Local therapy to distant metastatic sites in stage IV rhabdomyosarcoma

Mohan, Arvind; Venkatramani, Rajkumar; Okcu, M. Fatih; Nuchtern, Jed G.; Vasudevan, Sanjeev A.; Mahajan, Anita; Rainusso, Nino; Allen‐Rhoades, Wendy; Chintagumpala, Murali; Paulino, Arnold C.

doi:10.1002/pbc.26859

Cited by 26 publications

(26 citation statements)

References 13 publications

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“…For instance, Texas Children’s Hospital analysis of local therapy to metastases in stage IV rhabdomyosarcoma revealed a LC of 73% at a median follow-up of 2 years after irradiation of nonlung or nonbone marrow metastatic sites. 12 The majority of lesions in the above series received a total of 50.4 Gy in 28 fractions. Similarly, Kandula et al studied outcomes after irradiation of metastatic sites in patients with stage IV neuroblastoma and observed 23% local failures in 13 metastatic sites treated with at least 12 fractions of conventional RT to a median dose of 21.6 Gy.…”

Section: Discussionmentioning

confidence: 89%

“…Similar outcomes have been reported in studies where standard fractionation was used to treat metastatic sites. 12, 15, 21 In an analysis of survival after conventional RT for metastatic ES in children, Paulino et al reported 2-year rates for OS and PFS at 30.3% and 23.3%. 21 Rahn and colleagues observed a median survival of 6.5 months after palliative RT to the metastatic lesions in a cohort of 44 children with different primary malignancies.…”

Section: Discussionmentioning

confidence: 99%

“…At the same time, there is substantial evidence suggesting both favorable local control at metastatic sites and acceptable side effect profile with the use of conventional fractionation in pediatric cancers. 8–12 However, longer multi-week courses of palliative RT may have a deleterious impact on the quality of life of these often-debilitated patients and increase both psychosocial and economic burden on child’s caregivers.…”

Section: Introductionmentioning

confidence: 99%

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Short Hypofractionated Radiation Therapy in Palliation of Pediatric Malignancies: Outcomes and Toxicities

Lazarev

Kushner

Wolden

2018

International Journal of Radiation Oncology*Biology*Physics

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Section: Discussionmentioning

confidence: 89%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Short Hypofractionated Radiation Therapy in Palliation of Pediatric Malignancies: Outcomes and Toxicities

Lazarev

Kushner

Wolden

2018

International Journal of Radiation Oncology*Biology*Physics

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“…In childhood RMS, treatment and prognosis vary widely according to a series of clinical characteristics such as histology and fusion status, having fusion-positive RMS patients a worse outcome compared to fusion-negative ones [24][25][26]. Distal metastases remains the most adverse prognostic factor despite continuous improvement of the diagnostic capabilities and the use of more intense treatments [27][28][29]. Within this group of patients, outcome further differs significantly when multiple risk factors are combined, such as patient's age, primary tumor site, number of metastases, and bone or bone marrow involvement [30,31].…”

Section: Discussionmentioning

confidence: 99%

Prognostic Value of Circulating IGFBP2 and Related Autoantibodies in Children with Metastatic Rhabdomyosarcomas

et al. 2020

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Insulin-like growth factor-binding protein 2 (IGFBP2) is a tumor-associated protein measurable in patients’ biopsies and blood samples. Increased IGFBP2 expression correlates with tumor severity in rhabdomyosarcoma (RMS). Thus, we examined the plasmatic IGFBP2 levels in 114 RMS patients and 15 healthy controls by ELISA assay in order to evaluate its value as a plasma biomarker for RMS. Additionally, we looked for the presence of a humoral response against IGBFP2 protein measurable by the production of anti-IGFBP2 autoantibodies. We demonstrated that both circulating IGFBP2 protein and autoantibodies were significantly higher in RMS patients with respect to controls and their combination showed a better discriminative capacity. IGFBP2 protein identified metastatic patients with worse event-free survival, whereas both IGFBP2 and anti-IGFBP2 antibodies negatively correlated with overall survival. Our study suggests that IGFBP2 and anti-IGFBP2 antibodies are useful for diagnostic and prognostic purposes, mainly as independent negative prognostic markers in metastatic patients. This is the first study that reports a specific humoral response in RMS plasma samples and proves the value of blood-based biomarkers in improving risk assessment and outcome of metastatic RMS patients.

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“…Radiotherapy in combination with chemotherapy is an effective modality for treatment of lung metastases in pediatric tumors 1,2. Stereotactic body radiotherapy (SBRT) delivers conformal and ablative treatment in 1–5 fractions, while sparing the volume of heart and lung irradiated.…”

Section: Introductionmentioning

confidence: 99%

<p>Efficacy and tolerability of stereotactic body radiotherapy for lung metastases in three patients with pediatric malignancies</p>

Deck¹,

Eastwick²,

Sima³

et al. 2019

OTT

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Purpose: To report a case series of 3 pediatric patients treated with Stereotactic Body Radiation Therapy (SBRT) for lung metastases. Patients and methods: Three patients (ages 9, 11, and 21) received SBRT for rhabdoid tumor, Ewing sarcoma, and Wilms tumor histologies, respectively. SBRT doses were 37.5–50 Gy in 3–5 fractions treating twelve lesions. Results: Three patients (ages 9, 11, and 21) received photon SBRT for pulmonary metastases. The patients were as follows: 1) 21-year-old male with favorable histology Wilms tumor and 1 lesion treated, 2) 11-year-old female with Ewing sarcoma and 1 lesion treated for relapse after previous whole lung radiation (15 Gy), and 3) 9-year-old female with rhabdoid tumor of the left thigh with 10 lesions treated over a two-year period. Median dose delivered was 40 Gy (range, 37.5–50 Gy), delivered in a median of 4 fractions (range, 4–5) of a median of 10 Gy per fraction (range, 9.4–10 Gy). Within a minimum follow-up of 1.9 years (range 1.9–4 years), local control for all 13 treated metastases is 100% without any observed acute toxicities. One possible late toxicity (grade 2 rib fracture) developed 1.3 years following SBRT for treatment of a peripheral lesion (rhabdoid tumor) in an area of disease progression and was managed conservatively. Two patients are surviving 2.9 years (Wilms tumor) and 1.9 years (Ewing sarcoma) after SBRT, and one (rhabdoid tumor) expired 2 years after her final course (4 years after initial SBRT). Two patients (rhabdoid tumor and Ewing sarcoma) suffered disease progression outside of the treated lesions and one patient (Wilms tumor) is without evidence of disease and has not required whole lung irradiation or further systemic therapy. Conclusion: SBRT appears effective and well tolerated for pediatric lung metastases, however further studies are warranted.

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Local therapy to distant metastatic sites in stage IV rhabdomyosarcoma

Abstract: Local treatment to all metastatic sites was associated with improved PFS and OS at 5 years. The use of WLI improved pulmonary control in patients with lung metastasis. We recommend an aggressive approach including local therapy to DMS in children with stage IV RMS.

Cited by 26 publications

References 13 publications

Short Hypofractionated Radiation Therapy in Palliation of Pediatric Malignancies: Outcomes and Toxicities

Short Hypofractionated Radiation Therapy in Palliation of Pediatric Malignancies: Outcomes and Toxicities

Prognostic Value of Circulating IGFBP2 and Related Autoantibodies in Children with Metastatic Rhabdomyosarcomas

<p>Efficacy and tolerability of stereotactic body radiotherapy for lung metastases in three patients with pediatric malignancies</p>

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