.6 yrs (9 patients), confirming higher diagnostic accuracy of68Ga-DOTATATE-PET-CT.Figure 1.13 patients underwent surgery; in two cases surgery was not possible due to tumor location, so pharmacological support with phosphate supplements and calcitriol was started; a patient underwent to TC-guided radiofrequency ablation. After surgery, 7 patients experienced a complete remission, 3 had a persistence of the disease, and 3 an overtime relapse, even after a longstanding normalization of PS (6 years). After surgical tumor removal, PS significantly increased in few days (from 1.36±0.39 to 2.9±1.1, p=0.0001), while iFGF-23 levels tended to rapidly decreased (from 396.6±707.3 to 62.8±78.4). Before the introduction of68Ga-DOTATATE-PET-CT, 6 patients underwent to imaging-guided closed biopsy to confirm tumor localization; by using68Ga-DOTATATE-PET-CT only 2 subjects had closed biopsy. Furthermore, in our population only patients who had biopsy to detect the lesion (7 patients) had relapses compared to patients who did not.Conclusion:To our knowledge, this is the widest European cohort of patients affected by TIO reported in the last two decades. We confirm an important delay between symptoms onset and diagnosis. To locate tumor, a stepwise approach is recommended, starting with a thorough medical history and physical examination, followed by functional imaging, preferring68Ga-DOTATATE-PET-CT. Tumor biopsy is not recommended due to the potential cell spilling. Surgery is considered the only definitive treatment, aiming to a wider excision. Active surveillance is always needed, due to the possible relapses, even after a long period of complete clinical and biochemical remission.Disclosure of Interests: :Chiara Crotti: None declared, Francesca Bartoli: None declared, Maria Manara Consultant of: Consultant and/or speaker for Eli-Lilly, MSD, Sanofi-Genzyme, Novartis, Alfa Wasserman and Cellgene, Speakers bureau: Consultant and/or speaker for Eli-Lilly, MSD, Sanofi-Genzyme, Novartis, Alfa Wasserman and Cellgene, Primo Andrea Daolio: None declared, Francesca Zucchi: None declared, Roberto Caporali Consultant of: AbbVie; Gilead Sciences, Inc.; Lilly; Merck Sharp & Dohme; Celgene; Bristol-Myers Squibb; Pfizer; UCB, Speakers bureau: Abbvie; Bristol-Myers Squibb; Celgene; Lilly; Gilead Sciences, Inc; MSD; Pfizer; Roche; UCB, Luigi Sinigaglia: None declared, Massimo Varenna: None declared

scholarly journals FGF-23 and Phosphate in Children with Chronic Kidney Disease: A Cross-Sectional Study in Kazakhstan

Medicina ◽  
2020 ◽  
Vol 57 (1) ◽  
pp. 15
Author(s):  
Altynay Balmukhanova ◽  
Kairat Kabulbayev ◽  
Harika Alpay ◽  
Assiya Kanatbayeva ◽  
Aigul Balmukhanova
Keyword(s):  
Chronic Kidney Disease ◽  
Kidney Disease ◽  
Fibroblast Growth Factor 23 ◽  
Cross Sectional Study ◽  
Sectional Study ◽  
Cross Sectional ◽  
Ckd Stage ◽  
Advanced Stages ◽  
Fgf 23 ◽  
Stage 1

Background and objectives: Chronic kidney disease (CKD) in children is a complex medical and social issue around the world. One of the serious complications is mineral-bone disorder (CKD-MBD) which might determine the prognosis of patients and their quality of life. Fibroblast growth factor 23 (FGF-23) is a phosphaturic hormone which is involved in the pathogenesis of CKD-MBD. The purpose of the study was to determine what comes first in children with CKD: FGF-23 or phosphate. Materials and Methods: This cross-sectional study included 73 children aged 2–18 years with CKD stages 1–5. We measured FGF-23 and other bone markers in blood samples and studied their associations. Results: Early elevations of FGF-23 were identified in children with CKD stage 2 compared with stage 1 (1.6 (1.5–1.8) pmol/L versus 0.65 (0.22–1.08), p = 0.029). There were significant differences between the advanced stages of the disease. FGF-23 correlated with PTH (r = 0.807, p = 0.000) and phosphate (r = 0.473, p = 0.000). Our study revealed that the elevated level of FGF-23 went ahead hyperphosphatemia and elevated PTH. Thus, more than 50% of children with CKD stage 2 had the elevating level of serum FGF-23, and that index became increasing with the disease progression and it achieved 100% at the dialysis stage. The serum phosphate increased more slowly and only 70.6% of children with CKD stage 5 had the increased values. The PTH increase was more dynamic. Conclusions: FGF-23 is an essential biomarker, elevates long before other markers of bone metabolism (phosphate), and might represent a clinical course of disease.

scholarly journals Increased Levels of sRAGE in Diabetic CKD-G5D Patients: A Potential Protective Mechanism against AGE-Related Upregulation of Fibroblast Growth Factor 23 and Inflammation

2017 ◽  
Vol 2017 ◽  
pp. 1-9 ◽  
Author(s):  
Elena Dozio ◽  
Valentina Corradi ◽  
Elena Vianello ◽  
Elisa Scalzotto ◽  
Massimo de Cal ◽  
...  
Keyword(s):  
Cardiovascular Risk ◽  
Growth Factor ◽  
Fibroblast Growth Factor ◽  
Cardiac Remodeling ◽  
Glycated Albumin ◽  
Fibroblast Growth Factor 23 ◽  
Protective Mechanism ◽  
Fibroblast Growth ◽  
Increased Risk ◽  
Fgf 23

Advanced glycation end products (AGEs) may induce cardiac remodeling in kidney disease by promoting fibroblast growth factor 23 (FGF-23) expression. Since AGEs are increased in diabetes mellitus (DM), our first aim was to evaluate the existence of any potential association between AGEs, FGF-23, inflammation, and increased cardiovascular risk in DM patients on dialysis (CKD-G5D). Secondarily, we explored the potential role of the soluble receptor for AGEs (sRAGE) as a marker of heart failure. Levels of glycated albumin (GA), sRAGE, c-terminal FGF-23 (cFGF-23), brain natriuretic peptide (BNP), and inflammatory mediators were compared between DM and non-DM CKD-G5D patients. The levels of sRAGE, cFGF-23, BNP, and proinflammatory markers were over the ranges of normality in both DM and non-DM groups. Only GA and sRAGE levels were increased in DM compared to non-DM patients. Plasma levels of sRAGE and CRP were the only independent predictors of BNP concentration. In conclusion, in DM CKD-G5D patients, sRAGE appeared to be a marker of cardiac remodeling. Indeed, its increase could be a potential protective mechanism against the increased risk of cardiovascular complications related to AGEs and inflammation. The causal relationship between sRAGE and cardiovascular risk in these patients needs to be further confirmed by mechanistic studies.

scholarly journals Insufficiency Fracture in the Para-Acetabulum, with Features Mimicking those of a Malignant Bone Tumor

2008 ◽  
Vol 1 ◽  
pp. CCRep.S803
Author(s):  
Akio Sakamoto ◽  
Takuaki Yamamoto ◽  
Kazuhiro Tanaka ◽  
Shuichi Matsuda ◽  
Tatsuya Yoshida ◽  
...  
Keyword(s):  
Bone Tumor ◽  
Signal Intensity ◽  
Insufficiency Fracture ◽  
Hip Pain ◽  
Pelvic Bone ◽  
High Signal ◽  
Insufficiency Fractures ◽  
Mineral Density ◽  
Abnormal Signal ◽  
High Index Of Suspicion

Para-acetabular insufficiency fractures are rare and exceedingly difficult to diagnose without a high index of suspicion, since the images mimic those of bone tumors. We herein present the case of a 55-year-old woman who suffered from hip pain with subacute onset. She had undergone a hysterectomy-ovariectomy due to endometriosis when she was 41 years old. Her bone mineral density was normal due to supplemental treatment with female hormones. About 3 months after onset, she was referred to our institute with a diagnosis of pelvic bone tumor. Plain radiographs and computed tomography showed irregular osteosclerosis in the para-acetabulum. Bone scintigraphy demonstrated uptake in the para-acetabulum. Magnetic resonance imaging showed abnormal signal with low-signal intensity on T1-weighted images and high-signal intensity on T2-weighted images throughout the entire hemipelvic bone. Since the pain continued for more than 3 months, open biopsy was undertaken and the lesion was found to be non-neoplastic. Six months after onset, the pain disappeared. The clinical course suggested a diagnosis of insufficiency fracture in the para-acetabulum. Para-acetabular insufficiency fractures should always be considered in cases of hip pain, even in patients with prolonged symptoms.

scholarly journals MP594THE RELATIONSHIP BETWEEN FIBROBLAST GROWTH FACTOR-23 (FGF-23) AND SELECTED CLINICAL AND LABORATORY PARAMETERS

2017 ◽  
Vol 32 (suppl_3) ◽  
pp. iii648-iii649
Author(s):  
Danuta Fedak ◽  
Marek Kużniewski ◽  
Marcin Krzanowski ◽  
Paulina Dumnicka ◽  
Wladyslaw Sulowicz
Keyword(s):  
Growth Factor ◽  
Fibroblast Growth Factor ◽  
Fibroblast Growth Factor 23 ◽  
Fibroblast Growth ◽  
Laboratory Parameters ◽  
Fgf 23

scholarly journals Fibroblast Growth Factor 23 and Blood Pressure in Older Adults

Hypertension ◽  
2020 ◽  
Vol 76 (1) ◽  
pp. 236-243
Author(s):  
David A. Drew ◽  
Ronit Katz ◽  
Stephen Kritchevsky ◽  
Joachim H. Ix ◽  
Michael G. Shlipak ◽  
...  
Keyword(s):  
Blood Pressure ◽  
Older Adults ◽  
Growth Factor ◽  
Fibroblast Growth Factor 23 ◽  
Fibroblast Growth ◽  
Antihypertensive Medications ◽  
Incident Hypertension ◽  
Blood Pressures ◽  
The Mean ◽  
Fgf 23

FGF-23 (fibroblast growth factor 23) regulates phosphorus and vitamin D. Elevated FGF-23 is associated with incident hypertension in young- and middle-aged adults, but there is limited data in older adults. Serum FGF-23 was measured using an intact ELISA assay in 2496 participants of the Healthy Aging and Body Composition Study. The association between FGF-23 and prevalent hypertension (self-reported and confirmed by use of antihypertensive medications) and number of antihypertensive medications was determined. The associations between FGF-23 and incident hypertension, and diastolic and systolic blood pressure trajectories were evaluated over 10 years. Models were adjusted for demographics, estimated glomerular filtration rate and albuminuria, cardiovascular disease risk factors, and measures of mineral metabolism. The mean (SD) age was 75 (3) years, with 51% women, and 40% black participants. The prevalence of hypertension at baseline was 75% and the mean systolic and diastolic blood pressures were 134 (21) mm Hg and 70 (12) mm Hg, respectively. The majority of participants without hypertension at baseline developed incident hypertension (576 of 1109 or 52%). In adjusted models, each 2-fold higher FGF-23 was associated with prevalent baseline hypertension (odds ratio=1.46 [1.24–1.73]) and greater number of blood pressure medications (IRR=1.14 [1.08–1.21]) but not with baseline diastolic or systolic blood pressure. In fully adjusted longitudinal analyses, a 2-fold higher FGF-23 was associated with incident hypertension (hazard ratio=1.18 [1.03–1.36]) and worsening systolic blood pressures (β=0.24 [0.08–0.40] mm Hg per year increase), but not with diastolic blood pressures (β=0.04 [−0.04 to 0.12] mm Hg per year increase). Higher FGF-23 concentrations are associated with prevalent and incident hypertension as well as rising systolic blood pressures in community-living older adults.

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