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.

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)

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.

Periosteum: characteristic imaging findings with emphasis on radiologic-pathologic comparisons.

The periosteum covers most bone structures. It has an outer fibrous layer and an inner cambial layer that exhibits osteogenic activity. The periosteum is a dynamic structure that plays a major role in bone modeling and remodeling under normal conditions. In several disorders such as infections, benign and malignant tumors, and systemic diseases, the osteogenic potential of the periosteum is stimulated and new bone is produced. The newly formed bone added onto the surface of the cortex adopts various configurations depending on the modalities and pace of bone production. Our aim here is to describe the anatomy, histology, and physiology of the periosteum and to review the various patterns of periosteal reaction with emphasis on relations between radiological and histopathological findings. A careful evaluation of the periosteal reaction and appearance of the underlying cortex, in combination with the MRI, clinical, and laboratory data, provides valuable information on lesion duration and aggressiveness, thereby assisting in the etiological diagnosis and optimizing patient management. A solid reaction strongly suggests a benign and slow-growing process that gives the bone enough time to wall off the lesion. Single lamellar reactions occur in acute and usually benign diseases. Multilamellar reactions are associated with intermediate aggressiveness and a growth rate close to the limit of the walling-off capabilities of the bone. Spiculated, interrupted, and complex combined reactions carry the worst prognosis, as they occur in the most aggressive and fast-growing diseases: the periosteum attempts to create new bone but is overwhelmed and may be breached.

Sclerotic multiple myeloma with an unusual sunburst periosteal reaction occurring in the sternum.

Multiple myeloma is a disseminated neoplastic monoclonal gammopathy that usually affects the skull, clavicle, rib, pelvis, spinal column, and proximal portions of the humerus and femur. The initial manifestation of multiple myeloma in the sternum is rare. The classic radiological presentations of multiple myeloma are multiple "punched-out" areas of bone destruction, expansile lytic lesions, and generalized osteoporosis. Primary sclerotic presentation is rare and occurs in only 3 % of cases. A sclerotic multiple myeloma with a sunburst periosteal reaction occurring in the sternum has not been reported in the English literature. We report a case of sclerotic multiple myeloma of a 49-year-old woman. In the sternum, the lesion displayed extensive sclerosis mixed with mottled lytic areas with a sunburst periosteal reaction occurring in the periphery, which radiologically mimicked an osteosarcoma. Multiple focal areas of sclerosis were also found in the right clavicle, pelvis, multiple ribs, and vertebrae.

Voriconazole-associated soft tissue ossification: an undescribed cause of glenohumeral joint capsulitis.

Voriconazole-related periostitis has been increasingly described in the literature over the last several years as a recognizable disease entity, especially in lung transplant patients. This relationship should be considered when approaching immunosuppressed patients presenting with diffuse bone pain and imaging findings of periostitis. We present a case of voriconazole-associated periostitis, capsular and enthesial ossification and glenuhumeral capsulitis in a patient with a hematologic malignancy. To the authors' knowledge, soft tissue ossification associated with voriconazole has not been described in the radiology literature.

Five-day, low-level laser therapy for sports-related lower extremity periostitis in adult men: a randomized, controlled trial.

Periostitis in the lower leg caused by overexercise is a universal problem in athletes and runners. The purpose of this study was to observe the functional improvement of the lower limbs upon rehabilitation low-level laser therapy (LLLT). All medical data were gathered from enrolled adults with sports-related lower leg pain. A total of 54 patients underwent triple-phase bone scans using skeletal nuclear scintigraphy, which confirmed periostitis in their lower limbs. The patients were then randomly divided into two groups: one group received laser therapy (N = 29) and the other group (N = 25) received an equivalent placebo treatment (a drug or physical therapy). Treatment protocol commenced with rehabilitation intervention and LLLT was performed three times daily for 5 days at a dosage of 1.4 J/cm(2). A Likert-type pain scale was used to evaluate the severity of pain. Balance function, including postural stability testing (PST) and limits of stability (LOS), was also performed to evaluate the function outcome. Patients experienced a significant improvement in pain by day 2 or day 5 after starting LLLT, but here was no significant difference in pain scale between the measurements before (baseline) and after LLLT. Comparing the PST, the group differences of dynamic vs. static testings ranged from -18.54 to -50.22 (compared 12, 8, 4, 3, 2, 1 to 0, all p < 0.0001), and the PST after LLLT were 3.73 units (p = 0.0258) lower than those of before LLLT. Comparing the LOS, the group differences of dynamic vs. static testing were similar to those in PST, and the relationship between LOS and groups only varied with the direction control during dynamic testing in direction at backward/right vs. right (p < 0.0001). LLLT had a positive effect on proprioception in patients with lower limb periostitis. Larger, better controlled studies are needed to determine what specific effects LLLT has on the function of proprioception.

Florid reactive periostitis of the metacarpal and phalanx: 2 case reports.

Florid reactive periostitis is a rare benign fibro-osseous proliferative lesion of soft tissue and is mainly composed of fibrous connective tissue and mature bone. The lesion usually occurs in the finger and seldom in the metacarpal. The lesion mimics malignant and infectious disorders, thus often leading to inappropriate treatment. Radiography usually shows an ossified mass attached to the underlying hand bones without interruption of the cortex. We report 1 case of florid reactive periostitis with unossified mass and underlying bone marrow edema in the metacarpal of hand and 1 case with adjacent cortical destruction in the phalanx. In addition, we describe the imaging findings of computed tomography CT and magnetic resonance imaging MRI and discuss their diagnostic values compared with routine radiographic examination.

Goldbloom's syndrome - a case report.

The Goldbloom's syndrome (GS) is a rare clinical condition of unknown aetiology, occurring exclusively in the pediatric population. It consists in an idiopathic periosteal hyperostosis with dysproteinemia, whose symptoms can mimic a neoplastic disease. We present a case report illustrating the diagnostic challenge of this condition. The exclusion of the common causes of bone pain, associated with generalized periostitis and increased gammaglobulins suggested the diagnosis of GS. The self-limited symptoms, the resolution of radiological findings in four months and the normalization of laboratory abnormalities within ten months, allowed to establish definitely the diagnosis of GS. GS must be considered when diffuse bone pain, prolonged fever and weight loss are present after exclusion of malignant disease with bone involvement.

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.

Paranasal sinus infection causing orbital subperiosteal abscess: surgical management of this devastating entity.

BACKGROUND: Orbital infection is an uncommon devastating infection and is usually a complication of paranasal sinus infection. Without appropriate treatment, orbital infection may lead to serious complications, even death. Prompt treatment is mandatory to avoid visual loss or intracranial complications. The literature shows that initially, intravenous antibiotics should be administered, and after 48 h, if no improvement appears, the affected orbit and the sinuses must be surgically drained. The authors describe two cases of orbital cellulitis with a brief literature review. CASE REPORT: The authors describe two cases of orbital abscess caused by paranasal sinus infection. In case 1, the patient presented a decreased visual acuity associated with ophthalmoplegia of the right eye. In case 2, the patient presented a decreased visual acuity. Thus, administration of intravenous antibiotic combined with surgical drainage was performed. After surgical procedure, eye movements were normalized in case 1, and in both patients, the visual acuity returned to normal parameters. DISCUSSION: The authors recommend early surgical drainage with parenteral antibiotic administration and careful postoperative observations by monitoring the signs and symptoms of the orbital complaint.