Recurrent osteomyelitis with proliferative periostitis after segmental resection and reconstruction of the mandible: a case report.

A 50-year-old patient presented with a two-year history of chronic osteomyelitis of the left mandibular body. It was treated by wide segmental resection of the left hemimandible and reconstruction with a free vascularized fibular graft. Six months after surgery, the patient returned with pain, swelling, and moth-like lesions in the transplant in combination with appositional bone formation surrounding the ossified fibular bone. Radiographic and histological examination led to the diagnosis of a recurrent osteomyelitis with proliferative periostitis affecting the resected and reconstructed mandible. Application of ibandronate led to a significant symptom decrease.

Spectrum of voriconazole-associated periostitis in clinical characteristics, diagnosis and management.

PURPOSE: Previous knowledge about the relationship between voriconazole exposure and periostitis was mainly based on limited case reports and few retrospective studies. The purpose of this study was to assess the clinical characteristics, diagnosis and management of voriconazole-associated periostitis. METHODS: Case reports and case series from 1998 to November 30, 2021 on periostitis induced by voriconazole were collected for retrospective analysis. RESULTS: Forty four patients (18 male and 26 female) from 34 studies were included in total. The median age was 58 years (29-74). The majority of patients had undergone organ transplantation (50.0%) or suffered from hematologic malignancy (31.81%). The median onset time of symptoms was 6 months after the start of voriconazole. The most common initial symptom was diffuse skeletal pain (68.28%) which can be severe and even disabling (66.7%). Ribs (37.21%), femurs (32.56%), scapulae (25.58%), humerus (23.26%), and clavicle (23.26%) were the common involved locations. Most cases were accompanied by different degrees of elevated serum alkaline phosphatase and fluoride level, while some presented with elevated bone-specific alkaline phosphatase. The main radiological features included periosteal reaction and multifocal high radiotracer uptake on bone scintigraphy. The formation of new bone was characterized with bilateral, irregular, nodular, as well as high density. The resolution of symptoms was observed with discontinuation of voriconazole in all patients, of whom 18 patients (52.94%) were relieved within a week. Itraconazole, posaconazole or isavuconazole were safe alternatives to voriconazole in voriconazole-induced periostitis. CONCLUSION: Voriconazole-induced periostitis is an infrequent complication characterized by bone inflammation involving one or multiple skeletal areas. Bony pain, elevated serum alkaline phosphatase as well as fluoride level are suspicious signs during voriconazole treatment.

Chronic Osteomyelitis With Proliferative Periostitis of the Mandible in a Child: Report of a Case Managed by Immunosuppressive Treatment.

BACKGROUND: Osteomyelitis with proliferative periostitis is a relatively uncommon inflammatory condition of the jaws, mainly characterized by periosteal formation of reactive bone. It primarily affects children and adolescences, also referred to as Garre's osteomyelitis, more frequently involving the molar region of the mandible. Cases lacking an obvious source of infection may have an immunologically mediated etiopathogenesis, falling under the spectrum of primary chronic osteomyelitis or chronic recurrent multifocal osteomyelitis (CRMO). CASE REPORT: Herein, we present a case of chronic osteomyelitis in a 6.5-year-old girl, who suffered from recurrent painful episodes of swelling of the mandible for the last 2 years, previously requiring hospitalization and administration of intravenous (IV) antibiotics and NSAIDs with limited responsiveness. The biopsy showed features consistent with osteomyelitis with proliferative periostitis. The patient was initially managed with an IV combination antibiotic regimen with only partial improvement. The possibility of an autoimmune mechanism in the context of primary chronic osteomyelitis or CRMO was considered, and immunosuppressive therapy (TNF inhibitor etanercept along with corticosteroids and methotrexate) was administered, resulting in clinical resolution. CONCLUSIONS: Osteomyelitis and its childhood variants are relatively rare and their management presents several challenges. Although typically treated with administration of antibiotics, possibly along with surgical intervention, other treatment modalities may be necessary for resilient and persistent cases. In a subset of cases, especially in the absence of local infectious factors, immunologically mediated mechanisms may play an important role and appropriate immunosuppressive therapy may be effective.

[A man with painful shins]. / Een man met pijnlijke scheenbenen.

A 55-year-old man was evaluated at the outpatient rheumatology clinic with painful shins since 6 weeks. He also had a maculopapular rash on his trunk. Bone scintigraphy showed bilateral tibia periostitis. Serologic testing for syphilis was positive matching active infection. The diagnosis secondary syphilis with bilateral tibia periostitis was made.

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.

A vexing case of bone pain in a renal transplant recipient: Voriconazole-induced periostitis.

Immunosuppression increases the risk of opportunistic infections including fungal infections in solid organ transplant recipients. Voriconazole is used to treat invasive aspergillus infections but prolonged usage may rarely lead to periostitis. Increased plasma fluoride concentration leading to osteoblastic upregulation is thought to be the catalyst, and symptom reversal occurs with discontinuation of the offending agent. A renal transplant recipient who was on voriconazole for invasive aspergillosis developed diffuse debilitating symmetrical bone pain. Having ruled out other neurological, metabolic, and drug etiologies, voriconazole-induced periostitis was diagnosed. Increased plasma fluoride level was documented, but bone scan was non-specific. A therapeutic discontinuation of voriconazole and switch to posaconazole provided rapid symptom resolution. The patient accidently restarted voriconazole as an outpatient resulting in the same symptomology, and thus provided further evidence that this was drug related. Voriconazole-induced periostitis is a described entity in immunosuppressed solid organ transplant patients who are treated with a prolonged course of voriconazole. This case study is novel in that it demonstrates drug induced periostitis in a renal transplant recipient who developed debilitating periostitis within a short time after starting voriconazole and equally rapid resolution once it was discontinued. We conclude that patients treated with voriconazole should be routinely monitored for periostitis.

Caso clínico: hipofosfatasia de la niñez. Seguimiento clínico / Clinical case: childhood hypophosphatasia. Clinical follow-up

La hipofosfatasia (HP) es una enfermedad congénita, causada por mutaciones con pérdida de función en el gen ALPL que codifica la isoenzima no específica de tejido de la fosfatasa alcalina (TNSALP). Su expresión clínica es muy variable, desde casos de muerte intraútero por alteración grave de la mineralización ósea, hasta casos solo con caída prematura de la dentición. Se presenta el caso clínico de un varón al que se le diagnosticó odontohipofosfatasia a los 30 meses por pérdida temprana de piezas dentarias y niveles anormalmente bajos de fosfatasa alcalina, sin signos de raquitismo ni deformidades óseas. Durante su seguimiento, hasta los 13 años, presentó síntomas compatibles con HP infantil leve, como cansancio al caminar, incoordinación en la marcha y dolor en miembros inferiores que aumentaban con la actividad física. Ante la aparición de edema bimaleolar y poca respuesta al tratamiento con calcitonina y antiinflamatorios, se descartaron patologías infecciosas o reumáticas o ambas y se diagnosticó, por biopsia de tibia y peroné, periostitis sin detección de cristales de pirofosfato. Los controles radiológicos durante su evolución mostraron ensanchamiento metafisario en muñeca, falta de remodelado de metacarpianos, hojaldrado perióstico en tibia y peroné e hipomineralización en metáfisis tibiales, con "lenguas radiolúcidas" características de HP. Como conclusión, la hipofosfatasia debe considerarse como una entidad clínica para descartar en niños que presentan pérdida temprana de dientes. La presencia de este cuadro clínico es en general suficiente para realizar el diagnóstico de HP de la niñez. (AU)

Hypophosphatasia (HP) is a congenital disease, caused by mutations with loss of function in the gene ALPL that encodes the non-specific tissue isoenzyme of alkaline phosphatase (TNSALP). Its clinical expression displays considerable variability, from cases of intrauterine death due to severe alteration of bone mineralization, to cases with only early loss of teeth. We report the case of a male, diagnosed as odontohypophosphatasia at 30 months of age due to early loss of teeth and abnormally low levels of alkaline phosphatase, without signs of rickets or bone deformities. During follow-up, up to 13 years of age, he presented symptoms consistent with mild infantile HP such as tiredness when walking, lack of gait coordination, and pain in lower limbs, especially after physical activity. Due to the appearance of bimalleolar edema and poor response to treatment with calcitonin and anti-inflammatory drugs, infectious and / or rheumatic pathologies were ruled out. Periostitis without pyrophosphate crystal detection was diagnosed by tibial and fibular biopsy. Radiological controls during follow up showed metaphyseal wrist enlargement, lack of remodeling of metacarpals, periosteal flaking in the tibia and fibula and hypomineralization in the tibial metaphysis, with "radiolucent tongues"; characteristic of HP. In conclusion, hypophosphatasia should be considered as a clinical entity in children who present early loss of teeth. The presentation of this clinical case is generally sufficient to make the diagnosis of childhood HP. (AU)

Chronic non-suppurative mandibular osteomyelitis with proliferative periostitis: A review.

Chronic non-suppurative osteomyelitis (CNSO) is a chronic bone disease and may be associated with a reparative periosteum entity called proliferative periostitis (PP). This condition rarely affects the maxillofacial region. Mandibular cases were already described for an infectious dental cause, often with an "onion skin" radiographic aspect, but some rare reported cases showed no obvious etiology. They represent a challenge for diagnosis because of possible misdiagnosis leading to unsuccessful or inappropriate treatment attempts. An uncommon case of mandibular CNSO associated with PP in a 9-year-old boy with no obvious infectious or inflammatory causes is reported. Clinical and radiographic examinations revealed a swelling in the left hemimandible, associated with multiple osteolytic areas inside both medullary and newly formed periosteal bone and cortical bone perforations. Recovery signs were seen after a 22-month follow-up period, and radiographic signs of bone healing were observed. From this new case report, a review of the literature was performed on reported cases of mandibular CNSO with PP, and discussed the etiological, clinical, radiologic, and therapeutic aspects of this pathology. This work highlights the importance of considering CNSO with PP in the differential diagnosis of one-sided painless mandibular swellings, even in the absence of an obvious cause.

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.

Periostitis Ossificans Arising in the Mandibular Bone of a Young Patient: Report of an Unusual Case and Review of the Literature.

Periostitis ossificans, also known as Garré osteomyelitis, is a specific type of chronic osteomyelitis that forms new bone under the periosteum resulting from a periosteal reaction to chronic inflammation or infections. It commonly affects the mandible secondary to odontogenic infection. The therapeutic approach involves eliminating the infectious cause and antibiotic administration. This report describes an unusual case of periostitis ossificans arising from the mandible of an 11-year-old boy. The cause of infection was correlated with a lower right unerupted third molar, which had no obvious connection with the oral cavity. The histologic diagnosis was chronic osteomyelitis with proliferative periostitis. The patient has been followed for 1 year, without any evidence of recurrence. Periostitis ossificans can be diagnostically problematic, and various conditions must be considered in the differential diagnosis.

Florid reactive periostitis of the long bone: A case series of seven patients.

BACKGROUND: Florid reactive periostitis (FRP) is a rare benign periosteal lesion, which mostly involves the tubular bones of the extremities. FRP of the long bone is especially rare. We here report a case series of long bone FRP and also discuss the differential diagnosis and clinical outcome of our series. PATIENTS AND METHODS: In total, four female and three male patients with long bone FRP and a mean age of 25.1 years were evaluated for this study, from which four upper extremity and three lower extremity FRP lesions were identified. Patients were classified according to the clinical, radiological and pathological manifestations of the lesion. RESULTS: Periosteal reaction and edema around the lesions were observed in all cases. Calcified mass, bone marrow edema and cortical erosion were observed in six out of seven patients. Moreover, two lesions with peripheral mineralization and zoning appearances were observed. Limb rest and anti-inflammatory therapy proved to be sufficient treatment in all cases. The lesions were spontaneously resolved in all cases, leaving a residual exostosis. CONCLUSION: Our report indicates that short bone and long bone FRP could reveal different characteristics in some aspects, including the choice of the therapeutic approach. In spite of the favorable clinical outcome of long bone FRP, its differentiation from more aggressive lesions, especially osteosarcoma and osteomyelitis, should still be taken into consideration. In addition, according to our evidence, the implication of the zonal pattern in differential diagnosis of FRP and myositis ossificans (MO) could be re-evaluated.

Florid reactive periostitis in the fifth phalange of a professional boxer: A case report.

RATIONALE: Florid reactive periostitis is a rare, benign bone and chondrogenic lesion that develops most frequently in the phalanges of the hands. Although the definitive cause of florid reactive periostitis is unknown, the major inciting factor is generally considered to be trauma, including repetitive minor trauma. PATIENT CONCERNS: We present a case of florid reactive periostitis affecting two contiguous phalangeal bones of the left fifth phalange of a 23-year-old male professional boxer. The patient had experienced chronic pain around the metacarpophalangeal joint of the left fifth phalange when punching with the left hand; this pain was improved but not resolved after conservative treatment. INTERVENTIONS: Plain radiography of the fifth phalange revealed a bulging bone lesion on the volar side of the proximal phalanx and metacarpal of the left fifth phalange. Computed tomography also showed raised bony lesions on the volar side of these bones without bony destruction or intraregional calcification. Surgery was performed and histopathology revealed that the bone specimens comprised a mixture of fibrous connective tissue and relatively immature bone covered by bland osteoblasts. DIAGNOSES: These findings were suggestive of a benign bone formation process, and the lesion was diagnosed as florid reactive periostitis. OUTCOMES: The pain and tenderness in the left fifth phalange experienced during boxing had completely resolved by 6 months postoperatively, and 12 years postoperatively the patient had full range of motion and no recurrence of pain. LESSONS: Traction force, such as those associated with "leading jabs," may induce repetitive minor trauma and subsequent periosteal damage, resulting in the gradual development of bulging bone ridges on the volar surface of the proximal fifth phalange and metacarpus of the hand.

Periostitis ossificans (Garrè's osteomyelitis): An unusual case.

Garrè's sclerosing osteomyelitis is a specific type of chronic osteomyelitis that mainly affects children and young adults and is commonly associated with odontogenic infection. The paper describes an unusual case of Garrè's osteomyelitis in an 11-year-old boy, in whom the condition arose following poor oral hygiene and periodontal problems in relation to permanent mandibular left first molar. Clinically the patient presented with bony hard, nontender swelling and the occlusal radiograph revealed pathognomic feature of "onion skin" appearance.

Case 215: voriconazole-induced periostitis.

History A 74-year-old woman presented with multifocal bone pain, including pain in multiple ribs, bilateral shoulders, and bilateral hips. The pain began several months before presentation and was quite severe, ultimately necessitating control with narcotics. At examination, strength in both lower extremities was slightly reduced, sensation and reflexes were intact, and range of motion was full, though painful. There were no notable constitutional symptoms of fever or weight loss. Laboratory work-up was remarkable for elevated alkaline phosphatase level (277 U/L [4.6 mkat/L]). The patient had undergone left lung transplantation 8 years prior for pulmonary fibrosis. A thorough pulmonary work-up for the cause of fibrosis, which included gathering an exposure, occupational, allergy, and previous infectious history, and a rheumatoid work-up were negative. The patient's posttransplantation course was complicated by bronchiolitis obliterans from chronic rejection and by recent pulmonary embolism, for which she was undergoing anticoagulation therapy at the time of presentation. Additionally, the patient experienced repeated pulmonary infections with Aspergillus, leading to multiple hospitalizations and long-term antifungal prophylaxis with voriconazole. A bone scan from an outside hospital was reviewed, and further imaging was performed.