Osteochondrolipoma of the foot treated by surgical excision: a case report and literature review.

BACKGROUND: Osteochondromas, classified as a new benign subtype of lipomas and characterised by chondroid and osseous differentiation, are rare lesions that have been infrequently reported in previous literature. The maxillofacial region was reported as the most frequent localization, with infrequent occurrence in the lower limb. This paper represents the first documented case report of osteochondrolipoma in the foot. CASE PRESENTATION: A 51-year-old male patient presented with a chief complaint of right foot pain at the plantar aspect, accompanied by the observation of swelling between the first and the second metatarsal shafts. His complaint of pain and swelling started 10 and 4 years prior, respectively. Since their onset, both symptoms have progressed in nature. Imaging revealved a large mass exhibiting a nonhomogenous composition of fibrous tissue and bony structures. Surgical intervention through total excision was indicated. CONCLUSION: Osteochodrolipoma is a benign lesion that can affect the foot leading to decreased functionality of the foot due to the pain and swelling. Surgical excision is the recommended approach for this lesion, providing both symptomatic relief and confirmation of the diagnosis through histopathological examination.

Effect of Solitary Osteochondroma on Alignment and Length in the Lower Extremities.

BACKGROUND: There is a lack of information about the effects of untreated solitary osteochondroma (SO) on longitudinal growth of the lower extremities in children and adolescents. This study aimed to assess the coronal alignment and length of the lower extremity in patients with SO around the knee and to identify the factors related to the development of deformities. METHODS: We retrospectively reviewed 111 patients diagnosed with SO around the knee. The patients were classified into 2 groups depending on the location of the SO: 51 in the distal femur and 60 in the proximal tibia. Characteristics of the lesions, such as type, location, size, and distance from the joint line, were determined. Radiographic analysis of the lower limbs included mechanical lateral distal femoral angle, mechanical medial proximal tibial angle, whole-leg length, femoral length, and tibial length. RESULTS: The mean age at the time of diagnosis was 12.3±3.4 years. No statistically significant differences were found between the affected and contralateral sides for mechanical lateral distal femoral angle and mechanical medial proximal tibial angle in either the distal femur or the proximal tibia groups. In patients with femoral lesions, the femoral and whole-leg lengths were significantly shorter on the affected side than on the unaffected side ( P <0.001 and 0.002, respectively), and the mean differences were 2.1±3.6 and 2.1±4.4 mm, respectively. Univariate logistic regression analysis did not reveal any factors associated with limb length discrepancy (LLD). In patients with tibial lesions, no statistically significant differences were found in LLD. CONCLUSIONS: SOs around the knee did not cause clinically significant deformity of the lower extremity. However, in contrast to proximal tibia lesions, SO in the distal femur was associated with the shortening of the affected limb. Consideration should be given to the development of LLD in skeletally immature children with SO in the distal femur. LEVEL OF EVIDENCE: Level III-retrospective comparative study.

BPOP in early childhood following resection of osteochondroma: report of a case.

The diagnosis of an osteochondroma in the short bones of the extremities is atypical and the presentation in infancy is unusual. A 3-month-old female presented for evaluation of radial deviation of the right index finger present since birth. Radiographs showed a broad-based osseous outgrowth with the usual features of an osteochondroma arising from the base of middle phalanx. Initial corrective surgery at 22 months was followed by recurrence of the lesion. Another resection at 4 years confirmed a final diagnosis of BPOP (bizarre parosteal osteochondromatous proliferation). The subsequent pathologic diagnosis of BPOP appears to support the hypotheses concerning the etiology of BPOP as possibly arising from repeated trauma to the metaphysis.

Benign Brain and Spinal Tumors Originating from Bone or Cartilage.

Benign osseocartilaginous tumors of the spine are overall uncommon, representing between 1 and 13% of all primary bone tumors and less than 10% of all spinal tumors. Tumors in this category include osteoblastic lesions such as the related osteoid osteoma and osteoblastoma, and cartilage-forming lesions including osteochondroma, chondroma, and chondroblastoma. Aneurysmal bone cysts, giant cell tumors of bone, and eosinophilic granulomas also comprise benign tumors of the spine arising from bone. There is significant heterogeneity in the epidemiology, molecular biology, imaging features, and optimal treatment of these lesions. For example, osteoid osteoma is characterized by high expression of the cyclooxygenase enzymes, making it amenable to treatment with anti-inflammatory drugs initially, whereas other lesions such as osteoblastoma may require intralesional curettage or en bloc resection sooner. Generally, en bloc resection is preferred when possible to minimize risk of recurrence. Further, some tumors may arise in the setting of syndromic conditions, such as multiple chondromas arising in Ollier disease or Maffucci syndrome, or as part of genetic disorders, such as osteochondromas in the context of hereditary multiple exostosis. These lesions may present with local pain, cause neurological compromise or be discovered incidentally on routine imaging. The Enneking classification and Weinstein-Boriani-Biagini system are routinely used to classify lesions and assist in surgical planning. More novel techniques such as radiofrequency ablation and laser photocoagulation have been applied for the treatment of osteoid osteoma and may have utility in the treatment of other lesion types. A multidisciplinary approach is critical in the management of benign lesions of the spine, and both chemotherapeutic and surgical approaches are routinely used.

Surgical Guides and Prebent Titanium Improve the Planning for the Treatment of Dentofacial Deformities Secondary to Condylar Osteochondroma.

PURPOSE: To investigate current Computer-Aided Design and Computer-Aided Manufacturing (CAD/CAM) technologies applied in the treatment of dentofacial deformities secondary to condylar osteochondroma and introduce a modified method with additional pre-bent titanium miniplates to improve the accuracy of operation. METHODS: Literature review about the application of CAD/CAM in the treatment of condylar osteochondroma and secondary dentofacial deformities was conducted. And 8 patients with condylar osteochondroma and secondary dentofacial deformities were treated by the CAD/CAM cutting and drilling surgical guides as well as pre-bent titanium miniplates. Pre- and post-operative 3D-cephalometric measurement were recorded and the difference between virtual simulation and postoperative modeling images was measured. Follow-up and radiographic examinations were performed. RESULTS: A total of 17 studies (including 216 patients) about the application of CAD/CAM in the treatment of dentofacial deformities secondary to condylar osteochondroma have been reported since 2010, including the 8 present patients. In our study, all patients were satisfied with the surgical outcome, without obvious relapse or evidence of temporomandibular joint disorder or other complications during follow-up; all patients avoided condylar reconstruction and sagittal split of ramus osteotomy on the ipsilateral mandible side. Comparison between simulated plans and actual postoperative outcomes showed surgical simulation plan was accurately transferred to the actual surgery. CONCLUSIONS: The application of CAD/CAM cutting and drilling guides as well as pre-bent titanium plates could achieve more accurate and favorable outcomes, improving the clinical planning and surgical execution for patients with condylar osteochondroma and secondary dentofacial deformities.

Simultaneous Management of Condylar Osteochondroma and Secondary Malformation With a New Intraoral Approach.

PURPOSE: The present study aimed to investigate the clinical efficacy of simultaneous management of condylar osteochondroma and its secondary dentofacial deformities using an intraoral surgical approach. METHODS: Six patients with condylar osteochondroma were treated with intraoral vertical ramus osteotomies and condylar resection. The free rising branch was used for reconstructing the temporomandibular joint. The simultaneous orthognathic surgery and plastic surgery were performed sequentially to correct the secondary dentofacial deformities. The indexes of aesthetic symmetry, occlusion relationship, temporomandibular joint function, condylar height, and volume change were assessed in the subsequential follow up. RESULTS: The mean follow up period was 31 months. All patients had no tumor recurrence. The ipsilateral joint function, occlusal relationship, and facial symmetry were satisfied. The ipsilateral condylar reconstruction had no obvious bone resorption and the ramus height was maintained well. Postoperative assessment showed the preoperative design was accurately fulfilled. CONCLUSIONS: The simultaneous condylar osteochondroma resection and temporomandibular joint reconstruction using intraoral approach avoids extraoral scars and correct facial asymmetry without compromising the long-term joint function and occlusal relationship.

False aneurysm of the popliteal artery revealing a solitary osteochondroma of the distal femur in an 11-year-old boy.

An 11-year-old boy presented with pain in the right knee, intermittent reverse ischemia of the right foot and paraesthesia of the right toes. An angio-CT showed a false aneurysm of the right superior popliteal artery, and a solitary osteochondroma of the posterior aspect of the distal femur. Excision of the aneurysm and the osteochondroma was performed in two-stages. The patient was clinically well at 1-year follow up.

Popliteal Artery Thrombosis in a Young Patient Secondary to Proximal Tibial Osteochondroma.

Popliteal artery occlusion is mainly seen in elderly patients with late stage atherosclerotic occlusive disease. In young, nonsmoking patients, popliteal artery occlusion can be caused by a variety of other etiologies. The diagnosis is suspected clinically and confirmed with ultrasound, computed tomography angiogram (CTA) or angiography, which can also aid in understanding the underlying cause. We present a 40-year-old very active male, who developed progressive symptoms of claudication over a 4 months interval and was found to have a thrombosed popliteal artery secondary to external compression from a tibial osteochondroma. The patient was treated with in-situ saphenous vein bypass from the above knee popliteal artery to the anterior tibial artery. The bypass was widely patent at 24 months.

Bizarre Parosteal Osteochondromatous Proliferation (Nora's Lesion) of Mandible With Recurrence: Case Report and Review of Literature.

Bizarre parosteal osteochondromatous proliferation (BPOP) is a rare benign lesion exhibiting radiographic and histologic features that can be mistaken for malignancy. Most cases have been reported in the small tubular bones of the hands and feet, but involvement of the skull and jaws is extremely rare. Here, we present a case of BPOP involving the mandible in a 23-year-old male that, after initial excision, recurred within 18 months. To the best of our knowledge, this is only the third published case of BPOP arising in the mandible.

An unusual example of hereditary multiple exostoses: a case report and review of the literature.

BACKGROUND: Hereditary multiple exostoses (HME) is a rare skeletal disorder characterised by a widespread. distribution of osteochondromas originating from the metaphyses of long bones. CASE PRESENTATION: This case study examines a 55-year-old male cadaver bequeathed to the University of Liverpool who suffered from HME, thus providing an exceptionally rare opportunity to examine the anatomical changes associated with this condition. CONCLUSIONS: Findings from imaging and dissection indicated that this was a severe case of HME in terms of the quantity and distribution of the osteochondromas and the number of synostoses present. In addition, the existence of enchondromas and the appearance of gaps within the trabeculae of affected bones make this a remarkable case. This study provides a comprehensive overview of the morbidity of the disease as well as adding to the growing evidence that diseases concerning benign cartilaginous tumours may be part of a spectrum rather than distinct entities.

Bizarre Parosteal Osteochondromatous Proliferation (Nora Lesion) in Pediatric Phalanges.

Bizarre parosteal osteochondromatous proliferation, or Nora tumor, is an uncommon lesion affecting the tubular bones of the hands and feet. Normally arising from the cortical surface and periosteum of these bones, these lesions histologically consist of a hypercellular cartilaginous cap covering a bony stalk that is surrounded by ossified areas and spindle cell stroma. The differential diagnosis includes conditions involving the periosteum such as chondrosarcoma, parosteal osteosarcoma, osteochondroma, turret exostosis, and florid reactive periostitis. The only effective treatment is wide surgical excision; nevertheless, local recurrence rates are extremely high and may necessitate revision surgery. In the present study, we report 3 cases of Nora lesion located in the hand in pediatric patients. The diagnosis in these cases was challenging owing to their presenting symptoms and radiographic findings. The diagnosis was made based on characteristic findings noted on the radiographic images and was confirmed by histological examination following excision.

Simultaneous Condylar Reconstruction by Free Ramus Osteotomy Graft After Complete Condylectomy for Osteochondroma.

ABSTRACT: Mandibular condyle osteochondromas cause morphologic and functional disturbances. Multiple options exist for reconstructing the condylar segment following complete condylectomy. In this series, we describe 3 cases of mandibular condyle osteochondroma treated with complete condylectomy, orthognathic surgery, and a novel free ramus osteotomy graft. This is the first report to reconstruct the temporomandibular joint using a free ramus graft. Through this single-staged approach we were able to avoid recurrence, preserve function, and restore facial balance without a separate donor site or an alloplastic implant.

Osteochondroma : the local effects and the management.

Osteochondroma is a common benign bone tumour, presenting as a single or multiple lesions, pedunculated or sessile type, mainly in 1st-2nd decades and mostly in males. The presented case series, studied the local effect of (osteochondroma) at the end of long bones with best line of treatment (RQ). Eighteen patients with osteochondromas, with an age range of 9- 45 years, mean age 19.6 ± 11.147 years, were seen from October 2016 to November 2018. There were 8 cases with painful lesion due to (mechanical interaction) or (pressure on near by neurovascular bundle), or from (inflamed bursa). Four cases were asymptomatic, 3 cases had Growth disturbances of the fellow bone, 3 cases presented with neurological symptoms and one case had vascular complications. Osteochondromas may present with different types of local effects on its surrounding tissues. Surgical excision is the best line of treatment.

Femoral Injury Over the Site of Spontaneous Regression of an Osteochondroma in a Teenage Athlete.

ABSTRACT: The spontaneous regression of an osteochondroma is extremely rare. We report a case of medial femoral condyle impaction fracture over the site of spontaneous regression of a pedunculated osteochondroma discovered on advanced imaging after an acute injury in a 16-year-old male American football athlete. Although spontaneous regression of an osteochondroma has been described, the case presented reveals questions regarding resultant architectural changes to the bone after resorption, leaving it prone to injury. This is the first case that describes increased injury risk potential at the site of osteochondral regression.

Bilateral Proximal Tibiofibular Synostosis Caused by Osteochondroma in a 21-Year-Old Highly Active Male-First in Literature.

Background and Objectives: Up until now, only one case of unilateral proximal tibiofibular synostosis caused by osteochondroma has been reported. This report is the first well-documented bilateral case of proximal tibiofibular synostosis caused by an osteochondroma. Case Report: A 21-year-old, highly active male patient with bilateral proximal tibiofibular synostosis caused by an osteochondroma suffering from persistent knee pain is presented. As conservative methods had failed, the patient was treated by bilateral open resection of the connecting bone. Histopathological findings confirmed the preoperative diagnosis. The patient returned to sports three weeks after surgery and continued soccer training six weeks after surgery. Discussion: The case report presents the successful treatment of a bilateral proximal tibiofibular synostosis caused by an osteochondroma by bilateral open resection of the connecting bone.

Trevor's disease of the distal radioulnar joint in two children with achondroplasia.

Two children with achondroplasia who developed an abnormal bony outgrowth at the distal radioulnar joint (DRUJ), indistinguishable from an osteochondroma on histology, but the radiographic appearance, location, and asymmetry suggested the rare diagnosis of dysplasia epiphysealis hemimelica (DEH or "Trevor's disease"). One child experienced symptomatic relief with surgical excision and one was observed clinically due to lack of significant symptoms. These are the first presented cases of DEH in achondroplasia, both affecting the DRUJ. Due to the infrequency of DEH, more research is needed to better understand the potential connection to achondroplasia. For management, we suggest shared surgical decision making based on symptoms.

Tarsal tunnel syndrome secondary to osteochondroma of the calcaneus: a case report.

BACKGROUND: Tarsal tunnel syndrome is an entrapment neuropathy that can be provoked by either intrinsic or extrinsic factors that compresses the posterior tibial nerve beneath the flexor retinaculum. Osteochondroma, the most common benign bone tumor, seldom occur in foot or ankle. This is a rare case of tarsal tunnel syndrome secondary to osteochondroma of the sustentaculum tali successfully treated with open surgical excision. CASE PRESENTATION: A 15-year-old male presented with the main complaint of burning pain and paresthesia on the medial plantar aspect of the forefoot to the middle foot region. Hard mass-like lesion was palpated on the posteroinferior aspect of the medial malleolus. On the radiological examination, 2.5 × 1 cm sized bony protuberance was found below the sustentaculum tali. Surgical decompression of the posterior tibial nerve was performed by complete excision of the bony mass connected to the sustentaculum tali. The excised mass was diagnosed to be osteochondroma on the histologic examination. After surgery, the pain was relieved immediately and hypoesthesia disappeared 3 months postoperatively. Physical examination and radiographic examination at 2-year follow up revealed that tarsal tunnel was completely decompressed without any evidence of complication or recurrence. CONCLUSIONS: As for tarsal tunnel syndrome secondary to the identifiable space occupying structure with a distinct neurologic symptom, we suggest complete surgical excision of the causative structure in an effort to effectively relieve symptoms and prevent recurrence.

Unsuspected osteochondroma-like outgrowths in the cranial base of Hereditary Multiple Exostoses patients and modeling and treatment with a BMP antagonist in mice.

Hereditary Multiple Exostoses (HME) is a rare pediatric disorder caused by loss-of-function mutations in the genes encoding the heparan sulfate (HS)-synthesizing enzymes EXT1 or EXT2. HME is characterized by formation of cartilaginous outgrowths-called osteochondromas- next to the growth plates of many axial and appendicular skeletal elements. Surprisingly, it is not known whether such tumors also form in endochondral elements of the craniofacial skeleton. Here, we carried out a retrospective analysis of cervical spine MRI and CT scans from 50 consecutive HME patients that included cranial skeletal images. Interestingly, nearly half of the patients displayed moderate defects or osteochondroma-like outgrowths in the cranial base and specifically in the clivus. In good correlation, osteochondromas developed in the cranial base of mutant Ext1f/f;Col2-CreER or Ext1f/f;Aggrecan-CreER mouse models of HME along the synchondrosis growth plates. Osteochondroma formation was preceded by phenotypic alteration of cells at the chondro-perichondrial boundary and was accompanied by ectopic expression of major cartilage matrix genes -collagen 2 and collagen X- within the growing ectopic masses. Because chondrogenesis requires bone morphogenetic protein (BMP) signaling, we asked whether osteochondroma formation could be blocked by a BMP signaling antagonist. Systemic administration with LDN-193189 effectively inhibited osteochondroma growth in conditional Ext1-mutant mice. In vitro studies with mouse embryo chondrogenic cells clarified the mechanisms of LDN-193189 action that turned out to include decreases in canonical BMP signaling pSMAD1/5/8 effectors but interestingly, concurrent increases in such anti-chondrogenic mechanisms as pERK1/2 and Chordin, Fgf9 and Fgf18 expression. Our study is the first to reveal that the cranial base can be affected in patients with HME and that osteochondroma formation is amenable to therapeutic drug intervention.

Evaluation of Mandibular Symmetry in Patients With Condylar Osteochondroma Who Underwent Intro-Oral Condylar Resection and Simultaneous Bimaxillary Orthognathic Surgery.

Mandibular condylar osteochondroma (OC) can result in facial asymmetry, malocclusion, and temporomandibular joint dysfunction. The authors have previously demonstrated a novel method for conservative condylectomy and simultaneous orthognathic surgery for treatment of mandibular condylar OC. The purpose of this study was to evaluate the immediate improvement and long-term stability of mandibular symmetry in the treatment of condylar OC. Fifty-six patients with unilateral mandibular condylar OC combined with secondary facial asymmetry and malocclusion were enrolled in this retrospective study. The computerized tomography (CT) scans were acquired with the mandible in centric relation (CR) before surgery, 1 week and 12 to 18 months after surgery. The images were reconstructed and processed for the analysis with ProPlan CMF 2.1 software. After defining the skeletal landmarks and the reference planes, the chin deviation, chin rotation and mandibular asymmetry index were calculated. The operations and healing were uneventful and the patients showed no signs of recurrence or temporomandibular joint ankylosis during the follow-up. Facial symmetry was greatly improved right after the surgery in the chin deviation (from 9.2 to 1.7 mm, P < 0.01), chin rotation (from 11.2 to 2.3, P < 0.01) and the asymmetry index of three mandibular landmarks (Go, MF and Sg, all P < 0.01). It also showed a stable result after 12 to 18 months follow-up. We also established a novel measurement method, which showed that the combination of conservative condylectomy via the intraoral approach based on intraoperative navigation and simultaneous orthognathic surgery is effective for improving the facial symmetry when treat the mandibular condylar OC.

A Rare Case of Tibioperoneal Arterial Trunk Entrapment Caused by a Fibular Osteochondroma.

BACKGROUND: Osteochondromas or exostoses are the most common benign tumors of the bones, which appear during the growth period. The involvement of lower extremities is common, particularly in metaphyseal structures of the femur and humerus and around the knee joint. However, the incidence of the development of the tumor at the proximal fibula is rare. The most common signs and symptoms of the disease are pain, pulsatile mass, limb swelling, neurologic sequelae, bursa formation with subsequent bursitis, and impairment of the developmental growth. Vascular complications are rare, yet osteochondromas can cause catastrophic arterial complications threatening the life of the patient if they occur and can include vessel perforation and thrombosis, arterial thromboembolic events, and pseudoaneurysm formation. METHODS: We report a case of a 24-year-old male patient presenting with tibioperoneal arterial trunk entrapment caused by a massive osteochondroma derived from the proximal fibula. A combined vascular-orthopedic approach was initiated with the excision of the tumor, in which the neurovascular structures (tibial nerve, popliteal artery and vein, anterior tibial artery, and tibioperoneal trunk) were carefully recognized and protected. RESULTS: The postoperative course of the patient was uneventful. CONCLUSIONS: High clinical suspicion of a vascular complication should be raised by physicians when investigating a young patient with a suspected osseous tumor in the popliteal fossa and symptomatology of concurrent peripheral arterial disease. Prompt surgical management is crucial for the salvage of the affected limb in cases of arterial involvement. Furthermore, quick surgical exposure of the mass and regular postoperative follow-up check minimizes the risk of irreparable impairments and tumor recurrence.