© The Author (2008). Published by Oxford University Press. All rights reserved
Clinical Problems After Initial Unplanned Resection of Sarcoma
1 Department of Orthopedic Surgery, Osaka City University Graduate School of Medicine
2 Department of Orthopedic Surgery, Yodogawa Christian Hospital
3 Department of Orthopedic Surgery, Osaka City General Hospital, Osaka, Japan
For reprints and all correspondence: Manabu Hoshi, Department of Orthopedic Surgery, Osaka City University Graduate School of Medicine, 1-4-3 Asahi, Abeno-Ku, Osaka 545-8585, Japan. E-mail: hoshi{at}med.osaka-cu.ac.jp
Received April 17, 2008; accepted August 14, 2008
 |
Abstract |
|---|
 TOP Abstract INTRODUCTIONPATIENTS AND METHODSRESULTSDISCUSSIONReferences |
|---|
Objective: Unplanned resection of a sarcoma is often chosen in the early phase by general physicians without any imaging scrutiny. The present study aimed to highlight the clinical problems associated with unplanned resection of sarcomas.
Methods: Thirty-eight patients who underwent unplanned resection of a sarcoma and additional treatment were examined. The definite histological grading was high in 31 patients and low in 7 patients.
Results: The tumors were located in the depth of the subfascia in 13 patients. The maximal tumor sizes exceeded 5 cm in 16 patients. Preoperative MRI was only performed in six patients. The previous surgical margins were intralesional in 20 patients and marginal in 18 patients. Inappropriate transverse skin incisions were found in 21 patients. Extensive hematoma at the initial surgical site was seen in five patients. Thirty-three patients accepted additional wide resection due to the insufficient removal of malignancy. During an average follow-up of 42.7 months, seven patients died of lung and brain metastases.
Conclusions: On excision of any soft tissue tumor, surgeons should be aware of the potential risk for erroneous management of malignancy. If not, careless surgery may render the treatment protocol complicated and require excessive additional tissue resection with poor function and prognosis. Appropriate salvage treatment may have a significant role to play after unplanned resection of the sarcoma.
Key Words: surgery • sarcoma • unplanned resection
|
INTRODUCTION |
|---|
|
TOPAbstractINTRODUCTIONPATIENTS AND METHODSRESULTSDISCUSSIONReferences |
|---|
Sarcomas in soft tissue are rare and have been reported to comprise only 0.8% of all cancers (1). Consequently, they are apt to be ignored by general physicians and are surgically excised as benign tumors or inflammatory lesions without wide margins. When postoperative pathology reveals malignancy, or local recurrences or distant metastases are found, the patients are usually referred to specialists in musculoskeletal oncology and are often recommended to undergo additional wide resection to ensure local curability and adjuvant chemotherapy.
The present study examined the clinical courses of 38 such cases to address the problems associated with inadequate surgeries for soft tissue sarcomas in the initial stage. The standard procedures for the treatment of such cases are discussed.
|
PATIENTS AND METHODS |
|---|
|
TOPAbstractINTRODUCTIONPATIENTS AND METHODSRESULTSDISCUSSIONReferences |
|---|
A total of 38 soft tissue sarcoma patients who underwent inappropriate surgical resections without wide safety margins and required additional treatment from April 1982 to March 2007 were reviewed. The patients comprised 18 males and 20 females who were followed up for >10 months in our institutions. All tumors were surgically removed without consideration of the surgical margins in other hospitals. The patients' age ranged from 11 to 89 years (mean 49.4 ± 19.8 years). The median follow-up period was 42.7 ± 34.0 months (range 10–132 months). No patients were lost during the follow-up.
At the first visit to our institutions, the patients were evaluated radiologically using plain radiographs, local computed tomography (CT) and magnetic resonance imaging (MRI) with or without Gd enhancement. Distant metastases to the lungs were also checked by CT. Pathological sections of the originally excised tumors were reviewed by experts in pathology before the start of the additional treatment protocol. The principles of additional wide resection were in accordance with those previously described (2,3) and involved the creation of a 3-cm wide margin from the previous surgical scar. The reactive fibrous scar tissue, hematoma and edema in each lesion were mainly evaluated by CT, T2-weighted and Gd-enhanced T1-weighted MRI (Fig. 1a–d) with the aim of complete removal. After additional wide resection, we checked the gross and histological findings of the removed marginal tissues to detect residual tumors (Fig. 1e). If the surgical margin was judged to be inadequate, we added postoperative local radiation therapy and/or systemic chemotherapy.
|
If the lesion was assessed as unresectable by radiology or resection was likely to cause extensive loss of the major limb function, we recommended that the patients receive a new therapeutic modality of carbon ion radiotherapy (4) with or without systemic chemotherapy.
The clinical information was reviewed in the present study with a focus on the tumor location, tumor site, tumor size, presumptive diagnoses at the previous surgery, definitive diagnosis, histological grade of the sarcoma, interval between the initial and additional surgery and histological detection of malignant cells at the widely resected margin.
The clinical records of inappropriate initial procedures were investigated, in terms of the specialty of the initial physician, surgical margin of the initial tumor resection and available preoperative imaging data. After additional wide resection, the oncological outcome, including additional surgery, chemotherapy, radiation therapy and prognosis at final follow-up, was reviewed.
The 5-year survival and event-free rates were calculated by the Kaplan–Meier analysis, and the prognostic factors concerning location, depth, tumor size, histological grading, interval until additional surgery, residual tumor, surgical margin of initial resection, the way of skin incision, extensive hematoma, chemotherapy and initial metastases were examined. All calculations were performed with the aid of the StatView statistical package (Abacus Concepts Inc., CA, USA).
The patients and/or their families provided informed consent for the treatment procedures and permission to publish their medical data.
|
RESULTS |
|---|
|
TOPAbstractINTRODUCTIONPATIENTS AND METHODSRESULTSDISCUSSIONReferences |
|---|
The clinical data of the 38 patients who underwent unplanned resection of a sarcoma are listed in Table 1. The tumors were located in the thigh in 11 patients, upper arm in 8, forearm in 6, lower leg in 5, trunk in 5 and knee in 3. The tumors were located subcutaneously in 25 patients and inter/intramuscularly in 13. One tumor arose from the brachial plexus. The maximal sizes of the original tumors ranged from 1.0–18.0 cm (mean 5.6 ± 4.4 cm), and 16 of 38 patients (42.1%) had tumors >5 cm at their first visit to a physician.
|
As shown in Table 1, the list of presumptive diagnoses indicated that physicians tended to assume that the tumors were benign, except for three cases where a sarcoma was suspected. The preoperative presumptive diagnoses were non-specified benign tumor in 22 patients, lipoma in 3, epidermoid cyst in 3, hematoma in 2, giant cell tumor of the tendon sheath in 1, neurofibroma in 1, infectious disease in 1, swollen lymph node in 1 and desmoid in 1. However, the postoperative pathological diagnoses were malignant fibrous histiocytoma in 11 patients, liposarcoma in 8 (well-differentiated in 2, myxoid-round in 3, pleomorphic in 1 and de-differentiated in 2), synovial sarcoma in 5, leiomyosarcoma in 3, epithelioid sarcoma in 2, clear cell sarcoma in 1, low-grade myofibroblastic sarcoma in 1, malignant mixed tumor in 1, malignant peripheral nerve sheath tumor in 1, fibrosarcoma in 1, myofibrosarcoma in 1, epithelioid hemangioendothelioma in 1, malignant hemangiopericytoma in 1 and myoinflammatory fibroblastic sarcoma in 1. Grading of malignancy on histology was high in 31 patients and low in 7. The mean interval from the initial resection to additional wide resection was 108 ± 171 days, and the mean interval from the first visit to additional resection was 48.6 ± 66.1 days. It was calculated to be
2 months from the initial surgery to visit to us. The pathology of marginal tissue obtained during the additional wide resection revealed the presence of microscopic residual tumor cells in 23 of 35 cases (65.8%). The resections with erroneous margins and the directions of the skin incisions in the initial resection surgeries are shown in Table 2. Most of the patients underwent resection without adequate preoperative CT and/or MRI to make the presumptive diagnoses. Preoperative CT without MRI was performed in two cases, whereas MRI without CT was performed in one case and both CT and MRI were performed in five cases.
|
With regard to the specialty of the surgeons of the initial surgeries, 17 were orthopedic surgeons, 16 were general surgeons, 3 were plastic surgeons and 2 were dermatologists. Regarding the excision methods, 20 patients underwent intralesional excision and 18 underwent marginal excision. Incorrect transverse skin incisions were made in 21 cases (Fig. 2a). An extensive postoperative hematoma after the initial operation was observed in five cases (Fig. 2b).
|
The treatment programs and outcomes of the 38 cases are shown in Table 3. Additional wide resection was performed in 33 patients and amputation in 2, while carbon ion radiotherapy was applied to 2 patients without wide resection. Radiotherapy at a dose of 30 Gy per 10 fractions was carried out in one patient with pelvic and lung metastases at the first visit (Case 20). Systemic chemotherapy was carried out in 18 patients. After additional wide resection, the major skin and soft tissue defects were covered with a skin graft in 15 patients and a musculocutaneous flap in 3 (Fig. 3). During an average follow-up of 42.7 ± 34.0 months, 25 patients remained continuously disease-free, 4 patients showed no evidence of disease and 2 were alive with disease, whereas 7 patients died of sarcoma-related diseases. According to a Kaplan–Meier analysis, the overall 5-year survival rate was 76.3% and the overall 5-year event-free rate was 54.6% (Fig. 4). Among many various factors examined in the present study, only two factors of systemic chemotherapy and initial metastases significantly contributed to their prognoses. The 5-year survival rate of the patients receiving systemic chemotherapy was significantly lower than than those without systemic chemotherapy (P < 0.05), and initial distant metastases significantly reduced their 5-year survival rates (P < 0.01).
|
|
|
|
DISCUSSION |
|---|
|
TOPAbstractINTRODUCTIONPATIENTS AND METHODSRESULTSDISCUSSIONReferences |
|---|
Unplanned resection of a sarcoma has been discussed in several previous reports. Giuliano and Eilber (5) were the first authors to describe the term 'unplanned total excision', meaning excisional biopsies for presumed benign diseases. Noria et al. (6) further defined the term 'unplanned resection of a sarcoma' as an excisional biopsy or unplanned resection of a lesion without the benefits of preoperative imaging and without regard for the necessity of removing the lesion with a margin of normal tissue. We further considered that the clinical problems associated with unplanned resection of a sarcoma were applicable to surgical procedures without consideration of potential malignancy. Performance of an incorrect procedure usually results from a lack of awareness of the standard treatment for sarcomas (3). Education and standardization of the treatment for soft tissue tumors are essential for avoiding unplanned excision.
In the present study, the clinical problems after unplanned resection of a sarcoma were reviewed. Soft tissue malignant tumors can arise from any part of the body with quite low incidences. Therefore, physicians with specialties other than surgical oncology participate in the management of such tumors, especially when the tumor is located subcutaneously, and tend to perform simple tumor excisions without any concern for malignancy. In order to avoid such erroneous surgery, the initial physicians must be aware of the following clinical signs suggesting malignancy: (i) mass >5 cm in maximal size; (ii) rapid tumor growth; (iii) mass deeply located in the muscles; (iv) heterogeneous enhancement on contrast MRI; (v) sudden-onset pain in a painless mass (7). Preoperative CT and MRI examinations are essential for a presumptive diagnosis, especially when the clinical symptoms for malignancy are present.
The mean interval from the initial resection to additional wide resection was 108 ± 171 days. It seems to be too long, and it took >100 days from the initial surgery to visit to us in 23.7% (9 of 38). Interestingly, in these nine cases, initial physicians were three (33%) orthopedic surgeons, and five were general surgeons and one dermatologist. Performance of an incorrect procedure usually results from a lack of awareness of the standard treatment for sarcomas. After the pathological diagnosis of the resected specimen confirmed malignant, or after local recurrence or distant metastases were discovered, the patients should promptly be referred to oncologists familiar with musculoskeletal tumors. Additional wide resection should be planned as soon as possible in a specialized oncology institution.
A biopsy or an excisional biopsy is essential to determine the pathological diagnosis and create a management program for a musculoskeletal tumor, and should be carried out as carefully as possible, even if the tumor is benign (8). Previously described incorrect procedures (7–9) are: (i) transverse skin incision; (ii) excessive postoperative hematoma; (iii) insult to uninvolved joints; (iv) drainage away from the skin incision; (v) broad mattress skin sutures.
If the skin incision is not longitudinal, the procedure has the potential to expand the tumor contamination over several muscles. In 21 of the 38 cases described here, transverse skin incisions were made during the initial surgery, and such incisions must be avoided to prevent subsequent wide resection surgery, as extensive resection of muscle fibers that are oriented longitudinally and consequent postoperative loss of limb function are inevitable.
Postoperative extensive hematoma should also be avoided to prevent local dissemination of tumor cells. Tumor cells can expand with bleeding, which theoretically leads to an increased probability of tumor recurrence over a wide area. Five cases in the present series had a postoperative extensive hematoma after the initial surgery that may have resulted in residual tumor cells at the margin of the additional wide resection. In such cases, amputation may have to be recommended to obtain a strict safety margin, although fortunately no local recurrences were noted at final follow-up in this series. To avoid such incorrect surgical procedures, biopsies for tumors suspected of malignancy on preoperative imaging should be carried out in oncology centers, as previously recommended (8).
In additional wide resection, the determination of the surgical safety margin is not easy, as the complicated CT and/or MRI scans show postoperative reactive changes from the initial surgery (10–14). Sugiura et al. (15) proposed a surgical safety margin of 3 cm from high-signal intensity areas on T2 images and on enhanced CT to reduce local recurrence rates. Following these criteria, we designed a surgical safety margin of 3 cm from reactive high-signal intensity areas on T2-weighted MRI and Gd-enhanced areas on T1-weighted MRI. Once an unplanned resection has been completed, the surgical margin of the additional resection needs to be more extensive than the initial margin.
Three patients with unresectable sarcoma were recommended for treatment with carbon ion radiotherapy. This new treatment modality is expected to be preferable for controlling local recurrence of malignant tumors and may represent an alternative modality to additional wide resection (4). However, the efficacy of carbon ion radiotherapy for this purpose has not been fully elucidated.
Among the major risk factors of the 5-year survival rate, statistical significance was found only in systemic chemotherapy and initial metastases, not in location, depth, tumor size, histological grading, interval until additional surgery, residual tumor, surgical margin of initial resection, the way of skin incision and extensive hematoma. The patients receiving systemic chemotherapy had worse prognoses, rather than those without chemotherapy. Hence, systemic chemotherapy was, in most cases, administered to patients with a diagnosis of high-grade sarcoma with advanced stage. Actually, initial distant metastases also significantly decreased the 5-year survival rate in the present study.
Regarding the prognosis of patients who have received unplanned resection of a sarcoma, several reports from oncology centers have reported variable outcomes (15–20). Ueda et al. (16) described that the local recurrence rate was higher in patients who received inadequate initial resection than in patients who received successful primary tumor resection in the same institution. However, Fiore et al. (17) reported that there were no significant differences in the local relapse, metastasis and mortality rates between patients requiring a re-excision and those with a successful primary excision. Lewis et al. (18) reported that the disease-specific metastasis-free survival rate was lower in patients who underwent re-excision than in patients who received one successful primary operation. However, Kawaguchi previously reported that surgical treatment of the intralesional or marginal margin of a sarcoma results in insufficient curability (2,3). Unplanned resection of sarcoma leads to these unpreferable situations. If the salvage treatment could be performed appropriately, the oncological outcomes may not be influenced (15). In the present study, the 5-year survival rate in the present series was 76.3% and the outcomes were almost equivalent to those for sarcomas excised in one resection in previous reports (2,3), suggesting a significant role of additional wide resection in improving the treatment outcomes of unplanned resections compared with insufficient treatment without additional wide resection.
Conflict of interest statement
None declared.
|
References |
|---|
|
TOPAbstractINTRODUCTIONPATIENTS AND METHODSRESULTSDISCUSSIONReferences |
|---|
1 Jemal A, Siegel R, Ward E, Murray T, Xu J, Thun MJ. Cancer statistics, 2007. CA Cancer J Clin (2007) 57:43–66.
2 Kawaguchi N, Matumoto S, Manabe J. New method of evaluating the surgical margin and safety margin for musculoskeletal sarcoma, analysed on the basis of 457 surgical cases. J Cancer Res Clin Oncol (1995) 121:555–63.[CrossRef][Web of Science][Medline]
3 Kawaguchi N, Ahmed AR, Matsumoto S, Manabe J, Matsushita Y. The concept of curative margin in surgery for bone and soft tissue sarcoma. Clin Orthop Relat Res (2004) 419:165–72.[CrossRef][Medline]
4 Kamada T, Tsujii H, Tsuji H, Yanagi T, Mizoe JE, Miyamoto T, et al. Efficacy and safety of carbon ion radiotherapy in bone and soft tissue sarcomas. J Clin Oncol (2002) 20:4466–71.
5 Giuliano AE, Eilber FR. The rationale for planned reoperation after unplanned total excision of soft-tissue sarcomas. J Clin Oncol (1985) 3:1344–8.
6 Noria S, Davis A, Kandel R, Levesque J, O'Sullivan B, Wunder J, et al. Residual disease following unplanned excision of soft-tissue sarcoma of an extremity. J Bone Joint Surg Am (1996) 78:650–5.
7 Wong CK, Lam YL, So YC, Ngan KC, Wong KY. Management of extremity soft tissue sarcoma after unplanned incomplete resection: experience of a regional musculoskeletal tumour centre. Hong Kong Med J (2004) 10:117–22.[Medline]
8 Mankin HJ, Lange TA, Spanier SS. The hazards of biopsy in patients with malignant primary bone and soft-tissue tumors. J Bone Joint Surg Am (1982) 64:1121–7.
9 Siebenrock KA, Hertel R, Ganz R. Unexpected resection of soft-tissue sarcoma. More mutilating surgery, higher local recurrence rates, and obscure prognosis as consequences of improper surgery. Arch Orthop Trauma Surg (2000) 120:65–9.[Medline]
10 Enneking WF, Spanier SS, Goodman MA. A system for the surgical staging of musculoskeletal sarcoma. Clin Orthop Relat Res (1980) 153:106–20.[Medline]
11 Kaste SC, Hill A, Conley L, Shidler TJ, Rao BN, Neel MM, et al. Magnetic resonance imaging after incomplete resection of soft tissue sarcoma. Clin Orthop Relat Res (2002) 397:204–11.[CrossRef][Medline]
12 Davis AM, Kandel RA, Wunder JS, Unger R, Meer J, O'Sullivan B. The impact of residual disease on local recurrence in patients treated by initial unplanned resection for soft tissue sarcoma of the extremity. J Surg Oncol (1997) 66:81–7.[CrossRef][Web of Science][Medline]
13 Shapeero LG, Vanel D, Verstraete KL, Bloem JL. Fast magnetic resonance imaging with contrast for soft tissue sarcoma viability. Clin Orthop Relat Res (2002) 397:212–27.[CrossRef][Medline]
14 Stojadinovic A, Leung DH, Hoos A, Jaques DP, Lewis JJ, Brennan MF. Analysis of the prognostic significance of microscopic margins in 2,084 localized primary adult soft tissue sarcomas. Ann Surg (2002) 235:424–34.[CrossRef][Web of Science][Medline]
15 Sugiura H, Takahashi M, Katagiri H, Nishida Y, Nakashima H, Yonekawa M, et al. Additional wide resection of malignant soft tissue tumors. Clin Orthop Relat Res (2002) 394:201–10.[CrossRef][Medline]
16 Ueda T, Yoshikawa H, Mori S, Araki N, Myoui A, Kuratsu S, et al. Influence of local recurrence on the prognosis of soft-tissue sarcomas. J Bone Joint Surg Br (1997) 79:553–7.[CrossRef][Medline]
17 Fiore M, Casali PG, Miceli R, Mariani L, Bertulli R, Lozza L, et al. Prognostic effect of re-excision in adult soft tissue sarcoma of the extremity. Ann Surg Oncol (2006) 13:110–7.[CrossRef][Web of Science][Medline]
18 Lewis JJ, Leung D, Espat J, Woodruff JM, Brennan MF. Effect of re-resection in extremity soft tissue sarcoma. Ann Surg (2000) 231:655–63.[CrossRef][Web of Science][Medline]
19 Manoso MW, Frassica DA, Deune EG, Frassica FJ. Outcomes of re-excision after unplanned excisions of soft-tissue sarcomas. J Surg Oncol (2005) 91:153–8.[CrossRef][Web of Science][Medline]
20 Peiper M, Zornig C, Schroder S. Re-excision of soft tissue sarcoma after inadequate initial operation. Br J Surg (1995) 82:278–9.[CrossRef][Web of Science][Medline]
CiteULike 
Connotea 
Del.icio.us What's this?
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||