Outcome and Patterns of Relapse in Childhood Parameningeal Rhabdomyosarcoma Treated With Proton Beam Therapy

Doyen, J.; Jazmati, Danny; Geismar, Dirk; Frisch, Sabine; Schleithoff, Stefanie Schulze; Vermeren, Xavier; Scheer, Monika; Blase, C.; Tippelt, Stephan; Timmermann, Beate

doi:10.1016/j.ijrobp.2019.08.005

Cited by 23 publications

(23 citation statements)

References 23 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Accordingly, our descriptive evaluations show a significantly higher median irradiation dose to the eye with protons when compared to photons. A high median radiation dose for proton irradiation can also be observed in other studies such as Doyen et al (median total prescribed dose 55.8 Gy) [26]. Therefore, the use of protons cannot be associated with any increased risk for toxicity within this study.…”

Section: Discussionsupporting

confidence: 73%

“…It is well known that in addition to the radiation dose used, the volume irradiated has a major impact on organs at risk (OARs) and this is minimized by new radiation methods [29]. Maximum doses are often determined for OARs to minimize sequelae (for example, < 55 Gy at the base of the skull, optic chiasm, optic nerve) [26]. Nevertheless, studies show that late effects occur even with these techniques [28].…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Evaluation of treatment-associated eye toxicity after irradiation in childhood and adolescence—results from the Registry of the Evaluation of Side Effects after Radiotherapy in Childhood and Adolescence (RiSK)

et al. 2021

Self Cite

View full text Add to dashboard Cite

Purpose The aim of the study is to evaluate treatment-related acute and late eye toxicity associated with radiation therapy in childhood and adolescence as correlated with RT (radiotherapy) doses. Methods From 2001 to 2016, a total of 1725 children and adolescents undergoing radiation therapy were prospectively documented in the Registry of the Evaluation of Side Effects after Radiotherapy in Childhood and Adolescence (RiSK). The RTOG/EORTC criteria were used to classify ocular acute and late effects. Uni- and multivariate analyses were carried out to evaluate the impact of patient age, pre-existing impairments, and radiation dose on ocular toxicity. Results Of all documented patients, 593 received dose to the eye and formed the basis of this analysis. In 435 patients, information on acute reaction was available and graded 1, 2, 3, and 4 in 49, 17, 0, and 2 patients, respectively. Information on late toxicity was available in 268 patients and graded 1, 2, 3, and 4 in 15, 11, 11, and 5 patients, respectively. The acute toxicity rate was significantly higher in children who received a maximum dose > 50 Gy to the eye (p < 0.001) and who had a pre-existing eye impairment (p < 0.001 in multivariate analysis). The development of late toxicity was significantly higher for patients experiencing acute toxicity and having received a radiation dose > 50 Gy. Conclusion Acute and late toxicity both correlate with high radiation dose to the eye (> 50 Gy) and acute toxicity additionally with pre-existing eye impairments.

show abstract

Section: Discussionsupporting

confidence: 73%

Section: Discussionmentioning

confidence: 99%

Evaluation of treatment-associated eye toxicity after irradiation in childhood and adolescence—results from the Registry of the Evaluation of Side Effects after Radiotherapy in Childhood and Adolescence (RiSK)

et al. 2021

Self Cite

View full text Add to dashboard Cite

show abstract

“…23,24 Even with these doses, local relapse remains the dominant form of failure for PM-RMS, with localized failure seen in approximately 70% of those who relapse on both European and North American cooperative group trials. The majority of local failures occur in the high dose region, [25][26][27] suggesting that marginal misses or volumetric coverage are not the issue. Furthermore, the utilization of a post-chemotherapy cone-down volume (to reduce the high-dose treatment volume) does not appear to compromise local control.…”

Section: Evidencementioning

confidence: 99%

“…Another series that used proton therapy for the treatment of RMS across primary sites reported no regional failures, with radiotherapy directed only to involved nodal levels. 25 In a retrospective review of 24 patients with alveolar PM-RMS in which radiation was directed only to the involved nodal level(s) with a boost to the involved nodes, 3 year regional control was 94%. 32 One patient with a node negative nasal cavity primary developed a submental nodal recurrence.…”

Section: Evidencementioning

confidence: 99%

Local control of parameningeal rhabdomyosarcoma: An expert consensus guideline from the International Soft Tissue Sarcoma Consortium (INSTRuCT)

Casey

Mandeville

Bradley³

et al. 2022

Preprint

Self Cite

View full text Add to dashboard Cite

The International Soft-Tissue Sarcoma Database Consortium (INSTRuCT) consists of a collaboration between the Children’s Oncology Group (COG) Soft Tissue Sarcoma Committee, the European pediatric Soft-Tissue Sarcoma Study Group (EpSSG), and the Cooperative Weichteilsarkom Studiengruppe (CWS). As part of the larger initiative of INSTRuCT to provide consensus expert opinions for clinical treatment of pediatric soft tissue sarcoma, we sought to provide updated, evidenced-based consensus guidelines for local treatment of parameningeal rhabdomyosarcoma using both existing literature as well as recommendations from the relevant cooperative group clinical trials. Overall, parameningeal rhabdomyosarcoma represents a distinctly challenging disease to treat given its location near many critical structures in the head and neck, frequently advanced local presentation, and predilection for local failure. Definitive chemoradiation remains the standard treatment approach for parameningeal rhabdomyosarcoma, with surgery often limited to biopsy or salvage therapy for recurrent disease. In this consensus paper, we specifically discuss consensus guidelines and evidence for definitive local management with radiotherapy, with a focus on imaging for radiotherapy planning, dose and timing of radiation, approach for nodal irradiation, various radiation techniques including proton therapy, and the limited role of surgical resection.

show abstract

“…Due to the atypical clinical features and bone marrow pathology, most patients are misdiagnosed with haematological malignancies or other solid tumours at initial diagnosis presentation, and these results in delayed treatment [ 4 ]. More notably, those patients often present with highly aggressive disease and poor prognosis, they either do not respond to conventional therapies or relapse after treatment [ 5 ]. To date, there are few published reports on the clinical characteristics and treatment outcomes of these children.…”

Section: Introductionmentioning

confidence: 99%

Clinical features and prognosis of paediatric rhabdomyosarcoma with bone marrow metastasis: a single Centre experiences in China

Jian

et al. 2021

BMC Pediatr

View full text Add to dashboard Cite

Background The aim of this study was to summarize the clinical characteristics, therapeutic effects and prognosis of patients with rhabdomyosarcoma (RMS) and bone marrow metastasis, improve the understanding of this disease. Method This was a single-institution retrospective study involving the children with RMS, who presented with bone marrow metastasis at initial presentation to our hospital between 1st, Jan, 2006 and 31st, Dec,2019. Follow-up concluded on 31st, Dec, 2020 and the clinical data were collected and analysed. Result Between 1st Jan 2006 and 31st Dec 2019, 13 eligible patients presented to our hospital, including 10 males and 3 females, these eligible patients accounted for 4.5% of all RMS patients. The median age at onset was 5.6 years (range 1.7-14 years). The patients not only had unfavourable primary sites, but also had multiple metastases. The bone marrow aspirate samples of the patients comprised 8-95% blast-like cells. Nine of 13 patients were misdiagnosed with haematological malignancies or other solid tumours. With respect to histology, four of 13 children were classified as embryonal RMS and nine as alveolar RMS. Eleven patients underwent PAX-FOXO1 fusion testing; eight had the POX- FOXO1 fusion gene. Immunohistochemically(IHC) analysis revealed that the tumour cells were positive for Desmin, Vimentin, Myo-D1 and Myogenin. More importantly, the patients had extremely poor prognoses, the median EFS was 12.0 months (range 3-28.3 months) and the median OS was 27.0 months (range6-46.2 months). Conclusion This study demonstrates that children with RMS and bone marrow metastasis usually exhibit atypical primary sites and multiple metastases, with presentation mimicking haematological malignancies or other solid tumors at initial presentation. Pathology and IHC analysis combined with POX-FOXO1 fusion gene detections can effectively confirm the diagnosis. These patients are more likely to relapse or progress during early treatment and are prone to intracranial metastasis. While multidisciplinary therapy combined with Temozolomide may prevent it, further prospective research is required to evaluate the therapeutic effects.

show abstract

Outcome and Patterns of Relapse in Childhood Parameningeal Rhabdomyosarcoma Treated With Proton Beam Therapy

Cited by 23 publications

References 23 publications

Evaluation of treatment-associated eye toxicity after irradiation in childhood and adolescence—results from the Registry of the Evaluation of Side Effects after Radiotherapy in Childhood and Adolescence (RiSK)

Evaluation of treatment-associated eye toxicity after irradiation in childhood and adolescence—results from the Registry of the Evaluation of Side Effects after Radiotherapy in Childhood and Adolescence (RiSK)

Local control of parameningeal rhabdomyosarcoma: An expert consensus guideline from the International Soft Tissue Sarcoma Consortium (INSTRuCT)

Clinical features and prognosis of paediatric rhabdomyosarcoma with bone marrow metastasis: a single Centre experiences in China

Contact Info

Product

Resources

About