Unusual Case of Extrapulmonary Metastatic Recurrence in a Patient With Osteosarcoma

Osteosarcoma (OS) is the most common nonhematolymphoid primary bone malignancy characterized by osteoid or new bone formation. Lungs and bones are the most common sites of metastases. We report a case where unusual sites of the soft tissue recurrence from OS were detected on restaging fluorine-18 fluorodeoxyglucose positron emission tomography/computed tomography scan done post 6 years of disease free interval.

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“…Patients with pulmonary recurrence have a better prognosis as compared to those with extrapulmonary involvement. [ 3 4 5 6 ]…”

Section: Discussionmentioning

confidence: 99%

Unusual sites of metastatic recurrence of osteosarcoma detected on fluorine-18 fluorodeoxyglucose positron emission tomography/computed tomography

et al. 2015

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“…Osteosarcoma had been pathologically divided into three groups depending on the predominant type of matrix (osteoid, fibrous or chondroid matrix) [1]. The presence of mineralized osteoid matrix may explain the osteosarcoma tissue affinity for 99m Tc-MDP.…”

Section: Discussionmentioning

confidence: 99%

“…Sometimes however, its role can be extended to encompass soft tissue status in certain tumors like osteosarcoma. Unusual sites of soft tissue metastasis had been reported in osteosarcoma including the pleura, pericardium, kidney, adrenal glands and nonregional lymph nodes [1]. These metastases are normally characterized with positive 99m Tc methylene diphosphonate (MDP) uptake.…”

Section: Introductionmentioning

confidence: 99%

Radionuclide Detection of Multiple Soft Tissue Metastases of Osteosarcoma Masquerading as Bone Metastasis

Takesh

Zoller

Ghazi³

et al. 2012

Med Princ Pract

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Objective: The aim of this work was to report an unusual case of multiple soft tissue metastases of osteosarcoma detected using bone scan and confirmed by CT scan. Clinical Presentation and Intervention: A 15-year-old patient with a history of osteogenic sarcoma presented with fatigue and loss of appetite in addition to a painless swelling of the distal left lower extremity. Bone scan and chest CT scan were performed for restaging purposes. The bone scan revealed multiple bone metastases and suspected tumor recurrence in the left lower extremity in addition to multiple extraosseous hot spots. The latter findings were assessed as ossified soft tissue metastases after considering the performed CT scan. Conclusion: This case showed the double role of bone scan in osteosarcoma in simultaneously assessing the bone status and tumor spread in soft tissue. The benefit of a comparison of bone scan findings with other modalities was also demonstrated.

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“…At the time of diagnosis, approximately 15% to 20% of patients have clinically detectable metastases and the 5-year overall survival rate of those patients is usually less than 30% [3][4][5][6][7]. The majority of osteosarcoma metastases occurre in the lung, while metastases to the extrapulmonary locations have been rarely reported, such as breast, abdominal viscera, chest wall, heart, penis, brain, subcutaneous tissue, skeletal muscle [8][9][10][11][12][13][14][15]. Among these extrapulmonary involvement, skeletal muscle metastases are extremity rare which account for only 1.6% of patients with metastases from osteosarcoma and there are only several relevant case reports that have been published to date [16].…”

Section: Introductionmentioning

confidence: 99%

Synchronous lung and multiple soft tissue metastases developed from osteosarcoma of tibia: a rare case report and genetic profile analysis

Zheng

Wang

Luo

et al. 2022

BMC Musculoskelet Disord

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Background Osteosarcoma is the most common primary malignant bone tumor with a highly metastatic propensity in children and young adolescents. The majority of metastases develope in the lung, while metastases to the extrapulmonary locations have rarely been discussed, especially in skeletal muscle. Case presentation We reported a young patient with pathologically diagnosed osteosarcoma of the right tibia who was initially treated with standard chemotherapy and complete surgical resection. However, pulmonary metastases and multiple soft tissue masses in skeletal muscle developed four years after the index surgical resection. Subsequently, a targeted next-generation sequencing assay based on an 806 oncogenes and tumor suppressor genes panel was performed to analyze genetic alterations in this patient with rare metastatic pattern. The genetic analysis revealed canonical somatic mutations of RB1 and germline variants of ALK (c.862 T > C), BLM (c.1021C > T), PTCH1 (c.152_154del), MSH2 (c.14C > A), RAD51C (c.635G > A). Using silico prediction programs, the germline variants of the MSH2 and RAD51C were predicted as “Possibly Damaging” by Polymorphism Phenotyping v2 (PolyPhen-2) and “Tolerated” by Sorting Intolerant from Tolerant (SIFT); BLM was classified as “Tolerated”, while the germline variant of ALK was predicted to be pathogenic by both PolyPhen-2 and SIFT. Conclusions Osteosarcoma with extrapulmonary metastases is rare, especially located in the skeletal muscle, which predicts a worse clinical outcome compared with lung-only metastases. The several novel variants of ALK, BLM, PTCH1 in this patient might expand the mutational spectrums of the osteosarcoma. All the results may contribute to a better understanding of the clinical course and genetic characteristics of osteosarcoma patients with metastasis.

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Unusual Case of Extrapulmonary Metastatic Recurrence in a Patient With Osteosarcoma

Cited by 5 publications

References 15 publications

Unusual sites of metastatic recurrence of osteosarcoma detected on fluorine-18 fluorodeoxyglucose positron emission tomography/computed tomography

Unusual sites of metastatic recurrence of osteosarcoma detected on fluorine-18 fluorodeoxyglucose positron emission tomography/computed tomography

Radionuclide Detection of Multiple Soft Tissue Metastases of Osteosarcoma Masquerading as Bone Metastasis

Synchronous lung and multiple soft tissue metastases developed from osteosarcoma of tibia: a rare case report and genetic profile analysis

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