Neonatal osteoblastic tumor with a novel PTBP1::FOSB fusion.

Congenital/neonatal bone neoplasms are extremely rare. We present the case of a patient with a neonatal bone tumor of the fibula that had osteoblastic differentiation and a novel PTBP1::FOSB fusion. FOSB fusions are described in several different tumor types, including osteoid osteoma and osteoblastoma; however, these tumors typically present in the second or third decade of life, with case reports as young as 4 months of age. Our case expands the spectrum of congenital/neonatal bone lesions. The initial radiologic, histologic, and molecular findings supported the decision for close clinical follow-up rather than more aggressive intervention. Since the time of diagnosis, this tumor has undergone radiologic regression without treatment.

Osteoid Osteoma Mimicking Arthritis.

ABSTRACT: Intra-articular osteoid osteoma may simulate arthritis, due to the intra-articular presence of prostaglandin, which leads to synovitis, joint effusion, pain, and high local temperature. Also, intra-articular osteoid osteoma may present with minimum or no cortical thickening. Therefore, a high suspicion is needed for a correct and early diagnosis. Perfusion weighted imaging, such as dynamic contrast-enhanced imaging, can aid in the localization of the tumor nidus, seen as an early arterial-phase focal enhancement after the gadolinium injection, with fast washout, as a result of its hypervascularity.

Arthroscopic Excision of Intra-Articular Osteoid Osteoma of the Hip: A Case Series.

PURPOSE: To identify the clinical features of intra-articular osteoid osteoma (OO) of the hip, to evaluate the clinical effect of arthroscopic excision for intra-articular OO, and to summarize the characteristics of revision cases of hip OO and the revision surgery under arthroscopy in these cases. METHODS: We retrospectively reviewed the data of 25 patients who underwent arthroscopic excision of hip OO. The case series included 10 patients who underwent revision surgery. Lesion location, presenting symptoms, and symptom duration were analyzed; postoperative improvement was assessed using the modified Harris Hip Score (mHHS) and International Hip Outcomes Tool (iHot-12) score. We examined the reasons for revision surgery and the characteristics of OO progression after the first surgery. RESULTS: The most common presenting symptom was groin pain that was relieved by nonsteroidal anti-inflammatory drugs (NSAIDs). Varying degrees of limitation of range of motion (ROM) were present in all patients. The osteosclerosis around the tumor nest on computed tomography (CT) scan is a characteristic radiographic feature in this disease. However, the classic radiographic feature was apparent on plain x-rays in only 2 of 25 patients. As a kind of efficient radiological method, magnetic resonance imaging (MRI) can help in distinguishing OO from femoroacetabular impingement (FAI), as the latter is characterized by a large effusion and bone marrow edema at the atypical site of impingement. For the patients who had only 1 arthroscopic resection, the mean (± standard deviation) mHHS and iHot-12 scores were 70.30 ± 9.06 (range 51 to 86) and 75.07 ± 7.69 (57 to 88), respectively. At last follow-up, the mean scores were 98.30 ± 2.15 (94 to 100) and 97.76 ± 2.04 (94 to 100). For revision cases, the mean mHHS and iHot-12 scores were 68.55 ± 3.77 (60 to 72) and 67.88 ± 5.39 (56 to 76). At last follow-up, the mean scores were 97.11 ± 2.47 (94 to 100) and 95.22 ± 1.78 (94 to 100). In the present study, 24 of 25 patients (96%) reached the minimal clinically important difference (MCID) of mHHS, and 21 of 22 patients (95.2%) reached the MCID of iHot-12. Among the revision patients, the most common misdiagnosis at first surgery was FAI. Another feature is that a wrong diagnosis or incomplete intra-articular OO resection can stimulate the tumor and cause an inflammatory reaction and rapidly progressive OA, necessitating prompt revision surgery for complete removal. The degree of joint degeneration was related to the time since the first operation. CONCLUSION: OO of the hip joint typically presents with pain and limited joint activity. Misdiagnosis as FAI or synovitis is common, and CT scan is very helpful for accuracy diagnosis. Arthroscopic excision appears to be an effective method for the treatment of OO of the hip joint. LEVEL OF EVIDENCE: IV, case series.

CT-guided radiofrequency ablation for osteoid osteomas: a systematic review.

OBJECTIVES: CT-guided radiofrequency ablation (CT-RFA) is considered to be the gold standard for treatment of osteoid osteoma (OO) yet treatment failures (TFs) continue to be reported. This systematic review was conducted to evaluate factors associated with TF, such as ablation time, lesion location, and patient age as well as evaluating how TF has trended over time. METHODS: Original studies reporting on patients undergoing CT-RFA of OO published between 2002 and 2019 were identified. TF was defined as patients with (1) recurrent or persistent pain +/- (2) imaging evidence of persistent OO. TFs were subdivided into those occurring after the index procedure (primary TF) or those occurring after repeat RFA (secondary TF). Subgroup analysis was performed for TF based on the study date (2002-2010 or 2010-2019), time duration of ablation at 90 °C (6 min or > 6 min), patient age, and tumor location (spinal vs. appendicular). RESULTS: Sixty-nine studies were included for a total of 3023 patients. The global primary TF rate was 8.3% whereas the secondary TF rate was 3.1%. The TF rate reported in studies published after 2011(7%) was about half that during the earlier time period 2002-2010 (14%). There was no statistical difference in TF corrected for age, OO location, or duration of ablation (respectively p = 0.39, 0.13, and 0.23). The global complication rate was 3%, the most frequent being skin burns (n = 24; 0.7%). CONCLUSIONS: A decrease in TF observed between 2011-2019 compared to 2002-2010 may reflect improvements in operator technique or advancements in equipment. Duration of ablation, patient age, or location of OO failed to significantly correlate with TF. KEY POINTS: • CT-guided radiofrequency ablation of osteoid osteomas is a safe technique with a low rate of treatment failure (8.3% failure rate after the primary radiofrequency reducing to 3.1% following a secondary treatment). • The treatment failure rate has decreased over time, possibly due to an improved understanding of the disease process, better technique, and advances in equipment. • Duration of ablation, patient age, or lesion location did not significantly correlate with treatment failure.

Osteoid osteoma in older adults: clinical success rate of percutaneous image-guided thermal ablation.

AIM: To assess the clinical success rate of percutaneous radiofrequency and laser ablation of osteoid osteomas in older adults. MATERIALS AND METHODS: Percutaneous radiofrequency or laser ablation was performed in 43 patients (35 years and older) with osteoid osteoma to achieve definitive cure in this retrospective study. The clinical success rate was defined as complete pain relief determined by numeric rating scale (NRS) scores (pre-procedural and post-procedural at 1-week, 6-week, and 2-year intervals). Tumour characteristics, technical success, procedural details, biopsy results, and complications were documented. RESULTS: Forty-four osteoid osteomas were ablated in 43 patients, and all procedures were technically successful. The clinical success rates were 95.2% (41/43), 93% (40/43), and 93.2% (41/44) at 1-week, 6-week, and 2-year post-procedural intervals. The acute complication rate was 2.3% (1/44; meningeal perforation following epidural anaesthesia). No delayed complication was documented. CONCLUSION: Osteoid osteomas are not unique to the paediatric and young population, and safe and effective definitive treatment of these benign tumours in older adults can be achieved by percutaneous radiofrequency and laser ablation with excellent patient outcomes.

A thoracic pain of difficult diagnosis. Unusual localization of osteoid osteoma.

Osteoid Osteoma (OO) is a benign tumor that can affect any age, but it occurs mostly in adolescents. Only few cases are reported in early infancy but very rare in advanced age. From our series of OO of the spine, we selected a rare case that combines many unusual features that makes diagnosis very difficult. A case of a painful thoracic syndrome in an old female patient due to an OO localized in the inferior edge of the left pedicle of T11 with engagement of the foramen was reported. The age of the patient, the absence of any typical clinical and diagnostic signs, such as nocturnal pain or side effects to NSAIDs administration, are unusual at presentation of OO. She presented instead, a type of pain to the chest that was stabbing, fulminating and radiating. The interest of the case is due to the association of a variety of clinical aspects that stimulate discussion as well as to the role of the modern investigative diagnostic process.

Unusual localization and presentation of osteoid osteoma mimicking juvenile spondyloarthritis: a case report.

BACKGROUND: Osteoid osteoma is a painful benign skeletal tumour of unknown aetiology. Most often it occurs in the long bones of extremities and responds well to nonsteroidal anti-inflammatory medications. However, unusual localization and atypical presentation of this tumour might present a diagnostic challenge, especially if symptoms mimic that indicative of juvenile spondyloarthritis. CASE PRESENTATION: A misdiagnosed ten-and-a-half-year-old girl with osteoid osteoma involving the distal phalanx of a little finger is presented. Her initial symptoms were pain and swelling of the little finger resembling dactylitis, while various imaging modalities showed signs of tenosynovitis, indicating a possible development of juvenile spondyloarthritis. Several trials of different non-steroid anti-inflammatory drugs gave no satisfactory results and ultrasound guided triamcinolone-hexacetonide injection provided only a short relief. Finally, almost three years after initial presentation, persistent clinical symptoms warranted repeated imaging that raised suspicion of an osteoid osteoma. Directed treatment with surgical intervention led to almost immediate and complete resolution of her symptoms. CONCLUSIONS: Osteoid osteoma should be suspected in case of a tender swelling of a digit in children and adolescents, regardless of initial imaging findings and clinical presentation. Early diagnosis and treatment of this benign condition can have a substantial impact on quality of life of patients and their families and protect them from many unnecessary diagnostic procedures and treatment.

Multicentric, multifocal, and recurrent osteoid osteoma of the hip: first case report.

BACKGROUND: Osteoid osteoma is a benign bone-forming tumour, which very unfrequently has multifocal or multicentric presentation. We report the first known case of a multicentric, multifocal and recurrent osteoid osteoma treated using radiofrequency ablation. CASE PRESENTATION: A 39-year-old man with two-year history of left hip pain was admitted at our Institution. The pain was more intense during the night and partially relieved by salicylates. Pelvis CT demonstrated two lytic lesions (8 and 7 mm, respectively) with surrounding sclerotic reactive bone, both with a central focal area of high attenuation, located in the femoral neck and along the anterior portion of the acetabulum, respectively. Both lesions had clinical and imaging findings consistent with multicentric osteoid osteoma. Thus, the two lesions were biopsied - with pathologic confirmation of osteoid osteoma - and treated using radiofrequency ablation. Hip pain decreased but did not disappear, actually increasing a few months after treatment. CT and MRI were performed showing a smaller lesion (5 mm) with the same imaging features, surrounded by marrow oedema, along the posterior column of the acetabulum. The lesion was considered suspicious for osteoid osteoma, overlooked on previous examinations. Therefore, a diagnosis of multicentric and multifocal osteoid osteoma was established. The new lesion was again treated with radiofrequency ablation with symptom disappearance. However, hip pain relapsed after 18 months, and CT and MRI showed an osteoid osteoma recurrence on the posterior column of the acetabulum, which was biopsied and successfully treated using radiofrequency ablation. CONCLUSIONS: To our knowledge, this is the first reported case of multicentric, multifocal, recurrent osteoid osteoma. Our case report highlights the importance of considering a diagnosis of multifocal osteoid osteoma when dealing with multifocal lytic lesions of the bone and with pain persistence after treatment. It also emphasises the combined role of CT and MRI in this setting.

Osteoid Osteoma in the Thumb of an Adolescent Patient.

Osteoid osteoma is a relatively common benign tumor of bone, typically presenting in the diaphysis of long bones during the second or third decades of life. This tumor is rarely reported in the hand and wrist, making up only approximately 10% of cases. When reported in the hand, osteoid osteoma tends to occur more frequently in the proximal phalanx of the index and middle fingers. We present the case of an osteoid osteoma in an adolescent male in the distal phalanx of the thumb. The presentation of this osteoid osteoma was atypical owing to its location and lack of characteristic clinical features, making the initial work-up and final diagnosis challenging and pointing to the importance of considering this diagnosis on a differential for painful bony tumors in the hand.

[Osteoid Osteoma of the Upper Extremity]. / Osteoidní osteom v oblasti horní koncetiny.

PURPOSE OF THE STUDY Osteoid osteoma (OO) of the upper extremity is a rare pathology representing a major diagnostic challenge. The patients are often times incorrectly treated due to a misdiagnosis and therefore the final management of symptoms is delayed by several months. During this time the pain, which is the main symptom, increases. The surgical treatment is a fast and efficient solution. This paper aims to highlight the specificity of the OO of the upper extremity, evaluate our study group, the time to diagnosis and the benefits of individual imaging techniques. MATERIAL AND METHODS In the period 2007-2017, a total of 8 patients with a histologically verified OO of the upper extremity were consulted and treated at the authors departments. The group of patients was retrospectively evaluated based on the medical reports, surgical protocols, imaging examinations, histology results of preoperative samples and a postoperative questionnaire. Pain was assessed on the Visual Analogue Scale (VAS) preoperatively, at 2 weeks, 2 months and at one year postoperatively and in January 2018. The mean follow-up period was 48 months (range 16-78). The presence of swelling, synovitis and a limited range of motion were clinically assessed. The removed tissue was tested in an accredited biopsy laboratory. RESULTS 8 patients with OO of the elbow, wrist and hand were treated in the period 2007-2017. In 5 cases the OO was located in the wrist (scaphoid, 2x trapezoid, hamate and capitate) and in 2 cases in finger phalanges (middle and distal phalanx). The mean age of the patients was 24.5 years (median 21.5, range 18-42). The male to female ratio was 7:1. The mean interval between the onset of symptoms and the final diagnosis was 16.25 months (median 15.5, range 12-25). The CT examination proved to be the most beneficial imaging techniques. A plain radiograph led to a diagnosis in 1 case only. The preoperative mean pain VAS value 9.25 dropped to VAS 1 at two weeks after the surgery. No recurrence was reported. DISCUSSION The main challenge in treating the OO of the upper extremity is the diagnosis. Despite of the availability of advanced imaging techniques, the interval from the onset of symptoms to correct diagnosis did not change over the past decades. As to the treatment, despite the development of new procedures the method of choice continues to be the surgical excochleation. CONCLUSIONS Osteoid osteoma of the upper extremity is a rare pathology which shall be considered in case of an increasing pain, especially in young men. A plain radiograph of the anatomically complex terrain of the hand and wrist is mostly not useful in diagnosis, whereas the CT examination can visualise even a small size nidus. The surgical excochleation results in pain relief and early functional recovery with swelling disappearance. To diagnose the OO is not difficult provided we take it into consideration. Key words:osteoid osteoma, upper extremity, hand, wrist, diagnosis.

Cystic bone tumors of the foot and ankle.

Bone tumors are relatively rare in the foot and ankle region. Many of them present as cystic lesions on plain films. Due to the relative rarity of these lesions and the complex anatomy of the foot and ankle region, identification of such lesions is often delayed or they get misdiagnosed and mismanaged. This review discusses the most common cystic tumors of the foot and ankle including their radiographic features and principles of management.

[Osteoid osteoma in the scaphoid bone as cause of radiocarpal pain in a 15-year-old patient]. / Osteoidosteom im Os scaphoideum als Ursache für radiokarpale Handgelenksbeschwerden bei einem 15-jährigen Patienten.

Osteoid osteomas are benign bone tumors which rarely occur in the hand and impose severe diagnostic problems. The course of the disease is often protracted before the patient receives an adequate surgical treatment. The case of an osteoid osteoma in the scaphoid bone of a 15-year-old patient is presented, who was completely symptom free after a true diagnostic odyssey by resection of the nidus and reconstruction by crest bone graft and spongiosaplasty.

Foot and Ankle Osteoid Osteomas.

Foot and ankle osteoid osteomas (OOs) are often cancellous or subperiosteal and rarely present with a periosteal reaction. Additionally, the large number of disorders included in the differential diagnosis and the nonspecific findings on radiographs complicate the diagnosis. We performed a manual search of the senior surgeon's hospitals' operating room records for the terms "benign bone tumor," "foot," "ankle," and "osteoid osteoma" from January 2003 until December 2014. Of 87 surgically treated patients with lower extremity OOs, 9 patients (11%) with foot or ankle OOs were included. The mean age at presentation was 21 (range 6 to 30) years; all 9 (11%) patients were male. The patients were evaluated for swelling, pain, trauma history, night pain, response to pain relievers, duration of complaints, and interval to diagnosis. The mean follow-up period was 48 ± 24 months, and no recurrences had developed. The mean American Orthopaedic Foot and Ankle Society scale score was 59.04 ± 11 before surgery and 91.56 ± 6 after surgery. The difference was statistically significant at p ≤ .0003. Most previous studies have been limited to case reports. The need for findings from a case series was an essential determinant of our decision to report our results. Patients usually have been treated conservatively, often for a long period. However, delays in treatment cause social, economic, and psychological damage. In conclusion, the presence of atypical findings on radiographs has resulted in a preference for magnetic resonance imaging instead of computed tomography; however, the diffuse soft tissue edema observed on MRI can lead to the use of long-term immobilization and a delay in the diagnosis.

Osteoid osteoma of the temporal bone manifesting as first bite syndrome and a meta-analysis combined with osteoblastoma.

The objectives of this study were to describe a case of osteoid osteoma arising from the temporal bone manifesting only as first bite syndrome as the sole clinical symptom, to perform a meta-analysis of previously reported cases, and to differentiate the clinical characteristics of osteoid osteoma from those of osteoblastoma arising from the temporal bone. In addition to our case, articles addressing osteoid osteoma or osteoblastoma arising from the temporal bone were selected using PubMed, Embase, and the Japan Medical Abstracts Society database (1954 through 2014), with no language preference. The database was searched using the keywords ["osteoid osteoma" OR "osteoblastoma" AND "temporal bone"]. After critical review of 88 studies, 10 cases of osteoid osteoma and 29 cases of osteoblastoma were selected; therefore, including the present case, a total of 40 cases were eligible for qualitative analyses. The mean size of osteoid osteoma was 1.2 cm, which was significantly smaller than that of osteoblastoma (5.1 cm). Radiologically, osteoid osteoma was associated with a lower prevalence of extension into more than two anatomically categorized spaces in comparison with osteoblastoma (P < 0.01). Again, a lower prevalence of erosion of the outer and/or inner tables of the skull in the osteoid osteoma cases was noted (P < 0.05). Conversely, there were no significant differences in the prevalence of representative clinical symptoms, including pain and swelling. According to the present systematic review, osteoid osteoma and osteoblastoma are clinically uniform other than their size or extension.

Unusual cause of the thumb basal joint pain: osteoid osteoma of the trapezium.

The trapezium is rare site of osteoid osteoma development. The diagnostic challenge lies in its rare occurrence, and requires differentiation from various disease entities causing thumb basal joint pain. We report the case of a 29-year-old male who presented with severe thumb basal joint pain. He was initially treated for calcific periarthritis because of concomitant calcifications around the thumb basal joint, but had undiscovered osteoid osteoma. A high index of suspicion to a patient with wrist pain unresponsive to prior treatment is necessary for diagnosis of osteoid osteoma.

[Diagnostics and treatment of osteoid osteoma]. / Diagnostik und Therapie des Osteoidosteoms.

BACKGROUND: Osteoid osteoma is the third most common benign bone tumor and typically induces pain that is worse at night. OBJECTIVE: To identify the epidemiological, pathogenetic, histological and radiological characteristics of osteoid osteoma and to present the broad variety of treatment options. MATERIAL AND METHODS: This review article summarizes relevant clinical studies and meta-analyses on this topic. RESULTS: Osteoid osteoma is characterized by a central nidus smaller than 1.5 cm in diameter with surrounding bone sclerosis. In the majority of cases, the tumor occurs in the long bones of the lower extremities and is predominantly manifested in patients aged between 5 and 25 years. Pain is mediated by prostaglandins, which stimulate afferent peripheral nerve fibers. Besides plain radiographs, thin-section computed tomography represents the gold standard of diagnostics but should be complemented by magnetic resonance or nuclear medicine imaging modalities. The conservative treatment consists of long-term therapy with non-steroidal anti-inflammatory drugs. Minimally invasive radiofrequency ablation of the nidus is the current operative treatment of choice. CONCLUSION: Success rates of radiofrequency ablation and other minimally invasive procedures are high while treatment costs and length of hospital stay are low. Thus, open surgical curettage is reserved for rare indications and en bloc excision of the nidus should only be performed in cases of recurrent lesions.

Intra-articular osteoid osteoma as a differential diagnosis of diffuse mono-articular joint pain.

BACKGROUND: The aim of this retrospective study was to investigate the frequency of intra-articular osteoid osteoma (iaOO) in a large study cohort and to demonstrate its clinical relevance as an important differential diagnosis of non-specific mono-articular joint pain. METHODS: We searched the registry for bone tumours of the University Medical Centre Hamburg-Eppendorf for osteoid osteomas in the last 42 years. Herein, we present three selected iaOO which were detected in the three major weight-bearing joints. Computed tomography (CT) or magnetic resonance imaging (MRI) scans were performed for initial diagnosis. RESULTS: Out of a total of 367 osteoid osteomas, 19 (5.2 %) tumours were localized intra-articularly. In all three presented tumours, a history of severe mono-articular pain was reported; however, the mean time to correct diagnosis was delayed to 20.7 months. Clearly, the nidus seen in CT and MRI images in combination with inconsistent salicylate-responsive nocturnal pain led to the diagnosis of iaOO. CONCLUSIONS: Rarely, osteoid osteoma can occur in an intra-articular location. In cases of diffuse mono-articular pain, iaOO should be considered both in large and smaller joints to avoid delays in diagnosis and therapy of this benign bone tumour.

Osteoid osteoma mimicking monoarticular juvenile idiopathic arthritis in a girl.

Osteoid osteoma (OO) is a benign osteogenic neoplasm, usually affecting children and young adults, that is typically characterized by nocturnal pain and response to non-steroidal anti-inflammatory drugs. OO is frequently misdiagnosed because it mimics juvenile idiopathic arthritis (JIA), bone infection or malignancy. Herein we report the case of a girl who presented with chronic monoarthritis of the knee mimicking JIA. After 1 year, OO of the femoral distal metaphysis was diagnosed. OO was treated with computed tomography-guided radiofrequency ablation with disappearance of the symptoms and resolution of the neoplasm. No recurrences have been observed 3 years after the treatment. This case highlights that intra-articular or juxta-articular OO should be suspected in the case of misleading symptoms and signs, such as swelling, lack of typical pain and synovial thickening on ultrasound; needle biopsy of the lesion is necessary in the case of confusing imaging.

Piezosurgery for Excision of Large Osteoid Osteoma.

Osteoid osteoma, a rare benign osteoblastic tumor first described by Jaffe in 1935, is characterized as a small but painful lesion that mostly affects younger people. Usually benign and harmless, osteomas are removed for pain or esthetic reasons.Piezoelectric surgery is also increasingly being used effectively in major and minor osseous oral and maxillofacial surgeries, in delicate areas. It is used regularly for various procedures, including sinus lift procedures, bone graft harvesting, osteogenic distraction, ridge expansion, inferior alveolar nerve decompression and lateralization, cyst removal, dental extraction, and impacted tooth removal.The following report presents a patient of intraoral excision of a large osteoid osteoma from lingual aspect of mandibular lower border in the body region using piezoelectric surgery.

[The intraarticular osteoid osteoma : A diagnostic and therapeutic challenge]. / Das intraartikuläre Osteoidosteom : Eine diagnostische wie therapeutische Herausforderung.

Intra- and, respectively, periarticular osteoid osteoma are accompanied by sympathical arthritis which itself can irreversibly destroy the cartilage of the joint. In contrast to other locations, intra- and periarticular osteoid osteoma are rare. Reactive and symptomatic accompanying arthritis may lead to irreversible cartilage destruction especially in chronic courses. Therefore early diagnosis and therapy is crucial. We present a case report and discuss these rare tumors by reviewing the literature.