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.

Fibular Tip Periostitis: New Radiographic Sign Predictive of Chronic Peroneal Tendon Subluxation-Dislocation in Pes Planovalgus.

BACKGROUND. Tearing of the superior peroneal retinaculum is a known cause of peroneal tendon subluxation-dislocation. However, with the exception of cortical avulsions at the fibular attachment, superior peroneal retinaculum injury and subsequent peroneal tendon subluxation-dislocation are typically radiographically occult. OBJECTIVE. The purpose of this study was to evaluate the previously undescribed association between radiographic fibular tip periostitis and MRI evidence of peroneal tendon subluxation-dislocation in patients with hindfoot valgus. METHODS. Thirty-five patients with radiographic fibular tip periostitis and 35 ageand sex-matched individuals without periostitis were selected from among 220 consecutively registered patients with hindfoot valgus who had undergone both ankle radiography and MRI. Studies were retrospectively assessed by two musculoskeletal radiologists in consensus and by two additional blinded radiologists independently for the presence of peroneal tendon subluxation-dislocation, presence of subfibular impingement, and hindfoot valgus angle measurements. Interobserver agreement and accuracy, sensitivity, and specificity in the detection of fibular periostitis, peroneal tendon subluxation-dislocation, and subfibular impingement were calculated, and Fischer exact, Mann-Whitney, kappa coefficient, and intraclass correlation tests were performed. RESULTS. Both the consensus and the two independent interpretations showed that the frequency of peroneal tendon subluxation-dislocation was significantly greater in the group with periostitis (62.9%, 65.7%, and 85.7%) than in the group without periostitis (5.7%, 0%, and 14.3%) (p < .001). The finding of periostitis was specific and highly sensitive for predicting peroneal tendon subluxation-dislocation. The frequency of subfibular impingement was also statistically higher in the group with periostitis than in the group without it (p < .001). The hindfoot valgus angle was statistically larger in the group with periostitis than in the control group (p = .01-.002) and among patients with versus those without peroneal tendon subluxation-dislocation (p = .002 to p < .001). The blinded readers had substantial or almost perfect agreement on all imaging interpretations (concordance rate, 82.9-95.7%; κ = 0.66-0.91). CONCLUSION. The radiographic finding of fibular tip periostitis in patients with hindfoot valgus can be a predictor of peroneal tendon subluxation-dislocation and may also suggest advanced hindfoot valgus and subfibular impingement. These radiographic associations should be recognized by the radiologist, and MRI may be recommended as clinically indicated. CLINICAL IMPACT. Chronic undiagnosed peroneal tendon subluxation-dislocation can be a persistent cause of lateral ankle pain, leading to further degeneration and the possibility of complete peroneal tendon tears. Distal fibular periostitis in patients with hindfoot valgus can be a reliable radiographic indicator of this entity and may suggest the presence of subfibular impingement.

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.

Chronic osteomyelitis with proliferative periostitis of the mandibular body: report of a case and review of the literature.

Chronic osteomyelitis with proliferative periostitis is a rare form of osteomyelitis that is characterised by new bone formation with periosteal reaction. It is also traditionally known as Garre's osteomyelitis. The common sources of infection of the jaw include dental caries associated with periapical periodontitis, periodontitis, fractures and nonodontogenic infections. Chronic osteomyelitis with proliferative periostitis mainly presents in younger patients. Here, we present a case of a 12-year-old patient with chronic osteomyelitis with proliferative periostitis with no definitive infection source such as pericoronitis, caries and periodontitis. Therapeutic measures involved surgical debridement and antibiotics. Disease remission and a normal facial symmetrical morphology were observed at the six-month follow-up.

Bone Scintigraphy in Poststreptococcal Periostitis With Dysproteinemia.

A 39-year-old man presented with severe bone pain in the tibiae and forearms in the wake of a poststreptococcal sepsis complicated with pneumonia and erysipelas 4 months earlier. Bone scintigraphy was indicative of periostitis of the tibia, ulna, and radius bilaterally, and in combination with the increased inflammatory parameters and dysproteinemia, the diagnosis of Goldbloom syndrome was made. Goldbloom syndrome is an idiopathic periosteal hyperostosis associated with dysproteinemia and elevated inflammatory parameters. Although it has only been described in children/adolescents, this case illustrates that, in the specific clinical and biochemical setting, it should also be considered in adults.

Leg Pain From Periostitis as the First Clinical Manifestation of Large-Vessel Vasculitis: A Case Report.

We report a case of a 47-year-old woman with isolated periostitis of the lower leg as a first manifestation of large-vessel vasculitis. The diagnosis was first suspected when a second magnetic resonance imaging of the lower leg approximately 4 months after onset of shin pain showed edema near intramuscular vessels. Isolated periostitis is a very rare skeletal manifestation of systemic vasculitis and easily could be misdiagnosed. This case demonstrates that maintaining a broad differential diagnosis for a common presenting complaint such as localized leg pain is imperative. LEVEL OF EVIDENCE: V.

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.

Sclerosis in bisphosphonate-related osteonecrosis of the jaws and its correlation with the clinical stages: study of 43 cases.

We analysed the degree of sclerosis in the different stages of bisphosphonate-related osteonecrosis of the jaws (BRONJ) and studied the relation between the grade of sclerosis, the clinical symptoms, and the depth of lucency. We compared 43 patients with mandibular BRONJ with a control group of 40 cases with no bony lesions. The presence of sclerotic bone, cortical irregularities, radiolucency, fragmentation or sequestration, periostitis, and narrowing of the mandibular canal were studied from computed tomographic (CT) scans using the program ImageJ 1.47v (National Institute of Health, Bethesda, USA) to measure the radiolucency, width of the cortices, and degree of sclerosis. Patients with BRONJ had more severe sclerosis than controls (p<0.01). There was also a significant difference among the different stages of BRONJ, with the highest values found in stage III (p=0.02). The degree of sclerosis differed according to sex, type of bisphosphonate, and the clinical characteristics such as pain, or suppuration, but not significantly so (p>0.05). We conclude that the degree of sclerosis increases with the clinical stage of BRONJ, and is correlated with the depth of lucency.

Multifocal periostitis as a complication of chronic use of voriconazole in a lung transplant recipient.

Fungal infections are common in solid organ transplantation. An increasing number of transplant recipients receive antifungal therapy for prolonged duration owing to invasive fungal infections. Herein, we describe a diagnosis of periostitis as a complication of chronic use of voriconazole in a lung transplant recipient. The patient was diagnosed with probable pulmonary aspergillosis and was treated with oral voriconazole for a total of 9 months. Evidence of multifocal periostitis was observed in the axial and appendicular skeleton. Early recognition of this phenomenon is important to prevent unnecessary tests and procedures. Prompt discontinuation of voriconazole should result in improvement of symptoms.

Florid reactive periostitis ossificans of long bones and digits associated with reaction in a patient with leprosy.

Florid reactive periostitis ossificans is a rare bone lesion usually occurring in the small, tubular bones of the hands and feet. This entity is a benign and aggressive periosteal reaction associated with soft tissue swelling that appears similar to a bone lesion that radiographically and clinically mimics an infectious or neoplastic process. Typically the lesions occurs in an adolescent or young adult and presents as a small area of painful swelling and erythema over the affected bone. The cause of florid reactive periostitis ossificans is not exactly known though many authors have postulated varied etiopathogenesis for the same condition. In this report, is a very rare and unusual example of this entity that has been observed in association with erythema nodosum leprosum (ENL) a type 2 lepra reaction in a Leprosy patient.

The association between periodontal disease and periosteal lesions in the St. Mary Graces cemetery, London, England A.D. 1350-1538.

Numerous studies have demonstrated significant associations between periodontal disease and many other diseases in living populations, and some studies have shown that individuals with periodontal disease are at elevated risks of mortality. Recent analysis of a medieval skeletal sample from London has also shown that periodontal disease was associated with increased risks of mortality in the past. This study examines whether periodontal disease is associated with periosteal lesions in a skeletal sample from the urban St. Mary Graces cemetery (n = 265) from medieval London. The results reveal a significant association between periodontal disease and periosteal lesions in the St. Mary Graces sample (i.e., individuals with periodontal disease were also likely to have periosteal lesions), and the association between the two is independent of age. The association between the two pathological conditions might reflect underlying reduced immune competence and thus heightened susceptibility to pathogens that cause periodontal disease or periosteal lesions, exposure to an environmental factor, or underlying heightened inflammatory responses.

Florid reactive periostitis of the forearm bones in a child.

Florid reactive periostitis is a pronounced periosteal reaction, usually affecting the hands and feet, for which there is no obvious cause. It is rare in children and in long bones. We report an unusual case of florid reactive periostitis in a ten-year-old girl that involved both bones of the forearm. The lesion resolved over a period of one year, leaving a residual exostosis. She developed a physeal bar in the distal ulna in the region of the lesion at one-year follow-up. This was thought to be a complication of the biopsy procedure and was treated by resection and proximal ulnar lengthening.

Syphilitic periostitis of the skull and ribs in a HIV positive patient.

We report the case of a HIV and syphilis co-infected patient who presented with headache and rash and was found to have syphilitic periostitis. Our case illustrates a rare manifestation of early syphilis and presents the diagnostic dilemmas that can arise in HIV and syphilis co-infected patients.

Bilateral stress fracture of the fibulae and periostitis of the tibiae.

OBJECTIVE: This study describes a unique case of bilateral stress fractures of the fibulae and provides a literature review. CLINICAL PRESENTATION AND INTERVENTION: A 16-year-old female badminton player presented with pain around the bilateral distal lateral legs. She had mild bilateral varus deformity at the knee joint, and the bilateral ankles showed valgus deformity in standing posture. Radiographs and computed tomography showed periosteal reactions on the bilateral distal fibulae. Technetium-99m bone scintigraphy demonstrated increased uptake in the bilateral distal fibulae and the bilateral middle third of the tibiae. A diagnosis of bilateral distal fibular stress fractures was made. She was advised to stop playing badminton until the symptoms disappeared. CONCLUSION: Varus deformity of the knee and valgus deformity of the ankle may have influenced the mechanism underlying bilateral symmetric stress fractures.

Hypertrophic osteoarthropathy manifested with isolated calcaneal periostitis in bone scintigraphy.

Hypertrophic osteoarthropathy (HOA) is an incompletely understood syndrome characterized by digital clubbing and periosteal proliferation of long bones and it is commonly associated with primary lung tumors. Bone scintigraphy is a sensitive method in detecting HOA and characteristic findings have been reported. We present the case of a man with newly diagnosed non-small cell lung cancer, unremarkable clinical examination and blood tests and no digital clubbing. During disease staging, however, bone scintigraphy showed intense calcaneal cortical proliferation bilaterally without involvement of other parts of the skeleton. Cortical reaction of both calcanei resolved significantly after chemotherapy. This case indicates that HOA may manifest with isolated calcaneal periostitis bilaterally, which is a new addition to the literature.