Abstract：
BACKGROUND:Pheochromocytoma (PCC) and paraganglioma (PGL) are uncommon neoplasms with high morbidity in advanced stages. Effective systemic treatments are limited. METHODS:A multisite phase 2 trial evaluated sunitinib in patients with progressive PCC/PGL. Patients received 50 mg orally for 4-6 weeks. RESULTS:Between May 2009 and May 2016, 25 patients were enroled. The median age was 50 years and 56% were male. Three patients (12%) received prior chemotherapy and 16 (64%) prior surgery. The DCR was 83% (95% CI: 61-95%) and median PFS 13.4 (95% CI: 5.3-24.6) months. Of 23 evaluable patients, 3 (13%) with germline mutations (SDHA, SDHB, RET) achieved a PR. The patient with mutated RET and MEN2A remains on treatment after 64 cycles. The median time on treatment was 12.4 (1-88.0) months. Grade 3 or 4 toxicities were as expected and manageable; fatigue (16%) and thrombocytopenia (16%) were most common. One patient with grade 3 hypertension and 2 with grade 3 cardiac events discontinued treatment. CONCLUSION:Although the primary endpoint of disease control was met, the overall response rate of sunitinib was low in unselected patients with progressive PCC/PGL. Patients with germline variants in RET or in the subunits of SDH may derive greatest benefit.
journal_name
Br J Cancerjournal_title
British journal of cancerauthors
O'Kane GM,Ezzat S,Joshua AM,Bourdeau I,Leibowitz-Amit R,Olney HJ,Krzyzanowska M,Reuther D,Chin S,Wang L,Brooks K,Hansen AR,Asa SL,Knox JJdoi
10.1038/s41416-019-0474-xsubject
Has Abstractpub_date
2019-06-01 00:00:00pages
1113-1119issue
12eissn
0007-0920issn
1532-1827pii
10.1038/s41416-019-0474-xjournal_volume
120pub_type
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