A successful implant-supported fixed prosthesis in a patient with osteopetrosis: A clinical report.

Osteopetrosis (marble bone disease) is a family of rare genetic disorders characterized by impaired osteoclast function leading to hyperdense, hypovascular, brittle bone. Typical imaging shows increased bone mass and thickened cortical and trabecular bone. Bones are more prone to fracture and osteomyelitis may develop. When considering dental implant placement in a patient with osteopetrosis, the potential for bony fracture and/or osteomyelitis should be considered along with the decreased likelihood of successful osseointegration because of hypovascularity. This clinical report describes an unusual imaging presentation and successful osseointegration of multiple dental implants supporting an implant-supported fixed mandibular prosthesis with long-term survival.

Application of VSD technique in adults with chronic osteomyelitis of the extremities combined with soft tissue defects.

To investigate the clinical application of vacuum sealing drainage (VSD) in chronic osteomyelitis of the extremities combined with soft tissue defects in adults. This study retrospectively included 32 adult patients with clearly diagnosed chronic osteomyelitis of the extremities combined with local soft tissue defects, and the trauma was covered by VSD after debridement, osteotomy, and vancomycin-laden bone cement filling of the occupancy, and the trauma was covered by selecting a suitable flap transfer repair according to the site and extent of the soft tissue defect after the trauma condition was suitable, and the secondary trauma was taken from the abdominal full-thickness skin free skin slice graft, according to whether the skin graft area was performed. The skin flap hematoma and infection rate, as well as the skin flap survival rate and implant fixation time were compared and analysed between the two groups. The primary outcome is the implant fixation time, and the secondary outcome is the skin fragment survival rate. In 32 patients, VSD was performed on the bone cement surface to cover the trauma, and 33.2 to 39.8 kPa continuous vacuum sealing drainage was set. The average VSD time duration before soft tissue coverage was 47.87 ± 23.14 days, and the average number of VSD use was 7.18 ± 3.23. The use of VSD before soft tissue coverage did not cause complications such as negative pressure could not be maintained, vacuum sealing drainage was not smooth, skin blistering, trauma. Among the 32 patients, 12 cases of soft tissue coverage were followed by trauma free skin grafting with packing + VSD, and 20 cases were fixed with packing alone, and the duration of continuous packing and fixation of free skin pieces in the VSD group was significantly less than that in the control group (P = .006). The survival rate was significantly higher than that of the control group (P = .019). VSD in adult patients with chronic osteomyelitis of the extremities combined with soft tissue defects can effectively improve the trauma condition, provide the possibility of second-stage soft tissue coverage, and significantly shorten the preparation time for soft tissue coverage. In addition, when soft tissue coverage trauma is performed, VSD combined with skin graft packing technique can significantly improve the survival rate of skin pieces, shorten the time of skin graft fixation.

Successful treatment of SAPHO syndrome (chronic nonbacterial osteomyelitis and acne) with anakinra and denosumab. Case report and review of therapy.

SAPHO is an acronym derived from capital letters of Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO). SAPHO syndrome is an umbrella term covering a constellation of bone lesions and skin manifestations. A 40-year-old male complained about his jaw and back pain, swelling of multiple joints and weight loss accompanied by physical deterioration and acne type skin lesions. Laboratory tests revealed abnormal elevation of inflammatory markers. Imaging studies illustrated multiple osteolytic bone lesions and paraosseal infiltrates. According to the set of criteria diagnosis of SAPHO syndrome was stated. The patient was treated with glucocorticoids and non-steroidal anti-inflammatory drugs (NSAIDs), but only high dose dexamethasone and prednisone were effective. Daily subcutaneous administration of anakinra at the dose of 100 mg was initiated due to limited response to more classical therapies. Because of planned mandibular osteosynthesis initiation of denosumab was preferred before bisphosphonates. Therapeutic response was confirmed by FDG-PET/MR after 5 months of anakinra and denosumab therapy, showing decreased accumulation of FDG in periosteal and paraosseal infiltrates. Inflammatory markers significantly decreased, bone pain deferred but skin manifestation receded only partially. Therefore the response was evaluated as partial remission.

Femoral osteomyelitis caused by oral anaerobic bacteria with mixed bacteremia of Campylobacter rectus and Parvimonas micra in a chronic periodontitis patient: a case report.

BACKGROUND: Campylobacter rectus is a gram-negative rod, and Parvimonas micra is a gram-positive coccus, both of which are oral anaerobes that cause chronic periodontitis. Chronic periodontitis can cause bacteremia and systemic diseases, including osteomyelitis. Hematogenous osteomyelitis caused by anaerobic bacteria is uncommon, and to date, there have been no reports of mixed bacteremia with C. rectus and P. micra. Here, we report the first case of osteomyelitis of the femur caused by anaerobic bacteria with mixed bacteremia of C. rectus and P. micra caused by chronic periodontitis. CASE PRESENTATION: A 75-year-old man with chronic periodontitis, hyperuricemia, and benign prostatic hyperplasia was admitted to the hospital with a fracture of the left femur. The patient had left thigh pain for 4 weeks prior to admission. Left femoral intramedullary nail fixation was performed, and a large amount of abscess and necrotic tissue was found intraoperatively. The cultures of abscess specimens were identified as P. micra, Fusobacterium nucleatum, and C. rectus. C. rectus and P. micra were also isolated from blood cultures. C. rectus was identified by matrix-assisted laser desorption/ionization time-of-flight mass spectrometry and 16 S ribosomal RNA sequencing. Sulbactam-ampicillin was administered for approximately 1 month, after which it was replaced by oral clavulanic acid-amoxicillin for long-term suppressive treatment. CONCLUSIONS: Only five cases of bloodstream infection with C. rectus have been reported, and this is the first report of mixed bacteremia with P. micra. Clinicians should consider that chronic periodontitis caused by rare oral anaerobic bacteria can cause systemic infections, such as osteomyelitis.

Clinical study on orthopaedic treatment of chronic osteomyelitis with soft tissue defect in adults.

To investigate the clinical application value of different flap transfer and repair techniques in adult patients with chronic osteomyelitis of limbs complicated with soft tissue defects. According to the characteristics and defects of 21 cases, different plastic surgery was applied, including debridement, negative pressure device, and tissue flap to cover wound. Among 21 cases of chronic osteomyelitis complicated with local soft tissue defect, 15 patients were repaired with sural neurotrophic musculocutaneous flap transfer, 2 patients were repaired with medial plantar skin flap transfer, 2 patients were repaired with ilioinguinal skin flap transfer, 1 patient was repaired with z-forming wound, and 1 patient was repaired with soleus muscle flap combined with full-thickness skin graft. All the 21 patients underwent bone cement implantation after dead bone osteotomy. Among them, 19 patients underwent bone cement replacement with 3D prosthesis within 6 months to 1 year after surgery, and 2 patients carried bone cement for a long time. Early intervention, thorough debridement, removal of necrotic or infection, and then selecting the appropriate wound skin flap coverage are important means of guarantee slow osteomyelitis wound healing and for providing a possible way to permanent prosthesis implantation subsequently.

Keratocystic Odontogenic Tumor Associated with Impacted Molar in the Maxillary Sinus Causing Osteomyelitis.

Keratocystic odontogenic tumors are cystic masses that arise from cells involved in tooth development. These lesions can be very locally aggressive and have a remarkable rate of recurrence. This combination of traits necessitates aggressive treatment and monitoring. They most commonly affect the mandible; an uncommon presentation is to involve the maxillary sinus. Here we present a case of a keratocystic odontogenic tumor that developed in association with an impacted third molar in the maxillary sinus that subsequently became infected and evolved to maxillary osteomyelitis.

Induced membrane technique using beta-tricalcium phosphate for reconstruction of femoral and tibial segmental bone loss due to infection: technical tips and preliminary clinical results.

PURPOSE: This study aimed to provide preliminary evidence regarding effectiveness of grafting beta-tricalcium phosphate (ß-TCP) combined with a cancellous autograft for treating nonunion of long bones in the lower extremity due to infection by evaluating clinical and radiological outcomes. METHODS: We retrospectively reviewed the clinical and radiological results in seven patients (six men, one woman; median age 39 years) treated by the induced membrane technique for nonunion of the femur or tibia due to infection. In the second stage of the procedure, the bony defect was filled with a combination of autologous cancellous bone and ß-TCP, which were mixed in approximately the same proportions. The time interval between the second stage of the procedure and bone healing was investigated. Radiographic characteristics including maximum bone gap and radiographic apparent bone gap were evaluated. RESULTS: The median follow-up period was 14 months. Bone healing was achieved in a median of six months after the second procedure. The median maximum bone gap and radiographic apparent bone gap were 55 mm and 34 mm, respectively. DISCUSSION: Use of ß-TCP, which has osteoconductive ability, with an autograft provided good clinical and radiological outcomes. The findings of this preliminary study suggest the potential of ß-TCP as a useful bone substitute for autografts in the induced membrane technique. CONCLUSIONS: Our findings suggest that ß-TCP may be an effective extender when using the induced membrane technique.

Chronic infection and infected non-union of the long bones in paediatric patients: preliminary results of bone versus beta-tricalcium phosphate grafting after induced membrane formation.

PURPOSE: Chronic infection (CO) and infected non-union of the long bones are relatively rare conditions in paediatric patients. Large bone defects secondary to these conditions can be managed with the induced membrane technique. The technique requires grafting of the bone void, although it is not yet established what bone substitute is the best option. The aim of this work was to evaluate the outcome and efficacy of treatment in children with CO and infected non-union of the long bones using the induced membrane technique and bone (BG) versus beta-tricalcium phosphate (BTP) grafting. METHODS: Eight skeletally immature patients with CO and infected non-union of the long bones were treated surgically between 2010 and 2017 by a combination of resection of necrotic infected bone, debridement of surrounding soft tissue, osteosynthesis using a stable internal fixation when needed, and application of antibiotic-laden cement (ALC) spacer inducing new membrane before final bone reconstruction with bone substitutes: BTP in five cases, BG (allograft and/or autologous graft) in three cases. A second surgical step, once inflammatory markers had normalized, consisted of ALC spacer removal, application of BG or BTP graft and concomitant stable osteosynthesis, if needed, if this had not been done during the first surgical stage. All the patients underwent clinical, laboratory and imaging evaluation before and after surgery. Antibiotics were adjusted according to culture and sensitivity. RESULTS: Mean patient age at time of diagnosis was 13 ± four years (range, 4-16) and all had at least a 12-month follow-up (range 12-60). Estimated time for induced membrane formation was significantly shorter in patients treated with BTP compared with BG: 3±1 vs. 10±2 (p = 0.02). This result was confirmed by multivariate analysis (p = 0.044) taking into account adjustment for age of patients and time after initial surgery. Time of final union was about 5.5 ± 4.1 months (range 2-66). At the last follow-up visit, bone had healed and all the patients had resumed daily living and sports activities. CONCLUSION: The induced membrane technique with BG or BTP graft can achieve bone healing in large bone defects secondary to CO and infected non-union in children and adolescents. The choice of bone substitute is important. Our preliminary results show graft integration and bone healing can be expected sooner if BTP is used as bone void filler.

[Treatment of Mandibular Fracture-Associated Infectious Pseudarthrosis Accompanied by Osteomyelitis in a Peritoneal Dialysis Patient].

Peritoneal dialysis can be performed at home, and the transfer of solutes in the blood and other body fluids is slow compared to hemodialysis, reducing the load on the circulatory organs and lessening the frequency of hospital visits. We encountered a male patient in his 70s on peritoneal dialysis for end-stage renal failure who developed obsolete mandibular fracture-associated pseudarthrosis accompanied by osteomyelitis, which was treated with noninvasive reduction and fixation using circumferential wiring after the resolution of inflammation. The inflammation was resolved by an intravenous drip infusion of ampicillin and lavage of the local region through the fistulated region during hospitalization, and sequestrum was removed under local anesthesia. After the disappearance of drainage from the fistula, the mandibular fracture was fixed with circumferential wiring (noninvasive reduction and fixation) using a mandibular resin base (occlusion is possible). For noninvasive reduction and fixation of a midline fracture, a 6-week fixation period is usually necessary after surgery, but in this case it was fixed for 3 months after surgery because of the presence of infection and bone defect. In jaw bone infection in patients on long-term dialysis, high sensitivity to infection and incomplete cure occur due to a decline in cell-mediated immunity, renal osteodystrophy (ROD), and chronic kidney disease (CKD)-mineral and bone disorder. In the present patient, infection complicated the odontogenic source of infection and fracture, which may have protracted the condition. When jaw bone infection is noted in a patient on long-term dialysis, it is important to closely cooperate with the dialysis physician and select the administration method and dose corresponding to the route of administration and metabolism of antimicrobial agents in order to minimize the influence on the renal function. For the local region, infection control by oral hygiene management and cleaning is important, targeting treatment and management while avoiding the use of any antimicrobial agent.

Odontoid osteomyelitis with atlantoaxial subluxation in an infant.

PURPOSE: To report a rare case of odontoid osteomyelitis with atlantoaxial subluxation in a 6-month-old infant. BACKGROUND: Odontoid osteomyelitis with atlantoaxial subluxation is extremely rare in children. Although several cases have been reported, there have been no studies concerning proper surgical drainage and immobilization in this disease. METHODS: A 6-month-old infant with odontoid osteomyelitis with atlantoaxial subluxation was surgically treated. The patient underwent a 3-month intravenous and oral antibiotic course and the Minerva body jacket cast was used for 3 months. Follow-up was carried out with computed tomographic scans and a cervical spine dynamogram. RESULTS: At 18 months post-surgery, the patient had completely recovered with no cervical instability. Computed tomographic scans revealed complete fusion of odontoid synchondrosis. The infant remained asymptomatic with a full range of head movement. CONCLUSION: Surgical drainage and proper immobilization appears to be a satisfactory treatment for pyogenic osteomyelitis of odontoid synchondrosis secondary to retropharyngeal abscess and atlantoaxial subluxation. LEVEL OF EVIDENCE: N/A.

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.

Associated aneurysmal bone cyst and cemento-osseous dysplasia: a case report and review of the literature.

The purpose of this case report is to describe a previously unpublished association between focal cemento-osseous dysplasia (FCOD) and an aneurysmal bone cyst (ABC) and review the literature with regard to associated benign fibro-osseous lesions and cysts. A 41-year-old woman without a history of trauma presented with asymptomatic swelling in the right side of the mandible. Radiographs of the region revealed a unilocular radiolucent area with radiopaque foci. After aspiration of the lesion was positive for serosanguineous fluid, complete excision of the lesion was performed. Microscopic examination revealed a hybrid ABC and FCOD. The 12-month follow-up showed significant bone repair and no signs of recurrence. A review of the English-language literature from 1980 to 2012 revealed 1 retrospective study, 4 case series, and 18 single-case reports on the topic of cemento-osseous dysplasias, fibro-osseous lesions, and aneurysmal bone cysts. Of 59 cases, none reported an association between an ABC and FCOD. Although fibro-osseous lesions do not require intervention, surgical excision is recommended when they are associated with cysts. This case, in which an ABC and FCOD were associated, reinforces the need for a careful diagnostic process in radiographically mixed lesions that respond positively to aspiration biopsy.

Florid Cemento-Osseous Dysplasia Simultaneous the Chronic Suppurative Osteomyelitis in Mandible.

INTRODUCTION: The florid cemento-osseous dysplasia is an uncommon condition nonneoplastic, of unknown cause with higher prevalence in melanodermic women, limited the maxillary bones, is characterized by the presence of dispersed and diffuse radiopaque calcifications, constituted of bone and dense cemento; however, when the bone is infected it induces the suppuration and formation of osseous sequestra, thus resulting in an osteomyelitis frame. CLINICAL REPORT: The patient was attended in a Dental Specialties Center in the state of Alagoas, Brazil, presenting on clinical examination edema and extra oral fistula with pus drainage in hemiface submandibular of the right side. Radiographically it was possible to observe area of sclerosis and osseous sequestra involving the right side region of the mandible body, and it increases zones of the bone density. In association with clinical data and complementary diagnosis examinations, the option of treatment adopted was the complete removal of the bone fragment, followed by adaptation and plate fixation and titanium screws to reduce the risk of mandibular fracture. OBJECTIVE: The aim of the present paper was to relate a clinical patient of florid cemento-osseous dysplasia simultaneous the chronic suppurative osteomyelitis, highlighting their clinical, radiographic, and histological characteristics, as well as their diagnosis and treatment.

Chronic recurrent multifocal osteomyelitis in association with pyoderma gangraenosum.

BACKGROUND: Chronic recurrent multifocal osteomyelitis (CRMO) is a rare acquired inflammatory skeletal disorder of unknown origin. CRMO was first described by Gideon in 1972 and mainly affects children and young adults of female gender. The CRMO is part of the clinical picture of non-bacterial Osteomyelitis (NBO) and typically presents a relapsing recurring course with both remission and spontaneous exacerbation. CRMO is typically encountered in the limbs and the metaphysis of long bones in particular. Usually the clinical symptoms include painful swellings of the affected regions. This case report describes the rare case of a CRMO of the mandible in association with pyoderma gangraenosum. CASE PRESENTATION: A 14-year old female caucasian patient, residing in the south of Germany, presented in the oncological outpatient clinic of our Department of Paediatrics and Adolescent Medicine in June 2014 complaining of increasing neck pain and progressive swelling at her left cheek ongoing for about 6 weeks. These symptoms had been occurring quarterly for 4 years, but had never been as pronounced. Blood biochemistry showed a moderately elevated CRP (35 mg/l) and a significantly increased blood sedimentation rate (BSR 48/120 mm). The panoramic radiograph, however, revealed a bone alteration in the left mandibular region. Further investigations confirmed the diagnosis of CRMO. CONCLUSION: The present case underlines the fact that rare diseases might occasionally present with even more rare symptoms. These occasions can obviously be considered to present a considerable diagnostic challenge.

Florid osseous dysplasia associated with multiple simple bone cysts: a patient with 22 years of follow-up.

Florid osseous dysplasia (FOD) has been described as a condition that typically affects the jaws of middle-aged women of African descent and usually presents as multiple radiopaque masses distributed throughout the jaws. The diagnosis is generally based on clinical and radiographic presentation, often involving various regions of the jaws without evident bone expansion. This article describes the course of FOD in a 27-year-old African-American woman during 22 years of follow-up, emphasizing the dynamic radiographic characteristics of this entity and unusual co-occurrence with multiple simple bone cysts. These data reinforce the importance of long-term follow-up in patients diagnosed with FOD.

A Pathological Fracture of the Mandible due to Osteomyelitis following a Full Dental Clearance.

Orofacial infections following dental extractions are a common referral to an oral and maxillofacial department as an emergency, especially when combined with swelling and limited mouth opening. The case presented demonstrates a rare complication of chronic suppurative osteomyelitis with bilateral pathological fractures of the mandible, which occurred following a staged dental clearance. CPD/Clinical Relevance: Dental extractions are one of the most common treatments carried out by oral surgeons and general dental practitioners. This case highlights a rare but encountered complication of routine oral surgery and demonstrates when it is necessary to make an immediate referral to the local oral and maxillofacial surgery unit.

Two Cases of Masticator Space Abscess Spreading from Infections of the Mandibular Teeth.

The masticator space is located between the masseteric fascia and the pterygoid muscle fascia. Here we report two cases of masticator space abscesses spreading from infections of mandibular teeth. Case 1 is an 85-year-old lady who were referred to Yokohama City University Hospital with a left-cheek swelling and trismus. An enhanced CT scan revealed an abscess extending from the left infratemporal fossa to the temporal fossa. A purulent discharge was observed from her left lower gingiva. We performed surgical drainage under general anesthesia. After infection control, the affected teeth were extracted. Case 2 is an 82-year-old lady who was administered oral bisphosphonate for osteoporosis. She presented to another hospital with fever, trismus and swelling anterior to the right ear after right lower tooth extraction. Because MRI revealed persistent osteomyelitis of her mandible even after antibiotic treatment, she was referred to us. Enhanced CT showed an abscess in the right infratemporal fossa. After surgical drainage similar to Case 1, antibiotics were administered for approximately 4 months to control the osteomyelitis.　 It is important to recognize that infections of the mandibular teeth can cause an abscess in the masticator space through the pterygomandibular and infratemporal spaces.

[CHRONIC OSTHEOMYELITIS OF THE FACIAL MIDDLE ZONE WITH SPREADING ON THE BONES OF CRANIAL BASE ON BACKGROUND OF NARCOTIC DEPENDENCE].

Own experience of treatment of a narcotic dependence patients, suffering osteomyelitis of jaws, was analyzed. In those patients, who have had exposed themselves towards toxic impact of narcotic substances (even after arrest of the consumption) for a long period of time, the inflammation spreading on the bones of facial middle zone and the skull base with occurrence of a life3threatening purulent3septic, including intracerebral, complications (cerebral аbscess, meningitis, sepsis) were observed. Additional division of cerebral abscess on open and closed forms for optimization of diagnosis of іntracranial complications was proposed.