Pulmonary Lymphangitic Sarcomatosis of Primitive Neuroectodermal/Ewing's Sarcoma

Mirant-Borde, Maria; Colón‐Otero, Gerardo; Menke, David M.; Lee, Augustine

doi:10.1378/chest.1118379

Cited by 6 publications

(7 citation statements)

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“…Such spread shares similar characteristics to PLC, but given the origin of malignancy, it is described as pulmonary lymphangitic sarcomatosis (PLS). [3][4][5] To our knowledge, no case of PLS from ES in a child has been reported, and only 1 such case has been reported in an adult, presenting in a 67year-old male. 5 Metastatic pulmonary lymphangitic infiltration is best evaluated with computerized tomography scans and classically appears as nodular interlobular septal thickening with preservation of secondary pulmonary lobule architecture.…”

Section: Discussionmentioning

confidence: 96%

“…Metastatic spread of sarcoma is typically hematologic 2 ; however there have been a few case reports describing lymphangitic spread. Such spread shares similar characteristics to PLC, but given the origin of malignancy, it is described as pulmonary lymphangitic sarcomatosis (PLS) 3–5 . To our knowledge, no case of PLS from ES in a child has been reported, and only 1 such case has been reported in an adult, presenting in a 67-year-old male 5 …”

Section: Discussionmentioning

confidence: 98%

“…PLS has a poor prognosis, with a 50% survival within 2 months following the onset of respiratory symptoms 3 . Given the scarcity of reported PLS cases, diagnosis may be delayed, and treatment recommendations are limited, though chemotherapy has shown to have modest survival benefits 3–5 …”

Section: Discussionmentioning

confidence: 99%

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Aggressive Presentation of Ewing Sarcoma Recurrence Demonstrates Rare Pattern of Pulmonary Lymphangitic Carcinomatosis

Mack,

Swenson,

Steele

et al. 2023

Journal of Pediatric Hematology/Oncology

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A 16-year-old male presented with a 3-month history of a right groin mass. Diagnostic workup, including MRI, revealed a mass involving the right femur. Subsequent biopsy confirmed Ewing-like sarcoma. Staging scans of the chest at that time were negative for metastatic disease. He was treated with interval compressed chemotherapy, including Vincristine, Doxorubicin, Cyclophosphamide, Ifosfamide, and Etoposide, as well as local surgical resection. Surgical margins were widely positive, and as such, he was treated with adjuvant radiation therapy. Two years following treatment, surveillance scans were found to have suspicious lung nodules, which were subsequently resected. Biopsy results revealed BCOR+, undifferentiated spindle cell sarcoma. Despite treatment with chemotherapy and radiation, his disease continued to progress over the following 3 years with worsening pulmonary metastatic burden complicated by pneumothorax and eventual recurrence in the right groin. His care was transitioned to palliative radiation for pain control. Repeat staging exams revealed a new pattern of lymphangitic spread of disease in the lungs consistent with pulmonary lymphangitic carcinomatosis (PLC), as seen in Fig. 1. The patient was later transitioned to hospice care and expired 1 month following the new imaging findings.

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Section: Discussionmentioning

confidence: 96%

Section: Discussionmentioning

confidence: 98%

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Aggressive Presentation of Ewing Sarcoma Recurrence Demonstrates Rare Pattern of Pulmonary Lymphangitic Carcinomatosis

Mack,

Swenson,

Steele

et al. 2023

Journal of Pediatric Hematology/Oncology

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“…According to a previous report, pulmonary lymphangitic carcinomatosis (PLC) most often coexists with adenocarcinoma (80%), while PLC induced by sarcomas are rare (3.6%) [2] . To the best of our knowledge, three previous reports have described the CT findings of the patients with PLS in detail; two patients with angiosarcoma, one with neuroectodermal/Ewing's sarcoma [ 1 , 3 , 4 ]. These findings included uneven thickenings of the interlobular septa and bronchovascular bundle, which were similar to the CT findings of PLC.…”

Section: Discussionmentioning

confidence: 99%

Pulmonary lymphatic involvement in metastatic uterine sarcomas: imaging and pathological appearance

Kitayama

Tanaka

Hiraki

et al. 2021

Radiology Case Reports

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Pulmonary lymphatic involvement of sarcomas is an extremely rare form of metastases. We report the computed tomography (CT) features of pathologically confirmed pulmonary lymphatic involvement from metastatic uterine sarcomas. The CT illustrated smooth or nodular thickenings of the interlobular septa and bronchovascular bundle. Moreover, ground-glass opacity along the interlobular septa was also detected. These findings suggest that lymphatic involvement has diagnostic value for detecting this rare form of metastatic sarcomas. We also discuss possible differential diagnoses in this case and review previous cases reporting pulmonary lymphatic involvement in metastatic sarcomas. To the best of our knowledge, this is the first report describing pulmonary lymphatic involvement in metastatic uterine sarcomas. Pulmonary lymphatic spread of sarcomas is a rare form of metastatic sarcomas, but it should be considered when these findings suggesting lymphatic involvement are detected on CT.

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“…This is a very aggressive neoplasm with poor prognosis depending on clinical metastasis being blood dissemination the most frequent route of metastases to the lungs and bone marrow. 14…”

Section: Discussionmentioning

confidence: 99%

Metastatic pulmonary dissemination as differential diagnosis of COVID-19 disease

et al. 2020

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A 13-year-old boy presented to hospital with 3-day self-limited fever, followed by dry cough, persistent asthenia and impaired general condition of 2 weeks’ duration. Blood analyses showed a severe inflammatory status and chest X-ray images were consistent with bilateral COVID-19 pneumonia. He developed an acute respiratory failure that required paediatric intensive care admission and non-invasive ventilation. A targeted COVID-19 treatment was initiated with hydroxicloroquine, corticosteroids, enoxaparine and a single dose of tocilizumab. Repeated serological tests and real-time reverse transcription PCR for SARS-CoV-2 were negative. Other infectious pathogens were also ruled out. Thoracic high resolution CT showed an intense bilateral pulmonary dissemination with lytic vertebral bone lesions. After diagnostic investigations, Ewing’s sarcoma with metastatic pulmonary dissemination was diagnosed. Nowadays, in the context of SARS-CoV-2 community pandemic, we cannot forget that COVID-19 clinical presentation is not specific and other entities can mimic its clinical features.

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Pulmonary Lymphangitic Sarcomatosis of Primitive Neuroectodermal/Ewing's Sarcoma

Cited by 6 publications

References 0 publications

Aggressive Presentation of Ewing Sarcoma Recurrence Demonstrates Rare Pattern of Pulmonary Lymphangitic Carcinomatosis

Aggressive Presentation of Ewing Sarcoma Recurrence Demonstrates Rare Pattern of Pulmonary Lymphangitic Carcinomatosis

Pulmonary lymphatic involvement in metastatic uterine sarcomas: imaging and pathological appearance

Metastatic pulmonary dissemination as differential diagnosis of COVID-19 disease

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