Iliac Crest Research Articles

Frontal plane pelvic kinematics during high velocity running: Association with hamstring injury history

IntroductionHamstring injuries are the most prevalent non-contact soft tissue injury in sports, with a larger portion of injuries being recurrent. The sagittal plane running kinematics correlated to hamstring injury history has been well documented. However, analysis of frontal plane kinematics allows for observation of stability and symmetry. This study aimed to examine the frontal plane running kinematics of elite collegiate level sprinters, with and without previous hamstring injury, compared to healthy counterparts. MethodsThirty-nine participants performed three 50-m sprints, with three inertial measurement unit sensors placed on the pelvis: one on each iliac crest and one on the sacrum. Participants were classified based on sex, competitive status, and injury history. To investigate differences based on group classification, the data were used to analyze mediolateral motion (relative magnitude of mediolateral acceleration) and asymmetry (difference in acceleration between right and left iliac crests) during each stance phase throughout the run. ResultsInjured sprinters displayed significantly greater mediolateral motion and asymmetry during stances than healthy counterparts. ConclusionsThis study demonstrates that frontal plane running stance dynamics are different in athletes with previous hamstring injury than healthy athletes. These athletes may benefit from rehabilitation strategies targeting postural control and stability during dynamic tasks.

Successful Implantation of a Ventricular Shunt With Termination in the Iliac Bone in the Adult Patient With Obstructive Hydrocephalus

BACKGROUND AND IMPORTANCE: With the exception of the 3 classic shunt placement options (ventriculoperitoneal, ventriculopleural, and ventriculoatrial), surgically feasible alternative sites for distal catheter placement remain limited and often require the assistance of an access surgeon. Tubbs et al suggested the possibility of intraosseous cerebrospinal fluid diversion in the ilium, noting that ilium infusion in cadaveric specimens was possible without the development of body edema or fluid overflow. Since this publication, limited case reports have been published on the success of ventriculo-ilium (VI) shunt placement. Here, we describe the technique used for successful VI shunt placement in 2 adult patients. CLINICAL PRESENTATION: Here, we describe 2 patients with differing etiologies of hydrocephalus (obstructive and nonobstructive) and complex medical and surgical problems precluding traditional distal shunt termini. Both patients underwent successful placement of a VI shunt with distal catheter placement into the right iliac crest using a small right-angle connector and small cranial fixation plate to prevent backout of the catheter. DISCUSSION AND CONCLUSION: We report the first demonstration of successful placement of a VI shunt in 2 adult patients with evidence of shunt functionality and improved neurological outcome. We propose that the placement of a VI shunt is an easy and viable option when more traditional shunt methods are not available for use.

Effect of Mixture of Recombinant Human Bone Morphogenic Protein-2 and Demineralized Bone Matrix in Lateral Lumbar Interbody Fusion.

This study aims to determine the optimal dose of recombinant-human bone morphogenic protein-2 (rhBMP-2) for successful bone fusion in minimally invasive lateral lumbar interbody fusion (MIS LLIF). Previous studies show that rhBMP is an effective alternative to autologous iliac crest bone graft, but the optimal dose remains uncertain. The study analyzes the fusion rates associated with different rhBMP doses to provide a recommendation for the optimal dose in MIS LLIF. Ninety-three patients underwent MIS LLIF using demineralized bone matrix (DBM) or a mixture of rhBMP-2 and DBM as fusion material. The group was divided into the following three groups according to the rhBMP-2 usage : group A, only DBM was used (n=27); group B, 1 mg of rhBMP-2 per 5 mL of DBM paste (n=41); and group C, 2 mg of rhBMP-2 per 5 mL of DBM paste (n=25). Demographic data, clinical outcomes, postoperative complication and fusion were assessed. At 12 months post-surgery, the overall fusion rate was 92.3% according to Bridwell fusion grading system. Groups B and C, who received rhBMP-2, had significantly higher fusion rates than group A, who received only DBM. However, there was no significant increase in fusion rate when the rhBMP-2 dosage was increased from group B to group C. The groups B and C showed significant improvement in back pain and Oswestry disability index compared to the group A. The incidence of screw loosening was decreased in groups B and C, but there was no significant difference in the occurrence of other complications. Usage of rhBMP-2 in LLIF surgery leads to early and increased final fusion rates, which can result in faster pain relief and return to daily activities for patients. The benefits of using rhBMP-2 were not significantly different between the groups that received 1 mg/5 mL and 2 mg/5 mL of rhBMP-2. Therefore, it is recommended to use 1 mg of rhBMP-2 with 5 mL of DBM, taking both economic and clinical aspects into consideration.

Implications of the composite free flap harvest site on quality of life after head and neck surgery: a prospective series

Quality of life (QOL) has become a primary determinant of the treatment outcome. There is a poor evidence base regarding the QOL implications of free flap harvest from the various different osseous composite donor sites. This prospective study assessed the impact of free flap harvest on QOL and compared QOL morbidity between fibula, scapula, and iliac crest (deep circumflex iliac artery; DCIA) donor sites in head and neck reconstructive surgery. This was a single-site prospective cohort clinical research study. Fifty-nine patients were recruited between 2017 and 2021; 30 underwent fibula flap reconstructive surgery, 17 scapula flap, and 12 DCIA flap. The patients were assessed using the University of Washington Quality of Life Questionnaire version 4 (UW-QOL v4) preoperatively and again at >12 months postoperatively. The results showed no significant change in the mean global QOL score postoperatively when compared to the preoperative baseline in any of the donor site groups. However, the mean postoperative scores for the appearance domain were significantly lower than the preoperative scores in all of the donor site groups. In addition, fibula flap patients had significantly reduced physical activity and recreation QOL domain scores postoperatively when compared to the preoperative scores.

Musculoskeletal Asymmetry and History of Pain in Healthy Adults: Variation in Both Genders in Twin Cities of Pakistan

Introduction: Musculoskeletal asymmetries are common and have important consequences for the individual and society. This study aims to determine musculoskeletal asymmetry in healthy adults.
 Materials and Methods: A total of 400 healthy individuals with no musculoskeletal pain at the time of data collection were included in the study. Two custom-made wooden devices were used to assess the asymmetry in the participants by measuring their height from the base of the device up to different levels of the body. The modified Nordic pain scale was used to take the history of any regional pain in the last 6 months.
 Results: The mean age of male and female subjects was 21.69±2.19 and 23.49±1.72 years, respectively. The dominant side of the body in the majority of both genders was the right side. The most commonly reported painful site was low back with 47(23.5) males and 43(21.5) females affected, followed by neck pain in 40(20) males and 45(22.5) females. Out of 400 participants, only 31(15.5) males and 12(6) females had symmetrical shoulders and 41(20.5) males and 17(8.5) females had symmetry at the level of iliac crests.
 Conclusion: Musculoskeletal asymmetries are present in healthy adults at various levels of the body with a history of musculoskeletal pain relatively more prevalent in females compared to males.

Electroacupuncture at GB20 improves cognitive ability and synaptic plasticity via the CaM-CaMKII-CREB signaling pathway following cerebral ischemia-reperfusion injury in rats.

This study aimed to investigate the effects of electroacupuncture (EA) on cognitive recovery and synaptic remodeling in a rat model of middle cerebral artery occlusion (MCAO) followed by reperfusion and explore the possible mechanism. Focal cerebral ischemia was modeled in healthy adult Sprague-Dawley rats by MCAO. The MCAO rats were classified into four groups: sham, MCAO, MCAO + GB20 (receiving EA at GB20) and MCAO + NA (receiving EA at a "non-acupoint" location not corresponding to any traditional acupuncture point location about 10 mm above the iliac crest). Neurological deficit scores and behavior were assessed before and during the treatment. After intervention for 7 days, the hippocampus was dissected to analyze growth-associated protein (GAP)-43, synaptophysin (SYN) and postsynaptic density protein (PSD)-95 expression levels by Western blotting. Bioinformatic analysis and primary hippocampal neurons with calcium-voltage gated channel subunit alpha 1B (CACNA1B) gene overexpression were used to screen the target genes for EA against MCAO. Significant amelioration of neurological deficits and learning/memory were found in MCAO + GB20 rats compared with MCAO or MCAO + NA rats. Protein levels of GAP-43, SYN and PSD-95 were significantly improved in MCAO + GB20-treated rats together with an increase in the number of synapses in the hippocampal CA1 region. CACNA1B appeared to be a target gene of EA in MCAO. There were increased mRNA levels of CACNA1B, calmodulin (CaM), Ca2+/calmodulin-dependent protein kinase type II (CaMKII) and cyclic adenosine monophosphate response element binding (CREB) and increased phosphorylation of CaM, CaMKII and CREB in the hippocampal region in MCAO + GB20 versus MCAO and MCAO + NA groups. CACNA1B overexpression modulated expression of the CaM-CaMKII-CREB axis. EA treatment at GB20 may ameliorate the negative effects of MCAO on cognitive function in rats by enhancing synaptic plasticity. EA treatment at GB20 may exert this neuroprotective effect by regulating the CACNA1B-CaM-CaMKII-CREB axis.

Correlation between the Tuffier's Line and the Spinopelvic Parameters

Tuffier’s line (TL) is a horizontal line that connects the two superior iliac crests and is used to determine L4 and L5 vertebrae levels. Spinopelvic parameters have been gaining further importance in the diagnosis and treatment planning of spinal diseases. This study aims to reveal whether there is an interaction between TL and spinopelvic parameters. We examined the 113 patients who consulted our clinic for low back pains. TL levels were divided into five categories as L4 body, L4 inferior endplate, L4-5 disc space, L5 superior endplate, and L5 body. In the study group, 70 of the cases (61.9%) were female, 43 (38.1%) were male, and the average age was 50.8 (21–77). TL was determined to pass through L4 body in 38.9% of cases (n=44) regardless of gender. It was observed that sacral slope (SS) and lumbar lordosis (LL) were affected by the changes in TL level (P0.05). SS and LL being affected by changes in TL levels indicates that TL should be assessed together with the spinopelvic parameters. To conclude, TL is a potential spinal parameter that should be included in the spinopelvic parameters.

Open lift-drill-fill-fix for medial osteochondral lesions of the talus: surgical technique.

Osteochondral lesions of the talus (OLT) with afragment on the talar dome that fail conservative treatment and need surgical treatment can benefit from in situ fixation of the OLT. Advantages of fixation include the preservation of native cartilage, a high quality subchondral bone repair, and the restoration of the joint congruency by immediate fragment stabilization. To improve the chance of successful stabilization, adequate lesion exposure is critical, especially in difficult to reach lesions located on the posteromedial talar dome. In this study we describe the open Lift, Drill, Fill, Fix (LDFF) technique for medial osteochondral lesions of the talus with an osteochondral fragment. As such, the lesion can be seen as an intra-articular non-union that requires debridement, bone-grafting, stabilization, and compression. The LDFF procedure combines these needs with access through amedial distal tibial osteotomy. Symptomatic osteochondral lesion of the talus with afragment (≥ 10 mm diameter and ≥ 3 mm thick as per computed tomography [CT] scan) situated on the medial talar dome which failed 3-6months conservative treatment. Systemic disease, including active bacterial arthritis, hemophilic or other diffuse arthropathies, rheumatoid arthritis of the ankle joint, and malignancies. Neuropathic disease. End-stage ankle osteoarthritis or Kellgren and Lawrence score3 or4 [3]. Ipsilateral medial malleolus fracture less than 6months prior. Relative contra-indication: posttraumatic stiffness with range of motion (ROM) < 5°. Children with open physis: do not perform an osteotomy as stabilization of the osteotomy may lead to early closure of the physis, potentially resulting in symptomatic varus angulation of the distal tibia. In these cases only arthrotomy can be considered. The OLT is approached through amedial distal tibial osteotomy, for which the screws are predrilled and the osteotomy is made with an oscillating saw and finished with achisel in order to avoid thermal damage. Hereafter, the joint is inspected and the osteochondral fragment is identified. The cartilage is partially incised at the borders and the fragment is then lifted as ahood of amotor vehicle (lift). The subchondral bone is debrided and thereafter drilled to allow thorough bone marrow stimulation (drill) and filled with autologous cancellous bone graft from either the iliac crest or the distal tibia (fill). The fragment is then fixated (fix) in anatomical position, preferably with two screws to allow additional rotational stability. Finally, the osteotomy is reduced and fixated with two screws. Casting includes 5weeks of short leg cast non-weightbearing and 5weeks of short leg cast with weightbearing as tolerated. At 10-week follow-up, aCT scan is made to confirm fragment and osteotomy healing, and patients start personalized rehabilitation under the guidance of aphysical therapist.

Reconstruction of calcaneum using the deep circumflex iliac artery bone graft following wide local excision due to Ewing’s sarcoma: A case report on a rare disease presentation and its unconventional management

Introduction: Ewing’s sarcoma of the calcaneum, although rare, is a significant health threat as it is quick to spread locally and systemically reducing the overall survival rates. Conventionally, below knee amputation was used as its only management. Currently, due to numerous advancements, wide local excision following reconstruction is the treatment of choice worldwide. Case Report: This case report provides clinical and functional outcomes of using deep circumflex iliac artery bone graft for calcaneal reconstruction. The follow-up duration in our study was two years. Normal ambulatory status, post-procedure, was achieved within nine months with a good range of motion at ankle joint both actively and passively. No donor site morbidity was seen and radiological imaging confirmed a healthy and viable graft with negative oncological margins. Conclusion: Our choice of graft proved to be a suitable option for calcaneal reconstructions as the curvature of iliac crest resembles calcaneal tuberosity. The ability of its osseous flap to naturally fit the area of defect makes it the most appropriate choice for the reconstruction of the heel of the foot.

Arthroscopic Bone Block Cerclage Suture Fixation of Fresh Distal Tibial Allograft for Anterior Glenoid Reconstruction

Glenoid bone loss in patients with recurrent anterior shoulder instability poses a unique challenge to treating surgeons. Various bone block procedures have been used to reconstruct the glenoid, including autologous coracoid transfer, iliac crest autograft, distal clavicle autograft, and distal tibia allograft. Distal tibia allograft has been increasingly used because of its advantages over autologous graft sources. Having started out as an open procedure, glenoid reconstruction with distal tibia allograft has now found its way to evolving into an arthroscopic procedure. Various techniques have been devised for arthroscopic distal tibia allograft fixation, including screw, suture anchor, and suture button. This technical note describes an arthroscopic distal tibia allograft fixation technique, using a transglenoid parallel drill guide and 2 cerclage tape sutures that are fastened using a tensioner. This technique provides strong graft fixation while avoiding the potential complications and technical challenges of metal screw fixation.

Tensile Mechanical Properties of Dry Cortical Bone Extracellular Matrix: A Comparison Among Two Osteogenesis Imperfecta and One Healthy Control Iliac Crest Biopsies.

Osteogenesis imperfecta (OI) is a genetic, collagen-related bone disease that increases the incidence of bone fractures. Still, the origin of this brittle mechanical behavior remains unclear. The extracellular matrix (ECM) of OI bone exhibits a higher degree of bone mineralization (DBM), whereas compressive mechanical properties at the ECM level do not appear to be inferior to healthy bone. However, it is unknown if collagen defects alter ECM tensile properties. This study aims to quantify the tensile properties of healthy and OI bone ECM. In three transiliac biopsies (healthy n = 1, OI type I n = 1, OI type III n = 1), 23 microtensile specimens (gauge dimensions 10 × 5 × 2 μm3) were manufactured and loaded quasi-statically under tension in vacuum condition. The resulting loading modulus and ultimate strength were extracted. Interestingly, tensile properties in OI bone ECM were not inferior compared to controls. All specimens revealed a brittle failure behavior. Fracture surfaces were graded according to their mineralized collagen fibers (MCF) orientation into axial, mixed, and transversal fracture surface types (FST). Furthermore, tissue mineral density (TMD) of the biopsy cortices was extracted from micro-computed tomogra[hy (μCT) images. Both FST and TMD are significant factors to predict loading modulus and ultimate strength with an adjusted R 2 of 0.556 (p = 2.65e-05) and 0.46 (p = 2.2e-04), respectively. The influence of MCF orientation and DBM on the mechanical properties of the neighboring ECM was further verified with quantitative polarized Raman spectroscopy (qPRS) and site-matched nanoindentation. MCF orientation and DBM were extracted from the qPRS spectrum, and a second mechanical model was developed to predict the indentation modulus with MCF orientation and DBM (R 2 = 67.4%, p = 7.73e-07). The tensile mechanical properties of the cortical bone ECM of two OI iliac crest biopsies are not lower than the one from a healthy and are primarily dependent on MCF orientation and DBM. © 2023 The Authors. JBMR Plus published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research.

Comparation of outcomes of iliac crest bone graft and accessory navicular bone graft in the Cotton osteotomy for pediatric and adolescent flexible flatfoot

Objective: To compare the outcomes between iliac crest and accessory navicular as different bone grafts in the Cotton osteotomy in the treatment of pediatric flexible flatfoot deformity. Methods: A retrospective cohort study. The clinical data of pediatric flexible flatfoot patients with symptomatic accessory navicular received operations from July 2018 to March 2022 in Beijing Tongren Hospital were retrospectively analyzed. All the patients were treated with Cotton osteotomy. According to the different sources of bone grafting in Cotton osteotomy, the patients were divided into iliac crest group and accessory navicular group. There were 12 patients (19 feet) in iliac crest group, including 9 males and 3 females with a median age M(Q1, Q3) of 11(11, 12) years and were followed-up for 36(6, 48) months. There were 9 patients (16 feet) in accessory navicular group, including 6 males and 3 females, with a median age M(Q1, Q3) of 11(11, 11) years and were followed-up for 12(6, 17) months. Radiographic evaluations were reviewed and compared between the two groups before surgical treatment and at final follow-up, included talo-1st metatarsal angle (T1MT), talonavicular coverage angle (TNCA), talocalcaneal angle (TCA) in weight bearing anteroposterior view, and Meary angle, calcaneal Pitch angle, Kite angle, cuneiform articular angle (CAA) in weight bearing lateral view, and hindfoot alignment angle (HAA) in calcaneal long axial view. Functional scores included American Orthopedic Foot and Ankle Society (AOFAS), visual analog scale (VAS), and Maryland scores were recorded and compared before and after the operation. The complications were also recorded. Results: Total of 21 patients were successfully followed in this study. The radiographic measurements in the two groups, such as T1MT, TNCA, Meary angle, Pitch angle, Kite angle, CAA, HAA, were all significantly improved at the last follow-up when compared with those before the surgery (all P<0.05); and the AOFAS, VAS, Maryland scores in both groups were all improved after the surgery (all P<0.05). There was a significant difference in CAA alteration (∆CAA) after the operation between the two groups, the ∆CAA in iliac crest group and the accessory navicular group was 6.0°±2.6° and 4.3°±1.3°, respectively (P=0.017). There was no significant differences in the improvement of other radiographic measurements between the two groups (all P>0.05). All the patients had a successful bone union. Two patients had donor site pain in the iliac crest group and were treated conservatively. Conclusion: Compared with iliac crest bone graft, accessory navicular bone graft could achieve comparable outcomes in radiographic measurements and functional scores in the treatment of pediatric flexible flatfoot with accessory navicular pain.

Comparisons and associations among anthropometric indices of first and second division and assistant soccer referees.

Body composition is an important predictor of performance and a key component of health and physical fitness. Therefore, the purposes of this study were to compare soccer referees of the first and second divisions and field assistant referees from Iran and to analyze associations of a body shape index (ABSI), body adiposity index (BAI), abdominal volume index (AVI), body roundness index (BRI), conicity index (ConI), and body mass index (BMI) with body fat percentage (%BF). A total of 270 male soccer referees from the first (n = 124) and second (n = 72) divisions and assistant referees (n = 74) participated in this study. Skinfold thickness (measured at the chest, biceps, triceps, subscapular, abdominal, iliac crest, and front thigh), height, weight, hip circumference, and waist circumference were assessed to evaluate waist-to-height ratio (WHtR), %BF, and also ABSI, BRI, BAI, ConI, and AVI according to the ISAK protocol. The main results indicated differences in WHR, WHtR, ABSI, BRI, AVI, ConI, and BF% with the assistant referees presenting higher values (p < 0.05). When considering the backward selection model, there were some associations with %BF in each group, specifically BMI, BAI, and ABSI in the first division; BMI, WHR, and ABSI in the second division; and BMI in the assistant referee group (all p < 0.05). The present study did not confirm the hypothesis that the first-division referees presented better body composition-related variables than the second division or assistant referees. Instead, it showed that the assistant referees that participated in both divisions showed a tendency to higher values which suggests that the level of division is not a major factor when analyzing body composition.

Subtalar Distraction Bone Block Arthrodesis for Calcaneus Fracture Malunion

Intra-articular calcaneus fractures are associated with a high rate of malunion resulting in hindfoot and ankle impingement, peroneal tendinitis, sural neuritis, arthritis, hindfoot varus or valgus deformity, loss of hindfoot height, and resulting gastrocnemius-soleus complex weakness and gait dysfunction. These issues can arise whether the patient’s injury is treated conservatively with non–weight-bearing precautions, neglected/missed, or treated surgically with open reduction and internal fixation. The sequelae of calcaneal malunions can arise early or late in this process. For patients undergoing subtalar arthrodesis following calcaneus fracture malunion, the use of a bone block wedge can assist with achieving a stable ,well-aligned, pain-free hindfoot. Patient selection, preoperative planning, careful surgical technique, and compliance with postoperative care pathways are critical to optimize patient outcomes and healing of this complex reconstruction. Recent evidence supports the utility of weight-bearing CT to assist with preoperative planning for subtalar arthrodesis. Wedges used in subtalar distraction arthrodesis include bulk autograft (tricortical iliac crest), allograft (pre-fashioned or intraoperatively fashioned wedges from femoral head allograft or other source), and metallic wedges. These wedges are typically secured with screws spanning and compressing the arthrodesis site, which is filled with a mixture of concentrated bone marrow aspirate, bone growth factors, and allograft bone. Concomitant procedures may be indicated to ensure the overall success of the procedure, such as extra-articular, calcaneal osteotomy for additional realignment purposes and/or arthrolysis. A review of the literature shows high rates of union following subtalar bone block arthrodesis with improved functional outcomes. Our preferred technique for a bone block distraction subtalar arthrodesis, described herein, includes the use of an allograft wedge. Ongoing advances, such as the development of novel materials and structures for use in subtalar bone block arthrodesis, but most importantly, the incorporation of weight-bearing CT technology and potential for preoperative CT navigation to make this procedure more predictable, are all important areas for further exploration and future research. Level of Evidence: Level—4.

The incidence of intraoperatively acquired pressure injuries in the park-bench position was reduced by applying soft silicone multilayer foam dressings.

The Park-Bench Position (PBP) is associated with a high incidence rate of intraoperatively acquired pressure injuries (IAPIs). Preventive measures must be established to prevent the development of IAPIs. We investigated the risk factors for PBP by applying a soft silicone multilayered foam dressing (SMD) under core temperature management to prevent IAPIs. We conducted a prospective, single-centre, open-label observational study of patients undergoing elective neurosurgery operations using PBP in a university hospital in Japan. The incidence rate of IAPIs in this study was compared with that in our two previous studies, in which a film dressing was applied and core temperature management was not performed. IAPIs developed in 90 patients (6.7%); in the lateral thoracic region in five patients and the iliac crest region in one patient. The operative time (every 1 h: p = 0.0001, OR: odds ratio 3.62, 95% CI: confidence interval 1.73-11.42) was significantly associated with the incidence of IAPIs. In our two previous studies, the incidence rate of IAPIs was 11.0% and 24.1%, respectively, when film dressing was used. SMD may weaken the involvement of risk factors in IAPIs.

Vascularized fibular flap and custom-made synthesis in post-traumatic ulnar diaphyseal pseudarthrosis: a case report

BackgroundAlthough isolated fractures of the ulnar shaft are considered common and relatively benign injuries, numerous complications can arise especially in the context of suboptimal care pathways. For pediatric patients, however, there is no single indication of the surgical approach. In the context of the management of these complications, it is known that the vascularized fibular graft has numerous advantages and indications in the treatment of recurrent pseudarthrosis. However, in revision surgery the frequent occurrence of anatomical subversions requires the use of fixation means adapted to the individual patient. We present a clinical case of an adult patient suffering from post-traumatic ulnar pseudarthrosis treated with autologous vascularized fibula grafts and 3D-planned custom-made plate.Case presentationA 38-year-old Ivorian woman came to our attention with a painful nonunion of the ulnar shaft and significant dysmorphism of the left forearm, with shortening and flexion of the limb as an outcome of unspecified road trauma in childhood. No alterations of the nerve compartment were reported. As far as detectable, she had undergone autologous bone grafting and implantation of questionable synthetic means, without acute treatment. Since we evaluated the patient (2012), we have performed two debridement surgeries, associated with autologous avascular bone graft from the iliac crest and plate fixation (2012 and 2014). In both cases, rupture of the fixation media was observed. In 2021, the pseudarthrosis was treated with a vascularized fibular bone graft. The subverted radius and ulna anatomy and poor bone quality required patient-specific reconstruction of the pseudarthrosic ulna from a 3D scan and the production of custom-made plate and screws, supported by the creation of special guides for drilling and by optimizing the positioning of screws with preoperative digital models. In the postoperative period, regular follow-up visits with X-rays evaluations were performed at 1, 3 and 6 months after surgery. No inflammatory reactions or local rejection were found. The fibula graft healed at the proximal ulnar junction six months after the operation while it took eight months to heal at the distal junction. Functionally, we observed a pain reduction and a range-of-motion preservation.ConclusionsThe multiple failures of diaphyseal reconstruction with avascular bone grafts have forced the indication to the vascularized fibular flap. This case is a unique experience but we believe that the association between vascularized bone graft and the potential for customization through 3D planning represents a valid surgical potentiality in complex cases of post-traumatic reconstruction.

OR29-02 Energy Deficient Osteoblasts Inefficiently Mineralize Developing Bone In Sheep With Hypophosphatasia

Abstract Disclosure: J.C. Bertels: None. M. McCord: None. L.G. White: None. S. Huggins: None. D. Landrock: None. E.D. Giles: None. L.J. Suva: None. D. Gaddy: None. Hypophosphatasia (HPP) is a rare genetic disorder characterized by low enzymatic activity of tissue nonspecific alkaline phosphatase (TNSALP), resulting in decreased bone mineralization, muscle weakness, and premature loss of deciduous teeth. All HPP phenotypes have been characterized in our CRISPR/Cas9 gene-edited sheep model of HPP. Previous studies identified small, abnormal mitochondria in HPP sheep cells, leading to the hypothesis that reduced bone mineralization may result from decreased osteoblast (OB) differentiation and cellular energetics. To test this idea, iliac crest bone biopsies and sternal bone marrow stromal cells (BMSCs) were harvested from 1 yr old WT and HPP sheep (n=3 per genotype). Histomorphometric analysis of bone biopsies from HPP sheep revealed increased unmineralized osteoid matrix compared to WT, indicative of decreased OB activity, whereas osteoblast and osteoclast number were unchanged. To determine whether BMSCs from HPP sheep were altered from WT in regard to differentiation capacity toward the adipocyte (AD) or OB lineage, 21 day cultures of BMSCs in OB or AD differentiation medium were performed. HPP BMSCs had significantly increased AD differentiation and similar OB differentiation compared to WT. Next, high resolution respirometry of undifferentiated and differentiating cells toward either the OB or AD lineage using Seahorse XF analysis to measure oxidative respiration and glycolysis on days 0, 7, 14 and 21 of both OB and AD differentiation was performed. Results demonstrated that both undifferentiated and differentiating HPP cells (to OB and AD) had significantly decreased oxygen consumption rates (OCR) and extracellular acidification rates (ECAR), reflective of diminished maximal and ATP-linked oxidative respiration and glycolysis. When measured, intracellular ATP levels in differentiating BMSCs, both undergoing osteoblastogenic and adiopogenic differentiation, showed a higher accumulation of ATP. Collectively, these data provide the first evidence that decreased bone mineralization in HPP sheep results from decreased BMSC and OB energetics through both oxidative respiration and glycolysis. In addition, the data demonstrate that reduced TNSALP activity also leads to enhanced bone marrow adipogenesis. Coupled with our previous results demonstrating reduced oxidative respiration and glycolysis in sheep skeletal muscle, these findings demonstrate a fundamental requirement for TNSALP activity in the maintenance of normal cellular respiration and purinergic metabolism in cells of the musculoskeletal system. Moreover, they provide important insight into the potential for targeting increased cellular respiration to alleviate the deleterious effects of HPP. Presentation: Sunday, June 18, 2023

SAT194 A Rare Case Of Untreated Primary Hyperparathyroidism With Extremely High PTH And Lytic Bone Lesions

Abstract Disclosure: M. Shah: None. K. Akinola: None. M. Bollineni: None. S.A. Milan: None. S. Challagulla: None. Background: Primary hyperparathyroidism (PHPT) typically presents with PTH levels two times above the upper limit of normal. Given the increase in routine calcium measurements, PHPT is usually diagnosed and treated early and skeletal complications are not frequent at presentation. We present a case of PHPT with severely elevated PTH levels and bone lesions mimicking metastatic disease. Clinical Case: A 30-year-old male with a history of untreated primary hyperparathyroidism presented with severe chest wall pain and abdominal pain. Initial laboratory tests revealed a calcium level of 13.3 mg/dL (normal 8.6 to 10.5 mg/dL), alkaline phosphatase (AP) of 747 IU/L (normal 24 to 130 IU/L), and PTH of 2485 pg/mL (normal 9-77 pg/mL). The patient was diagnosed with primary hyperparathyroidism years before this presentation, but was lost to follow up due to lack of insurance. His PTH was first noted to be elevated in 2013 at 266 pg/mL. CT of the chest, abdomen, pelvis showed a 2.8 x 3.0 x 3.3 cm anterior mediastinal mass and diffuse mixed lytic and sclerotic bone lesions consistent with metastatic disease. The patient underwent further imaging with CT 4D parathyroid which revealed a 1.1 cm left inferior thyroid nodule and an anterior mediastinal mass of unclear etiology. Given the high concern for malignancy, a biopsy of the mediastinal mass was performed and returned as hypercellular parathyroid tissue with no atypia. Bone biopsies of the sclerotic lesions of the iliac crest were consistent with osseous tissue with prominent bone remodeling and fibrous tissue; no neoplasm was seen. He was treated with IV fluids and zoledronic acid during this work up which only temporarily improved his high calcium levels. He was then placed on cinacalcet prior to surgery. He ultimately underwent resection of the anterior mediastinal mass and left inferior parathyroid gland via a cervical approach. The patient’s preoperative PTH level was 3455 pg/mL and decreased to 209 pg/mL immediately after resection. Resection of the other parathyroid glands was not done due to the high risk of postoperative hypocalcemia. Pathology of both lesions returned as intrathymic parathyroid adenoma with focal fibrosis. Postoperatively, the patient developed symptomatic hypocalcemia as low as 6.4 mg/dL. He required IV and oral calcium for approximately 3 weeks postoperatively, consistent with hungry bone syndrome. He was discharged on an oral regimen of calcium supplementation. Discussion: Long standing untreated PHPT can present with severely elevated PTH levels and skeletal manifestations commonly mistaken for metastatic lesions. As in this case, parathyroidectomy was the only effective treatment. However, it can be complicated by hungry bone syndrome, especially in a patient with significant bone remodeling as evidenced by high AP and lytic bone lesions. Presentation: Saturday, June 17, 2023

FRI696 Endocrine Shades Of Silicone Fillers: A Case Of Calcitriol-mediated Hypercalcemia

Abstract Disclosure: M.S. Martinez Cruz: None. T.G. Lozovoy Madsen-Barbosa: None. C.J. Haas: None. Introduction: In rare cases, foreign body granuloma (FBG) reaction is so vast that it leads to severe systemic disease, including increased granuloma-mediated production of calcitriol. Case Description: A 42-year-old woman was referred to the ED due to elevated serum calcium and worsening GFR on routine blood work. She endorsed a two-week of generalized weakness, myalgias, polyuria, polydipsia, and intermittent constipation with prior episodes of such symptomatology. On her first presentation, severe hypercalcemia (13.8 mg/dL), acute-on-chronic renal disease, a normal TSH, an appropriately low PTH, elevated PTHrP, a low 25-hydroxy vitamin D, and a normal 1,25 dihydroxy-vitamin D were noted. She was treated with IV fluids and zoledronic acid 4mg IV one time with subsequent normalization of calcium levels. Six months later, she was re-admitted with similar complaints. Additional blood workup was unremarkable, including urine and serum protein electrophoresis and kappa/lambda ratio. She once again presented with similar complaints; additional workup revealed a normal 25-hydroxy vitamin D, mildly elevated serum angiotensin-converting enzyme, and a negative QuantiFERON-TB gold, but a marked elevation of 1,25 dihydroxy-vitamin D (138 pg/mL). She admitted to subcutaneous silicone filler injections in the lips, cheeks, and hips applied by a non-healthcare provider six years ago. She described that one year after the injections, she had noted intermittently indurated erythematous areas in the skin of the hips and upper lateral thighs that were notably tender to palpation. Diagnostic imaging included a whole-body PET-CT that demonstrated moderate FDG uptake within the subcutaneous adipose tissue of the lateral aspect of bilateral gluteal regions extending from the iliac crests to the upper thighs. She was diagnosed with silicone filler injection-induced hypercalcemia secondary to granulomatous inflammation and initiated on prednisone 20 mg PO daily with progressive normalization in her hypercalcemia (9.7 mg/dl) a month after initiation of treatment. Unique features/Teaching points: FBG reactions occur more commonly against permanent fillers like silicone, especially if injected in larger volumes and highly vasculature areas like hips or buttocks. These reactions may occur years after the initial injection. In rare cases, significant granulomatous immune response leads to the overactivity of 1-alpha-hydroxylase in activated macrophages, resulting in uncontrolled overproduction of calcitriol and, subsequently, hypercalcemia. FBG reactions may also cause increased levels of PTHrP, further contributing to hypercalcemia. Treatment focuses on general hypercalcemia management and FBG remission, most effectively achieved with anti-inflammatory corticosteroid doses. Further studies are needed to evaluate long-term treatment efficacy. Presentation: Friday, June 16, 2023

Onchocerciasis-associated epilepsy: an explorative case-control study with viral metagenomic analyses on Onchocerca volvulus.

A high prevalence of onchocerciasis-associated epilepsy (OAE) has been observed in onchocerciasis-endemic areas with high ongoing Onchocerca volvulus transmission. However, the pathogenesis of OAE remains to be elucidated. We hypothesise that the O. volvulus virome could be involved in inducing epilepsy. With this study, we aim to describe the O. volvulus virome and identify potential neurotropic viruses linked to OAE. In Maridi County, an onchocerciasis endemic area in South Sudan with a high prevalence of OAE, we will conduct an exploratory case-control study enrolling 40 persons aged 12 years and above with palpable onchocerciasis nodules. Cases will be participants with OAE (n=20), who will be age- and village-matched with controls without epilepsy (n=20). For each study participant, two skin snips at the iliac crest will be obtained to collect O. volvulus microfilariae, and one nodulectomy will be performed to obtain adult worms. A viral metagenomic study will be conducted on microfilariae and adult worms, and the O. volvulus virome of persons with and without OAE will be compared. The number, size, and localisation of onchocerciasis nodules in persons with and without OAE will be described. Moreover, the pre- and post-nodulectomy frequency of seizures in persons with OAE will be compared. The protocol has been approved by the Ethics Committee of the University of Antwerp and the Ministry of Health of South Sudan. Findings will be disseminated nationally and internationally via meetings and peer-reviewed publications. ClinicalTrials.gov registration NCT05868551 ( https://clinicaltrials.gov/study/NCT05868551). 1.1, dated 09/05/2023.