Cancer Research and Treatment

Severe Hepatic Sinusoidal Obstruction Syndrome in a Child Receiving Vincristine, Actinomycin-D, and Cyclophosphamide for Rhabdomyosarcoma: Successful Treatment with Defibrotide: Aery Choi, Young Kyung Kang, Sewon Lim, Dong Ho Kim, Jung Sub Lim, Jun Ah Lee; Cancer Res Treat. 2016;48(4):1443-1447. Published online March 30, 2016; DOI: https://doi.org/10.4143/crt.2016.096

Hepatic sinusoidal obstruction syndrome (SOS) is a life-threatening syndrome that generally occurs as a complication after hematopoietic stem cell transplantation or, less commonly, after conventional chemotherapy. Regarding SOS in rhabdomyosarcoma patients who received conventional chemotherapy, the doses of chemotherapeutic agents are associated with the development of SOS. Several cases of SOS in rhabdomyosarcoma patients after receiving chemotherapy with escalated doses of cyclophosphamide have been reported. Here, we report on a 9-year-old female with rhabdomyosarcoma who developed severe SOS after receiving chemotherapy consisting of vincristine, actinomycin-D, and a moderate dose of cyclophosphamide. She was treated successfully with defibrotide without sequelae to the liver.

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Adjuvant recombinant thrombomodulin therapy for hepatopathy induced by vincristine, actinomycin D, and cyclophosphamide in pediatric rhabdomyosarcoma: A case report
Tetsuko Kobayashi, Maiko Noguchi, Hideki Nakayama, Reiji Fukano, Shouichi Ohga
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“Pre‐emptive strike”—the case for early treatment of hepatic sinusoidal obstruction syndrome with defibrotide
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Cyclophosphamide/dactinomycin/vincristine

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Incidence and Survival of Pediatric Soft Tissue Sarcomas: Comparison between Adults and Children: Sun Min Lim, Cheol Joo Yoo, Jung Woo Han, Yong Jin Cho, Soo Hee Kim, Joong Bae Ahn, Sun Young Rha, Sang Joon Shin, Hyun Cheol Chung, Woo Ick Yang, Kyoo-Ho Shin, Jae Kyung Rho, Hyo Song Kim; Cancer Res Treat. 2015;47(1):9-17. Published online August 21, 2014; DOI: https://doi.org/10.4143/crt.2013.157

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Purpose
Pediatric-type sarcomas such as rhabdomyosarcoma (RMS), Ewing sarcoma (EWS), primitive neuroectodermal tumor (PNET), and desmoplastic small round-cell tumor (DSRCT) are rare in adults, with limited studies on their prognosis and optimal treatment strategies. We aimed to examine the outcome of children and adult patients with RMS, EWS, PNET, and DSRCT and relevant prognostic factors. Materials and Methods We retrospectively reviewed 220 pediatric-type sarcoma patients at a single institution between 1985 and 2011. Comparisons were made in order to examine differences in demographics, disease characteristics, and survival. Survival analyses were performed using the Kaplan-Meier method with log-rank tests and Cox proportional hazards models. Results A total of 220 consecutive patients were identified at our institute. Median age was 15.6 years (range, 0 to 81 years) and there were 108 children (49%) and 112 adult patients (51%). According to histological classification, 106 patients (48.2%) had RMS, 60 (27.3%) had EWS, 50 (22.7%) had PNET, and 4 (1.8%) had DSRCT. With a median follow-up period of 6.6 years, the estimated median overall survival (OS) of all patients was 75 months (95% confidence interval [CI], 27.2 to 122.8 months) and median event-free survival (EFS) for all patients was 11 months (95% CI, 8.8 to 13.2 months). No significant difference in OS and EFS was observed between adults and children. In multivariate analysis, distant metastasis (hazard ratio [HR], 1.617; 95% CI, 1.022 to 2.557; p=0.040) and no debulking surgery (HR, 1.443; 95% CI, 1.104 to 1.812; p=0.012) showed independent association with worse OS. Conclusion Metastatic disease and no surgical treatment are poor prognostic factors for OS among pediatric-type sarcomas for both adults and children.

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Initial Response to Treatment was Highly Associated with the Prognosis of Childhood Rhabdomyosarcoma: A Retrospective Analysis of a Single Center Experience in Korea: Jeong A Park, Eun Kyung Kim, Hyoung Jin Kang, Hee Young Shin, Il Han Kim, Hyo Seop Ahn; Cancer Res Treat. 2008;40(3):111-115. Published online September 30, 2008; DOI: https://doi.org/10.4143/crt.2008.40.3.111

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Purpose

Following the introduction of a multimodal approach to diagnosis and treatment, the prognosis of rhabdomyosarcoma (RMS) has markedly improved over the last three decades. However, there are few data on treatment outcomes in Korean patients.

Materials and Methods

We performed a retrospective analysis of 77 patients with RMS diagnosed and treated at Seoul National University Children's Hospital between 1986 and 2005.

Results

The overall 5-year survival and event-free survival rates for all patients were 77% and 59%, respectively. The Intergroup Rhabdomyosarcoma Study clinical grouping and initial response to treatment (20-week response) were important prognostic factors.

Conclusions

The outcome of childhood RMS was closely associated with the initial staging and the initial response to treatment. Modulating therapies according to initial responses and risk factors is critical, and new treatment strategies for high-risk patients are needed.

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Adherence to Treatment, Response and Patterns of Failure in Pediatric Parameningeal Rhabdomyosarcoma: Experience From a Tertiary Cancer Care Center From India
Soumyajit Roy, Sushmita Pathy, Bidhu K. Mohanti, Subhash Chander, Ahitagni Biswas
Journal of Pediatric Hematology/Oncology.2017; 39(2): e62. CrossRef
Pediatric rhabdomyosarcoma in India: A single-center experience
Deepak Bansal, Anirban Das, Amita Trehan, Rakesh Kapoor, Naresh K. Panda, Radhika Srinivasan, Nandita Kakkar, Kushaljit S. Sodhi, Akshay K. Saxena, Katragadda Lakshmi Narasimha Rao
Indian Pediatrics.2017; 54(9): 735. CrossRef
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A Case of Pleomorphic Laryngeal Rhabdomyosarcoma in an Adult: Choong Won Lee, Yeong Mook Kim, Jeong Youl Oh, Jeong Suk Choi, Soon Chul Hwang, Ok Ji Paik; J Korean Cancer Assoc. 1994;26(4):665-670.

Abstract PDF: Rhabdomyosarcoma (RMS) is malignant tumor and develops from the embryonal mesenchyme which give rise to striated muscle. Laryngeal localization of RMS is extremely rare. Cel- lular differentiation may be very different in this tumor and attempts have been made to correlated the morphological patterns with clinical course. A 39-year-old male suffered from hoarseness and dyspnea, visited to the department of inter- nal medicine, Wallace Memorial Baptist Hospital. Local excisional biopsy under indirect laryn- goscopy was done. A biopsy with review and compare it with previous reports.

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A Case of Bladder Rhabdomyosaroma in Child: Hee Jong Jeung, Sam Ryong Lee, Kwang Sung Park, Soo Bang Ryu, Hoon Kook, Tai Ju Hwang; J Korean Cancer Assoc. 1996;28(3):604-611.

Abstract PDF: Embryonal rhabdomyosarcoma arises from the embryonal mesenchyme that differentiates into striated skeletal muscle, and about 15 to 20% of rhabdomyosarcomas are genitourinary in origin. Rhabdomyosarcoma of urogenital origin occurs at 2 age peaks: one in children between 2 and 6 year old and the other during adolescence between ages 15 and 19 year old. We report a case of embryonal rhabdomyosarcoma of the bladder in a 3-year-old male. He presented dysuria with straining and dribbling, and diagnosis was made by cystoscopy and biopsy. Since partial cystectomy was performed, and he has being received continuous Pulse VAC chemotherapy with radiotherapy.

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