Rapid and Complete Remission of Metastatic Adrenocortical Carcinoma Persisting 10 Years After Treatment With Mitotane Monotherapy: Case Report and Review of the Literature.

Abstract:

:Mitotane has been used for more than 5 decades as therapy for adrenocortical carcinoma (ACC). However its mechanism of action and the extent of tumor response remain incompletely understood. To date no cases of rapid and complete remission of metastatic ACC with mitotane monotherapy has been reported. A 52-year-old French Canadian man presented with metastatic disease 2 years following a right adrenalectomy for stage III nonsecreting ACC. He was started on mitotane which was well tolerated despite rapid escalation of the dose. The patient course was exceptional as he responded to mitotane monotherapy after only few months of treatment. Initiation of chemotherapy was not needed and he remained disease-free with good quality of life on low maintenance dose of mitotane during the following 10 years. A germline heterozygous TP53 exon 4 polymorphism c.215C>G (p. Pro72Arg) was found. Immunohistochemical stainings for IGF-2 and cytoplasmic β-catenin were positive. Advanced ACC is an aggressive disease with poor prognosis and the current therapeutic options remain limited. These findings suggest that mitotane is a good option for the treatment of metastatic ACC and might result in rapid complete remission in selected patients.

journal_name

Medicine (Baltimore)

journal_title

Medicine

authors

El Ghorayeb N,Rondeau G,Latour M,Cohade C,Olney H,Lacroix A,Perrotte P,Sabourin A,Mazzuco TL,Bourdeau I

doi

10.1097/MD.0000000000003180

subject

Has Abstract

pub_date

2016-03-01 00:00:00

pages

e3180

issue

13

eissn

0025-7974

issn

1536-5964

pii

00005792-201603290-00014

journal_volume

95

pub_type

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