Alessandro Agrillo

Surgical Treatment of TMJ Ankylosis : Our Experience (60 Cases)

Limitation of mouth opening can be caused by bony or fibrous ankylosis of the temporomandibular joint as sequela to trauma, infection, autoimmune disease, or failed surgery.Various procedures have been reported for treatment of temporomandibular joint (TMJ) ankylosis; this article aims to describe the diagnostic protocol and the surgical procedures adopted at the department of Maxillo-Facial Surgery of Rome University “La Sapienza”.Between 1980 and 2000, 123 patients affected by TMJ ankylosis came under our observation; 60 of them (25 females and 35 males of 30 years average age) underwent surgery; bilateral TMJ ankylosis was observed in 21 cases, right-sided in 20 cases, left-sided in 19 cases. In 12 cases coronoid processes were involved. Etiopathogenesis was traumatic in 48 cases, septic in 5 cases, auto-immune (RA and seronegative spondyloarthropathies) in 5 cases; after block removing, arthroplasty was performed with pedunculated flap of temporal muscle (10 cases), Silastic material (11 cases), or lyophilized dura mater (2 cases). Simple condylar shaving was used in the remaining 36 cases.All patients under treatment showed a distinctive improvement both in articular functionality and symptoms; secondary surgery was necessary in seven cases due to the onset of articular complications from previous surgery. Silastic removal was necessary in five cases due to its inducement of foreign body granuloma. Follow-up was performed at 12, 24, and 48 months and 5 years postoperatively.In our opinion the gold standard surgery of TMJ ankylosis today is represented by shaving of articular surfaces and subsequent arthroplasty with or without temporal muscle myofascial flap interposition, whereas the use of Silastic as alloplastic material could be associated to an increased persistence of the local symptoms and a higher risk of foreign body granuloma and it may favor ankylosis relapse and hinder rehabilitation.

Learning from experience. Proposal of a refined definition and staging system for bisphosphonate-related osteonecrosis of the jaw (BRONJ)

Dear Editor, It is the authors’ belief that the internationally accepted definition of bisphosphonate-related osteonecrosis of the jaws (BRONJ) (Ruggiero et al, 2009) has several limitations that prevent clinicians from being confident with the diagnosis of the disease. Following recognition of the non-exposed BRONJ clinical variant (Lazarovici et al, 2009), we all became aware that the presence of ‘exposed necrotic bone in the oral cavity’, as outlined in the American Association of Oral and Maxillofacial Surgery (AAOMS) case definition, is just one of the possible clinical manifestations of BRONJ and is not found in all BRONJ patients. As ‘bone exposure’ is certainly not the initial sign of BRONJ in most patients and a minimum of 6–8 weeks’ persistence is required to confirm the clinical suspicion, the final diagnosis is usually delayed for several weeks or months. Therefore, to date, it has been almost impossible to study the early phases of BRONJ. This delayed diagnosis can also explain, at least in part, why the disease is often refractory to the medical and surgical treatments commonly used. We believe that clinicians will benefit from a definition of BRONJ that contains only robust information, without considering any definite clinical picture or a binding time-frame (i.e. 6–8 weeks). As qualified members of the Expert Panel of the Italian Society for Maxillofacial Surgery (SICMF) and the Italian Society of Oral Pathology and Medicine (SIPMO) on Bisphosphonate-Related Osteonecrosis of the Jaws, we are submitting to the attention of the scientific community the following definition of BRONJ: ‘bisphosphonate related osteonecrosis of the jaw (BRONJ) is an adverse drug reaction described as the progressive destruction and death of bone that affects the mandible or maxilla of patients exposed to the treatment with nitrogen-containing bisphosphonates, in the absence of a previous radiation treatment’. Along with this new definition of BRONJ, we set up and propose a diagnostic work-up to be used to reach the final diagnosis. Although exposed necrotic bone in the oral cavity still remains the best indicator of BRONJ (Ruggiero et al, 2009), other non-specific signs and symptoms (Table 1) should raise the suspicion of BRONJ, even in a patient with a well-recognized dental or periodontal disease (Fedele et al, 2010). In short, BRONJ should be always investigated in a patient taking nitrogen-containing bisphosphonates (NBP), when one or more clinical signs are present. Because BRONJ is primarily a disease that affects the jawbone, we strongly believe that radiological examination is an important step of the diagnosis. However, as the radiological findings may be characteristic not only of BRONJ (Khan et al, 2008), these findings should always match the clinical picture, in order to progress from the clinical suspicion to the final diagnosis. This is of outmost importance as bone biopsies for histology are still not routinely advised for the risk of worsening the disease process. A schematic approach to the work-up for a diagnosis of BRONJ is proposed (Scheme 1), where computed tomography (CT) currently represents the most useful diagnostic tool because of its widespread use and accessibility for patients (Bianchi et al, 2007). Structural alteration of trabecular bone is a consistent finding of CT scans performed in patients with BRONJ (Table 2) (Arce et al, 2009). CT scans can clearly depict the degree of osteosclerosis of the affected site, visible as the loss of contrast definition between the endosteal cortex and the subjacent medullary bone (i.e. trabecular thickening and/or regional or diffuse osteosclerosis), with respect to the uninvolved bone tissue (Hutchinson et al, 2010). Osteosclerosis seems to characterize the early stages of disease and also precedes the occurrence of frank bone exposure in the oral cavity (Saia et al, 2010), and thus it should be searched for to provide an early diagnosis. Table 1 Non-specific clinical signs associated with bisphosphonate-related osteonecrosis of the jaws (BRONJ) Table 2 Non-specific computed tomography (CT) findings associated with bisphosphonate-related osteonecrosis of the jaws (BRONJ) Scheme 1 Diagnostic work-up for BRONJ. VAS, visual analogic scale Even though magnetic resonance imaging (MRI) provides more detailed information than CT on focal bone marrow alterations (Bisdas et al, 2008), the former could provide non-diagnostic results owing to magnetic artifacts caused by the presence of dental casting alloys (Shafiei et al, 2003) as well as to motion artifacts caused by the prolonged time necessary to scan the head and neck region (Lenz et al, 2000). The majority of BRONJ cases could be diagnosed and staged correctly by the combined use of clinical methods and CT, the latter method currently being more cost-effective and easily accessible than MRI. On the other hand, we believe that MRI should be used to evaluate cases difficult to identify with CT and to establish the real extent of jawbone and soft-tissue involvement in BRONJ patients who are candidates for surgical resection. The staging system of a bone disease that relies on the use of radiologic imaging for its diagnosis should also be based on the common radiologic features of the disease. The current AAOMS staging system (Ruggiero et al, 2009; Migliorati et al, 2011), which assigns patients to different stages of disease on the basis almost exclusively of clinical criteria, fails, in our opinion, to consider this important aspect. In this regard, we set up a combined clinical and radiological staging system with the aim of pooling BRONJ patients in different groups also based on the radiological extent of the disease. The staging system we propose (Table 3) differs from the 2009 AAOMS classification as follows: Table 3 Clinical and radiological staging system of bisphosphonate-related osteonecrosis of the jaws (BRONJ) The absence of Stage 0, so that BRONJ patients with exposed and non-exposed necrotic bone simply represent distinct clinical pictures within the same disease stage. The description of three stages (1–3) based on clinical and CT findings, ‘where Stage 1 includes patients with focal (alveolar bone) osteosclerosis, Stage 2 includes patients with diffuse (alveolar and basal bone) osteosclerosis and Stage 3 includes patients with clinical and radiological signs of advanced and complicated disease. Pain and purulent discharge are no longer used to distinguish between different disease stages. As these symptoms define only asymptomatic (a) and symptomatic (b) forms of BRONJ in patients within the same stage, exclusion of pain and purulent discharge as criteria would limit the ‘ping-pong’ effect (the migration of patients from Stage 1 to Stage 2, and vice versa) that we all experienced when using the AAOMS classification, as a result of the repeated use of antibiotic therapies to treat recurrent bone infections and associated pain. The presence of clinically detectable sequestra is no longer regarded as a sign of complicated disease, as it is for AAOMS Stage 3. In fact, it is a common experience that the spontaneous expulsion or surgical removal of bony sequestra often leads to dramatic, albeit somewhat temporary, clinical improvement, with mucosal epithelialization of the affected site (Ferlito et al, 2011). The definition, work-up and staging system proposed here might, as a whole, provide several practical benefits, in the order: anticipated BRONJ diagnosis and access of patients to therapies; a reduced need for extensive surgical resections with associated long and debilitating postoperative hospitalization; and increased overall efficacy of treatments and patients’ curability. The disadvantage of the additional cost of bone imaging (CT) for the diagnosis would be offset against the overall reduction in treatment costs. When a new disease is found, little is known of its medical features and behavior; this makes it necessary to avoid general definitions that may include potentially healthy patients (resulting in a higher false-positive risk). This also occurred at the start, when the actual BRONJ case definition was adopted. At present, with the growing bulk of research on BRONJ, we believe that it is necessary to enter a new phase where clinicians try to establish the initial stages of the disease process, in order to make an earlier diagnosis and improve treatment effectiveness. Although adjustments and updating will be necessary in the future, the definition, work-up and staging system proposed here could achieve this purpose.

Staging of osteonecrosis of the jaw requires computed tomography for accurate definition of the extent of bony disease

Management of osteonecrosis of the jaw associated with antiresorptive agents is challenging, and outcomes are unpredictable. The severity of disease is the main guide to management, and can help to predict prognosis. Most available staging systems for osteonecrosis, including the widely-used American Association of Oral and Maxillofacial Surgeons (AAOMS) system, classify severity on the basis of clinical and radiographic findings. However, clinical inspection and radiography are limited in their ability to identify the extent of necrotic bone disease compared with computed tomography (CT). We have organised a large multicentre retrospective study (known as MISSION) to investigate the agreement between the AAOMS staging system and the extent of osteonecrosis of the jaw (focal compared with diffuse involvement of bone) as detected on CT. We studied 799 patients with detailed clinical phenotyping who had CT images taken. Features of diffuse bone disease were identified on CT within all AAOMS stages (20%, 8%, 48%, and 24% of patients in stages 0, 1, 2, and 3, respectively). Of the patients classified as stage 0, 110/192 (57%) had diffuse disease on CT, and about 1 in 3 with CT evidence of diffuse bone disease was misclassified by the AAOMS system as having stages 0 and 1 osteonecrosis. In addition, more than a third of patients with AAOMS stage 2 (142/405, 35%) had focal bone disease on CT. We conclude that the AAOMS staging system does not correctly identify the extent of bony disease in patients with osteonecrosis of the jaw.

Up to a quarter of patients with osteonecrosis of the jaw associated with antiresorptive agents remain undiagnosed.

Recent data suggest that the traditional definition of bisphosphonate-associated osteonecrosis of the jaw (ONJ) may exclude patients who present with the non-exposed variant of the condition. To test the hypothesis that a proportion of patients with ONJ remain undiagnosed because their symptoms do not conform to the traditional case definition, we did a secondary analysis of data from MISSION (Multicentre study on phenotype, definition and classification of osteonecrosis of the jaws associated with bisphosphonates), a cross-sectional study of a large population of patients with bisphosphonate-associated ONJ who were recruited in 13 European centres. Patients with exposed and non-exposed ONJ were included. The main aim was to quantify the proportion of those who, according to the traditional case definition, would not be diagnosed with ONJ because they had no exposed necrotic bone. Data analysis included descriptive statistics, median regression, and Fishers exact test. A total of 886 consecutive patients were recruited and 799 were studied after data cleaning (removal or correction of inaccurate data). Of these, 607 (76%) were diagnosed according to the traditional definition. Diagnosis in the remaining 192 (24%) could not be adjudicated, as they had several abnormal features relating to the jaws but no visible necrotic bone. The groups were similar for most of the phenotypic variables tested. To our knowledge this is the first study in a large population that shows that use of the traditional definition may result in one quarter of patients remaining undiagnosed. Those not considered to have ONJ had the non-exposed variant. These findings show the importance of adding this description to the traditional case definition.

Surgical planning in reconstruction of mandibular defect with fibula free flap: 15 Patients

The authors present a descriptive work on the reconstruction of large oromandibular defects with revascularized fibula free flaps in a group of patients treated by surgical resection of bony and soft tissues of the mandible. The principal objective is preoperative planning for the best morphological, functional, and aesthetic result. The authors report 15 reconstructions of wide oromandibular defects by revascularized fibula free flaps. The average patient age was 42 years. The mean follow-up period was 18 months. Correct surgical planning reduces surgical timing, minimizes the chance of failure during the reconstruction, and contemporaneously increases treatment efficacy and the best functional and aesthetic results.

LeFort III advancement with and without osteogenesis distraction.

The LeFort III osteotomy is the surgical treatment performed in patients with mid-facial retrusions in craniofacial dysostoses such as Crouzon, Apert, Pfeiffer syndromes, etc. The first authors to report the accomplishment of this osteotomy were Gillies and Harrison8 in 1951, this technique was then resumed and improved by Tessier in 19671, who made five different variants mainly regarding the typology of the osteotomy concerning the lateral wall of the orbit. Recently, distraction techniques have been applied to the upper mid-face which foresees a gradual advancement of the mid-face through the osteogenetic distraction procedure. The purpose of this retrospective clinical outcome study is to evaluate and compare the two different surgical techniques through the experience acquired during the treatment of 15 cases of craniofacial dysostoses from 1990-2005, and through international literature. The standard surgery technique was performed in 5 of these patients, whereas the osteogenetic distraction technique was performed in the remaining 10. All patients were studied preoperatively through the acquisition of photographic images, cephalometric analysis of the skull, and the study on plaster models of the occlusion of the dental arches. The instrumental exams required before surgery are the following: telecranium X-rays in two projections, orthopanoramic X-rays, CT. The results of this study indicate that the osteodistraction technique represents the choice treatment in severe retrusions of the mid-face while the traditional surgical technique remains indicated in adult patients that are in need of moderate advancements.

Ozone therapy in extractive surgery on patients treated with bisphosphonates.

It is certain that oral extractive surgery is a remarkable trigger to avascular osteonecrosis of the jaw in patients treated with pyrophosphate analogous. This acquisition limits the use of endo-oral surgery in those patients, even when they have already developed the lesions. In this study, we present the results obtained in a group of 15 patients deriving from a 33-patient cluster with osteonecrosis of the jaw in treatment at our department with a new protocol based on ozone therapy. The object of this article is to demonstrate how dental extraction becomes possible in a patient with avascular bisphosphonate-related jaw osteonecrosis or in those who simply received pyrophosphate analogous when proper treatment with ozone therapy has been done.

Hypophosphaturic mesenchymal tumor of the ethmoid associated with oncogenic osteomalacia.

Oncogenic osteomalacia is an uncommon syndrome characterized by bone pain, proximal muscle weakness, hypophosphatemia, hyperphosphaturia, and a low plasma concentration of 1,25-dihydroxy-vitamin D. The disease affects both sexes at around 40 years of age, although it can sometimes affect children and adolescents. Generally, the syndrome is associated with a tumor, usually benign, of mesenchymal origin and is resolved after removal of the tumor; this syndrome can sometimes be associated with malignant tumors. These tumors seem to be histologically heterogeneous and are generally localized in soft tissues and bone. In this article, a case of oncogenic osteomalacia associated with a hypophosphaturic mesenchymal tumor of the ethmoid is reported in a 24-year-old man. After surgical and radical removal of the tumor, the patient noted a decrease in the clinical symptoms and signs.

Complex-type penetrating injuries of craniomaxillofacial region.

Craniofacial traumas are one of the most common clinical events of the 21st century. The possibility of associated injuries of the head and neck may also determine functional and cosmetic problems in these patients. The most frequent pathologic conditions observed are contusions, lacerations, abrasions, avulsions, and the inclusion of foreign bodies. In particular, penetrating injuries represent a rare but complex variety of craniofacial trauma. Generally, the penetrating material is stiff enough to cross through different anatomic structures during a particularly violent collision caused by a road or work accident or during an attack. The therapeutic strategy adopted for this type of patient depends mainly on diagnostic procedures such as skull radiograms in different projections, computerized tomography, magnetic resonance imaging, and, occasionally, echotomography. However, on arrival at the emergency department, the clinical conditions of the patient will determine the type of investigation to be carried out. Last, to prevent any postoperative infections, wide spectrum antibiotic therapy is advisable. Ideally, imaging should be repeated postoperatively to confirm resolution of the initial clinical condition. In this article, the authors describe three unusual clinical cases of patients with penetrating injuries of the head and face together with the protocol adopted for treatment of such complex craniofacial injuries. The three cases described demonstrate that, despite the initial appearance of penetrating wounds, a correct diagnostic assessment followed by a suitable therapeutic protocol can reduce cosmetic and functional defects to a minimum.

Evolution of bisphosphonate-related osteonecrosis of the jaw in patients with multiple myeloma and Waldenstrom's macroglobulinemia: a retrospective multicentric study

Bisphosphonates (BPs) are used intravenously to treat cancer-related conditions for the prevention of pathological fractures. Osteonecrosis of the jaw (BRONJ) is a rare complication reported in 4–15% of patients. We studied, retrospectively, 55 patients with multiple myeloma or Waldenstroms macroglobulinemia followed up from different haematological departments who developed BRONJ. All patients were treated with BPs for bone lesions and/or fractures. The most common trigger for BRONJ was dental alveolar surgery. After a median observation of 26 months, no death caused by BRONJ complication was reported. In all, 51 patients were treated with antibiotic therapy, and in 6 patients, this was performed in association with surgical debridement of necrotic bone, in 16 with hyperbaric O2 therapy/ozonotherapy and curettage and in 12 with sequestrectomy and O2/hyperbaric therapy. Complete response was observed in 20 cases, partial response in 21, unchanged in 9 and worsening in 3. The association of surgical treatment with antibiotic therapy seems to be more effective in eradicating the necrotic bone than antibiotic treatment alone. O2 hyperbaric/ozonotherapy is a very effective treatment. The cumulative dosage of BPs is important for the evolution of BRONJ. Because the most common trigger for BRONJ was dental extractions, all patients, before BP treatment, must achieve an optimal periodontal health.