Successful complete thoracoscopic resection of costal osteochondroma: A case report.

Osteochondroma rarely occurs in the ribs; therefore, the treatment is not standardized. There are few studies of resection via complete thoracoscopic surgery (CTS), although video-assisted thoracoscopic surgery with mini-thoracotomy has been reported. Herein, we report a case of costal osteochondroma managed with CTS. A 23-year-old woman presented to our hospital due to left chest pain. Chest computed tomography revealed a bone-like structure protruding into the thoracic cavity from the left fourth rib. Thus, surgery was performed to obtain a definitive diagnosis and provide appropriate treatment. The tumor was resected from the base at the border of the normal bone via CTS using three 5.5-mm ports. A pathological diagnosis of costal osteochondroma was made. The patient had an uneventful clinical course and did not present with a recurrence 1 year after surgery. Therefore, CTS can be a good approach for cases with slim and stalked costal osteochondroma.

Resection of the entire first rib for giant osteochondroma by trans manubrial approach: a case report and review of the literature.

BACKGROUND: First rib tumors are extremely rare. Its compression of neurovascularity can easily lead to severe complications such as thoracic outlet syndrome, so early surgical resection is crucial. However, there is no standardized approach to surgery. CASE PRESENTATION: A previously healthy 18-year-old Chinese male undergoes a chest computed tomography (CT) scan that incidentally reveals a raised calcified mass on the right first rib, which is most likely an osteochondroma when combined with magnetic resonance imaging (MRI). We achieved excellent results with resection and thoracic reconstruction by adopting an inverse L-shaped incision in the anterior chest and a longitudinal split of the sternum. CONCLUSIONS: Our practice provides great reference for the surgical management of first rib tumors.

Bizarre parosteal osteochondromatous proliferation (Nora lesion) involving the spine: a case report and systematic review.

Bizarre parosteal osteochondromatous proliferation (BPOP), also termed Nora lesion, is a rare, benign tumor most often located in the hands and feet. We herein present the second reported case of BPOP affecting the spine, an uncommon location. One year after surgical excision, the patient was pain-free and showed no evidence of recurrence. We reviewed a total of 323 cases of BPOP among 101 articles, providing the first systematic update on the latest knowledge of BPOP. The age of patients with BPOP ranges from 3 months to 87 years, peaking in the second and third decades of life. The hands are the most common location of BPOP (58.39%), followed by the feet (20.81%). Imaging features play a key role in the diagnosis of BPOP, but histopathologic diagnosis remains the gold standard. Differential diagnosis of BPOP should be based on the epidemiologic and clinical features as well as clinical examination findings. Surgical resection is the most extensively used treatment for BPOP. Recurrence is common (37.44%) and can be treated with re-excision. This article can deepen our understanding of BPOP and will be helpful for the diagnosis and treatment of BPOP in clinical practice.

Subungual Osteochondroma of the Great Toe: A Case Report.

Bony outgrowths of the distal phalanx of the great toe have been described in the literature but rarely. These subungual bony outgrowths can be caused by subungual exostosis or subungual osteochondromas. Both of these abnormalities are bony outgrowths with differences in the cartilage cap wherein the exostoses have fibrocartilage, and osteochondromas have hyaline cartilage. The subungual exostosis and osteochondroma that are protruding present symptoms of pain, redness, and deformed nail bed, whereas the nonprotruding osteochondromas have only a lump as the presenting symptom. In both conditions, excision of the lesion and curettage of the base helps prevent a recurrence. Curettage at the end of the excision of the bony outgrowth is required to avoid recurrence. After excision, the specimen should be sent for histopathologic examination to differentiate between the exostosis and osteochondromas, which are underreported in subungual locations, and to rule out malignant transformation. We present a 13-year-old girl with an isolated subungual nonprotruding exostosis of the great toe that was treated by excisional biopsy. The histopathologic examination confirmed it as osteochondroma, which is underreported.

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.

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.

Bizarre parosteal osteochondromatous proliferations of the temporal region: a case report.

Bizarre parosteal osteochondromatous proliferations (BPOPs) are distinct clinical-pathological entities that demonstrate combinations of atypical-appearing osseous and chondromatous tissues. These lesions are usually reactive in nature. Histopathologically, 'bizarre' cartilage is a characteristic feature of this lesion. BPOPs usually represent slow-growing painless bony hard protuberances that arise from the surface of affected bone cortices, typically the metacarpals, metatarsals, and phalanges. The occurrence of these lesions in the skull and jaws is sporadic. This case report highlights the clinical presentation, histopathological characteristics, and management of BPOP arising from the supraorbital rim in a 61-year-old female patient.

Recurrence of exostosis as a result of medication-induced bruxism: case study.

Introduction: Alveolar oral exostosis is a common, benign condition routinely found in dentistry. Clinical problems associated with exostoses are the maintenance of oral hygiene as well as the fabrication of prosthodontic appliances. Over time, exostoses may contribute to irritation and periodontal disease. Case description: The patient in this case study had a recurrence of exostoses and was bothered by consistent and prominent pain. She reported being a bruxer; her bruxism was exacerbated due to attention-deficit hyperactivity disorder and antidepressant medications. Discussion: The etiology behind the recurrence of exostosis is discussed. The most evident etiology seems to be persistence of medication-induced bruxism, specifically awake bruxism. Conclusion: It is necessary to take a proper history to identify the cause of the recurrence of exostosis. Dental hygienists can contribute to a better understanding of and provide better treatment options for patients who have medication-induced bruxism.

Introduction: L'exostose buccale alvéolaire est une affection bénigne courante couramment observée en dentisterie. Les problèmes cliniques associés aux exostoses sont le maintien de l'hygiène buccale ainsi que la fabrication d'appareils prosthodontiques. Avec le temps, les exostoses peuvent causer de l'irritation et des maladies parodontales. Description de cas: Dans cette étude de cas, la patiente présente des exostoses récurrentes et est dérangée par une douleur constante et proéminente. Elle a déclaré souffrir de bruxisme exacerbé par la prise de médicaments antidépresseurs et contre le trouble déficitaire de l'attention avec hyperactivité. Discussion: L'étiologie derrière la récurrence de l'exostose est abordée. L'étiologie la plus évidente semble être la persistance du bruxisme induit par les médicaments, en particulier le bruxisme diurne. Conclusion: Il est nécessaire d'obtenir les antécédents médicaux appropriés pour identifier la cause de la récurrence de l'exostose. Les hygiénistes dentaires peuvent contribuer à une meilleure compréhension et offrir de meilleures options de traitement aux patients atteints de bruxisme induit par les médicaments.

Secondary peripheral chondrosarcoma in multiple osteochondromas: a retrospective single-institution case series.

BACKGROUND: Multiple osteochondromas is genetic disorder characterized by the formation of multiple benign cartilage-capped bone tumors, named osteochondromas, during skeletal development. The most feared complication is the secondary peripheral chondrosarcoma, a malignant cartilaginous neoplasm that arises from the chondroid cap of pre-existent osteochondromas. We conducted a retrospective cohort study on patients diagnosed and followed up from 1960 to 2019 to describe the clinical and pathological features of individuals affected by peripheral chondrosarcoma in multiple osteochondromas, to evaluate follow up information and individual outcome and to compare the results with literature. Data, including age, gender, site, histological grade, cartilage cap thickness, surgical treatments, surgical margins, genotype mutational status as well as treatment details were captured from the hospital electronic health records and from Registry of Multiple Osteochondromas. In addition, a complete histological review of all hematoxylin and eosin (H&E)-stained sections has been performed by expert pathologists. RESULTS: One hundred five of the screened cases were included in the present study. The age at diagnosis of SPC ranges from 13 to 63, with median age at diagnosis of 34 years. The site most frequently affected by malignant degeneration was the pelvis (46 patients, 44%) with higher incidence in male patients (32 males vs.14 females). The second one was lower limbs (including femur, fibula, or tibia), identified in 35 patients. Histological information - available for 103 patients - showed: 59 patients with grade 1; 40 patients had a grade 2 and 4 patients had a grade 3. The most common surgical treatment was the complete resection, followed by debulking, amputation and partial resection. Most of cases did not have recurrence of the disease. Outcome in disease-free survival highlights that a worse course of the disease was associated with histological grade 2 or 3, and partial resection surgery. In most of analyzed cases (94%) a pathogenic variant was identified. CONCLUSIONS: In conclusion, the present study gives an overview of the secondary peripheral chondrosarcomas, confirming that this disease represents an impacting complication for multiple osteochondromas patients and suggests that malignant transformation can occur also in younger patient, in a not irrelevant number of cases.

Actualización de los tumores cartilaginosos según la clasificación de la OMS de 2020 / Update of cartilaginous tumours according to the WHO classification 2020

Los tumores cartilaginosos son un grupo amplio y heterogéneo de neoplasias caracterizadas por la presencia de una matriz condroide que presenta crecimiento lobular y patrones de calcificación en arcos y anillos o en palomitas de maíz. En RM destaca su hiperintensidad en las secuencias potenciadas en T2, y en las imágenes poscontraste, un relace lobulado o septal. En la clasificación de 2020 de la OMS, los tumores de estirpe condral se clasifican en benignos, intermedios o malignos. A pesar de los avances tecnológicos, siguen suponiendo un reto tanto para el radiólogo como para el patólogo, siendo la principal dificultad la diferenciación entre los tumores benignos y malignos, razón por la que requieren un abordaje multidisciplinar. Este trabajo recoge los principales cambios introducidos en la actualización de 2020, describe las características de imagen de los principales tumores cartilaginosos y proporciona las claves radiológicas para diferenciar entre tumores benignos y malignos.(AU)

Cartilaginous tumours are a large and heterogeneous group of neoplasms characterised by the presence of a chondroid matrix, with lobular growth and arcuate, ring-like or popcorn-like calcification patterns. MRI shows hyperintensity in T2-weighted sequences and a lobulated or septal relief in postcontrast images. In the WHO 2020 classification, chondral tumours are classified as benign, intermediate or malignant. Despite technological advances, they continue to pose a challenge for both the radiologist and the pathologist, being the main difficulty the differentiation between benign and malignant tumours, which is why they require a multidisciplinary approach. This paper describes the main changes introduced in the 2020 update, describes the imaging characteristics of the main cartilaginous tumours and provides the radiological keys to differentiate between benign and malignant tumours.(AU)

Medullary Compression by a Cervical Osteochondroma in a Patient with Multiple Hereditary Exostoses: A Case Report.

CASE: A 19-year-old man with Multiple Hereditary Exostoses presented with cervical pain without neurological symptoms and/or signs. Magnetic resonance revealed a large C2 osteochondroma, occupying a part of the medullary canal. He was submitted to an en bloc resection with hemilaminectomy without fusion. At the 1-year follow-up, he presented resolution of pain and no neurological symptoms or signs, without cervical instability or radiological signs of disease recurrence. CONCLUSION: Cervical osteochondroma is usually asymptomatic. Neurological compression and differentiation to chondrosarcoma are the main concerns. Surgical excision allows the local cure of the disease and is usually performed without fusion.

Pay Attention to the Osteochondromas in Fibrodysplasia Ossificans Progressiva.

BACKGROUND: Fibrodysplasia ossificans progressiva (FOP) is an extremely rare disease characterized by malformation of the bilateral great toes and progressive heterotopic ossification. The clinical features of FOP occur due to dysfunction of the bone morphogenetic protein (BMP) signaling pathway induced by the mutant activin A type I receptor/activin-like kinase-2 (ACVR1/ALK2) which contributes to the clinical features in FOP. Dysregulation of the BMP signaling pathway causes the development of osteochondroma. Poor awareness of the association between FOP and osteochondromas always results in misdiagnosis and unnecessary invasive operation. CASE PRESENTATION: In this study, we present a case of classical FOP involving osteochondroma. An 18-year-old male adolescent, born with deformity of bilateral big toes, complained multiple masses on his back for 1 year. The mass initially emerged with a tough texture and did not cause pain. It was misdiagnosed as an osteochondroma. After two surgeries, the masses became hard and spread around the entire back region. Meanwhile, extensive heterotopic ossification was observed around the back, neck, hip, knee, ribs, and mandible during follow-up. Osteochondromas were observed around the bilateral knees. No abnormalities were observed in the laboratory blood test results. Whole exome sequencing revealed missense mutation of ACVR1/ALK2 (c.617G > A; p.R206H) in the patient and confirmed the diagnosis of FOP. CONCLUSION: In summary, classical FOP always behaves as a bilateral deformity of the big toes, as well as progressive ectopic ossification and osteochondromas in the distal femur and proximal tibia. An understanding of the association between osteochondromas and FOP aids in diagnosis and avoids unnecessary invasive management in patients.

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.