Partial response to erlotinib in a patient with imatinib-refractory sacral chordoma
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Clinical Sarcoma Research Open Access
CASE REPORT
Partial response to erlotinib in a patient with imatinib‑refractory sacral chordoma Saurav Verma1, Surya Prakash Vadlamani1, Shamim Ahmed Shamim2, Adarsh Barwad3, Sameer Rastogi4* and S. T. Arun Raj2
Abstract Background: Chordoma is a rare, slow growing and locally aggressive mesenchymal neoplasm with uncommon distant metastases. It is a chemo-resistant disease with surgery and radiotherapy being the mainstay in treatment of localized disease. In advanced disease imatinib has a role. We report a case of metastatic sacral chordoma with symptomatic and radiological response to erlotinib post-progression on imatinib. Case presentation: A 48-year-old male with a sacral chordoma underwent partial sacrectomy followed by postoperative radiotherapy. Upon recurrence he received palliative radiotherapy to hemipelvis and was offered therapy with imatinib. However, the disease was refractory to imatinib and he was started on treatment with erlotinib—showing a partial response on imaging at two months. He is currently doing well at 13 months since start of erlotinib. Conclusions: As seen in previously reported cases, erlotinib is a therapeutic option in advanced chordoma, even in imatinib refractory cases and thus warrants exploration of its therapeutic role in prospective clinical trials. Keywords: Chordoma, EGFR, Erlotinib Background Chordoma is a rare mesenchymal neoplasm which arises from the remnants of primitive notochord [1]. It accounts for 2–4% of all malignant bone tumors and the median age of presentation is 59 years (range 19–70 years) with a male predominance [2, 3]. They occur exclusively in spine, predominantly at sacrococcygeal and sphenooccipital areas, at a median or paramedian location [4]. It is a slow growing locally aggressive tumor with uncommon distant metastases, athough it can cause compressive symptoms. Surgery significantly improves overall survival and is the primary modality of therapy for the localized disease [5]. Radiotherapy also plays a key role in the management of patients with localized chordoma, particularly in the *Correspondence: [email protected] 4 Sarcoma Medical Oncology Clinic, All India Institute of Medical Sciences, New Delhi, India Full list of author information is available at the end of the article
adjuvant setting after a full or subtotal resection, and as the primary treatment in unresectable disease. Chordoma responds poorly to cytotoxic chemotherapy. Azzarelli et al. in a case series of 33 patients, concluded that none of the chemotherapeutic regimen induced a significant tumor response [6]. This intrinsic chemoresistance of chordoma paved the way to different antitumor approaches. In patients with advanced disease, novel therapeutic strategies are needed to prolong survival and improve the quality of life. Imatinib, which has off-target effects acting through the inhibition of platelet-derived growth factor receptor beta (PDGFRβ), is the most thoroughly evaluated therapeutic agent in chordoma, based on the expr
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