A literature search was performed on August 1st, 2020, of the PubMed, Medline, and Cochrane Library databases, with the search covering studies published from 1990 to 2019. The eligibility criteria specified studies and case reports regarding patients with primary intra-osseous CSs involving the L-spine who received surgical interventions. Articles including descriptions of the surgery methods, follow-up time, recurrence, and outcomes were further selected and investigated manually. We searched the databases using three keywords, namely, “primary”, “chondrosarcoma”, and “spine”, filtered with the race of Human, which resulted in 160 articles being identified. We next conducted a screening of the abstracts of those articles, excluding those articles in which the lesion did not involve the L-spine, those not about surgical interventions, or those in which the tumors were extra-osseous. After reviewing the full-text articles, we further excluded some articles or cases due to the disease locations, distinct tumor types, or incomplete descriptions of the surgical methods used. As a result, 8 articles discussing 23 cases were selected.
The characteristics of the selected cases, including the present case, are listed in Table 1. The average age of the patients was 39.42 ± 19.8 year-old (Mean ± SD), with 13 being male and 11 being female. The most common CS locations were L4 and L5, both of which accounted for 20.8% of the total cases.
Table 1
List of primary intra-osseous lumbar CS cases receiving surgical intervention
No | Article | Location | Age | Sex (years) | Staging (Enneking) | OP method | Reconstruction | Adjuvant therapy |
1 | Gösling et al., 2013 10 | T11-L1 | 43 | M | | multilevel en bloc spondylectomy with resection and replacement of the aorta in two-stage surgery | yes, mesh | no |
2 | Boriani et al., 2000 11 | L4 | 44 | F | IB | en bloc | no | no |
3 | L4 | 20 | M | IB | en bloc | no | no |
4 | L3 | 34 | M | IB | en bloc | no | no |
5 | L2 | 31 | F | IB | en bloc (corpectomy with anterior spinal fusion) | yes, ISOLA® and carbon fiber prosthesis | no |
6 | L5 | 64 | M | IIB | en bloc | yes, ISOLA® 1 auto-graft | no |
7 | L3 | 68 | F | IB | en bloc | yes, ISOLA® 1 carbon fiber prosthesis | no |
8 | L5 | 32 | F | IB | en bloc | yes, ISOLA® 1 auto-graft | no |
9 | L2-L3 | 41 | M | IIB | en bloc | yes, ISOLA® 1 CFP | PBth |
10 | L4 | 23 | F | IB | piece meal excision | no | RT |
11 | L2-L3 | 33 | M | IB | piece meal excision | no | RT |
12 | L2 | 56 | M | IIB | piece meal excision | no | no |
13 | L5 | 21 | M | IB | piece meal excision | no | no |
14 | L2-L4 | 42 | M | IB | piece meal excision | no | no |
15 | L3 | 78 | F | IB | piece meal excision | yes, VSP | RT |
16 | Hsu et al., 2011 12 | T12-L2 | 52 | M | | two-stage en bloc (posterior laminectomy with TPS fixation, then anterior spondylectomy) | yes, distractible titanium cage | no |
17 | Marmor et al., 2001 13 | L4 | 64 | F | | two-stage en bloc (posterior laminectomy TPS then anterior lateral spondylectomy) | yes, methylmethacrylate cage | no |
18 | Matsuda et al., 2006 14 | L1-L2 | 44 | M | | two-stage en bloc (laminectomy and T11-T12-L3-L4 fixation, then anterior lateral spondylectomy of L1-L2 and partial T12 with titanium mesh cage) | yes, mesh | no |
19 | Schoenfeld, et al., 2012 15 | L | 64 | F | IB | en bloc (by posterior and anterior approach with posterior instrumentation) | | RT (and intraoperative RT) |
20 | | L | 40 | F | IIB | intralesional excision | | RT |
21 | Tasdemiroglu et al., 1996 16 | L1-L2 | 1 | F | | laminectomy and subtotal resection | no | RT and CT |
22 | | L5 | 12 | F | | laminectomy and gross total resection with TPS fixation | no | RT and CT |
23 | Zibis AH et al., 2010 17 | L5 | 9 | M | | two-stage en bloc (posterior laminectomy with TPS fixation, then paramedian retroperitoneal anterior approach corpectomy) | yes, titanium cage and bone graft | Neoadjuvant and adjuvant CT, and RT |
24 | Current case | L3 | 30 | M | IB | two-stage en bloc (posterior laminectomy with TPS fixation, then anterior spondylectomy) | yes, titanium cage and plate | no |
Table 1
List of primary intra-osseous lumbar CS cases receiving surgical intervention (continued)
No | Article | Intralesional | Patho grade | Histologic type | Follow-up (mo) | Recurrence (mo) | Complication | Survival |
1 | Gösling et al., 2013 | yes | low | Conventional | 48 | no | transient cord injury | NED |
2 | Boriani et al., 2000 | no | | Conventional | 207 | yes (48) | | NED |
3 | no | | Conventional | 169 | no | | NED |
4 | no | | Conventional | 163 | no | | NED |
5 | yes | | Conventional | 60 | yes (30) | | DOD |
6 | no | | Conventional | 45 | no | | DUD |
7 | no | | Conventional | 45 | no | | NED |
8 | no | | Conventional | 30 | no | | NED |
9 | yes | | Conventional | 30 | yes (12) | | NED |
10 | yes | | Conventional | 80 | yes (22) | | DOD |
11 | yes | | Conventional | 119 | yes (37) | | DOD |
12 | yes | | Conventional | 3 | progression | | DOD |
13 | yes | | Conventional | 2 | progression | | DOD |
14 | yes | | Conventional | 36 | yes (15) | | DOD |
15 | yes | | Conventional | 10 | progression | | DOD |
16 | Hsu et al., 2011 | yes | | Clear cell | 24 | no | | NED |
17 | Marmor, et al., 2001 | no | | | 6 (days) | no | | NED |
18 | Matsuda et al., 2006 | no | | Mesenchymal | 60 | no | | NED |
19 | Schoenfeld, et al., 2012 | no | | | 72 | no | | NED |
20 | | yes | | | 24 | yes (24) | | DOD |
21 | Tasdemiroglu et al., 1996 | yes | | Poorly differentiated mesenchymal | 20 | | | under CT |
22 | | no | | Mesenchymal | 20 | no | | NED |
23 | Zibis AH et al., 2010 | yes | | Mesenchymal | 108 | no | | NED |
24 | Current case | no | low | Conventional | 12 | no | | NED |
PBth, proton beam therapy; RT, radiotherapy; CT, chemotherapy; NED, no evidence of disease; DOD, death of disease; DUD, death unrelated with disease. |
Further analysis was performed, and the results are shown in Table 2. Among the 16 patients who underwent en bloc surgery, 7 received a two-stage surgery, while 9 received a one-stage surgery. The piece meal/subtotal group had a significantly lower survival rate (p < 0.001, using Chi-square test). The recurrence rate of that group was also higher (87.5%) than that of the en bloc group (18.8%). Two patients in the two-stage en bloc group received adjuvant chemotherapy due to the use of an intralesional approach or a contaminated margin, whereas 2 patients in the one-stage en bloc group received adjuvant chemotherapy according to the chemotherapy guidelines for pediatric sarcoma. The results showed a better prognosis in general following en bloc surgery, a finding which was compatible with those of previous studies.4,6−8
Table 2
Analysis of primary intra-osseous lumbar CS cases receiving surgical intervention
Intervention Type | Case number | Age (Mean ± SD) | Mean f/u time (mo) | Single location | Multiple location | Adjuvant Therapy | IL | Patho | Survival | Recurrence |
En bloc | all | 16 | 40.75 ± 18.5 | 68 | 12 | 4 | 4 | 5 | | | 3 |
| two-stage | 7 | 43.71 ± 19.5 | 45 | 4 | 3 | 2 | 4 | 2m, 1cc, 2c | all NED | 0 |
| one-stage | 9 | 38.44 ± 18.4 | 85 | 8 | 1 | 2 | 2 | 1cc, 8c | 1 DOD; 1DUD; 7NED | 3 |
Subtotal/Piece | | 8 | 36.75 ± 23.4 | 37 | 5 | 3 | 5 | 8 | 1m, 2c | 7 DOD, one under CT | 7 progressed |
IL, intralesional; m, mesenchymal; cc, clear cell; c, conventional; NED, no evidence of disease; DOD, death of disease; DUD, death unrelated with disease; CT, chemotherapy |
Nearly half of the patients in the en bloc surgery group received a two-stage surgery (43.7%), including the patient in our case. Patients with multiple segment disease were inclined to receive two-stage approaches (75%, 3 of 4 patients). In contrast, one-stage approaches were preferred for single segment lesions (66.7%, 8 of 12 patients). All the subtotal/piece meal (100%) approaches were intra-lesional; however, only 31.2% of the en bloc cases were treated using an intra-lesional route (2 one-stage and 4 two-stage cases).
The most common pathological type of osseous CS among the collected cases was the conventional type, which accounted for 50% of all the cases. Notably, 8 of 9 (88.9%) one-stage en bloc cases were the conventional type. However, some of the studies and case reports did not have any description of the histologic type of the CSs in question. The mean follow-up time for the two-stage group was 45 months, while that for the one-stage group was 85 months. There was no recurrence noted in the two-stage group, while the recurrence rate in the one-stage group was 33% (3 of 9 patients, with one death from the disease). However, there was no significant difference in the recurrence rate between these two groups (p = 0.09, Chi-square test). All of the en bloc cases (2/2) with the mesenchymal type of CS underwent a two-stage surgery but no subsequent recurrence or death occurred.