For these patients, 5‐year EFS and OS were 27.9% and 39.6%, respectively. Metastatic pleomorphic rhabdomyosarcoma to the mandible: report of a rare case and review of the literature. Among 110 adult patients with embryonal, alveolar, or NOS histology, from a series of 180 adults with RMS who were treated at a single institution during a 25‐year period and retrospectively reviewed, 5‐year EFS was in the 30% range. Chemotherapy uses anticancer, or cytotoxic, drugs to destroy cancer cells. Histiocyte-rich rhabdomyoblastic tumor: a report of two cases and a review of the differential diagnoses. A Population-Based Analysis of Survival for Sinonasal Rhabdomyosarcoma. PARP inhibitors affect growth, survival and radiation susceptibility of human alveolar and embryonal rhabdomyosarcoma cell lines. Most patients with pleomorphic RMS (86%) had tumors larger than 5 cm. A home run for rhabdomyosarcoma after 30 years: What now?. Four of the 11 patients who had local recurrence were alive after achieving a second or third CR. The primary tumor was larger than 5 cm in 77% of all cases. Even uncontrolled studies of adults could be useful, provided that they are designed in a way that makes them suitable for confirming the applicability in another age group, through specific treatment solutions, of principles established in large, controlled trials in pediatric populations. Treatment is usually with a combination of surgery, chemotherapy and radiation therapy. Alveolar rhabdomyosarcoma. and you may need to create a new Wiley Online Library account. Epithelioid rhabdomyosarcoma: A rare case report mimicking carcinoma breast. Staging of rhabdomyosarcoma according to intergroup rhabdomyosarcoma … Become a volunteer, make a tax-deductible donation, or participate in a fundraising event to help us save lives. Regarding the local treatment score, 72 patients received a score of 1, 26 received a score of 0.8 (due to delay in the timing of irradiation), 1 received a score of 0.7, 6 received a score of 0.6, and 5 received a score of 0. This conclusion disagrees with the view, expressed by some authors, that adult RMS is inherently different from pediatric RMS.16-24 Unsatisfactory treatment results even have raised doubts as to whether chemotherapy should be used at all to treat adults with RMS; Hawkins et al.21 recently concluded that there was no evidence that chemotherapy provided any survival benefit for adult patients with RMS. These differences may explain in part the less favorable outcome of adult patients overall, and they invite us to treat adults with intensive approaches.22, 23 But are intensive treatments feasible for adult patients? Most doctors recommend that people in the high-risk group be treated in a clinical trial testing new drugs and drug combinations. The remaining 22 patients died of disease at 2–47 months (median, 14 months) after diagnosis. Resource Utilization and Costs in Adolescents Treated for Cancer in Pediatric vs Adult Institutions. This type of rhabdomyosarcoma is usually diagnosed in middle-aged people. They were treated with a combination of standard vincristine, actinomycin‐D, and cyclophosphamide combined with ifosfamide and etoposide. Data regarding treatment and outcome were complete for 171 patients. Surgery was a mainstay of treatment for pleomorphic RMS and was correlated with survival. Their outcome was exceedingly poor in the current series, although this typically is the case in large pediatric series as well. Over a period of time spanning more than 25 years, patients were treated with a relatively consistent approach, which in most cases included surgery, chemotherapy, and radiotherapy. Compared with the subset of 143 patients with embryonal, alveolar, or NOS histotype, the pleomorphic RMS subgroup was composed of a greater proportion of males (70% vs. 52%), older patients (84% vs. 32% age > 30 years and 43% vs. 7% age > 60 years), and patients with primary tumor location in the extremities (65% vs. 17%). Radiotherapy administered at an adequate dose more than 4 months after incomplete resection or biopsy corresponded to a score of 0.8. Use the link below to share a full-text version of this article with your friends and colleagues. Complete response (CR) was defined as the complete disappearance of disease, partial response (PR) was defined as a tumor reduction of > 50%, and minor response (MR) was defined as a maximum tumor reduction of > 25%. Journal of Cancer Research and Clinical Oncology. Another approach that has been studied is to give higher doses of chemo, sometimes followed by a stem cell transplant. Treatment strategies and outcomes for spinal rhabdomyosarcoma: a series of 11 cases in a single center and review of the literature. EFS and OS were calculated according to the Kaplan–Meier method.11 Survival was evaluated from the date of histologic diagnosis before the first definite treatment to an event‐free final follow‐up, or to disease progression or recurrence (for EFS only) or death due to any cause (for both EFS and OS). For reprint requests, please see our Content Usage Policy. International Journal of Surgery Case Reports. In the current series, aside from the favorable outcome of patients who were treated according to current guidelines for pediatric RMS, the overall rate of response to chemotherapy was 85%. The Japanese Journal of Gastroenterological Surgery. Adolescents with Cancer in Italy: Improving Access to National Cooperative Pediatric Oncology Group (AIEOP) Centers. With regard to local treatment, a score of 1 was assigned in cases of complete surgical resection, as defined above (with or without radiotherapy); and in cases of incomplete resection (with microscopically involved margins or macroscopic residual tumor) or biopsy, provided that these procedures were followed by adequate radiotherapy. This type of rhabdomyosarcoma usually occurs in adults, in the arms or legs. WebMD provides details on its symptoms, diagnosis, treatment, and more. In most cases, radiation fields included the initial volume of the tumor plus 2–3 cm margins, as well as any involved lymph nodes. To maximize the likelihood that adult patients are treated according to these principles, protocols that include drug regimens and treatment combinations specifically designed for adults might be worth trying prospectively. One patient developed a second tumor (breast carcinoma) during CR, 10 years after diagnosis of RMS. I am so thankful for the doctors and nurses at Mass General Hospital in Boston. Primary Uterine Rhabdomyosarcoma in a 54-Year-Old Postmenopausal Woman. The American Cancer Society couldn’t do what we do without the support of our partners. Cancer.org is provided courtesy of the Leo and Gloria Rosen family. Five‐year EFS and OS were 32.9% and 45.7%, respectively. Treatment protocols adopted from pediatric programs but tailored to adults could increase adults' chances of receiving appropriate treatment; prospective studies are needed to test this idea. Follow‐up, as of September 2002, ranged from 8 to 260 months (median, 50 months). Nonetheless, we agree with Esnaola et al.22 and Little et al.23 in our opinion that this is not the case. The role of 18F-FDG PET/CT in diagnosis and staging of musculoskeletal soft tissue sarcomas. Not very much is known about why normal skeletal muscle cells become cancerous. Bilateral bone marrow aspirates and/or biopsies were performed in several cases of embryonal and alveolar RMS. International Journal of Clinical Oncology. It is possible that age simply affects the relative size of various prognostic subsets in adults; this holds true within the pediatric population as well, where age is considered an adverse prognostic factor.9 Therefore, we believe that there is no reason to treat adults with RMS according to different guidelines from those that have been established over the years in large pediatric trials. Chemotherapy is indicated for all patients with rhabdomyosarcoma, but the amount of chemotherapy and the duration of treatment can vary depending on risk factors. Working off-campus? Recent pediatric oncology studies report overall survival rates exceeding 70% for patients with localized rhabdomyosarcoma given risk-adapted multidisciplinary treatments, including surgery, radiotherapy, and multiagent chemotherapy in particular. Rhabdomyosarcoma: an overview and nursing considerations. Chemo is systemic therapy, meaning that the drugs enter the bloodstream and go throughout the body to destroy cancer cells. Patients who received adequate chemotherapy (score, 1) had a relatively good MRFS (Table 2). In contrast, RMS is exceedingly infrequent in adults: soft tissue sarcomas make up less than 1% of all adult malignancies, and RMS accounts for 3% of all soft tissue sarcomas.1. Journal of International Medical Research. What Happens After Treatment for Rhabdomyosarcoma? Objectives: Rhabdomyosarcoma is an exceedingly rare tumor in adults, and standard chemotherapy used for children is much less effective in adults. There is no doubt that childhood RMS is a distinct entity. This makes chemo useful for killing cancer cells that have spread to other parts of the body, even if they can’t be seen. Rhabdomyosarcoma is an exceedingly rare tumor in adults, and standard chemotherapy used for children is much less effective in adults. 2018. The total length of treatment usually ranges from 6 months to a year. Only nine patients did not respond to front‐line chemotherapy. However, this may be the critical point—which prognostic factors affect adult series as compared with pediatric series? 1). Together, we’re making a difference – and you can, too. Tongue Spindle Cell Rhabdomyosarcoma: A Rare Case Report and Literature Review. At pathology consultation, initial diagnoses of RMS were changed in 10 cases; 2 were changed to desmoplastic small round cell tumor, 2 to malignant peripheral nerve sheath tumor, and 1 each to liposarcoma, leiomyosarcoma, malignant hemangiopericytoma, clear cell sarcoma of soft parts, malignant fibrous histiocytoma, and melanoma. One hundred eighty patients were considered for the current study: 149 had localized disease, and 31 had metastases at diagnosis. Patients with noninvasive tumors, patients with small tumors, and patients who received complete surgery at diagnosis (IRS Group I) had the best reported outcomes. Not All the Spindle Cell Tumors in Oral Cavity Are Sarcomatoid Squamous Cell Carcinoma. Rhabdomyosarcoma (RMS) is a rare malignancy. This study examines short-term outcomes using doxorubicin, ifosfamide, and vincristine for adult rhabdomyosarcoma. Nonetheless, one might point out that it has been demonstrated in several tumors that adults can tolerate dose‐intense, multimodal treatments. Where Are Adolescents with Soft Tissue Sarcomas Treated? Evolving Services for Adolescents with Cancer in Italy: Access to Pediatric Oncology Centers and Dedicated Projects. Case presentation: We report our experience of a rare case of rhabdomyosarcoma of the bladder in a 45-year-old Moroccan man who was successfully managed with neoadjuvant chemotherapy and surgical excision of the mass; he was disease free at … This response rate substantially differs from the rate observed in adults with soft tissue spindle cell sarcomas (response rate < 50%) and clearly falls in the same range as the rate for pediatric small cell sarcomas (i.e., RMS and Ewing family tumors). Therapeutic outcome and prognostic factors in sinonasal rhabdomyosarcoma: a single-institution case series. Adult oncologists tend to treat pleomorphic RMS as nonpediatric soft tissue spindle cell sarcomas. Cervical rhabdomyosarcoma in an endocervical polyp of a 50 year old patient with intermenstrual bleeding. Rhabdomyosarcoma is a type of sarcoma.Sarcoma is cancer of soft tissue (such as muscle), connective tissue (such as tendon or cartilage), or bone.Rhabdomyosarcoma usually begins in muscles that are attached to bones and that help the body move, but it may begin in many places in … Chemotherapy appears to have the same activity in adult and pediatric RMS, and when chemotherapy is included in a regimen similar to those used to treat pediatric patients, the outcomes for adults and children with RMS are similar to each other. If larger areas of tumor remain after surgery (or if surgery couldn’t be done for some reason), chemo (along with radiation) can often shrink these areas. It is very rare in the pediatric population, accounting for less than 1% of all RMS cases in the IRS I–III studies. In the absence of controlled, prospective trials, which, given the extreme rarity of the disease, clearly are unfeasible for adults with RMS, we believe that the findings of the current study are sufficient for recommending that adults be treated according to the same principles that have dramatically improved the prognosis of children with RMS in recent decades. This type of rhabdomyosarcoma is … International Journal of Surgical Pathology. Table 6 shows the clinical characteristics of patients in the current series along with those of a group of 252 pediatric patients who concomitantly were enrolled in the ICG RMS‐88 study. Asian Pacific Journal of Cancer Prevention. Model of Care for Adolescents and Young Adults with Cancer: The Youth Project in Milan. (2003). Childhood rhabdomyosarcoma is a disease in which malignant (cancer) cells form in muscle tissue. The activity of chemotherapy against pleomorphic RMS may be closer to the observed activity against adult sarcomas, and the role of chemotherapy in multimodality treatment is less clear for pleomorphic RMS than for pediatric sarcomas.1 In the current series, data on response to chemotherapy were available in only two cases (one PR and one lack of response). Lymph node involvement and alveolar histology were associated with the worst reported outcomes. Age-Related Alterations in Immune Contexture Are Associated with Aggressiveness in Rhabdomyosarcoma. Embryonal rhabdomyosarcoma masquerading as a left vocal cord granuloma. For the cohort of patients with a score of 1, outcome (5‐year OS, 61.5%) was decidedly better than for other subgroups (Fig. Journal of Community Hospital Internal Medicine Perspectives. Nonetheless, all existing studies report a poorer outcome for adults compared with children, despite the extrapolation of multimodality treatment from pediatric experience. What does it take to outsmart cancer? ERKRANKUNGEN DES BLUTES UND DES GERINNUNGSSYSTEMS, SOLIDE TUMOREN UND PRINZIPIEN DER INTERNISTISCHEN ONKOLOGIE. Auflage. The authors attempted to stratify patients according to the degree to which they had been treated appropriately, based on current treatment guidelines for childhood RMS. Adolescent and Young Adult (AYA) Oncology in the United States. The survival benefit of radiotherapy in localized primary adult rhabdomyosarcoma. Giant intrascrotal embryonal rhabdomyosarcoma in an adult: a case report and review of the literature. Learn more about these partnerships and how you too can join us in our mission to save lives, celebrate lives, and lead the fight for a world without cancer. To augment existing data and assist in clarifying the issue of the applicability of childhood protocols to adults, we retrospectively analyzed the outcomes of 171 adult patients with diagnoses of RMS who were seen at the Istituto Nazionale Tumori (INT), Milan, Italy, over a 25‐year period. We were unable to retrospectively assess the dose intensity of the regimens used. The Youth Project at the Istituto Nazionale Tumori in Milan. For example, drugs can be given to help prevent or reduce nausea and vomiting. Primary cardiac rhabdomyosarcoma successfully treated with eribulin. CONCLUSIONS: Age, location, nodal status, and histologic subtype do not appear be associated with survival in adults with rhabdomyosarcoma treated with multimodal therapy. Help make it a reality. Pleomorphic rhabdomyosarcoma metastasis to small intestine causing intussusception. Treatment involved surgical resection, radiation therapy, and chemotherapy with doxorubicin, ifosfamide, and vincristine. This is why doctors can often give them higher doses of chemo to kill the tumor. In the statistical analysis, the local treatment score was cross‐checked with local recurrence–free survival (LRFS) and overall survival (OS). Five‐year EFS was 36.9% for patients who underwent complete resection, whereas no patient with unresectable tumor achieved 5‐year EFS; 5‐year OS rates for these 2 subgroups were 55.7% and 52%, respectively. With regard to chemotherapy, a score of 0 was assigned to all cases in which chemotherapy was not used. Tumor size and local invasiveness were strongly correlated with metastases at onset: all but one patient with M1 disease had primary tumors classified as T2b. Thirteen patients were assigned scores ranging from 0.4 to 0.6, due to their regimens or the duration of treatment. Patients with nonmetastatic embryonal, alveolar, or NOS histotype were analyzed separately from patients with pleomorphic RMS and patients with metastatic disease, as described below. Learn more. Okcu MF, Hicks J. Rhabdomyosarcoma in childhood and adolescence: Treatment. Rhabdomyosarcoma in adults: analysis of treatment modalities in a prospective single-center series. If you do not receive an email within 10 minutes, your email address may not be registered, Thirty‐one patients (17% of the entire series) had metastatic disease at diagnosis. Revue d'Oncologie Hématologie Pédiatrique. Pathologic aspects and proposal for a new classification—an Intergroup Rhabdomyosarcoma Study, Rhabdomyosarcoma: a new classification scheme related to prognosis, The Intergroup Rhabdomyosarcoma Study I: a final report, Current concepts of diagnosis and treatment of bone and soft tissue tumors, Intergroup Rhabdomyosarcoma Study‐IV: results for patients with nonmetastatic disease, Rhabdomyosarcoma and undifferentiated sarcoma in the first two decades of life: a selective review of Intergroup Rhabdomyosarcoma Study Group experience and rationale for Intergroup Rhabdomyosarcoma Study V, Non‐parametric estimation from incomplete observation, Prognosis in children with rhabdomyosarcoma: a report of the Intergroup Rhabdomyosarcoma Studies I and II, Treatment of non‐metastatic rhabdomyosarcomas in childhood and adolescence. The side effects depend on the type and doses of drugs, and the length of time they are given. Of the 16 patients with recurrences who did not receive chemotherapy as their first treatment (score, 0), 5 were alive without disease after salvage chemotherapy (MRFS, 39.4%; OS, 49.4%). Most patients (n = 104) received a multidrug regimen that included cyclophosphamide or ifosfamide, in addition to doxorubicin, epirubicin, or dactinomycin; most regimens also included vincristine. Clinicopathological characteristics and treatment outcomes of Chinese patients with genitourinary embryonal rhabdomyosarcoma. Sixteen patients in this group were males. Ten patients were alive with disease. At the American Cancer Society, we’re on a mission to free the world from cancer. Still, the observation of similar results in a distinct subgroup in the current series indicates that the question posed by the current study is worth answering. Systematic review and pooled survival analysis of 137 patients. Management of Low and Intermediate Risk Adult Rhabdomyosarcoma: A Pooled Survival Analysis of 553 Patients. Data regarding tumor response to chemotherapy were available in only two cases (one PR and one lack of response). Exploration of the factors that prevent most adult patients (approximately 60% in the current series) from receiving fully adequate treatment is called for. All so you can live longer — and better. Chemo is systemic therapy, meaning that the drugs enter the bloodstream and go throughout the body to destroy cancer cells. Consensus and controversies regarding the treatment of rhabdomyosarcoma. Cancer 2003;98:571–80. For RMS, chemo is typically given once a week for the first few months, and then less often. Because of this, RMS in adults is often harder to treat effectively. Metastasis of Rhabdomyosarcoma to the Male Breast: a Case Report with Magnetic Resonance Imaging Findings. The drugs which have demonstrated activity in rhabdomyosarcoma include vincristine, actinomycin, cyclophosphamide, ifosfamide, doxorubicin, carboplatin, etoposide, irin… Distinctive Head and Neck Bone and Soft Tissue Neoplasms. The group of patients with embryonal, alveolar, or NOS RMS included 110 patients with complete data regarding clinical findings, treatment, and outcome. Wilms tumor, medulloblastoma, and rhabdomyosarcoma in adult patients: lessons learned from the pediatric experience. Rhabdomyosarcoma (RMS) is a malignant tumor (\"cancer\") that arises from a normal skeletal muscle cell. Rhabdomyosarcoma in adults: 5-year outcome as a function of "pediatric vs. adult treatment." Radiotherapy was administered to 73 patients (66%). Age-based disparities in treatment and outcomes of retroperitoneal rhabdomyosarcoma. Of course, treatment appropriateness cannot be regarded as an independent variable. : extremities; GU: genitourinary. Soft tissue sarcomas in adolescents and young adults: a comparison with their paediatric and adult counterparts. After over a year of chemo, some radiation and 2 surgeries he is finally on his last week on chemo. ALVEOLAR RHABDOMYOSARCOMA OF THE MANDIBLE IN AN ADULT INVADING THE PAROTID GLAND: A RARE CASE REPORT. In principle, an overall score of 1 corresponded to a treatment regimen that was entirely consistent with current recommendations for treatment of pediatric RMS. Rhabdomyosarcoma is more common in children and teenagers than in adults. Embryonal Rhabdomyosarcoma of the Cervix in Young Women. The log rank test12 was used to compare the survival curves of the patient subgroups in a univariate analysis to ascertain the potential value of various prognostic factors. But so far it's not clear that either of these approaches is any better than standard chemo, and they can cause more side effects. Adult patients with RMS have a poorer overall survival than pediatric patients, and survival varies considerably across histological subtypes. For people in the high-risk group (which includes those with metastatic disease), the VAC regimen is the most common one used. View issue TOC Volume 98, Issue 3 1 August 2003 Pages 571–580 . Table 3 shows the estimated EFS and OS rates of patients stratified according to various clinical characteristics. Philadelphia, Pa: Lippincott Williams & Wilkins; 2016. Radiotherapy at a total dose < 45 Gy (after incomplete resection) corresponded to a score of 0.6. In: Pizzo PA, Poplack DG, eds. Rhabdomyosarcoma, Spindle Cell/Sclerosing Variant: A Clinical and Histopathological Examination of this Rare Variant with Three New Cases from the Oral Cavity. Radiation may also be employed when complete tumor resection has not been possible. Imaging of Nasal Cavity and Paranasal Sinus Tumors. Outcome of 449 adult patients with rhabdomyosarcoma: an observational ambispective nationwide study. With regard to adult RMS, much of the published literature is from single-institution series, which report on clinical parameters and survival data for adults with RMS. Head and neck area 2. Childhood Rhabdomyosarcoma Treatment (PDQ®). Impact of Rhabdomyosarcoma Treatment Modalities by Age in a Population-Based Setting. Successful treatment of adult pleomorphic rhabdomyosarcoma with bone invasion in the lower leg by chemotherapy and biological reconstruction: A case report. Rhabdomyosarcoma as a very rare tumor in adult: Case series. Six patients had metastases to multiple sites. Metastasis sites were lung (n = 11), bone (n = 6), omentum (n = 5), distant lymph nodes (n = 4), bone marrow (n = 3), soft tissue (n = 3), liver (n = 2), kidney (n = 1), suprarenal gland (n = 1), and brain (n = 1). Twenty‐one patients were alive at the time of the analysis (13 after achieving first CR). Research. Which treatments your doctor recommends will depend on the location of the cancer, the size of the tumor, whether the cells are aggressive, and whether the cancer is confined to one area (localized rhabdomyosarcoma) or it has spread to other areas of the body (metastatic rhabdomyosarcoma). Asia-Pacific Journal of Clinical Oncology. Imagine a world free from cancer. Your child may also be eligible to participate in … The American Cancer Society is a qualified 501(c)(3) tax-exempt organization. Low-dose maintenance chemotherapy given after standard intensive chemotherapy improves both disease-free survival and overall survival in children with rhabdomyosarcoma… This study examines short-term outcomes using doxorubicin, ifosfamide, and vincristine for adult rhabdomyosarcoma. The cases of 190 consecutive patients age 18 years or older who were diagnosed with RMS between 1975 and 2001 were collected from the institutional database of the Pathology Unit of the INT. In the current series, the proportion of patients with alveolar RMS was greater (31% [40% if pleomorphic RMS is excluded] vs. 27%); invasive tumors (65% vs. 44%) and large tumors (73% vs. 50%) were more common; and there was a higher rate of lymph node involvement (31% vs. 15%). Vincristine, Actinomycin and Cyclophosphamide (VAC) based chemotherapy is the current standard. More recently, ultrastructural, immunohistochemical, and molecular techniques have led to refinement of the criteria for diagnosis.26, 27 Pleomorphic RMS is an aggressive neoplasm that probably is closer, both biologically and clinically, to adult, high‐grade soft tissue sarcomas than to pediatric RMS. Consequently, 180 patients were available for the current analysis. Treatment Outcome and Predictors of Survival in Thai Adult Rhabdomyosarcoma Cases. This study examines short-term outcomes using doxorubicin, ifosfamide, and vincristine for adult rhabdomyosarcoma. Having more favorable prognostic indicators at onset probably allowed these patients to receive intensive treatment. Rhabdomyosarcoma in Children and Adolescents: Patterns and Risk Factors of Distant Metastasis. A Mass Filling the Right Atrium: Primary Cardiac Rhabdomyosarcoma. Combined Sclerosing and Spindle Cell Rhabdomyosarcoma in Previous Craniotomy Site: A Case Report and a Review of the Literature. 11073 Background: RMS typically occurs in children. Systemic chemotherapy is a routine component of treatment for several STS that occur predominantly in children (eg, rhabdomyosarcoma, Ewing sarcoma, and osteogenic sarcoma). Overall, 109 patients received radiotherapy. Access to clinical trials for adolescents with soft tissue sarcomas: Enrollment in European pediatric Soft tissue sarcoma Study Group (EpSSG) protocols. Next article in issue: The association of P-glycoprotein with response to chemotherapy and clinical outcome in patients with osteosarcoma. Rhabdomyosarcoma in Adults: New Perspectives on Therapy, Tumor size and resectability are prognostic factors, Pathologic review from the Swedish Cancer Registry, Advanced disease in 87% of patients Contains discussion of possible explanations for poor outcomes for adult patients with ‘pediatric’ tumors, 50% rate of response to CT 8 of 13 patients died of disease, Paratesticular site Unfavorable clinical presentation, disappointing outcome, Comparison between adults and children Age is an independent prognostic factor, Adult RMS behaves similarly to other adult sarcomas No evidence that CT is of any benefit, Metastatic relapse was the primary cause of failure CT response and survival are correlated, Tumor size is the principal prognostic factor Improved outcome in patients treated with CT (not statistically significant). Subcutaneous ectopic model right Atrium: primary Cardiac rhabdomyosarcoma 3 ) tax-exempt organization chemoresponsiveness of this rare Variant with new... 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