Therapeutic Management of a Substantial Pelvic Aneurysmatic Bone Cyst Including the Off-Label Use of Denosumab in a 35-Year-Old Female Patient

D Ntalos,J M Rueger,D M Thiesen,C Schlickewei,M Priemel,A S Spiro

doi:10.1155/2017/9125493

D Ntalos, J M Rueger + Show 4 more

Open Access

https://doi.org/10.1155/2017/9125493

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Abstract

Aneurysmal bone cysts (ABC) are benign bone tumors, which are highly vascularized. The main course of treatment is curettage followed by bone grafting or cement insertion. Still recurrence remains a main problem for patients. Denosumab is a monoclonal antibody, which acts as an inhibitor of the RANK/RANKL pathway, diminishing bone turnover. Recent case reports have shown that Denosumab can be a promising therapeutic agent for people suffering from therapy-resistant ABC. We report the case of a 35-year-old female patient presenting with a pronounced ABC of the pelvis. Since the tumor was inoperable, Denosumab was administered, leading to a significant shrinkage of the lesion, which allowed surgical intervention. Upon recurrence, Denosumab was restarted putting the patient once more into remission. Follow-up was four years overall with a clinical and radiological stable disease for fifteen months after final discontinuation of the monoclonal antibody. Therefore, our case further underlines the potential of Denosumab in the treatment of ABC.

Highlights

Aneurysmal bone cysts (ABC) were first described in 1942 by Jaffe and Lichtenstein and account for almost one per cent of all benign bone tumors [1, 2]
Treatment options for aneurysmal bone cysts comprise surgical resection or curettage followed by bone grafting or cement insertion as well as arterial embolization or direct injection of the cyst with sclerosing agents such as polidocanol or local injection of doxycycline [8,9,10]
The five existing cases show very promising results for Denosumab being a treatment option for ABC though, which is in accordance with our findings [12,13,14,15,16]

Summary

Introduction

Aneurysmal bone cysts (ABC) were first described in 1942 by Jaffe and Lichtenstein and account for almost one per cent of all benign bone tumors [1, 2]. Involvement of the pelvis is rather rare and occurs, depending on the study, in four to twelve per cent of the cases [2, 4, 5]. The pathogenesis of these lesions, having been known for more than seventy years, still remains unclear. The five existing cases show very promising results for Denosumab being a treatment option for ABC though, which is in accordance with our findings [12,13,14,15,16]

Case Report

Findings

Discussion