An open-label, prospective, observational study of the efficacy of bisphosphonate therapy for painful osteoid osteoma.

OBJECTIVES: To assess the efficacy of bisphosphonate therapy on bone pain in patients with osteoid osteoma (OO) (main objective), and to describe bisphosphonate-induced changes in nidus mineralisation and regional bone-marrow oedema (BMO). METHODS: A prospective, observational study was conducted from 2011 to 2014. Patients with risk factors for complications of percutaneous or surgical ablation or recurrence after ablation, were offered once monthly intravenous bisphosphonate treatment until significant pain alleviation was achieved. RESULTS: We included 23 patients. The first two patients received pamidronate and the next 21 zoledronic acid (mean, 2.95 infusions per patient). Bisphosphonate therapy was successful in 19 patients (83%), whose mean pain visual analogue scale score decreased by 76.7%; this pain-relieving effect persisted in 17 patients (74%) with a mean follow-up time of 36 months. Computed tomography (CT) demonstrated a mean nidus density increase of 177.7% (p = 0.001). By magnetic resonance imaging (MRI), mean decreases were 38.4% for BMO surface area and 30.3% for signal intensity (p = 0.001 and p = 0.000, respectively). CONCLUSIONS: In 17/23 patients with painful OO managed conservatively with bisphosphonates, long-term final success was achieved. Bisphosphonates may accelerate the spontaneous healing of OO. KEY POINTS: • 19/23 patients with OO managed with bisphosphonates experienced significant pain relief • Pain relief was sustained in 17/23 patients, mean follow-up of 36 months • CT demonstrated a significant increase in nidus mineralisation • MRI demonstrated a significant decrease in bone marrow oedema • Bisphosphonate therapy may accelerate the spontaneous healing of OO.

Multiple diaphyseal sclerosis (Ribbing disease): what about neridronate?

Sclerosing bone disorders are uncommon diseases and represent a diagnostic challenge. Osteocondensation is a bone alteration, involving both acquired and hereditary conditions. Multiple diaphyseal sclerosis (Ribbing disease) is an inherited condition. It is characterized by excessive proliferation of endosteal and periosteal osseous tissue at the diaphyses of long bones, especially of tibias and femurs. The conventional radiology depicts cortical thickening of diaphyses of long bones while bone scintigraphy shows characteristically an abnormal tracer concentration in the involved diaphyses. The magnetic resonance imaging (MRI) examination confirms the presence of sclerosis and reveals bone marrow edema in the diaphyses of the afflicted bones. Due to the lack of knowledge of the pathophysiology, the treatment is empirical with glucocorticoids or bisphosphonates. Concerning bisphosphonates, the literature reports are conflicting. We report the case of a patient that showed lack of response to intravenous neridronate within 1 year of treatment, both in terms of pain and persistence of bone marrow edema at MRI.

Conservative treatment for osteoid osteoma of the odontoid process of the axis: a case report.

BACKGROUND: Osteoid osteoma is a primary benign bone lesion, which constitutes about 10% of all primary benign bone tumors and 3% of all primary bone tumors. The spine is involved in 10% of the cases, and the lumbar spine is the most commonly affected whereas the tumor is rarely seen in the cervical spine. With regard to the osteoid osteoma being located at the odontoid process of the axis, limited cases have been reported in the literature. CASE PRESENTATION: An osteoid osteoma of the odontoid process of the axis was diagnosed by computed tomography in an 18-year-old male patient with a 3-month history of pain. The patient's parents refused surgery for fear of surgical risks and high expense. Considering the benign nature of osteoid osteoma, we prescribed celecoxib 200 mg per day to the patient. With the treatment, the patient's pain was alleviated gradually and the range of motion of the cervical spine also recovered to normal. At the two-year phone follow-up, the patient was free of symptoms. CONCLUSIONS: For this kind of benign tumor, conservative treatment plus close follow-up is applicable whereas surgery bears significant risks and a heavy economic burden.

Bisphosphonate treatment in inaccessible osteoid osteomas: An alternative therapeutic approach.

INTRODUCTION: Osteoid osteoma is a benign osteogenic tumour traditionally treated by surgical excision or percutaneous CT-guided procedures. We describe three cases of osteoid osteomas of which the locations were difficult to access, or for which the procedure was potentially unsafe, involving treatment with zoledronic acid infusions. CASE DESCRIPTION: We report here three male 28-to-31-year-old patients with no medical history who had osteoid osteomas located at the second cervical vertebra, the femoral head, and the third lumbar vertebra respectively. These lesions were responsible for inflammatory pain requiring daily treatment with acetylsalicylic acid. Given the impairment risk, all of the lesions were ineligible for surgical or percutaneous treatment. Patients were successfully treated by 3 to 6 monthly zoledronic acid infusions. All patients experienced complete relief of their symptoms allowing aspirin discontinuation, without any side effects. In the first two cases, CT and MRI control showed nidus mineralization and bone marrow oedema regression, correlating with the pain decrease. After 5years of follow-up, there had been no recurrence of the symptoms. CONCLUSION: In these patients, monthly 4mg zoledronic acid infusions have been safe and effective in the treatment of inaccessible osteoid osteomas.

Administration of nonsteroidal anti-inflammatory drugs accelerates spontaneous healing of osteoid osteoma.

INTRODUCTION: It has been reported that osteoid osteoma may heal spontaneously. METHOD: To elucidate the efficacy of conservative treatment with nonsteroidal anti-inflammatory drugs (NSAIDs) for osteoid osteoma, clinical courses of the 15 patients with osteoid osteoma conservatively treated with NSAIDs were observed. Twelve out of the 15 patients took a usual dose of NSAIDs regularly (regular group). RESULTS: Except for one patient, all the patients of the regular group maintained pain-free state. Eight out of the 12 patients of the regular group were free of pain even after discontinuing NSAIDs in the average of 18.3 months (range 2-36 months). Because one patient of the regular group required twice the usual dose to maintain pain-free state, we performed surgical excision. The remaining three patients of the regular group were asymptomatic and still taking NSAIDs. The healing rate of the osteoid osteoma with regular dose of NSAIDs was 8/12 (67%) at the time of this study, which may be improved up to 11/12 (92%). On the other hand, mean period of time until spontaneous diminution of pain in the 14 patients conservatively observed without NSAIDs so far reported in the literature was 75 months (range 24-180 months). CONCLUSION: Thus, osteoid osteoma is highly likely to heal spontaneously and administration of NSAIDs accelerates spontaneous healing. Therefore, conservative treatment with NSAIDs can be an important option other than surgical excision in treating osteoid osteoma.

[A girl with nocturnal cervical pain]. / Een meisje met onbegrepen nachtelijke nekklachten.

A 12-year-old girl consults the orthopaedic surgeon to exclude somatic causes of her cervical pain. One of her specific symptom is nocturnal pain, which responds well to NSAIDs. SPECT-CT showed an osteoid osteoma of the C3 pedicle. Osteoid osteoma is mostly a self-limiting disease in young adults.

Imaging features and differential diagnosis of multiple diaphyseal sclerosis: A case report and review of literature.

RATIONALE: Multiple diaphyseal sclerosis (MDS), known as Ribbing disease, is a rare congenital bone disease resulting from autosomal recessive inheritance. The case study involved a 22-year-old female patient who had been diagnosed with chronic sclerosing osteomyelitis due to lack of knowledge about MDS. Previous studies reported rarely on this condition. PATIENT CONCERNS: A 22-year-old female with MDS was analyzed. DIAGNOSES: MDS is characterized radiographically by a fusiform widening of the diaphyseal portion of the long bones, which is caused by a thickening of the cortex with obstruction of the medullary cavity. The pathologies are observed utilizing diagnostic imagery and are often difficult to identify. INTERVENTION: The patient was following a suggested regimen of oral celecoxib capsules at 200 mg/day for 6 days. OUTCOMES: The patient's diagnosis was revised to the rare condition of Ribbing disease by reviewing the clinical history and distinctive radiography images and because the symptoms were alleviated by celecoxib capsule. We also present a review of the literature on the diagnosis and differential diagnosis of MDS based on clinical and imaging features. LESSONS: MDS is rare and may often be initially misdiagnosed as another type of sclerosing bone dysplasia, thus, it is important to be aware of the existence of MDS. Once MDS is suspected, differential diagnosis should be performed to exclude other sclerosing bone dysplasias, taking into account clinical history, distinctive radiographic appearance, distribution, and laboratory and histopathologic findings. Laboratory evaluation and pathologic findings are nonspecific but assist in excluding other diagnoses. More evidence is needed to illustrate the effectiveness of medical or surgical treatments for patients with MDS.

Osteoid osteoma of the acetabulum: diagnosis and medical treatment.

We describe the medical management of an acetabular osteoid osteoma in an 11-year-old girl. There was a 2-year delay until proper diagnosis of the patient's pathology was made. Accurate localization of the nidus in the subchondral bone, involving the cartilage, is difficult on the basis of plain radiographs. All imaging methods, including computed tomography, MRI, and bone scintigraphy, were used to confirm the diagnosis. We proposed medical treatment to avoid possible severe complications from surgical intervention. At the patient's latest follow-up visit, after 2 years of medical treatment with NSAIDs, there is complete healing of the osteoid osteoma and there is no sign of recurrence.

Benign Tumors of the Spine: Has New Chemotherapy and Interventional Radiology Changed the Treatment Paradigm?

STUDY DESIGN: Clinically based systematic review. OBJECTIVE: To determine the role of (A) medical treatment and (B) interventional radiology as either adjuvant or stand-alone treatment in primary benign bone tumors of the spine. METHODS: A multidisciplinary panel of spine surgeons, radiation oncologists, and medical oncologists elaborated specific focused questions regarding aneurysmal bone cyst, giant cell tumor, and osteoid osteoma. Denosumab, bisphosphonate, interferon, bone marrow aspirate, doxycycline, thermal ablation, and selective arterial embolization were identified as areas of interest for the article. A systematic review was performed through MEDLINE and EMBASE. Recommendations based on the literature review and clinical expertise were issued using the GRADE system. RESULTS: The overall quality of the literature is very low with few multicenter prospective studies. For giant cell tumor, combination with Denosumab identified 14 pertinent articles with four multicenter prospective studies. Nine studies were found on bisphosphonates and six for selective arterial embolization. The search on aneurysmal bone cyst and selective arterial embolization revealed 12 articles. Combination with Denosumab, Doxycycline, and bone marrow aspirate identified four, two, and three relevant articles respectively. Eleven focused articles were selected on the role of thermal ablation in osteoid osteoma. CONCLUSION: Alternative and adjuvant therapy for primary benign bone tumors have emerged. Their ability to complement or replace surgery is now being scrutinized and they may impact significantly the algorithm of treatment of these tumors. Most of the data are still emerging and further research is desirable. Close collaboration between the different specialists managing these pathologies is crucial. LEVEL OF EVIDENCE: N/A.

Intramedullary fixation for congenital pseudarthrosis of the tibia.

Eighteen consecutively seen patients who had a congenital pseudarthrosis of the tibia were treated operatively. The mean age when the patients were first seen was four years. Seventeen previous procedures had failed: six patients had had one previous procedure; three, two previous procedures; and one, five previous procedures. At an average follow-up interval of ten years (range, three to nineteen years), healing with re-formation of the medullary canal was seen in thirteen of the eighteen tibiae, including one tibia that had united after a Boyd amputation. Five patients did not have healing of the tibia: four of them had a below-the-knee amputation, and one declined additional treatment. The average residual angulation was 12 degrees in the sagittal plane and 5 degrees in the coronal plane. Union occurred in ten of the thirteen patients who had been managed with intramedullary fixation. Of these thirteen, eight had been managed with intramedullary fixation, bone-grafting, and implantation of an electrical stimulator, and seven of them had union. Five of the thirteen patients had been managed with intramedullary fixation and bone-grafting, and three of them had union. Union occurred in one of two patients who had been managed with vascularized free fibular transfer, one who had been managed with delayed bone-grafting, and one who had been managed with a Boyd amputation. Four of the five patients who did not have union were subsequently managed with a below-the-knee amputation, and one patient refused additional treatment.

[Diagnostic image (93). A boy with a painful leg swelling. Osteoid osteoma]. / Diagnose in beeld (93). Een jongen met een pijnlijke crurale zwelling. Osteoïd osteoom.

A 13-year-old boy presented with a swelling on his left lower leg and suffered from pain, which was almost immediately relieved by acetylsalicylic acid. Roentgenographic and histologic appearance demonstrated an osteoid osteoma in the tibia.

Conservative treatment for patients with osteoid osteoma: a case series.

BACKGROUND: Osteoid osteoma is one type of benign bone tumor that may respond to conservative treatment. PATIENTS AND METHODS: A retrospective study of 11 patients with osteoid osteoma was performed. Initially, patients were treated with non-steroidal anti-inflammatories (NSAIDs), unless they continued to have intolerable pain, or if the patient requested surgical resection and radiofrequency ablation (RFA). RESULTS: Conservative therapy was successful for five patients. Three patients were treated with NSAIDs and the mean duration of pain was 22 months. The other two patients went into remission without medication by 9 and 24 months, respectively. Four patients underwent surgery after an average of 12 months. Two patients were treated with RFA after 47 and 11 months, respectively. CONCLUSION: Osteoid osteoma can be treated conservatively. Surgical resection and radiofrequency ablation should also be taken into consideration as an option when the results of conservative treatment are poor.

Treatment responses in five patients with Ribbing disease including two with 466C>T missense mutations in TGFß1.

OBJECTIVE: To assess 5-year treatment responses and TGFB1 gene abnormalities in five patients with ribbing disease. METHODS: PCR analysis and bidirectional sequencing of TGFß1 exons 1 through 7 were performed in all five patients. RESULTS: The five patients, four women and one man with a mean age of 34 years at symptom onset, shared the following features: severe diaphyseal pain predominating in the lower limbs with diaphyseal hyperostosis; increased radionuclide uptake at sites of pain and, in some cases at other cortical sites; asymmetric or asynchronous lesions; long symptom duration (5-18 years) despite a variety of treatments; and a delay of several years (2-15) between symptom onset and the diagnosis. Of our five patients, two had a heterozygous missense mutation in exon 2 of TGFß1 (c.466C>T, p.Arg156Cys, previously described in Camurati-Engelmann syndrome) and three had commonly found TGFß1 polymorphisms. Intravenous bisphosphonate therapy was used in all five patients but induced substantial improvements in a single patient. Of the three patients given bolus methylprednisolone therapy, two experienced a lasting response; the exception was one of the two women with a TGFß1 mutation. CONCLUSION: Considerable heterogeneity in the clinical presentations, genetic abnormalities, and treatment responses contribute to the diagnostic challenges raised by ribbing disease. Detailed genetic studies are needed.

Osteoma osteoide de rodilla simulando artritis psoriásica juvenil / Osteoid osteoma of the knee mimicking juvenile psoriatic arthritis

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Follow-up bone scintigraphy images of a patient with osteoid osteoma during medical treatment.

Osteoid osteoma is a benign bone tumour which is usually seen in long bones. Although the eventual treatment of this tumour is surgery, medical treatment is another option. We present the 3 years follow-up bone scintigraphy images of a patient with osteoid osteoma which show improvement by medical treatment.

Ribbing disease: a case report and literature review.

Ribbing disease (RD) is a rare bone dysplasia characterized by benign endosteal and periosteal new bone formation confined to the diaphysis of the long bones of the lower extremities in young adults. The etiology and optimal treatment for the disease are unknown. It is often initially diagnosed as a low-grade osteomyelitis or a bone-forming neoplasia. It may also be confused with other causes of increased bone density. The onset is usually after puberty and the most common presenting symptom is pain that does not resolve with medical treatment and sometimes is intolerable. We report the case of a 22-year old woman with clinical and radiological manifestations of RD. In spite of different medical treatment modalities, pain did not resolve and the patient consulted multiple physicians. Intramedullary reaming of the tibia was performed to relieve the severe pain. To the authors' knowledge, in this report we present a case of RD for the third time in the orthopaedic literature and also she is the second case in the English literature to undergo a definite surgical treatment modality as intramedullary reaming for the solution of her pain. Owing to the rarity of the disease we aimed to report the complete findings of our encounter with the disease and to emphasize the role of an orthopaedic surgeon in consultation and intervention for the treatment of intolerable pain which is the most important symptom of this disease.

Efecto del Etoricoxib en el dolor causado por el Osteoma Osteoide: descripción de un caso / Etoricoxib effect on pain caused by osteoid osteoma: description of a case

El osteoma osteoide es una tumoración ósea benigna frecuente cuya manifestación principal es el dolor nocturno que cede habitualmente con antinflamatorios no esteroideos (AINES). Presentamos el caso de una paciente, mujer de 23 años, con un osteoma osteoide en acetábulo izquierdo. Discutimos el manejo clínico y terapéutico de la paciente así como revisamos la bibliografía actual del tema

Osteoid Osteoma is a benign, common and bone-forming tumor whose main symptom is night pain that usually stop with nonsteroidal antiinflammatory drugs (NSAIDs). We present the case of a patient, 23 years old woman, with an Osteoid Osteoma in left acetabulum. The management and treatment are discussed and the current bibliography reviewed