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Functional outcome in amputation versus limb sparing of patients with lower extremity sarcoma: A matched case-control study
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Functional Outcome in Amputation Versus Limb Sparing of Patients With Lower Extremity Sarcoma: A Matched Case-Control Study Aileen M. Davis, PhD, Michael Devlin, MD, Anthony M. Grijjk, ABSTRACT. Davis AM, Devlin M, Griffin AM, Wunder JS, Bell RS. Functional outcome in amputation versus limb sparing of patients with lower extremity sarcoma: a matched casecontrol study. Arch Phys Med Rehabil 1999;80:61.5-8. Objective: To quantify the differences in physical disability and handicap experienced by patients with lower extremity sarcoma who required amputation for their primary tumor as compared with those treated by limb-sparing surgery. Design: Matched case-control study. Twelve patients with amputation were matched with 24 patients treated by limbsparing surgery on the following variables: age, gender, length of follow-up, bone versus soft-tissue tumor, anatomic site, and treatment with adjuvant chemotherapy. Patients: Patients who underwent above-knee amputation (AKA) or below-knee amputation (BKA) for primary softtissue or bone sarcoma, who had not developed local or systemic recurrence, and who had been followed up for at least 1 year since surgery. Main Outcome Measures: The Toronto Extremity Salvage Score (TESS), a measure of physical disability; the Shortform-36 (SF-36), a generic health status measure; and the Reintegration to Normal Living (RNL), a measure of handicap. Results: Mean TESS score for the patients with amputations was 74.5 versus 85.1 for the limb-sparing patients. (p = .15). Only the physical function subscale of the SF-36 showed statistically significant differences, with means of 45 and 71.1 for the amputation versus limb-sparing groups, respectively (p = .03). The RNL for the amputation group was 84.4 versus 97 for the limb-sparing group (p = .05). Seven of the 12 patients with amputations experienced ongoing difficulty with the soft tissues overlying their stumps. Conclusions: There was a trend toward increased disability for those in the amputation group versus those in the limbsparing group, with the amputation group showing significantly higher levels of handicap. These data suggest that the differences in disability between amputation and limb-sparing patients are smaller than anticipated. The differences may be more notable in measuring handicap. 0 1999 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation
From the University Musculoskeletal Oncology Unit (DE. Davis, Griffin, Wunder, Bell) and the Department of Physiatry (Dr. D&n), Mount Sinai Hospital and University of Toronto, Toronto, Canada. Submitted for publication July 23, 1998. Accepted in revised form December 8, 1998. No commercial party having a direct financial interest in the results of the research supporting this article has or will confer a benefit upon the. authors or upon any organization with which the authors are associated. Remint reauests to Aileen M. Davis. PhD. Mount Sinai Hosoital. 600 Universitv I Awn&, Suit; 476H, Toronto, Canada I&G 1x5. 0 1999 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation 0003.9993/99/8006-5140$3.00/O
BSc, Jay S. Wunder, MD, Robert S. Bell, MD
HEN SUFFICIENT MARGINS of resection surrounding the tumor can be achieved, limb-sparing surgery, as opposed to amputation, has become the standard of care in treating bone and soft-tissue sarcoma of the extremity.’ This standard has developed because of technologic advances in radiographic imaging and reconstructive surgery as well as the use of adjuvant therapies. Despite these advances, however, discussionspersist about whether an amputation would provide a superior functional result for specific patients. These discussions usually occur when the treatment team recognizes that there is significant risk of treatment morbidity and perceives that the functional result of the reconstructive procedure will be poor. Sugarbaker and associates2 published the landmark paper comparing the results of amputation versus limb-sparing surgery. The findings of 21 patients with soft-tissue sarcoma randomized to receive amputation plus chemotherapy (n = 9) or limb-sparing surgery plus irradiation and chemotherapy (IZ= 12) suggested that the anticipated improvements in quality of life of limb-sparing patients, as measured by a battery of standardized measures, was not substantiated. Individual components of the measures suggested that there was a trend toward improved sexual functioning and physical functioning in the amputation group based on subtest analysis of the various measures. However, the second phase of this study, using a second battery of tests to evaluate pain, mobility, treatment trauma, and sexual functioning, failed to confirm the results of the first phase. No differences were demonstrated between the amputation and limb-sparing groups in this second phase. Consequently, the discussions regarding the functional outcome of patients with amputations versus patients undergoing limbsparing surgery for extremity sarcoma persist. Recognizing the advances that have occurred in the treatment of patients with soft-tissue and bone sarcoma over the past two decades and the ongoing debate about limb sparing versus amputation in this population, our group endeavored to clarify the functional outcomes of these groups of patients. Working from the International Classification of Impairment, Disability and Handicap (ICIDH) definitions of disability and handicap,3 the purpose of this matched case-control study was to quantify the differences in physical disability and handicap experienced by patients with lower extremity sarcoma who required amputation for their primary tumor as compared with those who could be treated by limb-sparing surgery. The ICIDH defines disability as any restriction or lack of ability to perform an activity in the manner or within the range considered normal for a human being.3 Handicap is limitation in, or prevention of, a role that is normal (depending on age, gender, and social and cultural factors) for the individual3
w
METHODS The cases for this study were defined by patients who (1) were treated by above-knee amputation (AKA) or below-knee amputation (BKA) for a nonlocally recurrent bone or soft-tissue sarcoma that was otherwise unresectable, (2) had a minimum of Arch
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1 year of follow-up since their amputation, and (3) had not developed local or systemic recurrence. Between January 1986 and October 1995,26 patients with primary bone or soft-tissue sarcoma were treated by AKA or BKA. Nine of these patients died of progressive disease, four were alive with metastatic disease, and 13 were eligible for the study. During the same period, 239 patients with lower extremity tumor were treated with limb-sparing surgery. The decision to primarily amputate the extremity was determined by the inability to adequately resect the tumor and reconstruct a functional limb. For bone sarcoma, complete resection is necessary, whereas for soft-tissue sarcoma, gross tumor resection, leaving no more than a small microscopic burden of tumor that is amenable to control with adjuvant radiotherapy, is acceptable. Indications for amputation included encasement of the neurovascular structures by the tumor, tumor progression on chemotherapy, or inadvertent contamination of the surgical field from previous biopsy or surgery. Patients treated with limb-sparing surgery who remained disease free were chosen as matched controls for the amputation group. The matching variables were tumor site such that the alternative to salvage was the level of amputation of the matched case, age (within 5 years), gender, length of follow-up (within 12 months providing the minimum follow-up was 1 year), bone or soft-tissue tumor, and treatment with adjuvant chemotherapy. The ratio of limb-sparing patients matched to amputation subjects was 2:l to improve the power of the study. Of the 13 patients with amputations who were eligible for the study, however, one patient who underwent BKA for soft-tissue sarcoma did not return the questionnaires. Hence, 12 patients with amputations matched to 24 patients undergoing limbsparing surgery were the focus of the study. Subjects completed the Toronto Extremity Salvage Score (TESS), a measure of physical disability developed specifically for the extremity tumor population4,5; the Shortform(SF36), a generic health status measure,6 and the Reintegration to Normal Living (RNL), a measure of handicap developed for cancer patients7v8 The TESS consists of 30 items, and the patient rates the difficulty experienced completing an activity on a 5-point Likert scale.The final score is converted to a range from 0 to 100. The SF-36 consists of 36 items scored in 8 subscales (physical function, role-physical, bodily pain, general health, vitality, social function, role-emotional, and mental health). Each subscale score ranges from 0 to 100. The RNL consists of 11 items: moving around living quarters, moving around community, ability to take trips out of town, comfort with self-care needs, occupation in work activities, participation in recreational activities, participation in social activities, assumption of role in family, comfort with personal relationships, comfort with self in company of others, and ability to deal with life events. The items are rated by the subject on a visual analogue scale, with the RNL score ranging from 0 to 110. For the TESS, SF-36, and RNL, a higher score indicates a higher level of functioning. Matching of the patients in the amputation and limb-sparing groups for age and follow-up was based on an allowable range of 5 years and 12 months, respectively. To ensure that these ranges did not result in significant differences in the two patient groups, the variables of age and follow-up were analyzed to test for equality using paired t tests. The TESS, SF-36, and RNL scores for the two groups were compared using a paired t test. RESULTS The mean age of the amputation patients was 34.4 years (SD = 11.6) and the mean follow-up was 28.5 months Arch
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(SD = 15.1). The limb-sparing group had a mean age of 30.4 years (SD = 11.7) and follow-up was 30.5 months (SD = 25). These differences were not statistically significant. There were four women and eight men in the amputation group-5 AKA and 7 BKA. The amputation group consisted of five patients with soft-tissue sarcoma and seven with bone sarcoma. Four of the patients with high-grade osteosarcoma of bone were treated with chemotherapy. Although no patient in the amputation group with soft-tissue sarcoma received radiotherapy, nine of the ten patients with soft-tissue sarcoma and limb preservation received preoperative radiotherapy and 1 received postoperative radiotherapy. All patients in the amputation group healed primarily, whereas in the limb-preservation group there were four wound complications, one major complication that required the patient to be readmitted to the hospital for intravenous antibiotic therapy and three minor complications treated with dressings. Two patients in the limb-sparing group who had periosteal stripping of the distal tibia after preoperative radiotherapy developed stable hair-line fractures in the radiotherapy field that healed without intervention. The findings for the amputation and limb-sparing groups are summarized in table 1. As measured by the TESS, the amputation group had a mean physical disability score of 74.5 (SD = 19.7, range 31 to 95); whereas the limb-sparing group had a mean score of 85.1 (SD = 16.9, range 46 to 97). These differences were not statistically different (p = .15). Of the SF-36 subscales,only the physical function scoreswere significantly different (p value of .03). The amputation group had a mean score of 45 (SD = 28.7) and the limb-sparing group had a mean score of 71.1 (SD = 26.9). Handicap as measured by the RNL showed significant differences (p = .05), with the amputation group having a greater degree of handicap. The mean scores were 84.4 (SD = 16, range 62 to 110) for the amputation group versus 97 (SD = 15.1, range 67 to 110) for the limb-sparing group. In evaluating the amputation versus salvage patients on a case-bycase basis, we found that only one patient treated by limb sparing rated himself as more handicapped than his matched patient with amputation. In comparing the patients with AKA with their limb-sparing matches and then the patients with BKA with their matched controls, we found large differences in the scores of the two groups for most of the measures (table 2). These differences favored the limb-sparing group. As anticipated, the group of patients with AKA experienced more disability than the BKA group, as evidenced by lower scores on the TESS and the Table 1: Comparison for the Amputation
of Disability and Handicap Group and the Limb-Sparing Amputation (/I = 12) Mean (SD)
Toronto Extremity ShortformPhysical function
Salvage
Score
74.5
(19.7)
Scores Group
Limb-Sparing (n = 24) Mean (SD)
p Value
85.1 (16.9)
.I5
45.0
(28.7)
71.1
(26.9)
.03*
Role-physical Bodily pain General health
47.5 70.6 63.2
(46.4) (21.4) (28.8)
78.6 80.5 74.4
(35.2) (26.6) (20.3)
.09 .34 .31
Vitality Social function Role-emotional
57.0 (21.4) 78.8 (21.3) 86.7 (32.2)
69.6 (16.6) 88.4 (21.1) 84.5 (32.4)
.I5 .29 .87
70.0 84.4
78.6 (17.2) 97.0 (15.1)
.41 .05*
Mental health Reintegration to Normal * Statistically t test.
significant
Living group
differences
(28.7) (16.0)
in scores
based
on paired
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Table 2: Comparison, by Amputation Level, of Disability and Handicap Scores for the Patients With Amputations Matched With Limb-Sparing Controls Above Knee (n = 5) Mean (SD)
Limb Sparing (n = IO) Mean (SD)
BelOW Knee (n = 7) Mean (SD)
Limb Sparing (n= 14) Mean (SD)
63.7 (9.1)
84.8 (15.9)
75.6 (24.0)
85.7 (19.7)
Physical function Role-physical
38.3 (11.5) 25.0 (43.3)
47.8 (34.4)
71.7 (26.0) 83.3 (30.3)
Bodily pain General health
78.3 (24.9)
70.6 (29.3) 75.0 (40.1) 83.9 (23.8)
Toronto
Extremity
vage Shortform-
Sal-
Score
Vitality Social function Role-emotional Mental health Reintegration Living
62.0 (22.9) 46.7 (25.7) 66.7 (26.0) 89.0 (19.0)
72.9 (23.5) 69.4 (17.2) 85.9 (22.6) 79.1 (39.6)
34.7 (12.2) 84.3 (9.7)
57.1 (47.2) 67.3 (20.9) 63.7 (32.7) 62.1 (19.5) 83.9 (18.7)
76.0 (31.7) 76.3 (17.1) 70.0 (17.3)
77.5 (15.3)
85.7 (37.8) 85.1 (17.1)
91.7 (20.4) 91.7 (20.4) 80.0 (20.9)
96.1 (17.7)
85.1 (10.2)
98.2 (12.4)
to Normal
subscales of the SF-36. The scores for the RNL were virtually identical for the two groups (means of 84.3 and 85.1, respectively). Statistical analyses were not done on these subgroups because of the small number of subjects. Lastly, the patients with amputations were asked about prosthetic use. Seven of the 12 complained of “blisters,” “pressure sores,” or persistent difficulty with the fitting of their prostheses. One additional patient with an AKA experienced significant phantom-limb pain that limited walking. DISCUSSION The results of this study suggest that there is a trend toward increased disability-that is, restriction or lack of ability to perform an activity in the manner or within the range considered normal for a human being,3 in patients treated by amputation rather than limb-sparing surgery for bone and soft-tissue sarcoma of the lower extremity. This is evidenced by the lower scoreson the TESS and the physical function subscale of the SF-36. The TESS was developed based on the ICIDH3 definition of disability, whereas the SF-36 physical function subscale is a component of the generic health status measure.‘j Many of the items in the SF-36 physical function subscale represent physical disability, and there was a statistically significant difference between the two groups of patients. However, the underlying subscale structure of the SF-36 is not reproducible in the extremity tumor population,* using Ware’s6 methods such that the SF-36 scores must be interpreted with caution. The differences in handicap-that is, limitation in or prevention of a role that is normal (depending on age, gender, and social and cultural factors) for the individual3between the two groups were significantly different for the RNL, suggesting that patients with amputations experience greater limitations in normal role functions within their family, work, and social environments. One may theorize that the differences between amputation and limb sparing for lower extremity sarcoma are experienced by the patient at the level of handicap and less so at the level of disability, because the amputee group on a case-by-casebasis reported lower scores on the RNL. The work of Postma and colleagues,9 Weddington and associates,10and Marsden and Swanson” would also support this hypothesis. Using self-report questionnaires, semistructured interviews, and visual analogue scales, Postma found that the patients with limb preservation reported physical
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complaints more frequently but the patients with amputations had a trend toward lower self-esteem and social isolation. The “physical complaints” are subsumed by the ICIDH definition of disability, whereas social isolation falls within the definition of handicap.3 Weddington’O evaluated a number of psychologic factors, none of which measured handicap. His work, similar to that of Postma and our work, could demonstrate no differences in disability between individuals with amputation and those treated with limb sparing. Marsdenl’ evaluated two cohorts of patients-one with amputation and one with limb sparing-and found no statistical differences in function between the groups based on the Musculoskeletal Tumor Society Rating Scale.r2 Marsden,” however, noted associations between concerns about appearance and economic hardship, suggesting that social and economic costs are high for the individual with an amputation. These findings again support the theory that differences between patients with amputation and those treated by limb sparing occur at the level of handicap. The interpretation of the work by Sugarbaker within this framework of disability and handicap is less clear, mainly owing to the conflicting results of the two phases of the study. Sugarbaker used the Sickness Impact Profile,13 a generic measure of health that evaluates sickness-relateddysfunction in sleep, eating, work, home management, recreation, ambulation, mobility, body care and movement, social interaction, alertness behavior, emotional behavior, and communication. Phase one of Sugarbaker’s study suggested that the persons with amputation experienced less difficulty in sexual relationships and in body care, suffered less emotional disturbance, and had better health care orientation than persons with limb preservation. However, if one looks at the subtest data of the SicknessImpact Profile13 that relate to handicap (home management, social interaction, and work), the subtest scores of patients with amputation reflect greater difficulty than those of patients with limb sparing in all but home management. These score differences did not reach statistical significance but a trend to differences in handicap is noted, with the patients with amputation having lower scores. Phase two of the study failed to show differences between the amputation and limb-sparing groups on several of disability items. The interpretation of the disability data of our study and those of Postma, Weddington,1o Marsden,” and Sugarbaker must also be considered on a statistical basis. All may lack statistical power to detect a difference in disability scores. For example, in our study, 30 patients with amputations matched with 60 patients with limb sparing would be required for the observed lo-point difference in the TESS scores to be significantly different at the .05 level with 80% power. The studies of Weddington, lo Postma, Marsden,” and Sugarbaker have similarly small sample sizes, suggesting a lack of statistical power. Of further concern, based on the current data, is the number of patients with amputation (7 of 12, 2 with AKA and 5 with BKA) who describe persistent problems with skin breakdown and fitting of their prostheses. These difficulties resulted in periods in which the patients were unable to wear their prosthetic limbs, which may influence the patients’ ratings of their disability and handicap. The reason for these prosthetic problems is unclear, but it may be related to difficulties achieving sufficient soft tissue over the bone or irregular contour to the stump. Studies that have evaluated outcomes in patients with amputation and those with limb sparing have used a variety of outcome measures; hence, any attempt to pool data to achieve a larger sample size would be impossible. The current trend Arch
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toward limb sparing over amputation likely prohibits a single center from accruing a large enough sample to evaluate with confidence the differences in disability between these two groups of patients. Consequently, a multicentered effort with all institutions collecting data using the same measures is required. The measures should evaluate both disability and handicap to test the hypothesis generated from this study that differences between patients with amputation and those treated by limb sparing for bone and soft-tissue sarcoma are most marked at the level of handicap and less so at the disability level. This distinction is important for the health care team in counseling patients who may be candidates for limb sparing from an oncologic point of view but who are considered at high risk for morbidity and poor functional outcome. References 1. Simon MA, Aschliman MA, Thomas N, Mankin HJ. Limb-salvage treatment versus amputation for osteosarcoma of the distal end of the femur. J Bone Joint Surg Am 1986;68:1331-7. 2. Sugarbaker PH. Barofskv I. Rosenbere SA. Gianola FJ. Oualitv of life’ assessment of pat&s in extremity sarcoma clinical Gals. Surgery 1982;91:17-23. 3. World Health Organization. International classification of impairment, disability and handicap. Geneva: WHO; 1980. 4. Davis AM, Wright JG, Williams JI, Bombardier C, Griffin AM, Bell RS. Development of a measure of physical function for patients with bone and soft tissue sarcomas. Qua1 Life Res 1996;5:508-16.
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5. Davis AM, Bell RS, Badley EM, Yoshida K, Williams JI. Evaluating functional outcome in lower extremity sarcoma patients: a comparison of four measures. Clin Orthop 1999;358:90100. 6. Ware JE Jr. SF-36 Health Survey: manual and interpretation guide. Boston (MA): Nimrod Press; 1993. 7. Wood-Dauphne SL, Opzoomer MA, Williams JI, Marchand B, Spitzer WO. Assessment of global function: the Reintegration to Normal Living Index. Arch Phys Med Rehabil 1988;69:583-90. 8. Wood-Dauphne, Williams JI. Reintegration to normal living as a proxy to quality of life. J Chron Dis 1987;40:491-502. 9. Postma A, Kingma A, De Ruiter J, Koops HS, Veth RPH, Goeken LNH, et al. Quality of life in bone tumor patients comparing limb salvage and amputation of the lower extremity. J Surg Oncol 1992;51:47-51. 10. Weddington WW Jr, Segraves KB, Simon MA. Psychological outcome of extremity survivors undergoing amputation or limb salvage. J Clin Oncol 1985;3: 1393-9. 11. Marsden FW, Swanson CE. Outcomes after multi-modality treatment of musculoskeletal tumours. Acta Orthop Stand Suppl 1997;273:101-5. 12. Enneking WF, Dunham W, Gebhardt MC, Malawar M, Pritchard DJ. A system for the functional evaluation of reconstructive procedures after surgical treatment of tumors of the musculoskeleta1 system. Clin Orthop 1993;286:241-6. 13. Bergner M, Bobbit RA, Kressel S, Pollard WE, Gilson BS, Morris JR. The Sickness Impact Profile: conceptual formulation and methodology for the development of a health status measure. Int J Health Serv 1976;6:393-415.
Functional Outcome in Amputation Versus Limb Sparing of Patients With Lower Extremity Sarcoma: A Matched Case-Control Study Aileen M. Davis, PhD, Michael Devlin, MD, Anthony M. Grijjk, ABSTRACT. Davis AM, Devlin M, Griffin AM, Wunder JS, Bell RS. Functional outcome in amputation versus limb sparing of patients with lower extremity sarcoma: a matched casecontrol study. Arch Phys Med Rehabil 1999;80:61.5-8. Objective: To quantify the differences in physical disability and handicap experienced by patients with lower extremity sarcoma who required amputation for their primary tumor as compared with those treated by limb-sparing surgery. Design: Matched case-control study. Twelve patients with amputation were matched with 24 patients treated by limbsparing surgery on the following variables: age, gender, length of follow-up, bone versus soft-tissue tumor, anatomic site, and treatment with adjuvant chemotherapy. Patients: Patients who underwent above-knee amputation (AKA) or below-knee amputation (BKA) for primary softtissue or bone sarcoma, who had not developed local or systemic recurrence, and who had been followed up for at least 1 year since surgery. Main Outcome Measures: The Toronto Extremity Salvage Score (TESS), a measure of physical disability; the Shortform-36 (SF-36), a generic health status measure; and the Reintegration to Normal Living (RNL), a measure of handicap. Results: Mean TESS score for the patients with amputations was 74.5 versus 85.1 for the limb-sparing patients. (p = .15). Only the physical function subscale of the SF-36 showed statistically significant differences, with means of 45 and 71.1 for the amputation versus limb-sparing groups, respectively (p = .03). The RNL for the amputation group was 84.4 versus 97 for the limb-sparing group (p = .05). Seven of the 12 patients with amputations experienced ongoing difficulty with the soft tissues overlying their stumps. Conclusions: There was a trend toward increased disability for those in the amputation group versus those in the limbsparing group, with the amputation group showing significantly higher levels of handicap. These data suggest that the differences in disability between amputation and limb-sparing patients are smaller than anticipated. The differences may be more notable in measuring handicap. 0 1999 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation
From the University Musculoskeletal Oncology Unit (DE. Davis, Griffin, Wunder, Bell) and the Department of Physiatry (Dr. D&n), Mount Sinai Hospital and University of Toronto, Toronto, Canada. Submitted for publication July 23, 1998. Accepted in revised form December 8, 1998. No commercial party having a direct financial interest in the results of the research supporting this article has or will confer a benefit upon the. authors or upon any organization with which the authors are associated. Remint reauests to Aileen M. Davis. PhD. Mount Sinai Hosoital. 600 Universitv I Awn&, Suit; 476H, Toronto, Canada I&G 1x5. 0 1999 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation 0003.9993/99/8006-5140$3.00/O
BSc, Jay S. Wunder, MD, Robert S. Bell, MD
HEN SUFFICIENT MARGINS of resection surrounding the tumor can be achieved, limb-sparing surgery, as opposed to amputation, has become the standard of care in treating bone and soft-tissue sarcoma of the extremity.’ This standard has developed because of technologic advances in radiographic imaging and reconstructive surgery as well as the use of adjuvant therapies. Despite these advances, however, discussionspersist about whether an amputation would provide a superior functional result for specific patients. These discussions usually occur when the treatment team recognizes that there is significant risk of treatment morbidity and perceives that the functional result of the reconstructive procedure will be poor. Sugarbaker and associates2 published the landmark paper comparing the results of amputation versus limb-sparing surgery. The findings of 21 patients with soft-tissue sarcoma randomized to receive amputation plus chemotherapy (n = 9) or limb-sparing surgery plus irradiation and chemotherapy (IZ= 12) suggested that the anticipated improvements in quality of life of limb-sparing patients, as measured by a battery of standardized measures, was not substantiated. Individual components of the measures suggested that there was a trend toward improved sexual functioning and physical functioning in the amputation group based on subtest analysis of the various measures. However, the second phase of this study, using a second battery of tests to evaluate pain, mobility, treatment trauma, and sexual functioning, failed to confirm the results of the first phase. No differences were demonstrated between the amputation and limb-sparing groups in this second phase. Consequently, the discussions regarding the functional outcome of patients with amputations versus patients undergoing limbsparing surgery for extremity sarcoma persist. Recognizing the advances that have occurred in the treatment of patients with soft-tissue and bone sarcoma over the past two decades and the ongoing debate about limb sparing versus amputation in this population, our group endeavored to clarify the functional outcomes of these groups of patients. Working from the International Classification of Impairment, Disability and Handicap (ICIDH) definitions of disability and handicap,3 the purpose of this matched case-control study was to quantify the differences in physical disability and handicap experienced by patients with lower extremity sarcoma who required amputation for their primary tumor as compared with those who could be treated by limb-sparing surgery. The ICIDH defines disability as any restriction or lack of ability to perform an activity in the manner or within the range considered normal for a human being.3 Handicap is limitation in, or prevention of, a role that is normal (depending on age, gender, and social and cultural factors) for the individual3
w
METHODS The cases for this study were defined by patients who (1) were treated by above-knee amputation (AKA) or below-knee amputation (BKA) for a nonlocally recurrent bone or soft-tissue sarcoma that was otherwise unresectable, (2) had a minimum of Arch
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1 year of follow-up since their amputation, and (3) had not developed local or systemic recurrence. Between January 1986 and October 1995,26 patients with primary bone or soft-tissue sarcoma were treated by AKA or BKA. Nine of these patients died of progressive disease, four were alive with metastatic disease, and 13 were eligible for the study. During the same period, 239 patients with lower extremity tumor were treated with limb-sparing surgery. The decision to primarily amputate the extremity was determined by the inability to adequately resect the tumor and reconstruct a functional limb. For bone sarcoma, complete resection is necessary, whereas for soft-tissue sarcoma, gross tumor resection, leaving no more than a small microscopic burden of tumor that is amenable to control with adjuvant radiotherapy, is acceptable. Indications for amputation included encasement of the neurovascular structures by the tumor, tumor progression on chemotherapy, or inadvertent contamination of the surgical field from previous biopsy or surgery. Patients treated with limb-sparing surgery who remained disease free were chosen as matched controls for the amputation group. The matching variables were tumor site such that the alternative to salvage was the level of amputation of the matched case, age (within 5 years), gender, length of follow-up (within 12 months providing the minimum follow-up was 1 year), bone or soft-tissue tumor, and treatment with adjuvant chemotherapy. The ratio of limb-sparing patients matched to amputation subjects was 2:l to improve the power of the study. Of the 13 patients with amputations who were eligible for the study, however, one patient who underwent BKA for soft-tissue sarcoma did not return the questionnaires. Hence, 12 patients with amputations matched to 24 patients undergoing limbsparing surgery were the focus of the study. Subjects completed the Toronto Extremity Salvage Score (TESS), a measure of physical disability developed specifically for the extremity tumor population4,5; the Shortform(SF36), a generic health status measure,6 and the Reintegration to Normal Living (RNL), a measure of handicap developed for cancer patients7v8 The TESS consists of 30 items, and the patient rates the difficulty experienced completing an activity on a 5-point Likert scale.The final score is converted to a range from 0 to 100. The SF-36 consists of 36 items scored in 8 subscales (physical function, role-physical, bodily pain, general health, vitality, social function, role-emotional, and mental health). Each subscale score ranges from 0 to 100. The RNL consists of 11 items: moving around living quarters, moving around community, ability to take trips out of town, comfort with self-care needs, occupation in work activities, participation in recreational activities, participation in social activities, assumption of role in family, comfort with personal relationships, comfort with self in company of others, and ability to deal with life events. The items are rated by the subject on a visual analogue scale, with the RNL score ranging from 0 to 110. For the TESS, SF-36, and RNL, a higher score indicates a higher level of functioning. Matching of the patients in the amputation and limb-sparing groups for age and follow-up was based on an allowable range of 5 years and 12 months, respectively. To ensure that these ranges did not result in significant differences in the two patient groups, the variables of age and follow-up were analyzed to test for equality using paired t tests. The TESS, SF-36, and RNL scores for the two groups were compared using a paired t test. RESULTS The mean age of the amputation patients was 34.4 years (SD = 11.6) and the mean follow-up was 28.5 months Arch
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(SD = 15.1). The limb-sparing group had a mean age of 30.4 years (SD = 11.7) and follow-up was 30.5 months (SD = 25). These differences were not statistically significant. There were four women and eight men in the amputation group-5 AKA and 7 BKA. The amputation group consisted of five patients with soft-tissue sarcoma and seven with bone sarcoma. Four of the patients with high-grade osteosarcoma of bone were treated with chemotherapy. Although no patient in the amputation group with soft-tissue sarcoma received radiotherapy, nine of the ten patients with soft-tissue sarcoma and limb preservation received preoperative radiotherapy and 1 received postoperative radiotherapy. All patients in the amputation group healed primarily, whereas in the limb-preservation group there were four wound complications, one major complication that required the patient to be readmitted to the hospital for intravenous antibiotic therapy and three minor complications treated with dressings. Two patients in the limb-sparing group who had periosteal stripping of the distal tibia after preoperative radiotherapy developed stable hair-line fractures in the radiotherapy field that healed without intervention. The findings for the amputation and limb-sparing groups are summarized in table 1. As measured by the TESS, the amputation group had a mean physical disability score of 74.5 (SD = 19.7, range 31 to 95); whereas the limb-sparing group had a mean score of 85.1 (SD = 16.9, range 46 to 97). These differences were not statistically different (p = .15). Of the SF-36 subscales,only the physical function scoreswere significantly different (p value of .03). The amputation group had a mean score of 45 (SD = 28.7) and the limb-sparing group had a mean score of 71.1 (SD = 26.9). Handicap as measured by the RNL showed significant differences (p = .05), with the amputation group having a greater degree of handicap. The mean scores were 84.4 (SD = 16, range 62 to 110) for the amputation group versus 97 (SD = 15.1, range 67 to 110) for the limb-sparing group. In evaluating the amputation versus salvage patients on a case-bycase basis, we found that only one patient treated by limb sparing rated himself as more handicapped than his matched patient with amputation. In comparing the patients with AKA with their limb-sparing matches and then the patients with BKA with their matched controls, we found large differences in the scores of the two groups for most of the measures (table 2). These differences favored the limb-sparing group. As anticipated, the group of patients with AKA experienced more disability than the BKA group, as evidenced by lower scores on the TESS and the Table 1: Comparison for the Amputation
of Disability and Handicap Group and the Limb-Sparing Amputation (/I = 12) Mean (SD)
Toronto Extremity ShortformPhysical function
Salvage
Score
74.5
(19.7)
Scores Group
Limb-Sparing (n = 24) Mean (SD)
p Value
85.1 (16.9)
.I5
45.0
(28.7)
71.1
(26.9)
.03*
Role-physical Bodily pain General health
47.5 70.6 63.2
(46.4) (21.4) (28.8)
78.6 80.5 74.4
(35.2) (26.6) (20.3)
.09 .34 .31
Vitality Social function Role-emotional
57.0 (21.4) 78.8 (21.3) 86.7 (32.2)
69.6 (16.6) 88.4 (21.1) 84.5 (32.4)
.I5 .29 .87
70.0 84.4
78.6 (17.2) 97.0 (15.1)
.41 .05*
Mental health Reintegration to Normal * Statistically t test.
significant
Living group
differences
(28.7) (16.0)
in scores
based
on paired
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Table 2: Comparison, by Amputation Level, of Disability and Handicap Scores for the Patients With Amputations Matched With Limb-Sparing Controls Above Knee (n = 5) Mean (SD)
Limb Sparing (n = IO) Mean (SD)
BelOW Knee (n = 7) Mean (SD)
Limb Sparing (n= 14) Mean (SD)
63.7 (9.1)
84.8 (15.9)
75.6 (24.0)
85.7 (19.7)
Physical function Role-physical
38.3 (11.5) 25.0 (43.3)
47.8 (34.4)
71.7 (26.0) 83.3 (30.3)
Bodily pain General health
78.3 (24.9)
70.6 (29.3) 75.0 (40.1) 83.9 (23.8)
Toronto
Extremity
vage Shortform-
Sal-
Score
Vitality Social function Role-emotional Mental health Reintegration Living
62.0 (22.9) 46.7 (25.7) 66.7 (26.0) 89.0 (19.0)
72.9 (23.5) 69.4 (17.2) 85.9 (22.6) 79.1 (39.6)
34.7 (12.2) 84.3 (9.7)
57.1 (47.2) 67.3 (20.9) 63.7 (32.7) 62.1 (19.5) 83.9 (18.7)
76.0 (31.7) 76.3 (17.1) 70.0 (17.3)
77.5 (15.3)
85.7 (37.8) 85.1 (17.1)
91.7 (20.4) 91.7 (20.4) 80.0 (20.9)
96.1 (17.7)
85.1 (10.2)
98.2 (12.4)
to Normal
subscales of the SF-36. The scores for the RNL were virtually identical for the two groups (means of 84.3 and 85.1, respectively). Statistical analyses were not done on these subgroups because of the small number of subjects. Lastly, the patients with amputations were asked about prosthetic use. Seven of the 12 complained of “blisters,” “pressure sores,” or persistent difficulty with the fitting of their prostheses. One additional patient with an AKA experienced significant phantom-limb pain that limited walking. DISCUSSION The results of this study suggest that there is a trend toward increased disability-that is, restriction or lack of ability to perform an activity in the manner or within the range considered normal for a human being,3 in patients treated by amputation rather than limb-sparing surgery for bone and soft-tissue sarcoma of the lower extremity. This is evidenced by the lower scoreson the TESS and the physical function subscale of the SF-36. The TESS was developed based on the ICIDH3 definition of disability, whereas the SF-36 physical function subscale is a component of the generic health status measure.‘j Many of the items in the SF-36 physical function subscale represent physical disability, and there was a statistically significant difference between the two groups of patients. However, the underlying subscale structure of the SF-36 is not reproducible in the extremity tumor population,* using Ware’s6 methods such that the SF-36 scores must be interpreted with caution. The differences in handicap-that is, limitation in or prevention of a role that is normal (depending on age, gender, and social and cultural factors) for the individual3between the two groups were significantly different for the RNL, suggesting that patients with amputations experience greater limitations in normal role functions within their family, work, and social environments. One may theorize that the differences between amputation and limb sparing for lower extremity sarcoma are experienced by the patient at the level of handicap and less so at the level of disability, because the amputee group on a case-by-casebasis reported lower scores on the RNL. The work of Postma and colleagues,9 Weddington and associates,10and Marsden and Swanson” would also support this hypothesis. Using self-report questionnaires, semistructured interviews, and visual analogue scales, Postma found that the patients with limb preservation reported physical
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complaints more frequently but the patients with amputations had a trend toward lower self-esteem and social isolation. The “physical complaints” are subsumed by the ICIDH definition of disability, whereas social isolation falls within the definition of handicap.3 Weddington’O evaluated a number of psychologic factors, none of which measured handicap. His work, similar to that of Postma and our work, could demonstrate no differences in disability between individuals with amputation and those treated with limb sparing. Marsdenl’ evaluated two cohorts of patients-one with amputation and one with limb sparing-and found no statistical differences in function between the groups based on the Musculoskeletal Tumor Society Rating Scale.r2 Marsden,” however, noted associations between concerns about appearance and economic hardship, suggesting that social and economic costs are high for the individual with an amputation. These findings again support the theory that differences between patients with amputation and those treated by limb sparing occur at the level of handicap. The interpretation of the work by Sugarbaker within this framework of disability and handicap is less clear, mainly owing to the conflicting results of the two phases of the study. Sugarbaker used the Sickness Impact Profile,13 a generic measure of health that evaluates sickness-relateddysfunction in sleep, eating, work, home management, recreation, ambulation, mobility, body care and movement, social interaction, alertness behavior, emotional behavior, and communication. Phase one of Sugarbaker’s study suggested that the persons with amputation experienced less difficulty in sexual relationships and in body care, suffered less emotional disturbance, and had better health care orientation than persons with limb preservation. However, if one looks at the subtest data of the SicknessImpact Profile13 that relate to handicap (home management, social interaction, and work), the subtest scores of patients with amputation reflect greater difficulty than those of patients with limb sparing in all but home management. These score differences did not reach statistical significance but a trend to differences in handicap is noted, with the patients with amputation having lower scores. Phase two of the study failed to show differences between the amputation and limb-sparing groups on several of disability items. The interpretation of the disability data of our study and those of Postma, Weddington,1o Marsden,” and Sugarbaker must also be considered on a statistical basis. All may lack statistical power to detect a difference in disability scores. For example, in our study, 30 patients with amputations matched with 60 patients with limb sparing would be required for the observed lo-point difference in the TESS scores to be significantly different at the .05 level with 80% power. The studies of Weddington, lo Postma, Marsden,” and Sugarbaker have similarly small sample sizes, suggesting a lack of statistical power. Of further concern, based on the current data, is the number of patients with amputation (7 of 12, 2 with AKA and 5 with BKA) who describe persistent problems with skin breakdown and fitting of their prostheses. These difficulties resulted in periods in which the patients were unable to wear their prosthetic limbs, which may influence the patients’ ratings of their disability and handicap. The reason for these prosthetic problems is unclear, but it may be related to difficulties achieving sufficient soft tissue over the bone or irregular contour to the stump. Studies that have evaluated outcomes in patients with amputation and those with limb sparing have used a variety of outcome measures; hence, any attempt to pool data to achieve a larger sample size would be impossible. The current trend Arch
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toward limb sparing over amputation likely prohibits a single center from accruing a large enough sample to evaluate with confidence the differences in disability between these two groups of patients. Consequently, a multicentered effort with all institutions collecting data using the same measures is required. The measures should evaluate both disability and handicap to test the hypothesis generated from this study that differences between patients with amputation and those treated by limb sparing for bone and soft-tissue sarcoma are most marked at the level of handicap and less so at the disability level. This distinction is important for the health care team in counseling patients who may be candidates for limb sparing from an oncologic point of view but who are considered at high risk for morbidity and poor functional outcome. References 1. Simon MA, Aschliman MA, Thomas N, Mankin HJ. Limb-salvage treatment versus amputation for osteosarcoma of the distal end of the femur. J Bone Joint Surg Am 1986;68:1331-7. 2. Sugarbaker PH. Barofskv I. Rosenbere SA. Gianola FJ. Oualitv of life’ assessment of pat&s in extremity sarcoma clinical Gals. Surgery 1982;91:17-23. 3. World Health Organization. International classification of impairment, disability and handicap. Geneva: WHO; 1980. 4. Davis AM, Wright JG, Williams JI, Bombardier C, Griffin AM, Bell RS. Development of a measure of physical function for patients with bone and soft tissue sarcomas. Qua1 Life Res 1996;5:508-16.
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5. Davis AM, Bell RS, Badley EM, Yoshida K, Williams JI. Evaluating functional outcome in lower extremity sarcoma patients: a comparison of four measures. Clin Orthop 1999;358:90100. 6. Ware JE Jr. SF-36 Health Survey: manual and interpretation guide. Boston (MA): Nimrod Press; 1993. 7. Wood-Dauphne SL, Opzoomer MA, Williams JI, Marchand B, Spitzer WO. Assessment of global function: the Reintegration to Normal Living Index. Arch Phys Med Rehabil 1988;69:583-90. 8. Wood-Dauphne, Williams JI. Reintegration to normal living as a proxy to quality of life. J Chron Dis 1987;40:491-502. 9. Postma A, Kingma A, De Ruiter J, Koops HS, Veth RPH, Goeken LNH, et al. Quality of life in bone tumor patients comparing limb salvage and amputation of the lower extremity. J Surg Oncol 1992;51:47-51. 10. Weddington WW Jr, Segraves KB, Simon MA. Psychological outcome of extremity survivors undergoing amputation or limb salvage. J Clin Oncol 1985;3: 1393-9. 11. Marsden FW, Swanson CE. Outcomes after multi-modality treatment of musculoskeletal tumours. Acta Orthop Stand Suppl 1997;273:101-5. 12. Enneking WF, Dunham W, Gebhardt MC, Malawar M, Pritchard DJ. A system for the functional evaluation of reconstructive procedures after surgical treatment of tumors of the musculoskeleta1 system. Clin Orthop 1993;286:241-6. 13. Bergner M, Bobbit RA, Kressel S, Pollard WE, Gilson BS, Morris JR. The Sickness Impact Profile: conceptual formulation and methodology for the development of a health status measure. Int J Health Serv 1976;6:393-415.