Open Access

Epidemiological characteristics of primary spinal osseous tumors in Eastern China

  • Zhenhua Zhou1,
  • Xudong Wang1,
  • Zhipeng Wu1,
  • Wending Huang1 and
  • Jianru Xiao1Email author
Contributed equally
World Journal of Surgical Oncology201715:73

DOI: 10.1186/s12957-017-1136-1

Received: 25 September 2016

Accepted: 22 March 2017

Published: 4 April 2017

Abstract

Background

Primary spinal osseous tumors are rare, yet they represent a difficult treatment paradigm because of the complexities of tumor resection and significant resistance to chemotherapy and radiation therapy. The geographic distribution of primary spinal osseous tumors throughout the world appears to be quite variable, with a very low incidence reported in Asian countries.

Methods

Data on 1209 cases of primary spinal osseous malignant and benign tumor cases diagnosed during the 20-year period of 1995 through 2015 in eastern China were analyzed.

Results

In 780 cases (64.5%), the lesion was benign and in 429 (35.5%) was malignant. The commonest primary malignant tumors were chordoma (9.8% of all cases) followed by plasma cell myeloma (8.5% of all cases). The most common benign tumor was hemangioma (28.1% of all cases) followed by giant cell tumor of bone (15.7% of all cases) and osteoblastoma (4.4% of all cases). The benign tumors affected men in 33.8% of cases and women in 30.7% of cases, the malignant tumors affected men in 23.7% of cases and women in 11.8%. The mean age (mean ± SD) in the benign group was 34.7 ± 19.8 years and in the malignant group was 47.4 ± 16.5 years. Related symptoms were pain (54.4%), radiculopathy (12.9%), cord compression (9.2%), mass (5.7%), pathological fracture (4.7%), deformity (2.1%), and weight loss (1.9%). The anatomical locations included almost every vertebra of the spine. The thoracic spine (38.1%) was the most common location of the tumors, followed by the cervical spine (27.4%) and lumbar spine (18.4%).

Conclusions

Compared with other similar series reported in the literature from the other countries, our results obtained in a developing country were different in some degree. This large series of primary spinal osseous tumors may reflect fairly well their real incidence and provide a sufficiently detailed perspective on epidemiologic studies of primary spinal osseous tumors in eastern China.

Keywords

Spine Primary osseous tumors Epidemiology

Background

Primary spinal osseous tumors are uncommon; previous reports showed that spinal osseous tumors were comprising 6 ~ 10% of all bone tumors [15]. Little was known about the etiology of spine osseous tumors. Published case reports focused on spine neoplasm usually had a limited case number, and few reports described epidemiological characteristics of spine osseous tumors. However, it is important to understand the etiology of spinal osseous tumors and information regarding the epidemiology of primary spine osseous tumors.

The present study performed an epidemiological analysis of 1209 consecutive tumors of osseous spine registered in 3 collaborating state bone tumor database (Eastern China) between 1995 and 2015. To our knowledge, the present study is the first report of epidemiological data concerning spinal osseous tumors in Asian population. Our findings showed that epidemiological features of primary spinal osseous tumors in xanthoderm, with respect to relative frequency and distribution of the various histologic types, as well as the clinical data, and compare the results with other epidemiological findings from different geographic locations around the world, which could provide valuable clues for epidemiology of primary spine osseous tumors in Asian.

Methods

Data sources

Data for this study were obtained from Bone Tumors and Nervous System Tumors Biobank of Shanghai (BT&NSTBS), Bone Tumor Sample Databases and Digital Information Platform of Shanghai (BTSD&DIPS), and Shanghai Biobank Network of Common Human Tumor Tissue (SBNCHTT). One thousand two hundred nine cases of spinal osseous tumors registered in abovementioned 3 database between 1995 and 2015 were selected for this study. Data collected for each patient included personal information such as name, age, sex, anatomical site of the tumor, and clinical and histological diagnoses. In the case of recurrent tumors, the histological appearance of the original and the recurrent tumors was compared and was considered as only one case. The diagnoses were re-evaluated according to the criteria suggested for the 2013 WHO histological classification [6].

Statistics

SPSS17.0 software (SPSS Inc., Chicago, IL) was used for statistical analysis of experimental data. Descriptive statistics was performed to calculate the frequency and percentages of variables mentioned before. Age was stratified into various groups at 10-year intervals.

Results

The histological types of the spinal osseous tumors are listed in Table 1 and Fig. 1. Of these, 64.5% (780 cases) were benign and 35.5% (429 cases) were malignant, The most common histological type of benign tumors was hemangioma accounting for 28.1% of all tumors (340 cases), followed by giant cell tumor (15.7%; 190 cases), osteoblastoma (4.4%; 53 cases), aneurysmal bone cyst (2.9%; 35 cases), eosinophilic granuloma (3.9%; 47 cases), osteochondroma (3.8%; 46 cases), solitary bone cyst (2.1%; 25 cases), osteoid osteoma (1.4%; 17 cases), fibroma (1.3%; 16 cases), lipoma (0.6%; 7 cases), and fibrous dysplasia (0.3%; 4 cases). Of malignant tumors, chordoma was the most common malignant tumor (9.8% of all tumors, 119 cases), followed by plasma cell myeloma multiple myeloma (8.5%,103 cases), chondrosarcoma (5.2%, 63 cases), malignant lymphoma (4.5%; 54 cases), malignant neurilemmoma (2.5%; 30 cases), primitive neural ectodermal tumor (PNET)\Ewing’s sarcoma (1.4%; 17 cases), malignant fibrous histiocytoma (1.3%; 16 cases), osteosarcoma (1.1%; 13 cases), and other sarcomas such as angiosarcoma, fibrosarcoma, liposarcoma, and leiomyosarcoma (less than 1%).
Table 1

Frequency, age, and gender distribution of primary spine osseous tumors

Type of tumor

Number (%)

Male

Female

Male vs female

Age range (years)

Mean ± SD (age)

No.

%a

No.

%a

No.

%a

Male

Female

Total

Benign tumors

780

64.5

409

33.8

371

30.7

52.4 vs 47.6%

9–79

33.7 ± 19.2

35.9 ± 20.6

34.7 ± 19.8

Hemangioma

340

28.1

174

14.4

166

13.7

51.2 vs 48.8%

12–79

47.1 ± 14.3

51.5 ± 12.9

49.2 ± 13.7

Giant cell tumor

190

15.7

91

7.5

99

8.2

47.9 vs 52.1%

15–66

34.5 ± 11.0

32.9 ± 12.1

33.6 ± 11.5

Eosinophililc granuloma

47

3.9

36

3.0

11

0.9

76.6 vs 23.4%

10–56

23.7 ± 14.3

29 ± 17.6

23.7 ± 14.7

Osteoblastoma

53

4.4

28

2.3

25

2.1

52.8 vs 47.2%

9–54

32.6 ± 10.5

26.5 ± 15.0

28.7 ± 13.2

Fibroma

16

1.3

10

0.8

6

0.5

62.5 vs 37.5%

26–76

41.6 ± 18.2

50.5 ± 15.0

44.9 ± 17.1

Osteoid osteoma

17

1.4

8

0.7

9

0.7

47.1 vs 52.9%

16–67

31.2 ± 22.3

39.0 ± 16.8

34.6 ± 19.8

Osteochondroma

46

3.8

28

2.3

18

1.5

60.9 vs 39.1%

13–64

34.0 ± 16.6

38.7 ± 20.8

36.1 ± 18.4

Solitary bone cyst

25

2.1

8

0.7

17

1.4

32.0 vs 68.0%

15–64

29.5 ± 15.2

48.8 ± 11.9

41.1 ± 16.2

Lipoma

7

0.6

5

0.4

2

0.2

71.4 vs 28.6%

23–63

41.8 ± 16.5

31.0 ± 11.3

38.7 ± 15.2

Aneurysmal bone cyst

35

2.9

19

1.6

16

1.3

54.3 vs 45.7%

10–62

25 ± 12.5

36.4 ± 15.3

30.4 ± 14.8

Fibrous dysplasia

4

0.3

2

0.2

2

0.2

50.0 vs 50.0%

25–32

29.5 ± 3.5

27 ± 2.8

28.3 ± 3.0

Malignant tumors

429

35.5

286

23.7

143

11.8

66.7 vs 33.3%

8–81

48.3 ± 16.7

46.3 ± 16.3

47.4 ± 16.5

PNET/ Ewing’s sarcoma

17

1.4

9

0.7

8

0.7

52.9 vs 47.1%

11–46

26 ± 13.3

25.8 ± 7.9

25.9 ± 10.9

Chordoma

119

9.8

79

6.5

40

3.3

66.4 vs 33.6%

27–81

56.6 ± 15.0

53.3 ± 13.3

55.5 ± 14.1

Malignant fibrous histiocytoma

16

1.3

11

0.9

5

0.4

68.8 vs 31.2%

29–67

51 ± 14.5

50.7 ± 1.9

50.9 ± 11.3

Liposarcoma

1

0.0

1

0.0

0

0.0

NA

44

44

NA

44

Osteosarcoma

13

1.1

5

0.4

8

0.7

38.5 vs 61.5%

14–60

40.2 ± 21.0

25.8 ± 11.7

33.6 ± 18.2

Angiosarcoma

10

0.8

7

0.6

3

0.2

70.0 vs 30.0%

36–73

54.3 ± 11.3

45.0 ± 7.8

51.5 ± 10.9

Malignant neurilemmoma

30

2.5

15

1.2

15

1.2

50.0 vs 50.0%

8–71

40.4 ± 16.3

50.3 ± 12.1

45.3 ± 15.0

Plasma cell myeloma

103

8.5

75

6.2

28

2.3

72.8 vs 27.2%

10–76

49.8 ± 15.3

57.2 ± 12.6

52.0 ± 14.8

Malignant lymphoma

54

4.5

30

2.5

24

2.0

55.6 vs 44.4%

10–77

47.2 ± 21.0

46.5 ± 18.7

46.9 ± 19.6

Leiomyosarcoma

1

0.0

1

0.0

0

0.0

NA

55

55

NA

55

Chondrosarcoma

63

5.2

52

4.3

11

0.9

82.5 vs 17.5%

20–68

46.1 ± 14.5

37.3 ± 14.8

44.0 ± 14.7

Fibrosarcoma

2

0.2

1

0.0

1

0.0

50.0 vs 50.0%

30–61

61

30

45.5 ± 21.9

Total

1209

100

695

57.5

514

42.5

57.5 vs 42.5%

8–81

39.1 ± 16.9

39.6 ± 16.7

39.3 ± 16.8

NA not applicable

a% in all tumors

Fig. 1

Distribution of primary spine osseous tumors. Distribution of a all primary spine osseous tumor, b benign spine osseous, and c malignant spine osseous tumor cases by histological type

Of the benign tumors, 20.5% were situated in the cervical spine, 26.1% in the thoracic spine, 12.0% in the lumbar spine, and 5.4% in the sacral spine. Of the malignant tumors, 6.9% were situated in the cervical spine, 12.0% in the thoracic spine, 6.3% in the lumbar spine, and 10.7% in the sacral spine. Of all tumors, 27.4% were situated in the cervical spine, 38.1% in the thoracic spine, 18.4% in the lumbar spine, and 16.1% in the sacral spine (Table 2).
Table 2

Distribution of spine osseous tumors by location

Type of tumor

Cervical spine

Thoracic spine

Lumbar spine

Sacral spine

Totals

No.

%

No.

%

No.

%

No.

%

No.

%

Benign tumors

257

20.5

328

26.1

151

12.0

68

5.4

804(780)a

64.0

Hemangioma

104

8.3

149

11.7

86

6.8

11

0.9

350(340)a

27.9

Giant cell tumor

44

3.5

87

6.9

23

1.8

36

2.9

190

15.1

Eosinophililc granuoma

20

1.6

22

1.6

5

0.4

0

0.0

47

3.7

Osteoblastoma

17

1.4

30

2.4

5

0.4

1

0.0

53

4.2

Fibroma

17

1.4

5

0.4

5

0.4

3

0.2

30(16)

2.4

Osteoid osteoma

12

1.0

2

0.2

3

0.2

0

0.0

17

1.4

Osteochondroma

21

1.7

12

1.0

11

0.9

2

0.2

46

3.7

Solitary bone cyst

8

0.6

2

0.2

5

0.4

10

0.8

25

2.0

Lipoma

5

0.4

1

0.0

0

0.0

1

0.0

7

0.6

Aneurysmal bone cyst

9

0.7

16

1.3

6

0.5

4

0.3

35

2.8

Fibrous dysplasia

0

0.0

2

0.2

2

0.2

0

0.0

4

0.3

Malignant tumors

87

6.9

151

12.0

80

6.3

134

10.7

452(429) a

36.0

PNET/Ewing’s sarcoma

1

0.0

4

0.3

9

0.7

3

0.2

17

1.4

Chordoma

29

2.3

5

0.4

5

0.4

80

6.4

119

9.5

Malignant fibrous histiocytoma

0

0.0

8

0.6

3

0.2

5

0.4

16

1.3

Liposarcoma

0

0.0

0

0.0

0

0.0

1

0.0

1

0.0

Osteosarcoma

2

0.2

6

0.6

0

0.0

5

0.4

13

1.0

Angiosarcoma

1

0.0

12

1.0

4

0.3

0

0.0

17(10)

1.4

Malignant neurilemmoma

4

0.3

4

0.3

12

1.0

12

1.0

32(30) a

2.5

Plasma cell myeloma

27

2.1

50

4.0

26

2.1

9

0.7

112(103)

8.9

Leiomyosarcoma

0

0.0

0

0.0

0

0.0

1

0.0

1

0.0

Malignant lymphoma

9

0.7

26

2.1

18

1.4

6

0.5

59(54)a

4.7

Chondrosarcoma

14

1.1

36

2.9

2

0.2

11

0.9

63

5.0

Fibrosarcoma

0

0.0

0

0.0

1

0.0

1

0.0

2

0.2

Total

344

27.4

479

38.1

231

18.4

202

16.1

1256(1209) a

100

aMore than two anatomical position involved in the same patient

In our series, most tumors with different histological types showed a similar distribution in males and females, although chordoma (119 cases, M:F = 2:1), plasma cell myeloma (103 cases, M:F = 2.7:1), chondrosarcoma (63 cases, M:F = 4.7:1), and eosinophilic granuoma (47 cases, M:F = 3.3:1) affected more frequently males than females (Table 3). From a total of 1209 bone tumors, the mean age (mean ± SD) was 39.3 ± 16.8 years (range, 8–81 years), 57.5% (695 cases) of the tumors occurred in males and 42.5% (514cases) in females, with a mean age (mean ± SD) of 39.1 ± 16.9 and 39.6 ± 16.7 years. The mean age (mean ± SD) of benign tumors group was 34.7 ± 19.8 years (range, 9–79 years). Of 780 benign tumors, 409 cases (33.8% of all cases) occurred in males and 371 cases (30.7% of all cases) in females (M:F = 1.1:1), with a mean age (mean ± SD) of 33.7 ± 19.2 and 35.9 ± 20.6 years. The mean age (mean ± SD) of malignant tumors group was 47.4 ± 16.5 years (range, 8–81 years). Of 429 malignant tumors, 286 cases (23.7% of all cases) occurred in males and 143 cases (11.8% of all cases) in females, with a mean age (mean ± SD) of 48.3 ± 16.7 and 46.3 ± 16.3 years. The most commonly affected age group for benign tumors was the 31- to 40-year-old group (14.3%; 173 cases), followed by the 51- to 60-year-old group (11.4%; 138 cases), and by the 41- to 50-year-old group (11.2%; 136 cases). The most common age group affected by malignant bone tumors was the 41- to 50-year-old group (7.9%; 95cases), followed by the 61- to 70-year-old group (6.8%; 82 cases). A similar frequency was observed in the 31- to 40-year-old group (6.7%; 81cases) and the 51- to 60-year-old group (6.5%; 79 cases).
Table 3

Age distribution of patients with primary spine osseous tumors (years)

Type of tumor

0–10

11–20

21–30

31–40

41–50

51–60

61–70

71–80

81–90

Total no. (%)

No. (%)

No. (%)

No. (%)

No. (%)

No. (%)

No. (%)

No. (%)

No. (%)

No. (%)

Benign tumors

14(1.2%)

102(8.4%)

122(10.1%)

173(14.3%)

136(11.2%)

138(11.4%)

76(6.3%)

19(1.6%)

0(0.0%)

780(64.5%)

Hemangioma

1(0.0%)

14(1.2%)

18(1.5%)

62(5.1%)

78(6.5%)

96(7.9%)

53(4.4%)

18(1.5%)

0(0.0%)

340(28.1%)

Giant cell tumor

0(0.0%)

27(2.2%)

50(4.1%)

62(5.1%)

32(2.6%)

11(0.9%)

8(0.7%)

0(0.0%)

0(0.0%)

190(15.7%)

Eosinophililc granuloma

3(0.2%)

23(1.9%)

9(0.7%)

5(0.4%)

2(0.2%)

5(0.4%)

0(0.0%)

0(0.0%)

0(0.0%)

47(3.9%)

Osteoblastoma

6(0.5%)

11(0.9%)

14(1.2%)

11(0.9%)

5(0.4%)

6(0.5%)

0(0.0%)

0(0.0%)

0(0.0%)

53(4.4%)

Fibroma

0(0.0%)

0(0.0%)

4(0.3%)

4(0.3%)

2(0.2%)

2(0.2%)

3(0.2%)

1(0.0%)

0(0.0%)

16(1.3%)

Osteoid osteoma

0(0.0%)

5(0.4%)

3(0.2%)

2(0.2%)

2(0.2%)

3(0.2%)

2(0.2%)

0(0.0%)

0(0.0%)

17(1.4%)

Osteochondroma

2(0.2%)

12(1.0%)

8(0.7%)

9(0.7%)

5(0.4%)

5(0.4%)

5(0.4%)

0(0.0%)

0(0.0%)

46(3.8%)

Solitary bone cyst

0(0.0%)

2(0.2%)

3(0.2%)

5(0.4%)

6(0.5%)

6(0.5%)

3(0.2%)

0(0.0%)

0(0.0%)

25(2.1%)

Lipoma

0(0.0%)

0(0.0%)

2(0.2%)

3(0.2%)

1(0.0%)

1(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

7(0.6%)

Aneurysmal bone cyst

2(0.2%)

8(0.7%)

9(0.7%)

8(0.7%)

3(0.2%)

3(0.2%)

2(0.2%)

0(0.0%)

0(0.0%)

35(2.9%)

Fibrous dysplasia

0(0.0%)

0(0.0%)

2(0.2%)

2(0.2%)

0(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

4(0.3%)

Malignant tumors

5(0.4%)

17(1.4%)

36(3.0%)

81(6.7%)

95(7.9%)

79(6.5%)

82(6.8%)

32(2.6%)

2(0.2%)

429(35.5%)

PNET/Ewing’s sarcoma

0(0.0%)

5(0.4%)

7(0.6%)

3(0.2%)

2(0.2%)

0(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

17(1.4%)

Chordoma

0(0.0%)

0(0.0%)

3(0.2%)

17(1.4%)

27(2.2%)

23(1.9%)

32(2.6%)

15(1.2%)

2(0.2%)

119(9.8%)

Malignant fibrous histiocytoma

0(0.0%)

0(0.0%)

2(0.2%)

0(0.0%)

5(0.4%)

6(0.5%)

3(0.2%)

0(0.0%)

0(0.0%)

16(1.3%)

Liposarcoma

0(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

1(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

1(0.0%)

Osteosarcoma

0(0.0%)

6(0.5%)

2(0.2%)

0(0.0%)

3(0.2%)

2(0.2%)

0(0.0%)

0(0.0%)

0(0.0%)

13(1.1%)

Angiosarcoma

0(0.0%)

0(0.0%)

0(0.0%)

2(0.2%)

3(0.2%)

3(0.2%)

1(0.0%)

1(0.0%)

0(0.0%)

10(0.8%)

Malignant neurilemmoma

1(0.0%)

1(0.0%)

1(0.0%)

9(0.7%)

8(0.7%)

5(0.4%)

4(0.3%)

1(0.0%)

0(0.0%)

30(2.5%)

Plasma cell myeloma

2(0.2%)

0(0.0%)

3(0.2%)

17(1.4%)

26(2.2%)

22(1.8%)

24(2.0%)

9(0.7%)

0(0.0%)

103(8.5%)

Leiomyosarcoma

0(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

1(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

1(0.0%)

Malignant lymphoma

2(0.2%)

3(0.2%)

6(0.5%)

9(0.7%)

12(1.0%)

8(0.7%)

8(0.7%)

6(0.5%)

0(0.0%)

54(4.5%)

Chondrosarcoma

0(0.0%)

2(0.2%)

11(0.9%)

24(2.0%)

8(0.7%)

9(0.7%)

9(0.7%)

0(0.0%)

0(0.0%)

63(5.2%)

Fibrosarcoma

0(0.0%)

0(0.0%)

1(0.0%)

0(0.0%)

0(0.0%)

0(0.0%)

1(0.0%)

0(0.0%)

0(0.0%)

2(0.2%)

Total

19(1.6%)

119(9.8%)

158(13.1%)

254(21.0%)

231(19.1%)

217(17.9%)

158(13.1%)

51(4.2%)

2(0.2%)

1209(100%)

As the most common presenting symptom, pain was found in 54.4% (658/1209) of patients, affecting 35.0% (273/780) of the benign and 89.7% (385/429) of the malignant tumors. Mass (swelling) was seen in 4.0% (31/780) of benign tumors and 8.9% (38/429) of malignant tumors. One hundred fifty-six patients (12.9%) had the symptoms of radiculopathy. Of all cases, 9.2% (111/1209) of patients had signs of subtotal or complete cord compression. The cord compression symptoms included motor weakness (8.2%, 99/1209), sphincter dysfunction (0.7%, 8/1209), and paraplegia (0.3%, 4/1209). Pathological fracture was found in 4.7% (57/1209) of patients. Other symptoms included deformity (2.1%, 25/1209) and weight loss (1.9%, 23/1209) (Table 4).
Table 4

First presenting features when diagnosed with primary spine osseous tumors

 

Total no. of case

Pain

Mass (swelling)

Radiculopathy

Motor weakness

Sphincter dysfunction

Paraplegia

Pathological fracture

Deformity

Weight loss

Benign tumors

780

273(35.0%)

31(4.0%)

72(9.2%)

45(5.8%)

0

0

23(2.9%)

4(0.5%)

7(0.9%)

Hemangioma

340

18

2

1

0

0

0

0

0

1

Giant cell tumor

190

169

11

65

43

0

0

21

3

2

Eosinophililc granuloma

47

35

17

4

1

0

0

0

0

1

Osteoblastoma

53

2

0

0

0

0

0

0

0

0

Fibroma

16

4

0

0

0

0

0

0

0

0

Osteoid osteoma

17

3

0

0

0

0

0

0

0

1

Osteochondroma

46

13

0

0

0

0

0

0

0

1

Solitary bone cyst

25

2

1

0

0

0

0

0

0

0

Lipoma

7

0

0

0

0

0

0

0

0

0

Aneurysmal bone cyst

35

25

0

1

1

0

0

1

0

1

Fibrous dysplasia

4

2

0

1

0

0

0

1

1

0

Malignant tumors

429

385(89.7%)

38(8.9%)

84(19.9%)

54(12.6%)

8(1.9%)

4(0.9%)

34(7.9%)

21(4.9%)

16(3.7%)

PNET/Ewing’s sarcoma

17

16

1

0

0

0

0

1

0

0

Chordoma

119

101

15

43

35

7

2

12

6

3

Malignant

fibrous histiocytoma

16

13

10

0

0

0

0

1

0

0

Liposarcoma

1

1

0

1

0

0

0

1

0

0

Osteosarcoma

13

13

2

 

0

0

0

0

0

0

Angiosarcoma

10

9

1

1

0

0

0

0

0

0

Malignant neurilemmoma

30

28

0

28

15

1

0

0

0

0

Plasma cell myeloma

103

92

0

7

2

2

2

19

15

13

Leiomyosarcoma

54

51

7

3

1

0

0

0

0

0

Malignant lymphoma

1

1

0

0

0

0

0

0

0

0

Chondrosarcoma

63

58

0

0

0

0

0

0

0

0

Fibrosarcoma

2

2

2

1

1

0

0

0

0

0

Total

1209

658(54.4%)

69(5.7%)

156(12.9%)

99(8.2%)

8(0.7%)

4(0.3%)

57(4.7%)

25(2.1%)

23(1.9%)

In our series, 398 cases with malignant tumors and 187 cases with benign tumors undergone surgery. Two hundred seventy-one cases with malignancies received chemotherapy. Thirty-four cases with malignant tumors were given radiotherapy. And other treatments were performed to 6 malignant cases and 35 benign cases respectively (Additional file 1: Table S1).

Discussion

Primary bone tumors of the spine are representing only less than 10% of all bone neoplasms. Available reports on the epidemiologic features of primary osseous spine tumors were mostly among Americans and Europeans [711]. To our knowledge, the present report represents epidemiologic features based the largest series of spinal osseous tumors from Asian. However, there are some differences of epidemiological characteristics in our study and previous reports (Table 5) [8, 10]. In Kelley’s series [10], 23.02% tumors were benign and 76.98% tumors were malignant, plasma cell myeloma was the most common primary malignant tumor, accounting for 26%, followed by chordoma (22.22%) and osteosarcoma (9.52%). Osteoblastoma (5.56%) was the most common benign tumor, followed osteoclastoma (0.79%) and aneurysmal bone cyst (0.79%). In our series, benign tumors comprised 64.5% of primary tumors. The most frequent benign was hemangioma (28.1%), followed by giant cell tumor (15.7%) and osteoblastoma (4.4%). The most frequent malignant was chordoma (9.8%), followed by plasma cell myeloma (8.5%) and chondrosarcoma (5.2%). These findings were not consistent with results from previous studies based in Caucasian, which would be influenced by geographical and racial variations in different reports.
Table 5

World literature of population-based studies on the epidemiology of primary spine osseous tumor

 

This study

Simon P. Kelley

S. Boriani

Author’s country

China

UK

Italy

Year of publication

2016

2007

1995

Period of study

1995–2015 (20 years)

1958–2000 (42 years)

1946–1992 (46 years)

No. of cases

1209

127

366

Gender

Male: 695 Female:514

Male: 66 Female:61

Not reported

Predominant presenting symptom

Pain

Pain

Not reported

Percentage of benign tumors

64.5%

22.8%

56.8%

Percentage of malignant tumors

35.5%

77.2%

43.2%

Most common type of benign tumors

Hemangioma

Osteoblastoma (7/127)

Eosinophilic granuloma

Most common type of malignant tumors

Chordoma

Plasma cell myeloma

Plasma cell myeloma

Most common segment of spine affected

Thoracic spine (479/1256)a

Thoracic spine (48/127)

Lumbar spine (181/366)

Mean age at presentation (range)

39.3 ± 16.8 (range 8–81 years)

42 (range 7–76 years)

Not reported

aMore than two anatomical position involved in the same patient

As the most frequent benign primary spine osseous tumor in present study, hemangioma was accounting for approximately one third of all primary tumors. During the past 20 years (1995–2015), 340 (28.1% of all cases) cases of spinal hemangioma were diagnosed, and 350 vertebrae were involved. This incidence of spinal hemangioma was significantly higher than that in other country [12], which suggested different epidemiologic features of spinal hemangioma in Asians. Giant cell tumor (GCT) of bone was considered a benign osteolytic tumor with variable aggressiveness and accounted for approximately 5% of all bone tumors [4, 13, 14]. In our study, GCTs accounted for 15.7% of spine osseous lesions, this incidence was markedly higher than that reported in other studies [1519]. Almost of GCTs occurred in the second to fourth decades, with a slight female predilection (M:F = 1:1.1) in the present series. The female predilection of GCT is supported by the earlier reports [17, 2022]. Furthermore, GCTs also showed predilection for the thoracic vertebra of spine in our study, which is not in accordance with other reports [19, 23]. Osteoblastoma was the third most common benign tumor in our study, with a relative frequency of 4.4% which was higher compared with the previous report [24]. In Herman M. Kroon’s and Richard A. Mcleod’s reports [25, 26], osteoblastoma obviously affected more males than females (64:34 and 87:36 respectively). In our study, we only observed a slight male predilection (M:F = 1:1.1). The most involved location was the thoracic spine, which is consistent with those reported from Europe and America [27, 28].

Chordoma was the most common malignant tumor which occurred more in males than in females in the present study. We found a significant predilection for males (M:F = 79:40), in agreement with reports by S Boriani (M:F = 37:15) [29], Silvia Stacchiotti (M:F = 91:47) [30], and Johannm Bjornsson (M:F = 27:13) [31]. It is worth noting that female preponderance (17:22) of chordoma was reported in Sweden [32]. In our series, chordomas were typically seen in adults and elderly, tended to occur in the sacrum (9.5%, 119 cases), which is consistent with the previous reports. In the present study, plasma cell myeloma accounted for 8.5% (103 cases) of all cases, representing the second most frequent primary malignant osseous tumor of spine, and often occurring in thoracic spine (4.0%, 50 vertebrae were involved). All plasma cell myeloma cases were found in the 4th–7th decade of life with a male predominance (M:F = 2.7:1). Previous studies reported that chondrosarcoma accounted for 3–12% of all spine primary tumors [3336]. In S Boriani’s report, the lumbar spine was the most frequently involved location (15/22, 68%) [33]. However, in the present study, thoracic spine was the most common location involved with chondrosarcoma (2.9%, 36 cases), and only 2 cases were observed in the lumbar spine. Most patients commonly encountered chondrosarcoma after the age of 20, with a great male predilection (M:F = 4.7:1).

Surgery is the first choice for the pain and neurological symptoms caused by spine tumors. Surgery can completely or partly alleviate tumor compression to the spinal cord, establish a tumor-free solid spine and relieve pain. In primary malignant tumors of the spine, total/partial laminectomy, total/partial vertebral body resection, and piecemeal resection and curettage, in addition to the surgical procedures described above, can be used. Chemotherapy and radiotherapy after surgery were used to reduce the risk of the cancer relapse or shrinks some malignant tumor such Ewing’s sarcoma that cannot be completely removed with surgery. Many benign silent (no symptom) tumors such as hemangioma were found in health checkup, and most of them did not receive any treatment. Although the WHO defines GCT as a benign osteolytic neoplasm, GCT displays the characteristics of both malignant and benign tumors and actually represents a benign tumor with the potential of malignancy [3739]. In our series, most cases of GCT undergone total spondylectomy, vertebrectomy, piecemeal resection or curettage. Some cases of GCT received other therapy such as bisphosphonate treatment after surgery.

Conclusions

Our study results represent data on the epidemiology of spinal osseous tumors in a large population of patients. We think that these data may reflect the epidemiological features of spine osseous tumors in Eastern China. For geographical and racial variations, the incidence of these neoplasms in a developing country is partly different from that found in other countries. There were several limitations in this study. As medical imaging technology have improved greatly during the study time span, the diagnosis sensitivity of primary spinal osseous tumors should lead to different prevalence in different time. Although the prognosis of spinal osseous tumors and longitudinal changes could not be fully assessed due to the nature of a retrospective study, we hope our work could provide useful epidemiological information as these data may have important implications for public health programs.

Declarations

Acknowledgements

Not applicable.

Funding

The study was funded by the Bone Tumors and Nervous System Tumors Biobank Project of Shanghai (12DZ2295103), Bone Tumor Sample Databases and Digital Information Platform Project of Shanghai (08DZ2292800), and Shanghai Biobank Network of Common Human Tumor Tissue (12DZ2295100).

Availability of data and materials

The manuscript does not refer to any new software, application, or tool. The authors do not wish to share data analyzed in this manuscript as no such consent was provided by the patients treated and no approval of the Bioethics Committee was obtained.

Authors’ contributions

ZZ wrote the manuscript. ZZ and XW performed data analysis. ZW and WH collected and evaluated information of cases. JX conceived of the study and participated in its designation. All authors read and approved the final manuscript.

Competing interests

The authors declare that they have no competing interests.

Consent for publication

Not applicable.

Ethics approval and consent to participate

All procedures performed in studies involving human participants were in accordance with the ethical standards of the Clinical Research Ethics Committee of Second Military Medical University and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. Human data were obtained with informed consent, and this study was approved by the Clinical Research Ethics Committee of the Second Military Medical University.

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Authors’ Affiliations

(1)
Department of Orthopaedic Oncology, Changzheng Hospital,, The Second Military Medical University

References

  1. Polednak AP. Primary bone cancer incidence in black and white residents of New York State. Cancer. 1985;55:2883–8.View ArticlePubMedGoogle Scholar
  2. Yeole BB, Jussawalla DJ. Descriptive epidemiology of bone cancer in greater Bombay. Indian J Cancer. 1998;35:101–6.PubMedGoogle Scholar
  3. Barbosa CS, Araujo AB, Miranda D. Incidence of primary benign and malignant neoplasms and bone pseudotumoral lesions. An epidemiologic analysis of 585 cases diagnosed at the Faculdade de Medicina of the Universidade Federal de Minas Gerais. AMB Rev Assoc Med Bras. 1991;37:187–92.PubMedGoogle Scholar
  4. Baena-Ocampo Ldel C, Ramirez-Perez E, Linares-Gonzalez LM, Delgado-Chavez R. Epidemiology of bone tumors in Mexico City: retrospective clinicopathologic study of 566 patients at a referral institution. Ann Diagn Pathol. 2009;13:16–21.View ArticlePubMedGoogle Scholar
  5. Ciftdemir M, Kaya M, Selcuk E, Yalniz E. Tumors of the spine. World J Orthop. 2016;7:109–16.View ArticlePubMedPubMed CentralGoogle Scholar
  6. Fletcher CDM UK, Fletcher F, editors. World Health Organization classification of tumors. Pathology and genetics of tumors of soft tissue and bone. Lyon: IARC Press; 2013.Google Scholar
  7. Chi JH, Bydon A, Hsieh P, Witham T, Wolinsky JP, Gokaslan ZL. Epidemiology and demographics for primary vertebral tumors. Neurosurg Clin N Am. 2008;19:1–4.View ArticlePubMedGoogle Scholar
  8. Boriani S, Biagini R, De Iure F, Andreoli I, Campanacci L, De Fiore M, Zanoni A. Primary bone tumors of the spine: a survey of the evaluation and treatment at the Istituto Ortopedico Rizzoli. Orthopedics. 1995;18:993–1000.PubMedGoogle Scholar
  9. Benezech J, Fuentes JM. Primary tumors of the spine. A multicenter cooperative study. Neurochirurgie. 1989;35:317–22.PubMedGoogle Scholar
  10. Kelley SP, Ashford RU, Rao AS, Dickson RA. Primary bone tumours of the spine: a 42-year survey from the Leeds Regional Bone Tumour Registry. Eur Spine J. 2007;16:405–9.View ArticlePubMedGoogle Scholar
  11. Dreghorn CR, Newman RJ, Hardy GJ, Dickson RA. Primary tumors of the axial skeleton. Experience of the Leeds Regional Bone Tumor Registry. Spine (Phila Pa 1976). 1990;15:137–40.View ArticleGoogle Scholar
  12. Doppman JL, Oldfield EH, Heiss JD. Symptomatic vertebral hemangiomas: treatment by means of direct intralesional injection of ethanol. Radiology. 2000;214:341–8.View ArticlePubMedGoogle Scholar
  13. Gupta R, Seethalakshmi V, Jambhekar NA, Prabhudesai S, Merchant N, Puri A, Agarwal M. Clinicopathologic profile of 470 giant cell tumors of bone from a cancer hospital in western India. Ann Diagn Pathol. 2008;12:239–48.View ArticlePubMedGoogle Scholar
  14. Goldenberg RR, Campbell CJ, Bonfiglio M. Giant-cell tumor of bone. An analysis of two hundred and eighteen cases. J Bone Joint Surg Am. 1970;52:619–64.View ArticlePubMedGoogle Scholar
  15. Sanjay BK, Sim FH, Unni KK, McLeod RA, Klassen RA. Giant-cell tumours of the spine. J Bone Joint Surg (Br). 1993;75:148–54.View ArticleGoogle Scholar
  16. Balke M, Schremper L, Gebert C, Ahrens H, Streitbuerger A, Koehler G, Hardes J, Gosheger G. Giant cell tumor of bone: treatment and outcome of 214 cases. J Cancer Res Clin Oncol. 2008;134:969–78.View ArticlePubMedGoogle Scholar
  17. Campanacci M, Baldini N, Boriani S, Sudanese A. Giant-cell tumor of bone. J Bone Joint Surg Am. 1987;69:106–14.View ArticlePubMedGoogle Scholar
  18. Luther N, Bilsky MH, Hartl R. Giant cell tumor of the spine. Neurosurg Clin N Am. 2008;19:49–55.View ArticlePubMedGoogle Scholar
  19. Leggon RE, Zlotecki R, Reith J, Scarborough MT. Giant cell tumor of the pelvis and sacrum: 17 cases and analysis of the literature. Clin Orthop Relat Res 2004;423:196–207.Google Scholar
  20. Zhen W, Yaotian H, Songjian L, Ge L, Qingliang W. Giant-cell tumour of bone. The long-term results of treatment by curettage and bone graft. J Bone Joint Surg (Br). 2004;86:212–6.View ArticleGoogle Scholar
  21. Olivera P, Perez E, Ortega A, Terual R, Gomes C, Moreno LF, Duenas A, De La Garza J, Melendez-Zajgla J, Maldonado V. Estrogen receptor expression in giant cell tumors of the bone. Hum Pathol. 2002;33:165–9.View ArticlePubMedGoogle Scholar
  22. Saiz P, Virkus W, Piasecki P, Templeton A, Shott S, Gitelis S. Results of giant cell tumor of bone treated with intralesional excision. Clin Orthop Relat Res. 2004;424:221–6.Google Scholar
  23. Ozaki T, Liljenqvist U, Halm H, Hillmann A, Gosheger G, Winkelmann W. Giant cell tumor of the spine. Clin Orthop Relat Res. 2002;401:194–201.Google Scholar
  24. Dahlin DC. Bone tumors: general aspects and an analysis of 3987 cases. Second edition. Springfield, Illinois: Charles C Thomas, Pbulisher; 1967.Google Scholar
  25. Kroon HM, Schurmans J. Osteoblastoma: clinical and radiologic findings in 98 new cases. Radiology. 1990;175:783–90.View ArticlePubMedGoogle Scholar
  26. McLeod RA, Dahlin DC, Beabout JW. The spectrum of osteoblastoma. AJR Am J Roentgenol. 1976;126:321–5.View ArticlePubMedGoogle Scholar
  27. Kirwan EO, Hutton PA, Pozo JL, Ransford AO. Osteoid osteoma and benign osteoblastoma of the spine. Clinical presentation and treatment. J Bone Joint Surg (Br). 1984;66:21–6.View ArticleGoogle Scholar
  28. MacLellan DI, Wilson Jr FC. Osteoid osteoma of the spine. A review of the literature and report of six new cases. J Bone Joint Surg Am. 1967;49:111–21.View ArticlePubMedGoogle Scholar
  29. Boriani S, Bandiera S, Biagini R, Bacchini P, Boriani L, Cappuccio M, Chevalley F, Gasbarrini A, Picci P, Weinstein JN. Chordoma of the mobile spine: fifty years of experience. Spine (Phila Pa 1976). 2006;31:493–503.View ArticleGoogle Scholar
  30. Stacchiotti S, Casali PG, Lo Vullo S, Mariani L, Palassini E, Mercuri M, Alberghini M, Pilotti S, Zanella L, Gronchi A, Picci P. Chordoma of the mobile spine and sacrum: a retrospective analysis of a series of patients surgically treated at two referral centers. Ann Surg Oncol. 2010;17:211–9.View ArticlePubMedGoogle Scholar
  31. Bjornsson J, Wold LE, Ebersold MJ, Laws ER. Chordoma of the mobile spine. A clinicopathologic analysis of 40 patients. Cancer. 1993;71:735–40.View ArticlePubMedGoogle Scholar
  32. Bergh P, Kindblom LG, Gunterberg B, Remotti F, Ryd W, Meis-Kindblom JM. Prognostic factors in chordoma of the sacrum and mobile spine: a study of 39 patients. Cancer. 2000;88:2122–34.View ArticlePubMedGoogle Scholar
  33. Boriani S, De Iure F, Bandiera S, Campanacci L, Biagini R, Di Fiore M, Bandello L, Picci P, Bacchini P. Chondrosarcoma of the mobile spine: report on 22 cases. Spine (Phila Pa 1976). 2000;25:804–12.View ArticleGoogle Scholar
  34. Camins MB, Duncan AW, Smith J, Marcove RC. Chondrosarcoma of the spine. Spine (Phila Pa 1976). 1978;3:202–9.View ArticleGoogle Scholar
  35. Kaim AH, Hugli R, Bonel HM, Jundt G. Chondroblastoma and clear cell chondrosarcoma: radiological and MRI characteristics with histopathological correlation. Skeletal Radiol. 2002;31:88–95.View ArticlePubMedGoogle Scholar
  36. Resnick DKM, Greenway GD. Tumors and tumor-like lesions of bone: imaging and pathology of specific lesions. In: Resnick D, editor. Diagnosis of bone and joint disorders. Philadelphia: Saunders; 2002. p. 3897.Google Scholar
  37. Chakarun CJ, Forrester DM, Gottsegen CJ, Patel DB, White EA, Matcuk Jr GR. Giant cell tumor of bone: review, mimics, and new developments in treatment. Radiographics. 2013;33:197–211.View ArticlePubMedGoogle Scholar
  38. Raskin KA, Schwab JH, Mankin HJ, Springfield DS, Hornicek FJ. Giant cell tumor of bone. J Am Acad Orthop Surg. 2013;21:118–26.View ArticlePubMedGoogle Scholar
  39. Cowan RW, Singh G. Giant cell tumor of bone: a basic science perspective. Bone. 2013;52:238–46.View ArticlePubMedGoogle Scholar

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© The Author(s). 2017

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