Central Giant Cell Granuloma Treated with Intralesional Corticosteroid Injections and Bisphosphonates: A Long-Term Follow-Up Case Study.

Central giant cell granuloma (CGCG) is a benign intraosseous lesion of the head and neck with potential for aggressive and locally destructive behaviour. Lesions of the maxilla tend to expand more than those of the mandible due to the thinner cortices and spongy tissue of this location. Surgical removal is the most common treatment; however, it may be disfiguring in aggressive cases, especially for lesions located in the maxilla. Alternative treatments, such as intralesional corticosteroid injections, have been performed with satisfactory results. We report a case of a 12-year-old female patient with a CGCG of the left maxilla that was treated with 40 doses of intralesional triamcinolone acetonide infiltrations combined with alendronate sodium and calcium carbonate. Clinical and imaging follow-up over 12 years demonstrates improvement in the patient's condition.

Chronic non-healing ulcer of the oral cavity: tuberculosis or carcinoma?

OBJECTIVE: Tuberculosis is a chronic granulomatous lesion, which primarily has an affinity for the lungs. It can involve other sites like lymph nodes, kidney, oral cavity. Infection of the oral cavity by M. tuberculosis can be as a Primary infection or as a Secondary infection. Primary presentation of oral tuberculosis is in the form of the chronic non healing ulcer. A Primary infection or an Asymptomatic Secondary infection can impose a great diagnostic dilemma, as it may mimic neoplasia. Here we present a case of a 32-year-old asymptomatic female with secondary infection.

Complicaciones infecciosas severas del territorio maxilofacial: presentación de casos clínicos / Severe infectious complications of the maxillofacial territory: clinical cases presentation

RESUMEN: Las infecciones en el territorio maxilofacial, son cuadros frecuentes, de origen polimicrobiano, con manifestaciones clínicas muy variables y que están asociadas a múltiples vías de ingreso de los microorganismos al territorio. Un gran porcentaje de estas infecciones se origina en la cavidad oral, principalmente en lesiones bacterianas que sufren los dientes. La compleja anatomía de la cabeza y el cuello, permiten que muchas de estas infecciones se diseminen por espacios profundos, llegando a comprometer órganos o regiones anatómicas adyacentes, que pueden llevar a cuadros clínicos de alto riesgo vital. Los casos clínicos presentados en este artículo corresponden a pacientes tratados en el hospital San Juan de Dios a causa de procesos infecciosos del territorio maxilofacial, por equipos multidisciplinarios.

ABSTRACT: Infections in the maxillofacial territory are frequent cases of polymicrobial origin, with very variable clinical manifestations and are associated with multiple entering pathways of microorganisms in the territory. A large percentage of these infections originate in the oral cavity, mainly in bacterial lesions that undergo experienced by the teeth. The complex anatomy of the head and neck allows many of these infections to spread through deep spaces, leading to compromising adjacent organs or anatomical regions, which can lead to high-risk clinical conditions. The clinical cases presented in this article correspond to patients treated at the San Juan de Dios hospital because of infectious processes of the maxillofacial territory, by multidisciplinary teams.

Langerhans cell histiocytosis of the maxillae in a child treated only with chemotherapy: a case report.

BACKGROUND: Langerhans cell histiocytosis is a sporadic disease caused by an uncontrolled pathogenic clonal proliferation of dendritic cells that have Langerhans cell characteristics. New treatment protocols provided by the HISTSOC-LCH-III (NCT00276757) trial show an improvement in the survival of children with langerhans cell histiocytosis. CASE PRESENTATION: We report a case of Langerhans cell histiocytosis, which presented as an osteolytic lesion of the left pre-maxillae enclosing the deciduous incisor and canine in a 7-month-old white Italian boy. He was treated with chemotherapy. He achieved complete remission after 7 months and after 24 months no signs of recurrence were observed. CONCLUSIONS: As a result of this treatment, anesthetic sequelae and loss of teeth were avoided; in addition, we prevented a loss of the vertical dimension of occlusion.

Case of disseminated histoplasmosis in a HIV-infected patient revealed by nasal involvement with maxillary osteolysis.

BACKGROUND: Disseminated Histoplasmosis (DH) is a rare manifestation of Acquired Immune Deficiency Syndrome (AIDS) in European countries. Naso-maxillar osteolysis due to Histoplasma capsulatum var. capsulatum (Hcc) is unusual in endemic countries and has never been reported in European countries. Differential diagnoses such as malignant tumors, cocaine use, granulomatosis, vasculitis and infections are more frequently observed and could delay and/or bias the final diagnosis. CASE PRESENTATION: We report the case of an immunocompromised patient infected by Human Immunodeficiency Virus (HIV) with naso-maxillar histoplasmosis in a non-endemic country. Our aim is to describe the clinical presentation, the diagnostic and therapeutic issues. A 53-year-old woman, originated from Haiti, was admitted in 2016 for nasal deformation with alteration of general condition evolving for at least 6 months. HIV infection was diagnosed in 2006 and classified at AIDS stage in 2008 due to cytomegalovirus infection associated with pulmonary histoplasmosis. At admission, CD4 cell count was 9/mm3. Surgical biopsies were performed and ruled out differential or associated diagnoses. Mycological cultures identified Hcc and Blood Polymerase Chain Reaction (PCR) for Hcc was positive. The patient was given daily Amphothericin B liposomal infusion during 1 month. Hcc PCR became negative in the blood under treatment, and then oral switch by itraconazole was introduced. Antiretroviral treatment was reintroduced after a 3-week histoplasmosis treatment. Normalization of naso-maxillar mucosa enabled a palatal prosthesis. CONCLUSION: Naso-maxillar histoplasmosis is extremely rare; this is the first case ever reported in a non-endemic country. Differential diagnoses must be ruled out by conducting microbiologic tools and histological examinations on surgical biopsies. Early antifungal treatment should be initiated in order to prevent DH severe outcomes.

[Correctly adDRESS the cause of hemophagocytic lymphohistiocytosis]. / Pièges diagnostiques d'un syndrome d'activation macrophagique.

Hemophagocytic lymphohistiocytosis (HLH) is a rare and severe syndrome usually associated with a cytotoxicity deficiency, which leads to an excess of immune response driven by activated macrophages and cytotoxic T cells. In children, HLH can be genetic, as part of a familial lymphohistiocytosis, or secondary: the most frequent causes are systemic-onset juvenile idiopathic arthritis, hematological malignancies, and severe infections, especially with Ebstein-Barr virus or leishmaniosis. We report on the case of a 3-year-old girl with no past medical history, who presented inaugural Pseudomonas aeruginosa maxillary osteitis, with secondary HLH. The rarity of this osteitis, the characteristics of the pathogen, and the onset of HLH oriented the diagnosis toward primary immunodeficiencies, malignancies, or systemic diseases. Steroids were initiated at 2mg/kg/day and were very effective in improving the systemic symptoms. Antibiotic therapy was continued unchanged. A few days after discontinuation of steroids, while the patient was still under antibiotics, she presented with erythroderma. Skin biopsy revealed eosinophil infiltrate in line with the diagnosis of a drug reaction with eosinophilia and systemic symptoms (DRESS), even though we only observed very transient eosinophilia, up to 0.98G/L, during HLH. Stopping antibiotics normalized the symptoms without using systemic corticosteroids. Patch tests confirmed an allergy to piperacillin. These atypical manifestations of DRESS underline that causative diagnosis of HLH is challenging, and DRESS syndrome should be considered.

Inlay osteotome sinus floor elevation with concentrated growth factor application and simultaneous short implant placement in severely atrophic maxilla.

Sinus floor elevation with simultaneous implant placement in severely atrophic maxilla is challenging. The aim of this retrospective study was to evaluate the short-term performance of modified osteotome sinus floor elevation (OSFE) with concentrated growth factor (CGF) application and concurrent placement of a short implant in cases with residual bone height (RBH) of 2-4 mm. Twenty-five short implants were installed in 16 patients with mean RBH of 3.23 mm using modified OSFE with CGFs from January 2012 to April 2014. Postoperatively, the implants were clinically evaluated, and vertical bone gain (VBG) was measured using cone beam computed tomography. The mean duration of follow-up was 19.88 months (12-32 months). All the implants were stable with an overall survival rate of 100%. The mean VBG immediately after surgery was 9.21 mm. Six months later, significant reduction of alveolar bone height (2.90 ± 0.22 mm) was found (P < 0.05). During the second 6-month period, further alveolar bone resorption (0.14 ± 0.11 mm) was noted but without significance (P > 0.05). Within the limits of this study, modified OSFE with CGF application and simultaneous short implant placement could yield predictable clinical results for severely atrophic maxilla with RBH of 2-4 mm.

Clinical Use of Deferoxamine in Distraction Osteogenesis of Irradiated Bone.

The deleterious effects of radiotherapy, including hypovascularity and hypocellularity, have made distraction of irradiated bones challenging. Animal studies, however, have demonstrated adjunctive measures such as the administration of deferoxamine to significantly improve bone regeneration across irradiated distraction gaps. In this report, the authors demonstrate, for the first time, enhanced bone formation following deferoxamine application in a patient following distraction of a previously irradiated maxilla. Computed tomography imaging of the pterygomaxillary buttress on the side of administration revealed significantly increased bone area and density relative to the contralateral buttress. This is the first presentation of clinical deferoxamine use to promote bone formation following irradiated bone distraction and highlights the promise for this adjunctive measure to make outcomes after distraction of irradiated bone more reliable.

Myricetin Prevents Alveolar Bone Loss in an Experimental Ovariectomized Mouse Model of Periodontitis.

Periodontitis is a common chronic inflammatory disease, which leads to alveolar bone resorption. Healthy and functional alveolar bone, which can support the teeth and enable their movement, is very important for orthodontic treatment. Myricetin inhibited osteoclastogenesis by suppressing the expression of some genes, signaling pathways, and cytokines. This study aimed to investigate the effects of myricetin on alveolar bone loss in an ovariectomized (OVX) mouse model of periodontitis as well as in vitro osteoclast formation and bone resorption. Twenty-four healthy eight-week-old C57BL/J6 female mice were assigned randomly to four groups: phosphate-buffered saline (PBS) control (sham) OVX + ligature + PBS (vehicle), and OVX + ligature + low or high (2 or 5 mg∙kg(-1)∙day(-1), respectively) doses of myricetin. Myricetin or PBS was injected intraperitoneally (i.p.) every other day for 30 days. The maxillae were collected and subjected to further examination, including micro-computed tomography (micro-CT), hematoxylin and eosin (H&E) staining, and tartrate-resistant acid phosphatase (TRAP) staining; a resorption pit assay was also performed in vitro to evaluate the effects of myricetin on receptor activator of nuclear factor κ-B ligand (RANKL)-induced osteoclastogenesis. Myricetin, at both high and low doses, prevented alveolar bone resorption and increased alveolar crest height in the mouse model and inhibited osteoclast formation and bone resorption in vitro. However, myricetin was more effective at high dose than at low dose. Our study demonstrated that myricetin had a positive effect on alveolar bone resorption in an OVX mouse model of periodontitis and, therefore, may be a potential agent for the treatment of periodontitis and osteoporosis.

Disease Stage and Mode of Therapy Are Important Determinants of Treatment Outcomes for Medication-Related Osteonecrosis of the Jaw.

PURPOSE: The treatment of patients with medication-related osteonecrosis of the jaw (MRONJ) is challenging. The purpose of the present study was to estimate the frequency and identify the factors associated with clinical improvement during treatment. PATIENTS AND METHODS: We designed and implemented a retrospective cohort study and enrolled a sample of subjects diagnosed with MRONJ between 2004 and 2015. The primary predictor variables were a set of heterogeneous variables grouped into the following categories: demographic (age and gender) and clinical (location of necrosis, therapy duration, medication type, disease stage, and treatment type). The primary outcome variable was the treatment outcome, defined as stable or worse and improved or healed. The descriptive, bivariate, and multiple logistic statistics were computed, and statistical significance was defined as P < .05. RESULTS: The sample included 337 subjects with a mean age of 68.9 years. Of the 337 subjects, 256 were women (76%). A total of 143 patients (42.2%) experienced spontaneous necrosis. Twenty-four (7.1%) had had exposure to targeted antiangiogenic agents. Those with stage 1 or 2 disease were more likely to have better outcomes than those with stage 3 disease (stage 1, adjusted odds ratio [OR] 3.4, P = .005; stage 2, adjusted OR 2.2, P = .03). Treatment type was a significant variable. Subjects undergoing surgery were 28 times more likely to have a positive outcome than those receiving nonoperative therapy (adjusted OR 28.7, P < .0001). CONCLUSIONS: Subjects with MRONJ who presented with less severe disease or who underwent operative treatment were most likely to have improvement or complete healing of their MRONJ-related lesions.

Osteoradionecrosis of the Posterior Maxilla: A New Approach Combining Erbium: Yttrium Aluminium Garnet Laser and Bichat Bulla Flap.

Osteoradionecrosis (ORN) of the jaw is a complication of radiation therapy for head and neck cancers. We report a case of ORN of the posterior maxilla treated with Er: YAG laser and a pedicled buccal fat pad (bichat bulla adipose) flap. A 69-year-old man presented complaining of pain on left maxilla. He had received high-dose radiotherapy (90 Gy) for squamous cell carcinoma of the left soft palate 2 years earlier. Clinical and radiographic examinations revealed ORN of the left maxillary molar region and maxillary sinusitis. Daily home care consisted of 0.9% saline irrigation and 0.8% H2O2 gel application. Sequestrectomy and tooth extraction were followed by debridement with Er: YAG laser and repair with a pedicled buccal fat pad flap. Complete resolution of ORN and maxillary sinusitis was established one year postsurgically. The excellent clinical outcome suggests that Er: YAG laser debridement and pedicled buccal fat pad flap are a viable option to treat ORN of the posterior maxilla.

Fibroblast growth factor-2 promotes healing of surgically created periodontal defects in rats with early, streptozotocin-induced diabetes via increasing cell proliferation and regulating angiogenesis.

AIM: To evaluate the effects of fibroblast growth factor (FGF)-2 on the healing of surgical periodontal defects in rats with early, streptozotocin-induced diabetes. MATERIALS AND METHODS: Fifty Wistar rats were assigned to streptozotocin-induced diabetes or non-diabetes group. Periodontal defects were surgically created at maxillary first molars. Defects were treated with hydroxypropyl cellulose (HPC) or FGF-2 with HPC. Defect fill was evaluated by microcomputed tomography. Histological and immunohistochemical analyses were performed. RESULTS: Compared to vehicle alone, FGF-2 treatment yielded significantly greater bone volume and trabecular thickness in diabetes group. Diabetes group displayed reduced new bone formation and significantly longer epithelial down-growth compared to non-diabetes group. In diabetes group, FGF-2 treatment increased PCNA-positive cells and new bone formation after 2 weeks and suppressed epithelial down-growth, but new cementum formation was minimal even after 4 weeks. In diabetes group, overexpression of vascular endothelial growth factor was evident in cells within connective tissue, and no significant enhancement was observed by FGF-2 treatment. FGF-2 increased the expression of α-smooth muscle actin in diabetes group. CONCLUSIONS: Treatment of surgical periodontal defects in diabetic rats with the single application of FGF-2 provided beneficial effects primarily on new bone formation via increasing cell proliferation and regulating angiogenesis.

Combination of LED light and platelet-derived growth factor to accelerate dentoalveolar osteogenesis.

AIM: This study aimed to evaluate the adjunctive effect of LED light in platelet-derived growth factor (PDGF)-aided dentoalveolar osteogenesis. MATERIAL AND METHODS: Full-thickness osseous wounds were created on rat maxillae and were either unfilled or filled with poly-(D,L-lactide) and poly-(D,L-lactide-co-glycolide) microspheres encapsulating PDGF. Animals received daily 660 ± 25 nm LED light irradiation at 0, 10 (LD), or 20 (HD) J/cm(2) , were killed at days 4-28 (n = 6/group/time) and evaluated by microcomputed tomography (micro-CT), histology, and the expressions of osteopontin and tartrate-resistant acid phosphatase (TRAP). RESULTS: Greater osteogenesis was noted in the PDGF-treated defects at day 14. Under the LED light irradiation, osteogenesis was significantly greater in both LD and HD groups of the non-PDGF-treated defects, but only in the LD group of the PDGF-treated defects. No significant differences in osteogenesis among groups were noted at day 28. Greater bone marrow space was noted in the LED light-irradiated specimens, especially in the PDGF-treated defects at both time points. Osteopontin was significantly promoted in the LD group at both time points, and TRAP was significantly promoted in all LED light-irradiated groups at day 28. CONCLUSION: LED light could an adjunct to promote early PDGF-aided dentoalveolar osteogenesis by facilitating the osteoblast-osteoclast coupling.

Adjuvant aqueous ozone in the treatment of bisphosphonate induced necrosis of the jaws: report of two cases and long-term follow-up.

Bisphosphonate induced necrosis of the jaws (BONJ) does not have a unique protocol of treatment and many therapeutic approaches have been arising in oral medicine with debatable results. A male and a female attended the University Oral Surgery Clinic presenting oral bone lesions induced by intravenous and oral bisphosphonates respectively as complications of dental extraction. Treatment included daily mouthwashes and weekly intra oral irrigations with 4 mg/L of aqueous-ozone, antibiotic therapy and sequential superficial debridment for sequestrectomies. Long-standing follow-ups showed complete mucosa covering of exposed bone area and resolution of purulent secretion. Antibacterial and antifungal properties of aqueous ozone may have played important roles in the treatment. The outcome measured intra oral examination and panoramic radiographs of the affected bone. The application of aqueous ozone daily mouthwashes and weekly professional irrigation were safe; free from adverse effects, easily of handling and worked as an important adjuvant therapeutic strategy for the treatment of BONJ.

Teriparatide therapy for bisphosphonate-related osteonecrosis of the jaw: a case report and narrative review.

Bisphosphonates (BP), potent antresorptive agents, play a key role in managing osteolytic bone disorders including osteoporosis, Paget's disease, bone metastasis, and multiple myeloma. However, their long-term administration is associated with increased risk for bisphosphonate-related osteonecrosis of the jaw (BRONJ) development. At present, there is no curative therapy for BRONJ, and patients are often treated palliatively with antibiotics, antimicrobial mouth rinses, and debridement of necrotic bone. This article highlights a new treatment modality that may be beneficial to a subset of osteoporosis patients suffering from BRONJ. Here we report a BRONJ case that was initially unresponsive to conservative treatment, but subsequently responded to teriparatide (recombinant human PTH1-34) therapy.

[Bisphosphonate related osteonecrosis of the jaw and infection with Actinomyces]. / Osteonecrosis maxilar asociada a bisfosfonatos e infección por Actinomyces.

BACKGROUND AND OBJECTIVE: Bisphosphonate related osteonecrosis of the jaw (BRONJ) has raised considerable interest since its recent description. Its pathogenesis is not yet clarified; formerly it has been considered a non-infectious complication, but recent studies seem to implicate bacteria of the genus Actinomyces. The objective of this study is to analyze the cases of BRONJ in our institution. PATIENTS AND METHODS: Review of medical records of patients diagnosed of BRONJ in the Maxillofacial Surgery Unit of our hospital. RESULTS: We have found 11 cases of BRONJ in our hospital: 4 women taking oral alendronate or risendronate for osteoporosis and 7 cancer patients treated with intravenous zolendronic acid. All of them showed bone invasion by bacteria of the genus Actinomyces. Nine patients underwent prolonged treatment with amoxicillin with favourable clinical outcome in all of them, but 3 died of their malignancy. By contrast, one patient with beta-lactamic allergy and irregular treatment with erythromycin and tetracycline had a chronic evolution of the lesions. There was no information for other patient. CONCLUSIONS: Actinomyces play an important role in the development of BRONJ and specific antibiotic treatment improves the prognosis of this process.

Primary tuberculosis: an unusual finding in the oral cavity.

The unusual involvement of the oral cavity in tuberculosis and the non-specific nature of its presentations mean that diagnosis of tuberculosis is often delayed and is an unexpected finding. The aim of this paper is to present a case of primary tuberculosis and discuss the implications of the manifestations and diagnosis of oral tuberculosis. This paper presents an unusual case of a painless, papillary, erythematous lesion in the anterior region of a maxillary edentulous ridge. When the patient concerned was first seen by the author, the lesion had been present for six months. There was cervical lymphadenopathy and it was diagnosed initially as a malignant lesion. Eventually, after biopsy and ultrasound examination, the diagnosis of primary oral tuberculosis was reached. The patient was managed solely by anti-tubercular drug therapy.

Glucocorticoid and calcitonin receptor expression in central giant cell lesions: implications for therapy.

Central giant cell lesion is an uncommon benign jaw lesion, with uncertain aetiology, and variable clinical behaviour. Studies of molecular markers may help to understand the nature and behaviour of this lesion, and eventually may represent a target for pharmacological approaches to treatment. The aim of this study was to analyse the expression of glucocorticoid and calcitonin receptors in central giant cell lesions before and after treatment with intralesional steroid. Paraffin-embedded blocks from patients who underwent treatment with intralesional triamcinolone hexacetonide injections were stained immunohistochemically. Biological material from patients who underwent a surgical procedure after treatment were tested immunohistochemically. 18 cases (9 aggressive and 9 non-aggressive) were included. The difference in calcitonin receptor expression was not statistically significant between the aggressive and non-aggressive lesions and between the patients with a good response and those with a moderate/negative response to treatment. Glucocorticoid receptor expression in the multinucleated giant cells was higher in patients with a good response. It can be postulated that immunohistochemical staining for glucocorticoid receptors may provide a tool for selecting the therapeutic strategy. An H-score greater than 48 for glucocorticoid receptors in multinucleated giant cells predicted a good response in this study.

Multiple myeloma vs. breast cancer patients with bisphosphonates-related osteonecrosis of the jaws: a comparative analysis of response to treatment and predictors of outcome.

BACKGROUND: Multiple myeloma (MM) and breast cancer (BC) are the two most common diseases associated with bisphosphonates-related osteonecrosis of the jaws (BRONJ), for which different therapeutical approaches have been proposed. The aim of this study was to compare the clinical behaviour of BRONJ in patients with MM vs. BC and the time of healing in terms of clinical and symptomatological remission, following a standardized therapeutic protocol. METHODS: Twenty-six BRONJ patients (13 men with MM and 13 women with BC) were prospectively enroled and treated with a specific systemic and topical antibiotic therapy. Several predictors of outcome were also evaluated. RESULTS: Nine patients (69.2%) with BC and 10 patients (76.9%) with MM progressed towards a complete clinical remission (CR) in a mean healing time of 183.3 days [SD: 113.7; 95% confidence interval (CI): 95.95-207.7] and 372.0 days (SD: 308.0; 95% CI: 151.7-592.3) (P = 0.776), respectively. The clinical improvement was statistically significant (P = 0.0013 and P = 0.0014), as well as the assessment of pain (P = 0.0015 and P = 0.0015), in MM and BC group, respectively. Cox regression analysis revealed that just triggering events (P = 0.036) were found to be significant predictors of outcome of BRONJ healing. CONCLUSIONS: Both groups of cancer patients experienced clinical and symptomatological remission regardless their malignancy, but BC patients earlier than MM patients.

Identification and treatment of bisphosphonate-associated actinomycotic osteonecrosis of the jaws.

Osteonecrosis of the jaws (ONJ) is a condition characterized by necrotic exposed bone in the jaws of patients receiving intravenous or oral bisphosphonate therapy. A review of the medical and dental literature reveals that the pathoetiology of ONJ remains unknown and there is no established link that bisphosphonates are the primary cause of this bone pathology. However, there is clinical evidence that Actinomyces may play a critical role in the pathogenesis of bisphosphonate-associated ONJ. Identification and a prolonged course of oral antimicrobial therapy may lead to complete resolution of this actinomycotic osteonecrosis.