Post-traumatic cortical cystic lesions in children: three case reports and a systematic review of the literature.

Post-traumatic cortical cystic lesions are rare radiolucent lesions that appear as a complication of low-severity fractures in children. Their relevance lies in the fact that few cases of these lesions have been described to date, so they are little known among clinicians caring for children. Three case reports of well-circumscribed cortical lytic lesions detected at 2-4 months during the follow-up of non-displaced distal radius fractures in children aged 9, 7, and 2 years are presented. The consistent clinical history and typical radiological features allowed the accurate diagnosis of post-traumatic cortical cystic lesion, without the need for advanced imaging tests or biopsy. At 12-, 8- and 11-month follow-ups, respectively, the lesions either disappeared or decreased in size. This benign and self-limited lesion should be correctly recognized to avoid confusion with other diagnoses, advanced imaging tests or biopsies, and unnecessary parental concerns.

CLINICAL FEATURES OF CONNECTIVE TISSUE DYSPLASIA, OSGOOD-SCHLATTER DISEASE AND MULTIPLE CORTICAL DISORDERS IN A CHILD.

OBJECTIVE: The aim: To analyze scientific data on the problems of disorders of bone morphogenesis in children, in particular, non-ossifying fibroma. To analyze modern methods of diagnosis and treatment of this disease in the context of the latest scientific achievements. PATIENTS AND METHODS: Materials and methods: The latest data of scientists from the world's leading clinics describing various forms of fibrous skeletal lesions in children of differ¬ent ages were analyzed. We examined a boy with fibrous lesions of the cortical layer of the knee joint bones and a girl with a large fibrous focus of the distal femoral metaphysis, which resulted in a closed pathological bone fracture. Surgical treatment, osteosynthesis, marginal resection of the tumor, bone grafting and histological examination were conducted. CONCLUSION: Conclusions: Despite numerous studies of this disease, the etiopathogenesis of this disease has not been studied. There are no early symptoms of fibrous bone lesions in children. Diagnosis is possible only when a pathological fracture of the affected bone occurs due to extensive growth of fibrous tissue and a significant decrease in the mechanical strength of the bone. Most cases of recognition of such a disease occur accidentally when performing X-ray examinations for other reasons - bruises, sprains, arthralgias, osteochondropathy, infectious diseases, etc. It is known that boys are more mobile and require more frequent X-ray examinations for limb injuries than girls, so the likelihood of accidental detection of such changes increases significantly. Thus, long-term observations of children with fibrous bone lesions have shown that after 40 years, patients rarely developed malignant tumors - osteogenic sarcoma, fibrosarcoma, malignant fibroma of tubular bones, pelvic bones. There are no early symptoms of fibrous bone lesions in children. Recognition of such a disease occurs by chance when X-ray examinations are performed for other reasons.

Non-ossifying fibromas and fibrous cortical defects around the knee - an epidemiologic survey in a Japanese pediatric population.

BACKGROUND: The aim of the present study was to evaluate the prevalence of non-ossifying fibroma (NOF) and fibrous cortical defect (FCD) in a Japanese pediatric population and the association between the lesion size and pain. METHODS: This retrospective study, conducted across 10 Japanese institutions, included patients aged 5-15 years who had undergone standard antero-posterior and lateral view radiography of the knee. Using these radiographs, we diagnosed the lesion as a NOF or FCD. Patient demographics, including age, sex, the size and location of the NOF, and chief complaint were recorded. The lesion size was determined using radiographs. Student's t-test was used to compare the associations between the lesion size and spontaneous pain. RESULTS: A total of 6222 subjects (3567 boys and 2455 girls) were included in this study. The number of NOF and FCD cases was 143 and 437, respectively, and the prevalence of NOF and FCD was 2.3% and 7.0%, respectively. The average size of NOF and FCD was 22.1 mm (range: 4-102 mm) and 13.2 mm (range: 5-21 mm), respectively. Three patients (2.1%) had pathological fractures due to NOF. Of the 140 NOFs and 437 FCDs, we obtained complaints from the medical records of 126 and 393 patients, respectively. The number of patients with spontaneous pain or other problems with NOF was 68 (54%) and 58 (46%), respectively, that of patients with FCD was 195 (50%) and 198 (50%) patients, respectively. The lesion size was not associated with spontaneous pain in either lesion (p = 0.67 and p = 0.27, respectively). CONCLUSION: The prevalence of NOF and FCD around the knee was lower than that reported in previous studies. The prevalence of NOF increased and that of FCD decreased with advancing age. In both lesions, the lesion size may not be associated with pain.

[Benign and malignant giant-cell rich lesions of bone: Pathological diagnosis with special emphasis on recent immunohistochemistry and molecular techniques]. / Diagnostic des lésions osseuses riches en cellules géantes : démarche diagnostique et intérêt des nouvelles techniques complémentaires immuno-histochimiques et moléculaires.

The infiltration by numerous osteoclastic giant cells is a frequent finding in bone tumors and pseudo-tumors. Pathologists must integrate clinical and radiological data to achieve a correct diagnosis in bone pathology. Benign giant-cell rich lesions of bone encompass giant cell tumor of bone, aneurysmal bone cyst, chondroblastoma, brown tumor and fibrous cortical defect/non-ossifying fibroma. Amongst malignant neoplasms, variants of conventional osteosarcoma, undifferentiated pleomorphic sarcoma, leiomyosarcoma and bone metastasis must be discussed. Recently, new diagnostic markers, antibodies for immuno-histochemistry and genetic markers, have been developed and are helpful to diagnose such lesions.

[Radiological diagnosis of enchondroma protuberans of the humerus]. / Diagnóstico radiológico del encondroma protuberans humeral.

Enchondroma protuberans (EP) is an uncommon exophytic cartilaginous tumor described as an intramedullary osseous lesion that protrudes through a cortical defect in one of the sides of the affected bone and expands to the adjacent soft tissues. Due to its peculiar features on imaging studies, the main differential diagnosis is with osteochondroma, chondrosarcoma, and periosteal chondral tumors. In this article, we describe the imaging findings in two patients with EP in the humerus. We review the literature and discuss the imaging features that can enable a definitive diagnosis. The identification of the connection between the two components of the lesion through a cortical defect is a key finding for the diagnosis. Available imaging techniques, especially MRI, make it possible to establish the diagnosis in this type of lesions and thus to choose the appropriate treatment, reducing the chance of local recurrence and malignant transformation.

Early experience with percutaneous balloon kyphoplasty for treatment of osteoporotic vertebral compression fracture in Nepal: A case report.

Key Clinical Message: Balloon kyphoplasty is a promising treatment option for osteoporotic vertebral compression fractures with posterior cortical defect, offering pain relief, vertebral height restoration, and low risk of cement leakage. Abstract: Millions of people worldwide suffer from osteoporotic vertebral compression fractures (OVCFs) annually, which cause pain and functional limitations, particularly in the elderly. Conservative treatments such as pain management, rest, and medication are frequently used, while surgical options such as vertebroplasty and kyphoplasty are considered. We present a case of 68-year-old female with vertebral compression fracture of L1 vertebra with posterior cortical defect and posterior wall retropulsion. She was treated successfully with balloon kyphoplasty. Kyphoplasty appears to be a better option than vertebroplasty in cases with posterior cortical defect due to lower chance of cement leakage.

Non-ossifying Fibroma of Mandible in a Four-Year-Old Girl: A Case Report.

Non-ossifying fibroma (NOF) is not prevelant in the mandible. It appears mostly in the long tubular bones in children and adolescents. We are presenting a case of a four-year-old girl reported to the maxillofacial department with painless swelling over the lower right side of the jaw. On the orthopantomogram (OPG), a well-defined multilocular radiolucency with a sclerotic margin was present. On computed tomography (CT), an expansile lytic lesion with cortical thinning without a breach in cortical continuity was noted. By correlating clinical and radiological features, a diagnosis of odontogenic and/or osteogenic lesion was made. The patient was considered for an excisional biopsy with curettage. On histopathology, NOF was confirmed. On postoperative follow-up, there was no sign of recurrence, and bone regeneration was significant.

Giant cell tumor of bone in an eighteenth-century Italian mummy.

Giant cell tumor (GCT) of the bone is a locally aggressive and rarely metastasizing neoplasm. It is composed of neoplastic mononuclear stromal cells with a monotonous appearance admixed with macrophages and osteoclast-like giant cells. In a small subset of cases, GCT is malignant. Terminology previously related to this entity, and which is no longer supported by the World Health Organization, includes osteoclastoma and benign fibrous histiocytoma (BFH). Giant cells occur in numerous other pathologic conditions of the bone, which accounts for the misrepresentation of these non-GCT tumors in the early literature. Non-ossifying fibroma (NOF), aneurysmal bone cyst, and chondroblastoma have been erroneously labeled GCT for this reason. A single description of an ancient GCT was reported by Brothwell and Sandison and subsequently mentioned by Aufderheide and Rodrìguez-Martìn who were astonished that more of these tumors had not been identified in archaeological cases. To the best of our knowledge, no other cases of ancient GCT have been cited in the paleopathology literature. The study of this type of neoplasm in antiquity can be used as a means to better understand its characteristics and behavior and to expand the depth of time of the etiology of these lesions. We report a case of GCT of the left femur observed following the total body CT imaging of a partially mummified adult female, dating to eighteenth century.

Synovial Pit of the femoral neck: a rare disease with rare presentations.

Herniation pits are small benign oval lesions that were reported to be always lying within the super-lateral femoral neck, and were first described in 1982 by Michael J. Pitt. They are usually a unilateral incidental finding along with asymptomatic course. It was widely believed that herniation pits are a result of invagination of the overlying synovium into small cortical defects in the femoral neck. In our case; the mentioned lesions were found atypically bilaterally at the inferomedial aspect of the neck of femur of a 7-year old child. Radiological scans were efficient to obtain an adequate diagnosis whereas conservative management proved to be sufficient dealing with the lesions. Synovial pits may have atypical clinical and radiological course, and this can raise concerns especially with symptomatic hip that may encourage surgical interventions. However, due to benign course of these lesions, we do not recommend any surgical intervention for such lesions.

Guided tissue engineering for healing of cancellous and cortical bone using a combination of biomaterial based scaffolding and local bone active molecule delivery.

A metaphyseal bone defect due to infection, tumor or fracture leads to loss of cancellous and cortical bone. An animal model separating the cancellous and cortical healing was used with a combination of a macroporous gelatin-calcium sulphate-hydroxyapatite (Gel-CaS-HA) biomaterial as a cancellous defect filler, and a thin collagen membrane (CM) guiding cortical bone regeneration. The membrane was immobilized with bone morphogenic protein-2 (rhBMP-2) to enhance the osteoinductive properties. The Gel-CaS-HA cancellous defect filler contained both rhBMP-2 and a bisphosphonate, (zoledronate = ZA) to prevent premature callus resorption induced by the pro-osteoclast effect of rhBMP-2 alone. In the first part of the study, the CM delivering both rhBMP-2 and ZA was tested in a muscle pouch model in rats and the co-delivery of rhBMP-2 and ZA via the CM resulted in higher amounts of bone compared to rhBMP-2 alone. Secondly, an established tibia defect model in rats was used to study cortical and cancellous bone regeneration. The defect was left empty, filled with Gel-CaS-HA alone, Gel-CaS-HA immobilized with ZA or Gel-CaS-HA immobilized with rhBMP-2+ZA. Functionalization of the Gel-CaS-HA scaffold with bioactive molecules produced significantly more bone in the cancellous defect and its surroundings but cortical defect healing was delayed likely due to the protrusion of the Gel-CaS-HA into the cortical bone. To guide cortical regeneration, the cortical defect was sealed endosteally by a CM with or without rhBMP-2. Subsequently, the cancellous defect was filled with Gel-CaS-HA containing ZA and rhBMP-2+ZA. In the groups where the CM was doped with rhBMP-2, significantly higher number of cortices bridged. The approach to guide cancellous as well as cortical bone regeneration separately in a metaphyseal defect using two bioactive molecule immobilized biomaterials is promising and could improve the clinical care of patients with metaphyseal defects.

Effects of mechanical loading on cortical defect repair using a novel mechanobiological model of bone healing.

Mechanical loading is an important aspect of post-surgical fracture care. The timing of load application relative to the injury event may differentially regulate repair depending on the stage of healing. Here, we used a novel mechanobiological model of cortical defect repair that offers several advantages including its technical simplicity and spatially confined repair program, making effects of both physical and biological interventions more easily assessed. Using this model, we showed that daily loading (5N peak load, 2Hz, 60 cycles, 4 consecutive days) during hematoma consolidation and inflammation disrupted the injury site and activated cartilage formation on the periosteal surface adjacent to the defect. We also showed that daily loading during the matrix deposition phase enhanced both bone and cartilage formation at the defect site, while loading during the remodeling phase resulted in an enlarged woven bone regenerate. All loading regimens resulted in abundant cellular proliferation throughout the regenerate and fibrous tissue formation directly above the defect demonstrating that all phases of cortical defect healing are sensitive to physical stimulation. Stress was concentrated at the edges of the defect during exogenous loading, and finite element (FE)-modeled longitudinal strain (εzz) values along the anterior and posterior borders of the defect (~2200µÎµ) was an order of magnitude larger than strain values on the proximal and distal borders (~50-100µÎµ). It is concluded that loading during the early stages of repair may impede stabilization of the injury site important for early bone matrix deposition, whereas loading while matrix deposition and remodeling are ongoing may enhance stabilization through the formation of additional cartilage and bone.

Plasma Neutrophil Gelatinase-Associated Lipocalin as a Predictor of Renal Parenchymal Involvement in Infants With Febrile Urinary Tract Infection: A Preliminary Study.

BACKGROUND: Urinary tract infection (UTI) is the most common bacterial infection in infants. Renal parenchymal involvement is an important prognostic factor; however, early detection of parenchymal involvement in UTI may be difficult during infancy. This study aimed to assess whether a recently established biomarker of UTI, neutrophil gelatinase-associated lipocalin (NGAL), can serve as a useful marker for the detection of cortical defects (CD) and to determine the appropriate diagnostic cut-off value of NGAL in infants with febrile UTI. METHODS: Infants hospitalized for febrile UTI were divided into two groups according to the presence of cortical defects on dimercaptosuccinic acid (DMSA) scintigraphy. Among 64 enrolled infants, 43 (67%) had CD (UTI-CD) and 21 (33%) had no CD (UTI-ND). The white blood cell count, C-reactive protein, and plasma NGAL (pNGAL) levels were determined before antibiotic therapy and compared between the two groups. RESULTS: pNGAL level was significantly higher in the UTI-CD group than in the UTI-ND group (340 µg/L vs 214 µg/L, P=0.002). Multivariate analysis showed that pNGAL level was the only independent predictor of CD (odds ratio 2.759, P=0.039). In the ROC curve analysis, pNGAL showed the highest area under the curve (0.745; 95% confidence interval, 0.561-0.821; P=0.014). The appropriate cut-off value of pNGAL was 267 µg/L (sensitivity, 72.1%; specificity, 71.4%). CONCLUSIONS: pNGAL was found to be a useful marker for early prediction of renal parenchymal involvement in infants with febrile UTI.

Soluble VEGFR1 reverses BMP2 inhibition of intramembranous ossification during healing of cortical bone defects.

BMP2 is widely used for promotion of bone repair and regeneration. However, bone formation induced by BMP2 is quite variable. Bone forming progenitor cells in different locations appear to respond to BMP2 in different ways, and repair outcomes can vary as a consequence of modulating effects by other factors. In this study, we have examined the effects of VEGF on BMP2-induced repair of a cortical bone defect, a 1 mm diameter drill hole, in the proximal tibia of mice. Treatment of the defect with either a bolus of PBS or soluble VEGFR1 (sVEGFR1), a decoy receptor for VEGF, had the same effects on bone formation via intramembranous ossification in the defect and cartilage formation and injured periosteum, during the healing process. In contrast, treatment with BMP2 inhibited intramembranous bone formation in the defect while it promoted cartilage and endochondral bone formation in the injured periosteum compared with mice treated with PBS or sVEGFR1. The inhibitory effect of BMP2 on bone formation was unlikely due to increased osteoclast activity and decreased invasion of blood vessels in the defect. Most importantly, co-delivery of BMP2 and sVEGFR1 reversed the inhibition of intramembranous bone formation by BMP2. Furthermore, the decreased accumulation of collagen and production of bone matrix proteins in the defect of groups with BMP2 treatment could also be prevented by co-delivery of BMP2 and sVEGFR1. Our data indicate that introducing a VEGF-binding protein, such as sVEGFR1, to reduce levels of extracellular VEGF, may enhance the effects of BMP2 on intramembranous bone formation. © 2016 Orthopaedic Research Society. Published by Wiley Periodicals, Inc. J Orthop Res 35:1461-1469, 2017.

Incidental occurrence of an unusually large mastoid foramen on cone-beam computed tomography and review of the literature.

The incidental finding of an enlarged mastoid foramen on the right posterior mastoid region of temporal bone is reported, together with a discussion of its clinical significance. A 67-year-old female underwent the pre-implant assessment of a maxillary left edentulous region. A cone-beam computed tomographic (CBCT) image was acquired and referred for consultation. Axial CBCT slices revealed a unilateral, well-defined, noncorticated, low-attenuation, transosseous defect posterior to the mastoid air cells in the right temporal bone. The borders of the osseous defect were smooth and continuous. No other radiographic signs suggestive of erosion or sclerosis were noted in the vicinity. The density within the defect was homogenous and consistent with a foramen and/or soft tissue. The patient's history and physical examination revealed no significant medical issues, and she was referred to a neuroradiologist for a second opinion. The diagnosis of an enlarged mastoid foramen was made and the patient was reassured.

Bone Regeneration and Remodeling within a Unidirectional Porous Hydroxyapatite Bone Substitute at a Cortical Bone Defect Site: Histological Analysis at One and Two Years after Implantation.

Unidirectional porous hydroxyapatite (UDPHAp) is an artificial bone substitute with a unique microstructure consisting of 100-300-µm oval pores that present the material unidirectionally. UDPHAp has a compression strength of 14 MPa and a porosity of 75%, which promotes cell migration and capillary formation within the material. Despite these advantageous properties, bone remodeling and bone formation with UDPHAp remain unclear. To examine long-term remodeling and differences in bone formation based on the defect site, trapezoidal prism-shaped UDPHAp blocks were implanted into rectangular-shaped cortical bone defects in the proximal tibia of Japanese white rabbits. Histological analysis performed at 52 and 104 weeks after implantation revealed that bone and capillaries had formed within the implanted UDPHAp material. Bone formed within the UDPHAp implanted in the cortical defect of rabbit tibia and remodel up to two years. The percentage of new bone area within UDPHAp was larger in cortical lesions than that in medullary lesions. These findings suggest that UDPHAp is a promising material for the repair of non-critical-sized cortical bone defects.

Scintigraphic depiction of non-ossifying fibromas and the role of SPECT/CT.

Non-ossifying fibromas (NOF) are a benign entity of the developing bone, relatively common in children and young adults. Their location is most frequently metaphyseal. They are usually asymptomatic (unless associated to a fracture) and have a self-limited behavior, with spontaneous regression through a sclerotic consolidation. Plain X-ray is the main imaging tool for its diagnosis. However, an unclear X-ray may lead to further imaging studies. We present the case of a 17-year-old male with back pain and lower limb dysmetria referred for a bone scintigraphy to complete the diagnostic and assess disease extension and the subsequent MRI evaluation.

Fracture arising of Steinmann pin cortical defect 3 years after insertion: A case report and review of literature.

A plethora of complications have been reported after insertion of a Steinmann pin amongst which the commonest ones are infection and pin loosening. We present a literature review on pathological fractures arising out of cortical defects left behind by pin insertion and report a unique case where a Steinmann pin insertion in proximal tibia has given rise to a fracture as late as 3 years after insertion.

Prevalence and risk factors for renal scars in children with febrile UTI and/or VUR: a cross-sectional observational study of 565 consecutive patients.

PURPOSE: To determine prevalence and risk factors for renal scar in children referred for urologic assessment of febrile UTI and/or VUR. METHODS: Pre-determined risk factors for renal scar were prospectively recorded in consecutive patients referred for UTI/VUR. Age, gender, VUR grade, and reported number of febrile and non-febrile UTIs were analyzed with logistic regression to determine risk for focal cortical defects on non-acute DMSA. RESULTS: Of 565 consecutive children, 24 (4%) had congenital renal dysplasia and 84 (15.5%) had focal defect(s). VUR, especially grades IV-V, recurrent febrile UTI, and older age increased risk. For any age child with the same number of UTIs, VUR increased odds of renal defect 5.4-fold (OR = 5.4, 95% CI = 2.7-10.6, AUC = 0.759). CONCLUSIONS: Focal DMSA defects were present in 15.5% of 565 consecutive children referred for febrile UTI and/or VUR; 4% had presumed congenital reflux nephropathy without cortical defect. All VUR grades increased risk for these defects, as did recurrent febrile UTIs and older age. However, 43% with grades IV-V VUR and 76% with recurrent UTI had normal DMSA.