Successful treatment of metastatic alveolar rhabdomyosarcoma with <i>MGMT</i> gene promoter methylation by temozolomide‐based combination chemotherapy

Kinoshita, Mariko; Yamada, Akihiro; Sawa, Daisuke; Kamimura, Sachiyo; Miyachi, Mitsuru; Moritake, Hiroshi

doi:10.1002/pbc.26750

Cited by 7 publications

(7 citation statements)

References 18 publications

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“…In total, 5 available case reports were found. We summarize the case reports in Table 1 ( 7 , 8 , 15 – 17 ). Of these cases, male children seem to be the more vulnerable group, which is consistent with overall incidence (male:female ratio of 1.51, 95% confidence interval (CI): 1.27–1.80) ( 18 ).…”

Section: Discussionmentioning

confidence: 99%

Retroperitoneal alveolar rhabdomyosarcoma intruding into spinal canal: A case report and literature review

Zhang

Huang

et al. 2022

Front. Med.

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BackgroundRhabdomyosarcoma (RMS) is the most frequent soft sarcoma in children and adolescents. Alveolar rhabdomyosarcoma (ARMS) is a relatively rare subtype that is characterized by aggressive behavior and an unsatisfactory prognosis. An ARMS can arise anywhere but most commonly occurs at extremity sites with a very small fraction in the retroperitoneum. The utility of 2-Deoxy-2-[fluorine-18]-fluoro-D-glucose (18F-FDG) positron emission tomography combined with computed tomography (PET/CT) remains to be established in ARMS.Case ReportA 3-year-old female child was accidentally found with a large left upper abdominal mass for a day. CT examination indicated a huge soft tissue mass in the left retroperitoneum extending superiorly to the level of the left hilus renalis and inferiorly to the left acetabulum in the pelvic cavity, with intrusion into the lumbar foramens. 18F-FDG PET/CT found a mass in the left retroperitoneum from the level of T12 to the left acetabulum, with the maximum standardized uptake value (SUVmax) of about 7.0, and a CT value of about 39 HU, invading the left L3-5 intervertebral foramina and protruding into the spinal canal, with unclear boundary with the spinal cord. Retroperitoneal tumor resection and the repair operation of vascular exploration were performed. An ARMS was confirmed by postoperative biopsy, immunohistochemical staining, and genetic detection with the rupture of the fork head in rhabdomyosarcoma (FKHR). The patient received chemotherapy and was in a good condition with no recurrence and obvious complications.ConclusionRetroperitoneal ARMS is rare and indicates a poor outcome with the potential to involve vital organs and intrude into the spinal canal. Accurate diagnosis and staging using PET/CT would contribute to better risk stratifications and appropriate treatment individually.

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Section: Discussionmentioning

confidence: 99%

Retroperitoneal alveolar rhabdomyosarcoma intruding into spinal canal: A case report and literature review

Zhang

Huang

et al. 2022

Front. Med.

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“…MGMT promoter methylation has been described in 13% to 43% of soft-tissue sarcomas such as undifferentiated pleomorphic sarcomas and liposarcomas (47,48). Clinically, application of temozolomide in MGMT-methylated sarcomas has only been described in case reports and a small case series (49)(50)(51). The first clinical study in patients with sarcoma is currently ongoing, evaluating MGMT status of patients with Ewing sarcoma receiving temozolomide and irinotecan (NCT03542097).…”

Section: Discussionmentioning

confidence: 99%

DNA Methylation Profiling Identifies Distinct Clusters in Angiosarcomas

Weidema¹,

Geer

Koelsche³

et al. 2020

Clinical Cancer Research

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Purpose: DNA methylation profiling has previously uncovered biologically and clinically meaningful subgroups within many tumor types, but was not yet performed in angiosarcoma. Angiosarcoma is a rare sarcoma with very heterogeneous clinical presentations, which may be based on differences in biological background. In this exploratory study, DNA methylation profiling of 36 primary angiosarcoma samples from visceral, deep soft tissue, radiation-induced, and UV-induced localizations was performed.Experimental Design: Primary angiosarcoma formalin-fixed paraffin-embedded samples from visceral, soft tissue, radiationinduced, and UV-induced origin were collected from a nationwide search for angiosarcoma in the Netherlands. DNA was extracted for methylation profiling with the Illumina Infinium Methylatio-nEPIC array. Quality control assessment and unsupervised hierarchical clustering were performed. Copy-number profiles were generated and analyzed for chromosomal stability. Clinical data were obtained from the Netherlands Cancer Registry.Results: DNA methylation profiling by unsupervised hierarchical clustering of 36 angiosarcoma samples (6 visceral, 5 soft tissue, 14 radiation-induced, 11 UV-induced) revealed two main clusters (A and B), which were divided into four subclusters. The clusters largely corresponded with clinical subtypes, showing enrichment of UV-induced cases in cluster A1 and radiationinduced cases in cluster A2. Visceral and soft tissue cases almost exclusively fell into cluster B. Cluster A showed significantly increased chromosomal instability and better overall survival (22 vs. 6 months, P ¼ 0.046) compared with cluster B.Conclusions: In this novel methylation profiling study, we demonstrated for the first time four different angiosarcoma clusters. These clusters correlated with clinical subtype, overall survival, and chromosomal stability.

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“…Another approach is the use of quantitative RT-PCR (qRT-PCR) to investigate the differential expression and/or release of miRNAs by solid tumors. 33 studies investigated miRNA in the serum or plasma of 2293 patients (miRNAs are listed Supplementary Table 2) [ [34] , [35] , [36] , [37] , [38] , [39] , [40] , [41] , [42] , 44 , [46] , [47] , [48] , [49] , [50] , [51] , [52] , [89] , [90] , [91] , [92] , [93] , [94] , [95] , [96] , [97] , [98] , [99] , [100] , [101] , [102] , [103] , [104] ]. In addition to qRT-PCR, one study used ddPCR [42] and three studies used TaqMan low density array (TLDA) [ 38 , 48 , 51 ].…”

Section: Detection Of Tumor-derived Cells and Nucleic Acid In Periphe...mentioning

confidence: 99%

“…The diagnostic value of one or more miRNAs were often evaluated by calculating area under the curve (AUC) at a predefined cutoff level of receiver operating characteristics (ROC) curves differentiating miRNA expression levels of patients and controls. Six studies used medical imaging including MRI, CT, and 18 F-FDG-PET/CT to determine tumor size, treatment response, and/or relapse [ 34 , 89 , 93 , 96 , 97 , 103 ].…”

Section: Detection Of Tumor-derived Cells and Nucleic Acid In Periphe...mentioning

confidence: 99%

“…RT-PCR was used to detect ctRNA at diagnosis in 50% (n=42) of patients included in five studies [ 78 , 79 , 81 , 85 , 88 ]. Four studies observed good diagnostic value of varying miRNAs [ 42 , 49 , 89 , 90 ]. Further, when comparing analyses of tumor tissue and PB, concordance was observed with both ctDNA and ctRNA [ 67 , 69 , 88 ].…”

Section: Applications Of Liquid Biopsies In Pediatric Sarcomamentioning

confidence: 99%

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Detection of circulating tumor-derived material in peripheral blood of pediatric sarcoma patients: A systematic review

Kjær,

Vase,

Rossing

et al. 2023

Translational Oncology

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Successful treatment of metastatic alveolar rhabdomyosarcoma with MGMT gene promoter methylation by temozolomide‐based combination chemotherapy

Cited by 7 publications

References 18 publications

Retroperitoneal alveolar rhabdomyosarcoma intruding into spinal canal: A case report and literature review

Retroperitoneal alveolar rhabdomyosarcoma intruding into spinal canal: A case report and literature review

DNA Methylation Profiling Identifies Distinct Clusters in Angiosarcomas

Detection of circulating tumor-derived material in peripheral blood of pediatric sarcoma patients: A systematic review

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