Treatment for Gorham-Stout syndrome with a combination of teriparatide and denosumab.

Gorham-Stout syndrome is an aggressive, non-hereditary, and rare disease affecting bone metabolism. Its etiology and pathogenesis remain elusive. The syndrome manifests with diverse clinical symptoms, often leading to frequent misdiagnoses and presenting challenges in treatment. In this study, we report a case of cranial and maxillary osteolysis in a 47-year-old female patient with somatic mutations in the VEGF-A, VEGF-B, and VEGF-C genes and the EPHB4 gene. After treatment with bisphosphonates, this patient still had persistent resorption of the mandible, but switching to a teriparatide and denosumab combination yielded substantial improvement. This study is the first report to show that teriparatide combined with denosumab can be used to treat Gorham-Stout syndrome.

Medication-related osteonecrosis of the lower jaw without osteolysis on computed tomography images.

INTRODUCTION: Surgery is the standard treatment for medication-related osteonecrosis of the jaw (MRONJ). This study reviewed patients with mandibular MRONJ who underwent surgical treatment, and in particular the characteristics of non-osteolytic MRONJ with no evidence of osteolysis on CT were described. MATERIALS AND METHODS: We conducted a retrospective study of patients with mandibular MRONJ who underwent surgery between January 2016 and September 2022. Various clinical and imaging factors regarding treatment outcomes were investigated and analyzed. Additionally, the disease course of non-osteolytic MRONJ was examined in detail. RESULTS: This study included 55 patients (66 surgeries) with a mean age of 74.7. The primary disease was osteoporosis (24 patients) and malignancy (31 patients); the type of antiresorptive agent was bisphosphonate (BP) in 21 patients and denosumab (DMB) in 26. BP was initially administered; however, it was changed to DMB in eight patients. Preoperatively, the cumulative cure rates for all 66 surgeries were 72.8% at 1 year and 77.3% at 2 years. Cure rates were significantly lower in patients with malignancy, those without osteolysis, and those who underwent sequestrum removal or marginal mandibulectomy than those with osteoporosis, osteolysis, and segmental mandibulectomy. Non-osteolytic MRONJ was observed in eight patients, all with malignancy and receiving high-dose DMB. Only two patients were cured after the initial surgery, and most patients ultimately underwent segmental mandibulectomy. CONCLUSIONS: Surgical treatment yielded good treatment outcomes in most patients with mandibular MRONJ; however, the cure rate was lower in patients with malignancy who showed no osteolysis on CT images.

Effects of apical periodontitis treatment on hyperglycaemia in diabetes: A prospective cohort study.

AIM: This prospective cohort study was undertaken to evaluate the success rate of root canal treatment (RCT) in type 2 diabetes mellitus (T2DM) patients with targeted level and unachieved targeted level of glycaemic control as well as the impact of RCT on the glucose blood level in T2DM patients. METHODOLOGY: Patients needing RCT were divided into three groups: these without T2DM, that is, the control group (CG), those with targeted level of glycated haemoglobin HbA1c < 7% (TL A1c) and the third ones with unachieved targeted level (UTL A1c), that is, with HbA1c ≥ 7%. Before RCT, HbA1c and the periapical index (PAI) score were assessed, as well as 1 year later. RESULTS: Our results showed less favourable treatment results of RCT such as a reduction of radiographic lesions in T2DM patients, particularly in subjects with UTL A1c. The intergroup analysis of PAI score at the 12-month follow-up revealed a significant difference in TL A1C (p = .022) and CG (p = .001) with respect to UTL A1c. Total number of healed teeth (PAI≤2) at the 12-month after RCT in UTL A1c was significantly lower in comparison to CG (p = .008). Contrariwise, RCT may improve the glycaemic control in diabetic patients with UTL A1c after 12 months of posttreatment. Regression analysis showed that UTL A1c patients were more likely to have AP persistence after endodontic treatment (OR = 4.788; CI: 1.157-19.816; p = .031). CONCLUSIONS: T2DM retards the AP healing and conversely AP contributes to increasing the inflammatory burden in T2DM. RCT reduces the cumulative inflammatory burden in T2DM and thus may contribute to improvement of glycaemic control particularly in patients with UTL A1c.

Effect of vitamin E-enhanced highly cross-linked polyethylene on wear rate and particle debris in anatomic total shoulder arthroplasty: a biomechanical comparison to ultrahigh-molecular-weight polyethylene.

BACKGROUND: Particle-induced osteolysis resulting from polyethylene wear remains a source of implant failure in anatomic total shoulder designs. Modern polyethylene components are irradiated in an oxygen-free environment to induce cross-linking, but reducing the resulting free radicals with melting or heat annealing can compromise the component's mechanical properties. Vitamin E has been introduced as an adjuvant to thermal treatments. Anatomic shoulder arthroplasty models with a ceramic head component have demonstrated that vitamin E-enhanced polyethylene show improved wear compared with highly cross-linked polyethylene (HXLPE). This study aimed to assess the biomechanical wear properties and particle size characteristics of a novel vitamin E-enhanced highly cross-linked polyethylene (VEXPE) glenoid compared to a conventional ultrahigh-molecular-weight polyethylene (UHMWPE) glenoid against a cobalt chromium molybdenum (CoCrMo) head component. METHODS: Biomechanical wear testing was performed to compare the VEXPE glenoid to UHMWPE glenoid with regard to pristine polyethylene wear and abrasive endurance against a polished CoCrMo alloy humeral head in an anatomic shoulder wear-simulation model. Cumulative mass loss (milligrams) was recorded, and wear rate calculated (milligrams per megacycle [Mc]). Under pristine wear conditions, particle analysis was performed, and functional biologic activity (FBA) was calculated to estimate particle debris osteolytic potential. In addition, 95% confidence intervals for all testing conditions were calculated. RESULTS: The average pristine wear rate was statistically significantly lower for the VEXPE glenoid compared with the HXLPE glenoid (0.81 ± 0.64 mg/Mc vs. 7.00 ± 0.45 mg/Mc) (P < .05). Under abrasive wear conditions, the VEXPE glenoid had a statistically significant lower average wear rate compared with the UHMWPE glenoid comparator device (18.93 ± 5.80 mg/Mc vs. 40.47 ± 2.63 mg/Mc) (P < .05). The VEXPE glenoid demonstrated a statistically significant improvement in FBA compared with the HXLPE glenoid (0.21 ± 0.21 vs. 1.54 ± 0.49 (P < .05). CONCLUSIONS: A new anatomic glenoid component with VEXPE demonstrated significantly improved pristine and abrasive wear properties with lower osteolytic particle debris potential compared with a conventional UHMWPE glenoid component. Vitamin E-enhanced polyethylene shows early promise in shoulder arthroplasty components. Long-term clinical and radiographic investigation needs to be performed to verify if these biomechanical wear properties translate to diminished long-term wear, osteolysis, and loosening.

Melittin Treats Periprosthetic Osteolysis in a Rat Model by Inhibiting the NF-kB Pathway and Regulating the Ratio of Receptor Activator of Nuclear Factor Kappa B Ligand/Osteoprotegerin.

BACKGROUND: Aseptic loosening around the prosthesis is a common cause of failure in total joint arthroplasty. Polyethylene wear particles trigger the release of inflammatory factors by macrophages. Key mediators involved in osteoclastogenesis include interleukin-6, tumor necrosis factor-α, receptor activator of nuclear factor kappa B (RANK), RANK ligand (RANKL), and bone protection hormone (Osteoprotegerin [OPG]). The purpose of our experiment was to see whether melittin can slow down the release of inflammatory mediators through the NF-kB pathway, regulate the RANKL/OPG ratio, reduce osteoclast formation, and delay the onset of arthritis in rats. METHODS: A total of 20 male Sprague-Dawley rats (10 months, Specific Pathogen Free, 350 g ± 20 g) were randomly divided into 5 groups: sham group, model group, melittin concentration 1 group (0.2 mg/kg), concentration 2 group (0.4 mg/kg), and concentration 3 group (0.6 mg/kg). All rats were implanted with TA2 high-purity titanium rods. A drill was used to create a bone canal along the long axis of the femur in the intercondylar notch. The model group and experimental groups were exposed to polyethylene particles, while the sham group did not receive any particles. RESULTS: The melittin group exhibited significantly increased serum levels of serum P, calcium-phosphorus product, OPG, PINP, PINP/CTX-I, and OPG/RANKKL (P < .05). In the experimental group, micro computed tomography scanning results revealed a decrease in the amount of bone defect around the prosthesis. Immunofluorescence analysis demonstrated a decrease in the expression of IKKα and P65, while the expression of OPG showed an upward trend. Both Hematoxylin-Eosin and Tartrate-Resistant Acid Phosphatase staining revealed less osteoclast and inflammatory cell infiltration in bone resorption pits. CONCLUSIONS: Our study demonstrates that melittin has the ability to inhibit the NF-kB pathway in a rat model, and reduce the impact of RANKL/OPG, thereby delaying osteoclast activity and alleviating periprosthetic osteolysis.

No Revisions Attributable to Wear of Highly Cross-Linked Polyethylene Liners: A Long-Term Follow-Up Study.

BACKGROUND: There is a paucity of data beyond 15 years on the survivorship of total hip arthroplasty since the introduction of highly cross-linked polyethylene (HXLPE) liners. Our aim was to evaluate implant survivorship, liner wear rates, and clinical outcomes after primary total hip arthroplasty using HXLPE liners implanted between 1999 and 2002. METHODS: Between 1999 and 2002, 690 primary total hip arthroplasties utilizing 28-mm femoral heads and HXLPE liners of a single design were identified using our institutional total joint registry. Femoral heads were made of metal in 96% of cases and ceramic in 4%. The mean age was 56 years, 48% were women, and the mean body mass index was 30. Survivorship analyses were performed for the outcomes of implant revision, reoperation, and complications for the entire cohort. Linear HXLPE liner wear rates were determined on 197 hips with radiographs with more than 18.5 years of follow-up. RESULTS: At 20 years, survivorship free of revision was 94%, free of reoperation was 92%, and free of any complication was 81%. There were no documented wear-related revisions. The linear wear rate at a mean of 20.3 years postoperatively was 0.02 mm/y. There was no statistically significant difference in measured wear observed between the first available postoperative radiographs and those taken at the final follow-up. The use of elevated liners, patient body mass index, age, preoperative diagnosis, acetabular component inclination, and anteversion angles were not associated with increased wear rates. Mean Harris hip scores improved from 52 preoperatively to 90 at greater than 18.5 years CONCLUSIONS: Primary total hip arthroplasties using a single first-generation HXLPE liner demonstrated excellent survivorship and clinical outcomes at long-term follow-up with no wear-related revisions. Wear rates of HXLPE liners at 20 years are exceedingly low and are not significantly impacted by acetabular component position or patient-dependent variables such as BMI. LEVEL OF EVIDENCE: IV.

Homozygosity for a Rare Plec Variant Suggests a Contributory Role in Congenital Insensitivity to Pain.

Congenital insensitivity to pain is a rare human condition in which affected individuals do not experience pain throughout their lives. This study aimed to identify the molecular etiology of congenital insensitivity to pain in two Thai patients. Clinical, radiographic, histopathologic, immunohistochemical, and molecular studies were performed. Patients were found to have congenital insensitivity to pain, self-mutilation, acro-osteolysis, cornea scars, reduced temperature sensation, tooth agenesis, root maldevelopment, and underdeveloped maxilla and mandible. The skin biopsies revealed fewer axons, decreased vimentin expression, and absent neurofilament expression, indicating lack of dermal nerves. Whole exome and Sanger sequencing identified a rare homozygous variant c.4039C>T; p.Arg1347Cys in the plakin domain of Plec, a cytolinker protein. This p.Arg1347Cys variant is in the spectrin repeat 9 region of the plakin domain, a region not previously found to harbor pathogenic missense variants in other plectinopathies. The substitution with a cysteine is expected to decrease the stability of the spectrin repeat 9 unit of the plakin domain. Whole mount in situ hybridization and an immunohistochemical study suggested that Plec is important for the development of maxilla and mandible, cornea, and distal phalanges. Additionally, the presence of dental anomalies in these patients further supports the potential involvement of Plec in tooth development. This is the first report showing the association between the Plec variant and congenital insensitivity to pain in humans.

Therapeutic effects of curcumin liposomes and nanocrystals on inflammatory osteolysis: In vitro and in vivo comparative study.

Curcumin could inhibit periprosthetic osteolysis induced by wear debris and adherent endotoxin, which commonly cause prosthesis loosening and negatively influence the long-term survival of joint arthroplasty. However, its limited water solubility and poor stability pose challenges for its further clinical application. To address these issues, we developed curcumin liposomes for intraarticular injection, as liposomes possess good lubricant capacity and pharmacological synergy with curcumin. Additionally, a nanocrystal dosage form was prepared to enable comparison with the liposomes based on their ability to disperse curcumin effectively. A microfluidic method was used for its controllability, repeatability, and scalability. The Box-Behnken Design was employed to screen the formulations and flow parameters, while computational fluid dynamics was used to simulate the mixing process and predict the formation of liposomes. The optimized curcumin liposomes (Cur-LPs) had a size of 132.9 nm and an encapsulation efficiency of 97.1%, whereas the curcumin nanocrystals (Cur-NCs) had a size of 172.3 nm. Both Cur-LPs and Cur-NCs inhibited LPS-induced pro-inflammatory polarization of macrophages and reduced the expression and secretion of inflammatory factors. The mouse air pouch model further demonstrated that both dosage forms attenuated inflammatory cell infiltration and inflammatory fibrosis in subcutaneous tissues. Interestingly, the anti-inflammatory effect of Cur-LPs was more potent than that of Cur-NCs, both in vitro and in vivo, although the cellular uptake of Cur-NCs was quicker. In conclusion, the results demonstrate that Cur-LPs have great potential for the clinical treatment of inflammatory osteolysis and that the therapeutic effect is closely related to the liposomal dosage form.

FasL is a catabolic factor in alveolar bone homeostasis.

AIM: Fas ligand (FasL) belongs to the tumour necrosis factor superfamily regulating bone turnover, inflammation, and apoptosis. The appendicular and axial skeleton phenotype of mature Faslgld mice has been reported. The impact of FasL on the alveolar bone providing support for the teeth at mature stages under healthy and induced inflammatory conditions remains unknown. MATERIALS AND METHODS: We performed a phenotypical analysis of mice carrying the homozygous Faslgld mutation and wild-type (WT) mice (C57BL/6) under healthy conditions and upon ligature-induced periodontitis. After 12 days, micro-computed tomography analysis revealed the distance between the cement enamel junction and the alveolar bone crest. Additional structural parameters, such as the bone volume fraction (BV/TV) and the periodontal ligament space volume, were measured. Histological analyses were performed to visualize the catabolic changes at the defect site. RESULTS: Healthy Faslgld mice were found to have more periodontal bone than their WT littermates. Faslgld had no significant effect on inflammatory osteolysis compared to WT controls with ligatures. Histology revealed eroded surfaces at the root and in the inter-proximal bone in both strains. CONCLUSIONS: Our findings suggest that FasL is a catabolic factor in alveolar bone homeostasis but it does not affect the inflammatory osteolysis.

PLGA nanoparticles engineering extracellular vesicles from human umbilical cord mesenchymal stem cells ameliorates polyethylene particles induced periprosthetic osteolysis.

The wear particle-induced dissolution of bone around implants is a significant pathological factor in aseptic loosening, and controlling prosthetic aseptic loosening holds crucial social significance. While human umbilical cord mesenchymal stem cell-derived exosomes (HucMSCs-Exos, Exos) have been found to effectively promote osteogenesis and angiogenesis, their role in periprosthetic osteolysis remains unexplored. To enhance their in vivo application, we engineered HucMSCs-Exos-encapsulated poly lactic-co-glycolic acid (PLGA) nanoparticles (PLGA-Exos). In our study, we demonstrate that PLGA-Exos stimulate osteogenic differentiation while inhibiting the generation of reactive oxygen species (ROS) and subsequent osteoclast differentiation in vitro. In vivo imaging revealed that PLGA-Exos released exosomes slowly and maintained a therapeutic concentration. Our in vivo experiments demonstrated that PLGA-Exos effectively suppressed osteolysis induced by polyethylene particles. These findings suggest that PLGA-Exos hold potential as a therapeutic approach for the prevention and treatment of periprosthetic osteolysis. Furthermore, they provide novel insights for the clinical management of osteolysis.

Visualization of clinically silent, odontogenic maxillary sinus mucositis originating from periapical inflammation using MRI: a feasibility study.

OBJECTIVES: Maxillary sinus mucositis is frequently associated with odontogenic foci. Periapical inflammation of maxillary molars and premolars cannot be visualized directly using radiation-based imaging. The purpose of this study was to answer the following clinical question: among patients with periapical inflammatory processes in the maxilla, does the use of magnetic resonance imaging (MRI), as compared to conventional periapical (AP) and panoramic radiography (OPT), improve diagnostic accuracy? METHODS: Forty-two subjects with generalized periodontitis were scanned on a 3 T MRI. Sixteen asymptomatic subjects with mucosal swelling of the maxillary sinus were enrolled in the study. Periapical edema was assessed using short tau inversion recovery (STIR) sequence. Apical osteolysis and mucosal swelling were assessed by MRI, AP, and OPT imaging using the periapical index score (PAI). Comparisons between groups were performed with chi-squared tests with Yates' correction. Significance was set at p < 0.05. RESULTS: Periapical lesions of maxillary premolars and molars were identified in 16 subjects, 21 sinuses, and 58 teeth. Bone edema and PAI scores were significantly higher using MRI as compared to OPT and AP (p < 0.05). Using the STIR sequence, a significant association of PAI score > 1 and the presence of mucosal swelling in the maxillary sinus was detected (p = 0.03). CONCLUSION: Periapical inflammation and maxillary mucositis could be visualized using STIR imaging. The use of MRI may help detect early, subtle inflammatory changes in the periapical tissues surrounding maxillary dentition. Early detection could guide diagnostic criteria, as well as treatment and prevention.

Evaluation of 3D MRI for early detection of bone edema associated with apical periodontitis.

OBJECTIVES: To detect and evaluate early signs of apical periodontitis using MRI based on a 3D short-tau-inversion-recovery (STIR) sequence compared to conventional panoramic radiography (OPT) and periapical radiographs in patients with apical periodontitis. MATERIALS AND METHODS: Patients with clinical evidence of periodontal disease were enrolled prospectively and received OPT as well as MRI of the viscerocranium including a 3D-STIR sequence. The MRI sequences were assessed for the occurrence and extent of bone changes associated with apical periodontitis including bone edema, periradicular cysts, and dental granulomas. OPTs and intraoral periapical radiographs, if available, were assessed for corresponding periapical radiolucencies using the periapical index (PAI). RESULTS: In total, 232 teeth of 37 patients (mean age 62±13.9 years, 18 women) were assessed. In 69 cases reactive bone edema was detected on MRI with corresponding radiolucency according to OPT. In 105 cases edema was detected without corresponding radiolucency on OPT. The overall extent of edema measured on MRI was significantly larger compared to the radiolucency on OPT (mean: STIR 2.4±1.4 mm, dental radiograph 1.3±1.2 mm, OPT 0.8±1.1 mm, P=0.01). The overall PAI score was significantly higher on MRI compared to OPT (mean PAI: STIR 1.9±0.7, dental radiograph 1.3±0.5, OPT 1.2±0.7, P=0.02). CONCLUSION: Early detection and assessment of bone changes of apical periodontitis using MRI was feasible while the extent of bone edema measured on MRI exceeded the radiolucencies measured on OPT. CLINICAL RELEVANCE: In clinical routine, dental MRI might be useful for early detection and assessment of apical periodontitis before irreversible bone loss is detected on conventional panoramic and intraoral periapical radiographs.

Cyst formation and bony ingrowth inside coil-type open-architecture anchors used for arthroscopic remplissage: a volumetric computed tomographic study of 50 anchors.

BACKGROUND: The use of anchors in the proximal humerus during arthroscopic surgery can cause localized bone loss due to osteolysis and cyst formation. The purpose of this study was to use computed tomography (CT) to evaluate the incidence of implant-related bone loss and cyst formation after implantation of polyetheretherketone (PEEK) coil-type open-architecture anchors during remplissage for the management of Hill-Sachs defects (HSDs) in patients with shoulder instability. METHODS: This was a single-cohort, observational study with a minimum of 12 months of follow-up. Subjects undergoing arthroscopic instability surgery with HSD requiring remplissage were included. The volume of the bone defects and the degree of bony ingrowth into the anchor were measured on CT images. RESULTS: Thirty-one participants (28 males, 3 females; mean age 29.4 years, standard deviation [SD] 10.6) in whom 50 anchors (4.5-mm Healicoil PEEK double-loaded anchors) were used were evaluated with a CT performed at a mean of 14.1 (SD 3.74) months after surgery. Full bony ingrowth inside the anchor was found in 15 anchors (30%, range 17.8%-44.5%); clear ossification with a thin lucent rim was found in 10 anchors (20%, range 10.0%-33.7%); discontinuous ossification was found in 8 anchors (16%, range 7.2%-29.1%); and no ossification was observed inside 17 anchors (34%, range 21.2%-48.7%). Regarding bone defect size, no bone defect was identified in 15 anchors (30%, 95% CI 17.9%-44.6%), a partial bone defect was found in 17 anchors (34%, 95% CI 21.2%-48.7%), hole enlargement was found in 17 anchors (34%, 95% CI 21.2%-48.7%), and 1 anchor caused a cyst larger than twice the size of the hole made for anchor insertion (2%, 95% CI 0.1%-8.6%). At the 1-year evaluation, none of the participants presented recurrence or residual apprehension. CONCLUSION: The use of PEEK coil-type open-architecture anchors for remplissage during instability surgery caused large cystic lesions in less than 10% of anchors. There was full bony ingrowth in one-third of anchors, and partial cancellous bone ingrowth occurred in another third of anchors.

Long-Term Outcomes of Cementing Highly Cross-Linked Polyethylene Liners Into Well-Fixed Acetabular Shells in Revision Total Hip Arthroplasty.

BACKGROUND: Cementing a new liner into a secure, well-positioned metallic shell can be a less-invasive strategy in revision total hip arthroplasty (THA). This study aimed to report the mean 14-year outcomes of cementing highly cross-linked polyethylene (XLPE) liners into well-fixed acetabular shells in revision THAs. METHODS: This study reviewed a single-surgeon series of cementing XLPE liners into well-fixed acetabular components. Of the 52 hips (51 patients) evaluated, 48 hips (47 patients) that satisfied a minimum follow-up of 10 years were included. The Harris Hip score was used for clinical evaluation. Final hip radiographs were used to determine the extent of acetabular osteolysis and stability of the components. The mean age at index operation was 53 years (range, 32 to 72). The mean follow-up duration was 14 years (range, 10 to 18). RESULTS: The mean Harris Hip score improved from 58 points (range, 23-81) preoperatively to 91 points (range, 45-100) at the final evaluation (P < .001). A total of 3 acetabular rerevisions were performed, all for aseptic loosening of the outer shell. One postoperative dislocation occurred, but it was successfully treated with a closed reduction. Final radiographs showed a significant reduction in acetabular osteolysis (P < .001). Implant survivorship free from any rerevision was 93.3% (95% confidence interval, 85.9-100%) at 14 years. CONCLUSION: Cementing an XLPE liner into a well-fixed acetabular shell in revision THA demonstrated excellent clinical and radiographic outcomes at a mean of 14 years postoperatively. This technique could be a safe and durable option in the absence of XLPE liners compatible with preimplanted shells.

Outcomes After Revision Total Knee Arthroplasty From a Specific, Now-Recalled Implant System.

BACKGROUND: A specific total knee arthroplasty (TKA) implant system was recently recalled for high incidence of early polymeric wear and osteolysis. We analyzed the early outcomes of aseptic revision with these implants. METHODS: We identified 202 aseptic revision TKAs of this implant system performed at a single institution between 2010 and 2020. Revision indications included aseptic loosening (n = 120), instability (n = 55), and polymeric wear/osteolysis (n = 27). Components were revised in 145 cases (72%) and isolated polyethylene insert exchange occurred in 57 cases (28%). Kaplan-Meier and Cox proportional hazards analyses were used to determine survivorship free from all-cause rerevision and rerevision risk factors. RESULTS: At 2 and 5 years, survivorship free from all-cause rerevision was 89 and 76% in the polyethylene exchange cohort versus 92 and 84% in the component revision cohort (P = .5). At 2 and 5 years, survivorship in revision with components from the same manufacturer was 89 and 80% compared to 95 and 86% in revision with components from a different manufacturer (P = .2). Among re-revisions (n = 30), cones (37%), sleeves (7%), hinge/distal femoral replacement implants were frequently used (13%). Men had increased risk for rerevision (hazard ratio = 2.3, P = .04). CONCLUSION: In this series of aseptic revision TKAs performed on a now-recalled implant system, survivorship free from rerevision was lower than expected when components from the same manufacturer were utilized, but comparable to contemporary reports when both the components were revised with an alternative implant system. Metaphyseal fixation with cones and sleeves as well as highly constrained implants was frequently utilized at time of rerevision TKA. LEVEL OF EVIDENCE: Level IV.

Is Highly Cross-Linked Polyethylene Safe for Use in High-Flexion Posterior Stabilized Total Knee Arthroplasty?

BACKGROUND: Application of highly cross-linked polyethylene (HXLPE) to a posterior cruciate-substituting total knee arthroplasty (TKA) might add the risk of fracture and failure of the tibial polyethylene insert. The purpose of this study is to evaluate the long-term (up to 19 years) clinical and radiographic results of posterior cruciate-substituting TKAs with HXLPE or conventional polyethylene. METHODS: This study analyzed the results of 1,217 patients (444 men and 773 women; mean age of 65 ± 7 years, range, 31-85) (2,434 knees) who had received a NexGen LPS-Flex prosthesis with a conventional tibial insert in one knee and the same prosthesis with an HXLPE tibial insert in the contralateral knee. The mean duration of follow-up was 17 years (range, 15-19). RESULTS: The 2 groups did not differ significantly (P > .05) with regard to the clinical and radiographic results. No knee in either group had a fracture of the tibial polyethylene post or failure of the locking mechanism of the tibial polyethylene insert or osteolysis. Twenty-eight knees (2.3%) in the HXLPE group and 26 knees (2.1%) in the conventional polyethylene group were revised. The estimated survival rate at 17 years was 97.7% in the HXLPE group and 97.9% in the conventional polyethylene group. CONCLUSION: The data suggest that clinical and radiographic findings at a mean of 17 years after posterior cruciate-substituting TKA are the same for patients treated with HXLPE and those treated with conventional polyethylene.

Prominent osteolysis in the maxilla: case report of an odontogenic fibroma mimicking a cyst.

BACKGROUND: Odontogenic fibroma (OF) is a rare benign odontogenic tumor of ectomesenchymal origin, mostly affecting the tooth-bearing portions of the jaws in middle-aged patients. Whilst small lesions tend to be clinically asymptomatic, varying unspecific clinical symptoms occur with an increase in size and may mimic odontogenic or other maxillofacial bone tumors, cysts, or fibro-osseous lesions of the jaws. CASE PRESENTATION: A 31-year-old female patient presented with a hard, non-fluctuating protrusion in the vestibule of the upper right maxilla. It was visualized on cone beam computed tomography (CBCT) as space-occupying osteolysis with the displacement of the floor and facial wall of the maxillary sinus, mimicking a cyst-like lesion. The tissue was surgically removed and identified as an OF in the histopathological examination. One year after the surgery, restitution of regular sinus anatomy and physiological intraoral findings were observed. CONCLUSIONS: This case report emphasizes that rare entities, like the maxillary OF presented, often demonstrate nonspecific clinical and radiological findings. Nevertheless, clinicians need to consider rare entities as possible differential diagnoses and plan the treatment accordingly. Histopathological examination is essential to conclude the diagnosis. OF rarely recur after proper enucleation.

[Aseptic loosening due to osteolytic reaction to polyethylene of o total knee prosthesis]. / Cas clinique. Descellement aseptique sur réaction ostéolytique au polyéthylène d'une prothèse totale de genou.

Total knee replacement (TKR) is currently the gold standard in the surgical management of degenerative knee diseases. It is a reliable operation with 95 % good results at 10 years after surgery. Aseptic loosening is the primary cause of TKR failure. It results from a periprosthetic osteolytic reaction secondary to the host's reaction to the wear particle of the prosthesis and responsible for an imbalance in osteoformation and osteolysis. Polyethylene (PE) is responsible for the majority of periprosthetic reactions but the release of metal particles may play an underestimated role with the risk of systemic manifestations. The first clinical signs appear on average 7 years after prosthesis placement. Radio-clinical assessment may be normal in the early stages. Infectious process must be systematically excluded before concluding to another type of complication.

L'arthroplastie par prothèse totale de genou (PTG) est actuellement l'intervention de référence dans la prise en charge chirurgicale des pathologies dégénératives du genou. C'est une opération fiable, avec 95 % de bons résultats à 10 ans de l'intervention. Le descellement aseptique représente la première cause d'échec d'une PTG. Il résulte d'une réaction ostéolytique périprothétique secondaire à la réaction de l'hôte aux particules d'usure de la prothèse, responsable d'un déséquilibre en ostéoformation et ostéolyse. Le polyéthylène (PE) est responsable de la majorité de ces réactions, mais la libération de particules métalliques pourrait jouer un rôle sous-estimé avec des risques de manifestations systémiques. Les premiers signes cliniques apparaissent en moyenne 7 ans après la mise en place de la prothèse. Le bilan radio-clinique peut être normal au stade débutant. Un processus infectieux doit systématiquement être exclu avant de conclure à un autre type de complication.

Successful treatment of SAPHO syndrome (chronic nonbacterial osteomyelitis and acne) with anakinra and denosumab. Case report and review of therapy.

SAPHO is an acronym derived from capital letters of Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO). SAPHO syndrome is an umbrella term covering a constellation of bone lesions and skin manifestations. A 40-year-old male complained about his jaw and back pain, swelling of multiple joints and weight loss accompanied by physical deterioration and acne type skin lesions. Laboratory tests revealed abnormal elevation of inflammatory markers. Imaging studies illustrated multiple osteolytic bone lesions and paraosseal infiltrates. According to the set of criteria diagnosis of SAPHO syndrome was stated. The patient was treated with glucocorticoids and non-steroidal anti-inflammatory drugs (NSAIDs), but only high dose dexamethasone and prednisone were effective. Daily subcutaneous administration of anakinra at the dose of 100 mg was initiated due to limited response to more classical therapies. Because of planned mandibular osteosynthesis initiation of denosumab was preferred before bisphosphonates. Therapeutic response was confirmed by FDG-PET/MR after 5 months of anakinra and denosumab therapy, showing decreased accumulation of FDG in periosteal and paraosseal infiltrates. Inflammatory markers significantly decreased, bone pain deferred but skin manifestation receded only partially. Therefore the response was evaluated as partial remission.

A novel pathogenic variant p.Asp797Val in IFIH1 in a Japanese boy with overlapping Singleton-Merten syndrome and Aicardi-Goutières syndrome.

Pathogenic-activating variants of interferon induced with Helicase C domain 1 (IFIH1) cause Singleton-Merten (S-M) syndrome, which accompanies acro-osteolysis, loss of permanent teeth, and aortic calcification, as well as causing Aicardi-Goutières (A-G) syndrome, which shows progressive encephalopathy, spastic paraplegia, and calcification of basal ganglia. Recently, patients with overlapping syndromes presenting with features of S-M syndrome and A-G syndrome were reported. However, progression of clinical features of this condition has not been fully understood. We report a Japanese boy with a novel pathogenic IFIH1 variant who presented with clinical features of S-M syndrome and A-G syndrome.