Bone marrow biopsy in the initial staging of Ewing sarcoma: Experience from a single institution

Abstract

Background Ewing sarcoma (ES) is the second most common bone tumor in adolescents and children. Staging workup for ES includes imaging and bone marrow biopsy (BMB). The effective role of BMB is now under discussion. Procedure A monoinstitutional retrospective analysis reviewed clinical charts, imaging, and histology of patients with diagnosis of ES treated at the Rizzoli Institute between 1998 and 2017. Results The cohort included 504 cases of ES of bone; 137 (27%) had metastases at diagnosis, while the remaining 367 had localized disease. Twelve patients had a positive BMB (2.4%). Eleven had distant metastases detected at initial workup staging with imaging assessment: six patients presented with bone metastases, five with both bone and lung metastases. Only one patient with ES of the foot (second metatarsus) was found to have bone marrow involvement with negative imaging evaluation (0.3%). Conclusions On the basis of our data, we suggest reconsidering the effective role of BMB in initial staging workup for patients with ES with no signs of metastases by modern imaging techniques. In metastatic disease, the assessment of the bone marrow status may remain useful to identify a group of patients at very high risk who could benefit from different treatment strategies.

Role of bone marrow biopsy for staging new patients with Ewing sarcoma: A systematic review

“…This latter approach is consistent with conclusions drawn by several studies included in this review. 21,24,26,29 While some studies reported an association of BMM disease with primary tumors of the pelvis, 24,27 it is not clear if this finding represents sampling of primary tumor tissue during BMBA procedure. Based upon the available evidence, we do not recommend BM sampling in the setting of a primary pelvic tumor if diagnostic workup reveals no concern for metastatic lesions.…”

“…Though rare, we identified 18 reported cases of newly diagnosed ES patients with isolated BMM. 24,25,27,36,37 In eight cases, location of the primary tumor was provided, with 63% (5/8) reporting location in the appendicular skeleton. 24,25,27 On review of these individual cases, no significant associations to aid in their identification were observed.…”

“…Combined, these studies comprised 142 patients (median 23 patients/study; range16-80 patients). 24,27 Tumor diameter (≥80 mm) and LDH level at diagnosis (>2x ULN) were reported to be factors significantly related to BM involvement by Inagaki et al 29 Oberlin et al reported that fever at diagnosis was significantly associated with the presence of BMM. 27 One study analyzed the concordance between BM aspirate (BMA) and BM biopsy (BMB).…”

“…We identified 10 studies providing quantitative data describing the incidence of BMM in all unselected patients with newly diagnosed ES (Table 1). 21,[23][24][25][26][27][28][29][30][31] These studies ranged in year of publication from 1995 to 2020. Eight studies were completed in a retrospective manner, one was a phase 3 clinical trial, and one was a pilot trial.…”

“…(8/692) of patients with isolated BMM. 24,25,27 Ten additional patients with isolated BM metastatic disease were identified in the literature for a total of 18. Eight of these 18 cases reported the location of the primary tumor: three pelvic primary tumors and five located in the appendicular skeleton.…”