|Year : 2016 | Volume
| Issue : 2 | Page : 333-338
Epidemiological data and case load spectrum of patients presenting to bone and soft tissue disease management group at a tertiary cancer center
A Gulia, A Puri, S Chorge, PK Panda
Orthopedic Oncology Services, Department of Surgical Oncology, Tata Memorial Hospital, Parel, Mumbai, India
|Date of Web Publication||6-Jan-2017|
Orthopedic Oncology Services, Department of Surgical Oncology, Tata Memorial Hospital, Parel, Mumbai
Source of Support: None, Conflict of Interest: None
Introduction and Background: This study was conducted to know the spectrum and number of bone and soft tissue (BST) tumors presenting to our institute. We needed to assess the gap between the number of patients seen and infrastructure available, and based on this information, help formulate guidelines for optimum utilization of resources and to provide best possible evidence-based cancer care. Settings And Designs: This is a prospective observational study (epidemiological). Materials and Methods: This study included all new patients seen in BST-disease management group (DMG) in the year 2010. An audit form was devised to capture all the relevant information. A comparison of our data with other national and international studies was also done. Results: Out of total 31,951 new patients registered at our institute, 2007 patients availed BST-DMG services. Sixty percent were bone tumors and 36% were soft tissue tumors. In bone tumor, 66% were malignant, 15% were benign, and 19% were non-neoplastic. Osteosarcoma (43%) was the most common malignant tumor followed by primitive neuroectodermal tumor/Ewing's (27%) and chondrosarcoma (11%). Giant cell tumor was the most common benign bone tumor. Eighty-one percent of all soft tissue lesions were malignant, of which 75% were of mesenchymal origin and 25% were of cutaneous origin. Conclusion: This is an attempt to document the epidemiology of musculoskeletal tumors presenting to our institution while guiding the institute to frame and implement disease-specific protocols and generate further research questions. Continued data collection and follow-up can provide valuable information on long-term survival and treatment-related toxicities. This data (within limitations) may be extrapolated to national level to identify the need for infrastructure and human resources.
Keywords: Bone tumors, epidemiology, soft tissue sarcoma, tertiary cancer center
|How to cite this article:|
Gulia A, Puri A, Chorge S, Panda P K. Epidemiological data and case load spectrum of patients presenting to bone and soft tissue disease management group at a tertiary cancer center. Indian J Cancer 2016;53:333-8
|How to cite this URL:|
Gulia A, Puri A, Chorge S, Panda P K. Epidemiological data and case load spectrum of patients presenting to bone and soft tissue disease management group at a tertiary cancer center. Indian J Cancer [serial online] 2016 [cited 2019 Oct 18];53:333-8. Available from: http://www.indianjcancer.com/text.asp?2016/53/2/333/197734
| » Introduction|| |
Our institute being one of the largest referral centers in the subcontinent registers more than 30,000 new cases of suspected or proven cases of malignancy. The bone and soft tissue (BST) services at our institute are dedicated to provide evidence-based, feasible, and practically relevant care for the patients with bone, soft tissue, and cutaneous malignancies of the extremities, girdles, and axial skeleton. The management of these malignant disorders requires a multimodality approach. The treatment for such disorders often requires numerous investigations and various cross consultations, which may be time-consuming for the patients and effort-intensive for the treating physicians. Thus, an appropriate ratio of patient to treating doctors and the available resources is mandatory to achieve these goals.
In resource-challenged environments, there is always a gap between the necessary infrastructure, human resources, and the patient load. Hence, there is a necessity to use limited resources in the best possible way for arriving at a diagnosis without undue delay. This helps facilitate early patient management with proper allocation of resources. A hospital audit that analyzes the variety and spectrum of the patients seen can assist to streamline the efforts made to provide the best possible evidence-based care for all the patients.
A methodological approach was devised to collect the data of patients of BST tumors to know their incidence, age distribution, and associated prognostic factors. A prospective observational study was designed to evaluate the frequency, spectrum, and overall load of BST sarcomas presenting to our institute.
| » Materials and Methods|| |
All the patients seen for the 1st time in BST outpatient department (including new registration in BST-disease management group [BST-DMG] and referrals from other DMGs) were enrolled in the study and were followed up till the commencement of the treatment. The data of all the patients from January 01, 2010 to December 31, 2010 were prospectively collected and analyzed.
An audit form was devised to capture all relevant information including patient demographics, contact details, educational and economic status, symptomatology, site, details of previous treatment taken, outside diagnosis, stage of disease at presentation, past and family history of cancer, final diagnosis at our institute, and final advice on the treatment. These details were jointly filled by patient and the attending clinician. The audit form was divided into four phases. Phase I included the information pertaining to the present study. Phase II and III were designed to capture the information of the treatment of the patients, related delay in treatment, or associated complications. Phase IV was designed to collect the information on subsequent follow-ups.
The relevant information was collected as a hard copy of the audit form for each patient. This information was regularly updated on a weekly basis from case files and electronic medical record (EMR). All the information were entered in a specially designed data collection system (FileMaker Pro, version 10, FileMaker Inc., Apple subsidiary) to help in further analysis of the data. Regular monthly audits were done to check the completeness of the collected data. The cases with insufficient information were reviewed via EMR and case files. Patients were contacted through telephone and/or e-mails to fill lacunae in data, wherever possible.
Description statistics was performed using SPSS software version 21.0 for windows (SPSS Inc., Chicago, IL, USA).
| » Results|| |
The following details pertain to the subsets of data that were analyzed from Phase I of the data collection module. Between January 01, 2010 and December 31, 2010, a total of 31,951 new patients were registered at our institute, of which 1627 (5%) were registered primarily in the BST service [Figure 1]a. Three hundred and eighty new patients were also referred to us from other departments making it a total of 2007 new cases.
|Figure 1: (a) Case distribution, (b) bone and soft tissue cases, (c) Indian state-wise case distribution|
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Out of the total 2007 patients seen by the BST services, there were 700 women and 1307 men with a mean age of 38 years. About 1203 (60%) cases were bone tumors, 723 (36%) were soft tissue tumors, and details of primary site of affection were not known in 81 (4%) patients [Figure 1]b. These 81 patients had inadequate details and were thus excluded from further analyses.
Out of the total 1203 bone tumor cases, 15% (n = 181) were benign, 66% (n = 794) were malignant, and 19% (n = 228) were lesions of nonneoplastic (NNP) etiology [Figure 2]a. Majority of the cases were from the state of Maharashtra in Western India (47%) [Figure 1]c, where our hospital is located.
|Figure 2: Incidence of bone tumors, (a) etiology, (b) gender, (c) age group, (d) site|
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Benign bone tumors (n = 181) were more common in the males (59%) [Figure 2]b. Fifty-five percent (n = 58) belonged to third decade of life and about 80% (n = 141) of these occurred between 11 and 40 years [Figure 2]c. Tibia was the most common site of affection in 24.9% (n = 45) cases followed by femur in 23.2% (n = 42). Among these, giant cell tumor (GCT) of bone was the most common histological diagnosis followed by aneurysmal bone cyst (ABC) [Table 1].
Of the 794 malignant bone tumors seen, 70% (n = 553) were primary bone sarcomas and 30% (n = 241) were secondary malignancies to the bone. The incidence of primary malignant bone tumors (n = 553) was highest in the second decade (42%). Males were predominantly affected (67%). Femur was the most common site of affection in 32.5% (n = 180) cases followed by tibia in 18.4% (n = 102) [Figure 2]d. Osteogenic sarcoma (OGS) was the most common histopathological diagnosis followed by primitive neuroectodermal tumor (PNET)/Ewing's sarcoma of bone [Table 2].
OGS (n = 243) was more commonly seen in males (67.48%). About 59% of the patients were in the second decade of life. Femur was the most common site of involvement in 113 cases (46.5%) followed by tibia in 67 cases (27.5%), humerus in 24 cases (10%), and fibula in 13 cases (5%). Metastatic workup details were not available in 7% (n = 16) of patients. Out of remaining 227 patients, 75.33% of patients had localized disease (n = 171) and 24.6% (n = 53) had metastatic disease at presentation. Most common site of metastasis was the lung (84%) followed by skeleton (14%). One patient (2%) had both lung and skeletal metastasis at presentation.
PNET/Ewing's sarcoma was the second most common malignant bone sarcoma and largely affects male (67%). Of the total 147 cases, pelvis was the most common site of affection in 32 cases (21.7%) followed by femur in 19.7% (n = 29), tibia in 15% (n = 22), humerus in 9% (n = 14), radius in 2%, and ulna in 1%. Details of metastatic workup were not available in 5 patients. Out of remaining 142, 65% had localized and 35% had metastatic disease at presentation. Lung was the most common sites of metastasis in 74% (n = 37) cases followed by bone in 20% (n = 10) cases. Both lung and skeletal metastasis was seen in 6% (n = 3) patients.
Chondrosarcoma was the third most common primary malignant bone tumor. It was most commonly seen in the sixth decade (26.23%) and predominantly affecting males (69%). Most common site of affection was pelvis (48%, n = 29) followed by femur (18%), humerus (6%), and tibia (6%). Details of metastatic work up were not available in 7 cases (11%). Out of remaining 54 patients, 48 were nonmetastatic (89%) and 6 cases (11%) had metastatic disease at presentation.
Two hundred and forty-one patients were diagnosed with secondary metastatic disease of bone with primary in other systems. Metastatic bone disease, where we failed to identify a primary site, was the most common cause with 31% (n = 75) of patients falling into this category. When a detectable primary was present, lung was the most common site with 17% (n = 45) [Table 2].
Out of 723 soft tissue lesions, 82% (n = 587) were malignant, 12% (n = 88) were benign, and 6% (n = 48) were NNP lesions [Figure 2]a. Benign soft tissue lesions were most commonly seen in the fourth decade 26% (n = 23). These tumors were predominantly seen in males (61%). Fibromatosis was the most common diagnosis seen in 27% (n = 24) of patients.
Out of the 587 malignant soft tissue lesions, 551 were primary malignant soft tissue tumors and remaining (6%) were soft tissue secondary deposits from primary elsewhere (n = 36). Seventy-five percent (n = 413) of the primary malignant soft tissue lesions were of mesenchymal origin (soft tissue sarcomas) and 25% (n = 138) were of cutaneous origin. Among 413 soft tissue sarcomas, synovial sarcoma (22.5%, n = 93) was the most common histopathology. Males were predominantly affected (70%) with thigh as the most common site of involvement (34%) and were evenly seen among third, fourth, and fifth decades [Figure 3]a,[Figure 3]b,[Figure 3]c.
|Figure 3: Soft tissue lesions (a) mesenchymal tumors incidence, (b) site distribution, and (c) age distribution|
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Of the 93 patients with synovial sarcoma, 70% were males. The disease most commonly affects young adults in the third decade of life (34.4%). Most common site of affection was the thigh (29%, n = 31). Details of metastatic workup were not available in 13% (n = 12) of patients. Of remaining 81 patients, 74% (n = 60) patients had localized disease and 24% (n = 21) had metastatic disease at presentation.
Spindle cell sarcoma comprised 18.5% (n = 76) of all mesenchymal soft tissue malignancies. The disease was most common in male patients (73.68%) and the thigh was affected the most (42%, n = 32). Details of metastatic workup were not available in 6% (n = 5) of patients. Of remaining 69 patients, 69% (n = 48) patients had localized disease and 31% (n = 21) had metastatic disease at presentation.
Soft tissue Ewing's sarcoma was seen in 12% (n = 51) of patients. The most common site of affection was the thigh in 22 patients (43%). Peak incidence was seen in third decade (n = 15) with male preponderance (67%). Details of metastatic workup were not available in 10% (n = 5) of patients. Of the remaining 46 patients, 50% (n = 23) patients had localized disease and 50% (n = 23) had metastatic disease at presentation. Other soft tissue malignancies include pleomorphic sarcoma (8%, n = 32), leiomyosarcoma (7%, n = 31), liposarcoma (6%, n = 25), rhabdomyosarcoma (5%), dermatofibrosarcoma protuberans (5%), and epithelioid sarcoma (3%) [Figure 3]a.
Squamous cell carcinoma (n = 138) contributed to 54% of cutaneous malignancies with male predominance (70%). Details of metastatic workup were not available in 12% (n = 9) of patients. Of the remaining 65 patients, 80% (n = 52) patients had localized disease and 20% (n = 13) had metastatic disease at presentation.
Malignant melanoma (n = 50) constituted 36% of cutaneous malignancies with male predominance (76%) and most commonly seen in the sixth decade of life. Foot was most commonly affected in 68% cases. Among these, 44% (n = 22) patients had localized disease and 54% (n = 27) had metastatic disease at presentation.
Out of total 2007 cases, 276 cases (13.75%) were of NNP etiology. Out of this, 80% (n = 223) had other orthopedic conditions and about 20% (n = 53) had infections.
| » Discussion|| |
The present study represents an attempt to document the epidemiology of BST tumors presenting to our institution, a tertiary cancer referral center in India. We have also attempted to compare and contrast our data to existing Asian and Western literature. Our review of cases in a particular year shows that the age distribution and sites of presentation of bone tumors are similar to those of other published studies. Of the 794 malignant bone tumors seen, the percentage of primary tumors was higher than secondary lesions which is due to referral bias, ours being a tertiary referral center. There was double the number of males than females diagnosed with primary malignant bone tumors. Approximately, 41% of primary bone tumors and 9.2% of soft tissue tumors were seen in patients <20 years of age in our series, which was in concordance with the 30% and 10% rate seen in surveillance, epidemiology, and end results (SEER) data  and similar incidence in a study by Malhas et al., while according to a separate study in European population, they comprised 3–5% of tumors diagnosed in children below 15 years and 7–8% in adolescents between 15 and 19 years of age.
Among benign bone tumors, GCT was the most common diagnosis (n = 181, 56%). About one-fourth of the GCT's presenting to us was recurrent cases. ABC was the second most common tumor. This is contrast to most Asian as well as Western literature in which osteochondroma is the most common benign tumor and GCT being the second most common.,,,, This difference can be attributed to the referral bias as most benign tumors except for the more locally aggressive tumors such as GCT and ABC would not be referred to tertiary oncology centers.
Most common malignant bone tumor in our series was osteosarcoma (43%) followed by Ewing's sarcoma (27%) and chondrosarcoma (11%). Our results are in line with other Asian studies with osteosarcoma and Ewing's sarcoma being the two most common primary malignant tumors [Table 3].,,, This is in contrast to Western literature where chondrosarcoma is the second most common primary malignant tumor followed by Ewing's sarcoma.,, About 24.6% of osteosarcomas and 34% of Ewing's sarcomas were metastatic at presentation in our series, which is in accordance with the SEER database from 1991 to 2010.,
|Table 3: Comparison of incidences of primary malignant bone tumors in Indian population|
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In our series, 241 patients (30.5%) presented with secondary metastatic involvement of the bone. A known primary could not be identified in approximately one-third of them. Lung and breast were the most common sites of primary with 17% and 12% of cases, respectively. Galasko has reported that majority of skeletal metastasis are from breast malignancies followed by prostate, thyroid, and lungs with skeletal metastasis from gastrointestinal (GI) malignancies being the least common. This is in contrast to our data where skeletal metastasis from lungs is most common followed by breast and GI malignancies. Similar results were also reported by a study by Yücetürk et al., who also stated that lung and breast were the two most common sites of metastasis to bone. Of the 1203 patients with suspected bone tumor, only 16 patients under the age of 35 (1.3%) had secondary metastatic disease of bone, stressing the fact that secondary lesions in the bone are extremely rare in younger population and primary tumors of the bone should be the first differential while evaluating suspected bone tumor lesion in younger population. Similar findings were also reported by Malhas et al., who observed an incidence of 0.6% (17 of 2913) under the age of 35 years with metastatic disease of bone.
Out of all soft tissue tumors, 82% were malignant. Synovial sarcoma was the most common soft tissue malignancy (17%) in our series compared to malignant fibrous histiocytoma or liposarcoma, which are the most common soft tissue sarcomas reported in most other series.,, Fibromatosis was the most common benign tumor in our series seen in 28% of cases. This is in contrast to another large series where lipoma was the most common benign lesion. This can be again explained by the fact that ours being a tertiary referral center, most lipomas would be treated at other institutes.
The present study has its own limitations. This study is a hospital-based audit and is not a population-based registry and thus has an inherent selection bias. Our proportion of patients with individual histopathological diagnosis is not an absolute reflection of the actual numbers in the population. This is a reflection of cases seen in a tertiary referral center, which may include more malignant or aggressive cases as compared to benign ones. The same trend has been noticed in other tertiary referral centers across the globe.
| » Conclusion|| |
The present study is an attempt to assess the case load and epidemiology of musculoskeletal tumors presenting to our institute. Besides being a source of epidemiological data and disease stage of presentation, the study can help the institute guide policies aimed at ideal utilization of resources. Information gleaned from the various phases of the data collection module can help in formulating guidelines for referral of patients to specialized cancer treating centers to strike the right balance between patient load and resource availability. Based on these figures, health care providers can better estimate the need for treatment delivery systems aimed at providing a rapid diagnosis and early initiation of therapy. The study data can also be used as a nucleus for generation of further clinical and basic research questions which merit investigations. The continued data collection and follow-up of study patients can provide valuable information on long-term survival, long-term treatment-related toxicities, and incidence of second malignancies. Finally, to some extent, these data can be extrapolated to the national level to identify the need of infrastructure and human resources for providing evidence-based practically feasible best patient care across the nation.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| » References|| |
Malhas AM, Grimer RJ, Abudu A, Carter SR, Tillman RM, Jeys L. The final diagnosis in patients with a suspected primary malignancy of bone. J Bone Joint Surg Br 2011;93:980-3.
Howlader N, Noone AM, Krapcho M, Neyman N, Aminou R, Altekruse SF, et al
., editors. SEER Cancer Statistics Review, 1975-2009 (Vintage 2009 Populations). Bethesda, MD: National Cancer Institute; 2012.
Stiller CA, Bielack SS, Jundt G, Steliarova-Foucher E. Bone tumours in European children and adolescents, 1978-1997. Report from the automated childhood cancer information system project. Eur J Cancer 2006;42:2124-35.
Solooki S, Vosoughi AR, Masoomi V. Epidemiology of musculoskeletal tumors in Shiraz, South of Iran. Indian J Med Paediatr Oncol 2011;32:187-91.
Jain K, Sunila, Ravishankar R, Mruthyunjaya, Rupakumar CS, Gadiyar HB, et al.
Bone tumors in a tertiary care hospital of south India: A review 117 cases. Indian J Med Paediatr Oncol 2011;32:82-5.
Rao VS, Pai MR, Rao RC, Adhikary MM. Incidence of primary bone tumours and tumour like lesions in and around Dakshina Kannada district of Karnataka. J Indian Med Assoc 1996;94:103-4.
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.
Shah SH, Muzaffar S, Soomro IN, Pervez S, Hasan SH. Clinico-morphological pattern and frequency of bone cancer. J Pak Med Assoc 1999;49:110-2.
Blackwell JB, Threlfall TJ, McCaul KA. Primary malignant bone tumours in Western Australia, 1972-1996. Pathology 2005;37:278-83.
Miller BJ, Cram P, Lynch CF, Buckwalter JA. Risk factors for metastatic disease at presentation with osteosarcoma: An analysis of the SEER database. J Bone Joint Surg Am 2013;95:e89.
Galasko C. The anatomy and pathways of skeletal metastases. In: Weiss L, Gilbert A, editors. Bone Metastases. Boston: GK Hall; 1981. p. 49-63.
Yücetürk G, Sabah D, Keçeci B, Kara AD, Yalçinkaya S. Prevalence of bone and soft tissue tumors. Acta Orthop Traumatol Turc 2011;45:135-43.
Kransdorf MJ. Malignant soft-tissue tumors in a large referral population: Distribution of diagnoses by age, sex, and location. AJR Am J Roentgenol 1995;164:129-34.
Kransdorf MJ. Benign soft-tissue tumors in a large referral population: Distribution of specific diagnoses by age, sex, and location. AJR Am J Roentgenol 1995;164:395-402.
[Figure 1], [Figure 2], [Figure 3]
[Table 1], [Table 2], [Table 3]
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