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Periosteal ES rarely develops distant metastases. We report two patients of this rare ES location that were found on the distal tibial shaft and proximal femoral diaphysis of a 21-year-old man and an 8-year-old boy, respectively. Both patients were treated with neoadjuvant chemotherapy, wide resection, and adjuvant chemotherapy. One of our patients had lung metastases at the time of diagnosis and died 5 years later. The other patient presented intramedullary humeral bone metastasis 19 years after diagnosis. There has been no evidence of disease in the 26 years of follow-up. Close follow-up of periosteal ES is recommended because distant metastases may exceptionally occur, even several years after diagnosis.<\/jats:p>","DOI":"10.1177\/10668969241246473","type":"journal-article","created":{"date-parts":[[2024,5,6]],"date-time":"2024-05-06T00:55:03Z","timestamp":1714956903000},"page":"178-188","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":2,"title":["Periosteal Ewing Sarcoma with Distant Metastases: Report of Two Patients and Review of the Literature"],"prefix":"10.1177","volume":"33","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7113-045X","authenticated-orcid":false,"given":"Eva M.","family":"Pena-Burgos","sequence":"first","affiliation":[{"name":"Pr\u00edncipe de Asturias University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mar\u00eda C.","family":"D\u00edez-Corral","sequence":"additional","affiliation":[{"name":"Pr\u00edncipe de Asturias University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eduardo J.","family":"Ortiz-Cruz","sequence":"additional","affiliation":[{"name":"La Paz University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel","family":"Bernab\u00e9u","sequence":"additional","affiliation":[{"name":"La Paz University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mar","family":"Tapia-Vi\u00f1e","sequence":"additional","affiliation":[{"name":"La Paz University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andr\u00e9s","family":"Redondo","sequence":"additional","affiliation":[{"name":"La Paz University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antonio","family":"P\u00e9rez-Mart\u00ednez","sequence":"additional","affiliation":[{"name":"La Paz University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alberto","family":"Pel\u00e1ez","sequence":"additional","affiliation":[{"name":"La Paz University Hospital (IdiPAZ)"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Clara","family":"Venegas Mascar\u00f3","sequence":"additional","affiliation":[{"name":"Hospital La Paz Institute for Health Research - IdiPAZ"},{"name":"Hospital La Paz Institute for Health Research - IdiPAZ"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adela","family":"Escudero L\u00f3pez","sequence":"additional","affiliation":[{"name":"Hospital La Paz Institute for Health Research - IdiPAZ"},{"name":"Hospital La Paz Institute for Health Research - IdiPAZ"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jose J.","family":"Pozo-Kreilinger","sequence":"additional","affiliation":[{"name":"La Paz University Hospital"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2024,5,5]]},"reference":[{"key":"e_1_3_2_2_2","first-page":"323","volume-title":"WHO Classification of tumours of soft tissue and bone","author":"de \u00c1lava E","year":"2022","unstructured":"de \u00c1lava E, Lessnik S, Stamenkovic I. 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