Extra-mandibular Osteoradionecrosis after the Treatment of Head and Neck Cancer.

AIMS: Osteoradionecrosis (ORN) is a serious toxicity of head and neck radiotherapy. It predominantly affects the mandible. Extra-mandibular ORN is rare. The aim of this study was to report the incidence and outcomes of extra-mandibular ORNs from a large institutional database. MATERIALS AND METHODS: In total, 2303 head and neck cancer patients were treated with radical or adjuvant radiotherapy. Of these, extra-mandibular ORN developed in 13 patients (0.5%). RESULTS: Maxillary ORNs (n = 8) were a consequence of the treatment of various primaries (oropharynx = 3, sinonasal = 2, maxilla = 2, parotid = 1). The median interval from the end of radiotherapy to the development of ORN was 7.5 months (range 3-42 months). The median radiotherapy dose in the centre of the ORN was 48.5 Gy (range 22-66.5 Gy). Four patients (50%) healed in 7, 14, 20 and 41 months. All temporal bone ORNs (n = 5) developed after treatment to the parotid gland (of a total of 115 patients who received radiotherapy for parotid gland malignancy). The median interval from the end of radiotherapy to the development of ORN was 41 months (range 20-68 months). The median total dose in the centre of the ORN was 63.5 Gy (range 60.2-65.3 Gy). ORN healed in only one patient after 32 months of treatment with repeated debridement and topical betamethasone cream. CONCLUSION: Extra-mandibular ORN is a rare late toxicity and this current study provides useful information on its incidence and outcome. The risk of temporal bone ORN should be considered in the treatment of parotid malignancies and patients should be counselled. More research is required to determine the optimal management of extra-mandibular ORN, particularly on the role of the PENTOCLO regimen.

Malignant transformation of chronic osteomyelitis of mandible: A rare presentation.

ABSTRACT: Chronic osteomyelitis is a bone disease that is characterized by inflammatory processes, including necrosis of mineralized and marrow tissues, suppuration, resorption, sclerosis, and hyperplasia. The primary cause of chronic osteomyelitis is usually microbiologic, and it results from an odontogenic infection, postextraction complication, inadequate removal of necrotic bone, early termination of antibiotic therapy, inappropriate selection of antibiotics, diagnostic failure, trauma, inadequate treatment for a fracture, or irradiation to the mandible. The incidence of malignant transformation of chronic osteomyelitis in the head and neck region is very low and rare in developed countries. This is the first case report on the malignant transformation of chronic osteomyelitis of the mandible which discusses its etiopathogenesis and radiographic imaging.

Osteoradionecrosis after postoperative radiotherapy for oral cavity cancer: A retrospective cohort study.

OBJECTIVE: Osteoradionecrosis (ORN) is a severe late complication after radiotherapy but current knowledge on ORN risks in the setting of postoperative radiotherapy (PORT) is limited. We studied the incidence and risk factors of ORN in patients with oral cavity cancers (OCC, treated with PORT. PATIENTS AND METHODS: A retrospective cohort study was conducted including OCC patients (mainly squamous cell) treated with postoperative intensity modulated radiotherapy between 2010 and 2018 with > 1 year disease-free survival. Cumulative incidences of ORN were computed using the Kaplan Meier method. Clinical and dosimetric risk factors for mandibular ORN were evaluated using Cox regression models. RESULTS: Within our cohort (N = 227, median follow-up 49 months) we observed 46 cases of ORN, mainly in the mandible (n = 41). The cumulative incidence of mandibular ORN was 15.9 % (SE 2.5 %) at three years and 19.8 % (SE 3.0 %) at five years. At univariable analysis, smoking, mandibular mandibulotomy or segment resection, mean dose to the mandible, and mandible volume (%) ≥ 60 Gy (V60) were significantly associated with increased ORN risks. At multivariable analysis, smoking (HR 2.13, 95 %CI 1.12-4.06) and V60 (HR 1.02 per 1 % increase, 95 %CI 1.01-1.04) remained predictive factors. For active smokers with a high V60 ≥ 40 % we observed rapid ORN development with a 1-year incidence of 29 % vs 6 % for others (p < 0.01). CONCLUSION: OCC Patients treated with PORT are at high risk for mandibular ORN. We identified the mandibular volume receiving ≥ 60 Gy as the dominant risk factor, especially in active smokers. Limiting high-dose volumes at treatment planning may decrease ORN risks.

Bisphosphonate therapy in chronic diffuse sclerosing osteomyelitis/tendoperiostitis of the mandible: Retrospective case series.

This study aims to evaluate short-term and long-term results of bisphosphonate therapy in patients with diffuse sclerosing osteomyelitis/tendoperiostitis (DSO/TP) of the mandible. Eighteen patients (12 female, 6 male) aged 34.8 ± 22.2 years with DSO/TP of the mandible that were treated with bisphosphonates were included. In 16 patients, the bisphosphonate treatment led to remission with decrease of symptoms (pain, swelling of the cheek, trismus, tenderness of masticatory muscles) with a follow-up period of 4.5 (0.8-11.9) years between start of bisphosphonate treatment and latest follow-up consult. Of these, three patients were still in need of regular bisphosphonate therapy. Two patients were lost to follow-up. Bisphosphonate therapy is a treatment option for DSO/TP of the mandible that is associated with a high chance of remission of symptoms. Within the limitations of the study it seems that this treatment might be an effective second step in DSO/TP refractory to conservative treatment.

Osteosclerotic metaphyseal dysplasia, dysosteosclerosis or osteomyelitis? Paediatric case presentation with associated mandibular swelling and a review of the literature.

Osteosclerotic metaphyseal dysplasia (OMD) is an extremely rare form of osteopetrosis, which bears significant clinical similarities to dysosteosclerosis (DSS). We aim to present a rare case of OMD with mandibular swelling and osteomyelitis infection including diagnosis journey as well as management in 7-year-old patient. Literature review completed for OMD cases. Case report investigative methods include genetic testing, CT facial bones and MRI scan, orthopantogram and bone biopsies. An initial suspected diagnosis of DSS with chronic osteomyelitis was made. However, following genetic testing, a diagnosis of OMD was confirmed. Our patient underwent a surgical debulking procedure and antibiotic treatment. Less than 10 patients with this condition have been reported within the international literature. There is a wide range of presentation. OMD, DSS and osteomyelitis are all within a similar spectrum of bone conditions. Our understanding, regarding OMD, remains limited and, hence, further research is required to elucidate a thorough clinical picture.

Speech and Swallowing Outcomes Following Surgical Resection with Immediate Free Tissue Transfer Reconstruction for Advanced Osteoradionecrosis of the Mandible Following Radiation Treatment for Head and Neck Cancer.

Despite recent advances in the radiation techniques used for the treatment of head and neck cancer (HNC) including intensity-modulated radiotherapy (IMRT), mandibular osteoradionecrosis (ORN) remains a significant complication. Advanced stage ORN is managed surgically with resection and immediate free tissue transfer reconstruction. An evaluation of the functional speech and swallowing outcomes was undertaken for patients undergoing surgical management of advanced ORN. We retrospectively reviewed consecutive patients, at a single, tertiary cancer centre, who underwent surgical resection for advanced Notani grade III ORN. Outcomes investigated included use and duration of tracheostomy and swallowing and speech status using Performance Status Scale for Head and Neck Cancer Normalcy of Diet (PSS-NOD) and Understandability of Speech (PSS-Speech) at baseline and 3 months following surgery. Ten patients underwent surgical resection with free tissue transfer reconstruction between January 2014 and December 2019. Two patients required supplemental nutrition via a gastrostomy at three months post surgery. As per the PSS-NOD data half of the patients' (n = 5) diet remained stable (n = 2) or improved (n = 3) and half of the participants experienced a decline in diet (n = 5). The majority of patients had no speech difficulties at baseline (n = 8). The majority of patients' speech remained stable (n = 8) with two patients experiencing a deterioration in speech clarity following surgery. Well-designed studies with robust, sensitive multidimensional dysphagia and communication assessments are required to fully understand the impact of surgical management of advanced ORN using resection with free tissue transfer reconstruction.

Nutritional status in patients of mandibular osteoradionecrosis: A single-institution experience.

BACKGROUND: Mandibular osteoradionecrosis (ORN) is a devastating complication secondary to the radiotherapy of head and neck cancer. The nutritional status of ORN patients is compromised, but remains rarely studied. We aimed to evaluate the overall nutritional status of patients with ORN and explore the risk factors behind poor nutrition. METHODS: This is a single-institution cross-sectional study. Patients diagnosed with ORN were consecutively recruited in a tertiary teaching hospital from July 2017 to August 2019. Multiple laboratory markers and physical indicators were examined to profile their nutritional status. The potential risk factors of poor nutrition were explored by logistic regression. RESULTS: A total of 107 patients with ORN were recruited. Among them, almost all patients (95.3%) had at least one laboratory marker lower than the normal physiological range. A total of 40 (37.5%) patients were categorized as undernutrition, who had lower serum albumin (mean difference: 1.8 ± 0.8 g/L; p = .02), prealbumin (mean difference: 26.8 ± 10.8 mg/L; p = .02), and BMI (3.8 ± 0.4 kg/m2 ; p < .0001) compared to patients of normal nutrition. Notably, the multivariate logistic regression indicated that patients with semi-liquid diet had 14.41 (95% CI: 3.03-68.54, p = .001) times; patients with liquid diet had 5.70 (95% CI: 1.55-20.98, p = .009) times more likely to be in undernutrition, as compared to patients with regular diets. CONCLUSIONS: This is the first study characterizing the poor nutritional status in ORN patients. Patients having semi-liquid or liquid diets tended to have poorer nutritional status. The nutritional status of ORN patients should be underlined for professional nutritional supports so as to enhance their quality of life. More studies are warranted.

Unusually sited lower third molar.

We present the case of a 60-year-old woman who presented to our unit with left-sided facial swelling, pain and trismus. Initially managed as a parotitis by a different specialty, an ultrasound subsequently showed a collection deep to the parotid associated with an ectopic wisdom tooth within the mandibular posterior ramus/condyle and the patient was referred to our department. After treating the acute infection, the wisdom tooth was surgically removed. Our case highlights the importance of the clinician maintaining an open mind to differential diagnoses and details a technique for surgical removal of a tooth with difficult access.

Occurrence of nonarticular incidental findings on panoramic radiographs in painful temporomandibular disorders / Ocurrencia de hallazgos incidentales no articulares en ortopantomografias en los trastornos temporomandibulares dolorosos

Facial pain is one of the symptoms of temporomandibular disorders (TMDs) but can be associated with other pathological conditions. The present study retrospectively evaluated the occurrence of nonarticular incidental findings in panoramic radiographs in a group of patients with painful TMDs. Outpatients with a diagnosis of TMD were included and distributed into three groups: arthralgia, myalgia or arthralgia and myalgia, according to the Diagnostic Criteria for Temporomandibular Disorders (DC/TMD). Nonarticular incidental findings of their panoramic radiographs were classified in relation to pathological, dental and endodontic status. The dependency relationships among the variables were analyzed using the likelihood test. Sixty patients (38 women and 22 men; mean age: 36.9 years) were evaluated. There was a predominance of arthralgia plus disc displacement (43.4 %), followed by myopain plus arthralgia and disc displacement (38.3 %) and myopain (18.3 %). Pathologic radiographic changes such as bone loss, caries, maxillary sinus opacification and periapical lesions were frequent. Dental alterations such as the absence and altered position of teeth and impacted teeth were frequent. Endodontic changes such as periapical lesions with or without endodontic treatment were frequent. There was no significant difference between groups except for gyroversion. The incidental findings were compatible with caries, sinusitis, impacted tooth and periapical lesion, which may be associated with orofacial pain and could potentially be superimposed on the initial diagnosis, although this hypothesis was discarded. Findings such as the absence and altered position of teeth were also frequent, which may represent occlusal factors associated with TMDs.

El dolor orofacial es uno de los síntomas de los trastornos temporomandibulares (TTM), pero puede estar asociado con otras afecciones patológicas. El presente estudio evaluó retrospectivamente la aparición de hallazgos incidentales no articulares a través de ortopantomografías en un grupo de pacientes con TTM dolorosas. Se incluyeron pacientes con diagnóstico de TTM y se distribuyeron en tres grupos: artralgia, mialgia o artralgia y mialgia, de acuerdo con los criterios de diagnóstico para los trastornos temporomandibulares (DC/TMD). Los hallazgos incidentales no articulares de las ortopantomografias se clasificaron en relación al estado patológico, dental y endodóncico. Las relaciones de dependencia entre las variables se analizaron mediante la prueba de probabilidad. Fueron evaluados 60 pacientes (38 mujeres y 22 hombres; edad media: 36,9 años). Hubo predominio de artralgia más desplazamiento de disco (43,4 %), seguido de mialgia más artralgia y desplazamiento de disco (38,3 %) y mialgia (18,3 %). Las alteraciones radiográficas patológicas como pérdida ósea, carie dentaria, opacificación del seno maxilar y lesiones periapicales fueron frecuentes. Entre las alteraciones dentales, las impactaciones, malposiciones o ausencias dentarias fueron frecuentes. Entre las alteraciones endodóncicas, las lesiones periapicales frecuentes. No hubo diferencias significativas entre los grupos, excepto para la girosversión dentaria. Los hallazgos incidentales fueron compatibles con carie dentaria, sinusitis, diente impactado y lesión periapical, lo que podría estar asociado con el dolor orofacial y así estar sobrepuesto en el diagnóstico inicial, aunque esta hipótesis fue descartada. Hallazgos como la ausencia y la posición alterada de los dientes también fueron frecuentes, lo que puede representar factores oclusales asociados con TTM.

Non-surgical treatment of adults with chronic diffuse sclerosing osteomyelitis/tendoperiostitis of the mandible.

Non-surgical therapy has proved to be effective in chronic diffuse sclerosing osteomyelitis (DSO) of the mandible in children. Therefore we aimed to investigate the effect of non-surgical therapy in adult DSO patients. We included consecutive patients with DSO who received non-surgical therapy in our center. They all received occlusal splint therapy, counselling about the disease, and/or physiotherapy by a specialised team. The use of analgesics, preferably nonsteroidal anti-inflammatory drugs, was advised for symptomatic control during periods of exacerbation. Sixteen patients (11/5 female/male) aged 39.9 ± 15.0 years with DSO of the mandible were included. The mean duration of symptoms was 39.7 ± 26.3 months before referral to our center. Patients were treated with a broad range of treatments before referral. All patients underwent non-surgical treatment. In 12 patients this led to remission. Four patients still had complaints after 12 months of non-surgical therapy and started with intravenous bisphosphonate therapy. In our center, DSO of the mandible was successfully treated with non-surgical therapy, despite a long duration before referral and extensive pre-treatment. Considering this high success rate, we recommend this non-surgical approach as the first treatment option for DSO of the mandible. In case of persistence, alternative treatments such as bisphosphonates should be explored.

Symptomatic Mandibular Fibrous Dysplasia With Concurrent Triple X- and Premutation Stage Fragile-X-Syndrome: Case Report With Short Literature Survey.

BACKGROUND: Certain constitutive chromosomal abnormalities of the human X chromosome are relatively common in conspicuous neuropsychiatric findings. Although tumors or tumor-like lesions are occasionally reported in diseases of the X chromosome, they are numerically negligible, for example, in aneuploidy such as the triple X syndrome (TXS). CASE REPORT: A 16-year-old female patient with a known TXS and premutation stage of fragile X syndrome was referred by her dentist for diagnosis and treatment of unilateral cheek swelling. The examination of the psychologically conspicuous patient revealed a unilateral mandibular tumor with dysesthesia of the mental nerve. Surgical removal of soft, crumbly spongiosa over the nerve canal resulted in sufficient pressure release of the constricted nerve and restoration of epicritic sensitivity. Imaging findings and histological and molecular genetic examination revealed monostotic craniofacial fibrous dysplasia. CONCLUSION: Although the data in the literature do not give reason to suppose an accumulation of neoplasms in TXS, a numb chin syndrome should be a reason for detailed diagnostics. Careful diagnosis allows for customized therapy. This is the first report on the coincidence of TXS, fragile X syndrome, and fibrous dysplasia in a single individual.

Central giant cell lesion of the jaws: An updated analysis of 2270 cases reported in the literature.

PURPOSE: To review all available data published on central giant cell lesion (CGCL) of the jaws into a comprehensive analysis of its clinical/radiological features, with emphasis on the predictive factors associated with its recurrence. METHODS: An electronic search was undertaken in 5 databases (February/2018), looking for reporting cases of CGCLs. RESULTS: A total of 365 publications were included, comprising 2270 lesions. CGCLs were more prevalent in women and the mandible. Cortical bone perforation occurred in 50% of the cases. Marginal/segmental resection was more often performed in larger lesions, and drug therapy was more frequent in small lesions. Recurrence was reported in 232 of 1316 cases (17.6%). The recurrence rate of the aggressive lesions (22.8%) after surgical treatment was higher than non-aggressive lesions (7.8%). Four of 5 CGCLs showed partial/total regression with pharmacological treatment. Aggressive lesions showed a worse response to corticosteroids than non-aggressive lesions. For the lesions submitted to surgery as the first treatment, curettage, enucleation, or marginal resection in relation to segmental resection, aggressive lesions, cortical bone perforation, and tooth root resorption were associated with increased recurrence rate. Recurrence related to a combination of surgical/pharmacological treatment could not be evaluated due to the variety of protocols. CONCLUSIONS: Aggressive CGCLs recur more often than the non-aggressive ones. Despite sometimes showing poor response to corticosteroid injection or surgical curettage, a combination of both treatment strategies should be considered in aggressive cases to reduce morbidities associated with radical surgery. The best protocol to manage aggressive and non-aggressive lesions remains to be determined.

Fatal Bleeding in Conjunction with Mandibular Medication-related Osteonecrosis of the Jaw (MRONJ).

Here, we report a case of fatal bleeding in conjunction with mandibular medicationrelated osteonecrosis of the jaw (MRONJ). A 75-year-old Japanese man was referred to our department with osteonecrosis of the jaw due to bisphosphonate (BP) for multiple bone metastases from prostate cancer. Aggressive surgical intervention was ruled out due to a poor prognosis in terms of life expectancy. Death occurred due to hemorrhagic shock resulting from massive oral bleeding caused by necrosis of the mandible. Numerous reports have suggested that jaw necrosis is induced not only by BP, but also RANKL antibody, steroids, and molecularly-targeted agents. This suggests that the number of cases of MRONJ is likely to increase among elderly patients in whom general health is already poor. The American Association of Oral and Maxillofacial Surgery recommends aggressive treatment only in cases of stage 3 disease. Therefore, such a therapeutic strategy may only be available for cases of jaw necrosis in which the general health status of the patient is otherwise good. To prevent a life-threatening outcome in cases of MRONJ, physicians, who are responsible for determining the drug strategy, should cooperate with oral surgeons in determining the best therapeutic strategy.

Mandibular Osteonecrosis Associated With Raloxifene.

Osteonecrosis is a disease with diverse pathophysiology, clinical presentation, and management. It may be associated with some medications used to treat systemic issues with bone metabolism. A few cases of jaw bone osteonecrosis have been associated with raloxifene. In this paper, the authors present a clinical report of a 64-year-old woman who presented with a necrosis foci in the right alveolar ridge of the mandible, associated with continued raloxifene use.

Glandular odontogenic cyst: An updated analysis of 169 cases reported in the literature.

OBJECTIVE: To integrate the available data published on glandular odontogenic cyst (GOC) into a comprehensive analysis of its clinical/radiological and histopathological features. METHODS: An electronic search was undertaken in May/2017. Eligibility criteria included publications having enough clinical/radiological/histological information to confirm the diagnosis. RESULTS: Fifty-eight publications (169 GOCs) were included. The lesion was slightly more prevalent in men than in women. There was a high prevalence in the fifty/sixth decades of life, in the anterior regions, and in mandibles. Lesions were commonly associated with bone expansion (73%) and unilocular radiological appearance (61.5%). GOC was found to be associated with tooth displacement or an unerupted tooth (30.9%), cortical bone perforation (26%), presence of clinical symptoms (24.3%), root resorption (13.9%). Microscopic parameters most commonly were observed in GOCs-in at least 95% of the lesions: presence of hobnail cells, intraepithelial microcysts, epithelial lining with variable thickness. The presence of apocrine snouting was the microscopic parameter less often found (40.4%). CONCLUSION: Although the recurrence rate of GOCs is not as high as previously believed, it is a relevant phenomenon (21.6%). Adjunctive procedures after enucleation should be considered. None of the clinical/radiological and histopathological features evaluated had a statistically significant effect on the recurrence rate.

Conservative treatment of children with chronic diffuse sclerosing osteomyelitis/tendoperiostitis of the mandible.

Chronic diffuse sclerosing osteomyelitis (DSO) of the mandible is a rare disease of unknown aetiology. It has been suggested that overuse of the masticatory muscles, tendoperiostitis (TP), is a contributing factor for DSO. Therefore, we tested this hypothesis by treating consecutive children with conservative therapy. All patients were treated with conservative therapy, comprising occlusal splint therapy, physiotherapy, and/or disease counselling. Pain intensity on a visual analogue scale (VAS) and pain frequency in number of days per 3 months were recorded before the start of treatment, and at 3, 6, and 12 months after treatment initiation. Eleven children (seven girls, four boys, mean age: 11.55 ± 1.97 years) were included in this study. Six patients showed a decrease in pain intensity and pain frequency over time and they continued with conservative therapy. For the remaining five patients, bisphosphonate administration was initiated because of persistent severe pain - one after 3 months of conservative therapy, and the other four after 1 year of conservative therapy. The pain complaints of patients with DSO/TP decreased with conservative therapy, and 55% did not require additional therapy. This suggests that DSO/TP of the mandible is precipitated by muscle overuse.

Mandibular Gorham-Stout disease: A case report and literature review.

RATIONALE: Gorham-Stout disease (GSD) is characterized by aggressive bone resorption, proliferation of vascular or lymphatic vessels, and soft-tissue swelling. Bones that initially appear normal start to resorb, partially or completely. However, the etiology of GSD is unknown. PATIENT CONCERNS: A 29-year-old man with a chief complaint of toothache and mobility in the lower right mandible for the previous 1 year. DIAGNOSES: Gorham-Stout disease (GSD). INTERVENTIONS: The RANK-ligand inhibitor denosumab was suggested to use to inhibit the development of osteoclasts and slow mandibular resorption. In addition, we proposed resection of the remaining mandible and reconstruction via vascularized bone graft, after resorption of the mandible had become stationary. OUTCOMES: Regular follow-ups were advised to this patient to monitor the stability of bone resorption prior to any surgical intervention. LESSONS: We strongly recommend that every attempt should be made for early diagnosis and prompt effective medical and surgical management. The failure to do so results in further complications and poor prognosis.

High-flow paediatric mandibular arteriovenous malformations: case reports and a review of current management.

High-flow vascular malformations in the paediatric population are potentially life-threatening and are challenging to treat. This paper describes the management of three cases of mandibular arteriovenous malformations and reviews the contemporary management options for these serious lesions.

Monofocal Chronic Nonbacterial Osteomyelitis in the Mandible Accompanied With Mucocutaneous Disease.

Chronic nonbacterial osteomyelitis (CNO) is a bone disease with nonbacteria osteitis of unknown etiology. Symptoms, including pain, swelling, fever, etc, always result in misdiagnosis and wrong therapy. This study aims to introduce the clinical features and the therapy of patients with CNO accompanied with mucocutaneous disease. The authors retrospectively reviewed the patients who presented to our hospital from 2000 to 2016, the final sample including 3 patients with CNO accompanied with mucocutaneous disease, who presented repeated severe pain of bone, swelling, tenderness, and fever during attack stage with no evidence of bacterial infections. One patient underwent surgery, and all of them took the therapy of nonsteroidal anti-inflammatory drugs and immunosuppressive agents. There was no significant effect after taken surgery, but good results with drugs. The study suggested us that medical therapy sometimes better than surgery in CNO accompanied with mucocutaneous disease.