Ehlers-Danlos syndrome (EDS) and Marfan syndrome (MFS) are characterised by hypermobility of joints and cardiovascular morbidity, and typical orofacial signs and symptoms are associated with both. Basic knowledge of these should prevent late-stage diagnosis and enable adequate management. This case series comprises all EDS and MFS patients who consulted the Department of Oral and Maxillofacial Surgery at University Hospitals Leuven between 2005 and 2017. Thirty patients had EDS or MFS, and in seven the diagnosis was made based on temporomandibular dysfunction or craniofacial dysmorphism. Non-facial symptoms led to diagnosis in the remaining patients. Twenty-five interventions were followed by two infections and one haemorrhage. One patient developed trismus. No major complications were reported. The complications we observed did not seem to differ from those often seen in patients who did not have EDS or MFS, possibly as a result of our strictly applied precautions and postoperative instructions. However, our study of relevant publications shows that patients with EDS and MFS are a vulnerable group.
There is currently no widespread strategy for treating medication-related osteonecrosis of the jaw (MRONJ), so our aim was to evaluate retrospectively the outcome of a minimally invasive treatment protocol for patients with both MRONJ and cancer. We designed a retrospective cohort study of patients with cancer who had been diagnosed with MRONJ after treatment with denosumab or bisphosphonates given intravenously. Primary outcome measures were improvement in the clinical stage of MRONJ and the time course to its resolution. Secondary outcome measures included the incidence of risk factors and patterns of treatment. Seventy-nine patients with 109 lesions were enrolled, and their characteristics, presentation of the lesions, complications, and relations to previous oral interventions were recorded. Treatment depended on the stage of disease, and included conservative medical, and minimally-invasive surgical, procedures. There was complete healing and resolution of disease in 38/57 stage 1 lesions, 30/47 stage 2 lesions, and 3/5 stage 3 lesions. The symptoms improved in 16/47 stage 2 lesions, and 2/5 stage 3 lesions. Fifteen of the stage 1 lesions, and one of the stage 2 lesions, failed to respond. Despite the possibility of an aggressive approach to the treatment of MRONJ, conservative treatment remains the first line of defence as regression is obvious, with evidence of no evolution to a higher stage. In our experience surgical intervention is recommended in persistent stage 3 MRONJ that has failed to respond to conservative treatment.
HighlightsWe report a case from our academic center, where mandibular metastasis presented with pain and hypoesthesia in the right lower face.Metastasis to the oral cavity can be located in both the soft and bony tissues and comprise only 1% of all oral malignancies; however, it is clinically significant because it indicates widespread metastatic disease and an unfavorable prognosis.Most oral bony metastasis are located in the mandible, especially in the molar region.In most cases with oral metastasis, palliative treatment is indicated. Early detection of oral lesions could improve treatment outcome and survival.Despite treatment, the average survival after diagnosis of mandibular metastasis is 6–7 months. Early detection of oral lesions could improve treatment outcome and survival. A full diagnostic work-up is therefore of great importance.
We present the case of a 32-year-old male patient complaining of recurrent mandibular pain for 3.5 years. Panoramic radiography indicated increased cortical density of the mandibular lower border. Scintigraphy and single-photon emission computed tomography revealed metabolic hyperactivity in that region without pathologic lymph nodes. A bone biopsy specimen of the mandibular lower border did not have inflammation or cytologic atypia. Endocrinologic investigation confirmed secondary hyperparathyroidism as a result of hypovitaminosis D. Several weeks after starting therapy with oral vitamin D supplements, the symptoms completely disappeared. Increased cortical density is a rare manifestation of secondary hyperparathyroidism, which normally causes the lamina dura to vanish and produces a ground-glass appearance as a result of blurring of the trabecular bone pattern. Because focal hyperostosis can have multiple benign or malignant causes, radiologic examination of the jaw bones is indispensable for evaluating orofacial pain. Increased cortical density may be caused by metabolic diseases, requiring further investigations, including biopsy and blood analysis.
The need for secondary orthognathic surgery (OS) after a high condylectomy (HC) in patients with active unilateral condylar hyperplasia was assessed in 25 patients, reviewing patient characteristics and treatment planning. At 6-12 months after HC, 13 patients (52%) required secondary OS. The amount of mandibular dental midline shift before the HC (P = 0.037), and a dental crossbite that was present before the HC (P = 0.017) were significantly associated with the need for secondary OS. Overall, the mandibular dental midline coincided with the facial midline in eight patients (32%) at 2 weeks after HC. In 16% of the patients, no additional OS was needed despite this being the initial treatment plan. Additionally, the initially planned type of secondary OS was modified in six other patients. Early HC in skeletally immature patients provided very good results, both aesthetic and functional, with only two of them needing supplementary OS at 6-12 months after HC. The HC remains a valuable treatment in patients with active unilateral condylar hyperplasia, as it can eliminate the need for secondary OS and is very well tolerated by most patients.
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