Clinical Outcome of Parosteal Osteosarcoma

Won Seok Song; Dae-Geun Jeon; Wan Hyeong Cho; Chang Bae Kong; Sang Hyun Cho; Kwang Ryul Lee; Soo-Yong Lee

doi:10.5292/jkbjts.2013.19.1.20

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Song, Jeon, Cho, Kong, Cho, Lee, and Lee: Clinical Outcome of Parosteal Osteosarcoma

Original Article

J Korean Bone Joint Tumor Soc 2013;19(1):20-27.

Published online: 10 January 2013

DOI: https://doi.org/10.5292/jkbjts.2013.19.1.20

Clinical Outcome of Parosteal Osteosarcoma

Won Seok Song, Dae-Geun Jeon, Wan Hyeong Cho, Chang Bae Kong, Sang Hyun Cho, Kwang Ryul Lee, Soo-Yong Lee

Department of Orthopedic Surgery, Korea Cancer Center Hospital, Seoul, Korea

Correspondence to: Dae-Geun Jeon Department of Orthopedic Surgery, Korea Cancer Center Hospital, 215-4, Gongneung-dong, Nowon-gu, Seoul 139-706, Korea TEL: +82-2-970-1242 FAX: +82-2-970-2403 E-mail: dgjeon@kcch.re.kr

Received 25 June 2013 Revised 14 May 2013 Accepted 20 May 2013

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Purpose

The purpose of this study was to evaluate the oncologic outcomes of parosteal osteosarcoma (POS) and to ascertain the fates of patients after local recurrence (LR).

Materials and Methods

The authors retrospectively reviewed 22 POS patients with an average follow-up of 114 months (range: 36–235 months). Seven of the 22 patients were referred after LR. There were 17 Stage IB and 5 Stage IIB (G2, 2; dedifferentiation, 3). Tumors were located in the femur (11) and in other locations (11). Initial surgical margins were wide in 10, marginal in 5, and intralesional in 7. Correlations between clinico-pathologic variables and LR and clinical courses after LR were evaluated.

Results

The 10-year overall survival rate was 85.7%. Three (14%) patients developed distant metastasis and all of them succumbed to the disease. Nine (41%) patients developed LR. Tumor location, resection type, and surgical margin were found to be correlated with LR. At final follow-up, 7 of the 9 patients that experienced local failure achieved no evidence of disease.

Conclusion

A substantial risk of misdiagnosis exists, especially for POS in other than a femoral location. Recurrent tumor re-excision is possible in most cases; however, patients with an aggressive recurrence pattern deserve special attention.

Keywords: parosteal osteosarcoma, local recur

References

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Figure 1.

The 10-year overall and event free survival rates were determined using the Kaplan-Meier plot.

Figure 2.

Patient 21 was a 37-year-old woman who was misdiagnosed as Nora's lesion. (A) Initial anteroposterior radiograph shows an ossified mass on the posterolateral aspect of humerus. (B) This anteroposterior radiograph was taken after 4 episodes of intralesional procedure at referral hospital. Note ill-defined calcified nodules which were located around proximal humerus. (C) The patient underwent segmental excision and reconstruction with recycled autograft. Local recurrence was noted on this anteroposterior radiograph taken after 3 month later. Concomitant metastasis was also identified.

Figure 3.

A diagram shows the outcome in a meta-analysis of 21 locally recurrent patients. Nearly a half (10/21) of them underwent amputation to manage the local recurrence (LR, local recurrence; NED, no evidence of disease; DOD, dead of disease).

Table 1.

Patient Demographics and Treatment Outcomes

Case number	Age/ Gender	Anatomical site	Stage/Grade	Medullary invasion	Soft tissue mass	Surgery	Initial surgical margin	Recurrence	Outcome	Follow up (months)
1	20/M	Distal femur	IIB/dedifferentiated	+	+	En bloc excision	Wide	None	CDF	98
2	21/F	Proximal humerus	IIB/dedifferentiated	+	+	En bloc excision	Marginal	Local	NED	209
3	22/M	Distal femur	IB/grade 1	−	−	Hemicortical excision	Wide	None	CDF	93
4	23/M	Scapula	IB/grade 1	−	−	En bloc excision	Wide	None	CDF	131
5	23/F	Distal femur	IB/grade 1	−	−	En bloc excision	Wide	None	CDF	84
6	23/F	Proximal humerus	IIB/grade 2	+	+	En bloc excision	Marginal	Distant	DOD	48
7	24/F	Distal femur	IB/grade 1	−	−	Hemicortical excision	Marginal	None	CDF	69
8	25/F	Distal humerus	IB/grade 1	−	−	Hemicortical excision	Marginal	None	CDF	112
9	27/F	Distal femur	IB/grade 1	+	+	En bloc excision	Wide	None	CDF	204
10	30/M	Femur diaphysis	IB/grade 1	−	−	En bloc excision	Wide	None	CDF	203
11	30//F	Distal femur	IB/grade 1	−	+	En bloc excision	Wide	None	CDF	94
12	34/M	Proximal femur	IB/grade 1	+	−	En bloc excision	Wide	None	CDF	235
13	34/M	Distal tibia	IB/grade 1	+	−	En bloc excision	Wide	None	CDF	73
14	37/M	Distal femur	IIB/grade2	−	+	Hemicortical excision	Marginal	Local/Distant	DOD	36
15	68/M	Distal femur	IIB/dedifferentiated	+	+	Amputation	Wide	None	CDF	97
16^*	6/F	Talus	IB/grade 1	−	−	Lumpectomy	Intralesional	Local	NED	175
17^*	18/F	Proximal ulna	IB/grade 1	+	+	Lumpectomy	Intralesional	Local	NED	136
18^*	18/F	Distal femur	IB/grade 1	−	−	Lumpectomy	Intralesional	Local	NED	62
19^*	27/F	Proximal tibia	IB/grade 1	−	−	Lumpectomy	Intralesional	Local	NED	101
20^*	31/F	Proximal tibia	IB/grade 1	+	+	Lumpectomy	Intralesional	Local	NED	76
21^*	37/F	Proximal humerus	IB/grade 1	−	+	Lumpectomy	Intralesional	Local/ Distant	DOD	49
22^*	38/F	Distal radius	IB/grade 1	−	+	Lumpectomy	Intralesional	Local	NED	104

^* Referred patients. CDF, continous disease free; NED, no evidence of disease; DOD, dead of disease; AWD, alive with disease.

Table 2.

Patient and Tumor Characteristics of 9 Patients with Local Recurrence and 13 Patients without Local Recurrence

	Variables	Recurred (%)	Not recurred (%)	p-value
Age	≤30	5 (33.3%)	10 (66.7%)	0.38
Age	>30	4 (57.1%)	3 (42.9%)
Gender	Male	1 (12.5%)	7 (87.5%)	0.07
Gender	Female	8 (57.1%)	6 (42.9%)
Initial stage	IB	7 (41.2%)	10 (58.8%)	1.00
Initial stage	IIB	2 (40.0%)	3 (60.0%)
Histologic grade	Grade 1	7 (41.2%)	10 (58.8%)	0.93
	Grade 2	1 (50.0%)	1 (50.0%)
	Dedifferentiated	1 (33.3%)	2 (66.7%)
Tumor volume	Mean (range)	91.9 (0.9–125)	397.9 (12.6–4,552)	0.48
	≤50 ml	6 (40.0%)	9 (60.0%)	1.00
	>50 ml	3 (42.9%)	4 (57.1%)
Location	Femur	2 (18.2%)	9 (81.8%)	0.03
Location	Others	7 (63.6%)	4 (36.4%)
Medullary invasion	Yes	3 (33.3%)	6 (66.7%)	0.67
Medullary invasion	No	6 (46.2%)	7 (53.8%)
Soft tissue mass	Yes	6 (54.5%)	5 (45.5%)	0.39
Soft tissue mass	No	3 (27.3%)	8 (72.7%)
Surgery	En bloc/amputation	1 (9.1%)	10 (90.9%)	< 0.01
	Hemicortical	1 (25.0%)	3 (75.0%)
	Lumpectomy	7 (100.0%)	0 (0.0%)
Margin	Wide	0 (0.0%)	10 (100.0%)	< 0.01
	Marginal	2 (40.0%)	3 (60.0%)
	Intralesional	7 (100.0%)	0 (0.0%)
Total		9 (40.9%)	13 (59.1%)

Table 3.

Treatment and Outcome of Patients with Local Recurrence

Case number	Initial pathologic diagnosis	Type of initial operation	Time to 1st LR (months)	No. of intralesional or marginal procedure	Operation to achieve wide margin	Subsequent recurrence (months)	No. of subsequent recurrence	Metastasis^† (site)	Outcome	Follow up(months)
2	Dedifferentiated POS	En-bloc resection	10	1	Re-excision	36	1	−	NED	209
14	POS G2	Hemicortocal excision	13	1	En-bloc excision & tumor prosthesis	8	2	21 (lung)	DOD	36
16^*	Osteochondroma	Lumpectomy	18	4	Talectomy	39	1	−	NED	175
17^*	POS G1	Lumpectomy	15	2	En-bloc excision & recycled autograft	5	1	−	NED	136
18^*	POS G1	Lumpectomy	7	1	Hemicortocal excision	None	None	−	NED	32
19^*	POS G1	Lumpectomy	17	1	En-bloc excision & recycled autograft	40	1	−	NED	81
20^*	POS G1	Lumpectomy	10	1	En-bloc excision & recycled autograft	38	1	−	NED	56
21^*	Nora's lesion	Lumpectomy	21	3	Segmental resection & recycled autograft	3	1	38 (lung, soft tissue)	DOD	49
22^*	POS G1	Lumpectomy	20	2	En-bloc excision & recycled autograft	None	None	−	NED	84

^* Referred case

^† Months after initial treatment. LR, local recurrence; POS, parosteal osteosarcoma; NED, no evidence of disease; AWD, alive with disease.

Table 4.

Summary of Publications Concerning Parosteal Osteosarcoma

Author/Year	Patient number	Enneking stage(IB/IIB/III)	Histologic grade			Location		Surgical margin (1st OP)			Local recurrence (%)	Metastasis (%)	Overall survival	Mean FU duration (yr)
Author/Year	Patient number	Enneking stage(IB/IIB/III)	G1	G2	Dedifferentiated	Femur (%)	Others	Intralesional	Marginal	Wide	Local recurrence (%)	Metastasis (%)	Overall survival	Mean FU duration (yr)
Temple et al. 2000	38	25/12/1	26	11	1	29(76%)	9	2	19	17	4/38 (11%)	1/37(3%)	38/38 (100%)	6.75 (0.5–19)
Okada et al. 1994	226^*(67)	NA/NA/0	157	32	37	142(63%)	84	6	25	35	33/67(49%)	14/67(21%)	56/67(84%)	13 (2–41)
Ritschl et al. 1991	33	NA	23	9	1	23(70%)	10	10	3	20	11/33(33%)	5/33(15%)	29/33(88%)	8(2–23)
Han et al. 2008	21	7/14/2000	7	11	3	16(76%)	5	2	6	13	2/21(10%)	1/21(5%)	20/21(95%)	9.1(2.5–22)
Current study	22	17/5/0	17	2	3	11	11	7	5	10	9/22(41%)	3/22(14%)	3/22(86%)	9.5(3–19.5)

^* 226 patients were registered and outcomes of 67 (managed at that center) patients were presented. OP, operation; FU, follow up; G, grade; NA, not assessed.

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