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Local recurrence and assessment of sentinel lymph node biopsy in deep soft tissue leiomyosarcoma of the extremities

Local recurrence and assessment of sentinel lymph node biopsy in deep soft tissue leiomyosarcoma... Background: Leiomyosarcoma of deep soft tissues of the extremities is a rare malignant tumour treated primarily by surgery. The incidence of local recurrence and lymph node metastasis is uncertain and it is not known whether a sentinel lymph node biopsy is indicated in these tumours. Methods: A retrospective review of patients treated for extremity deep soft tissue leiomyosarcoma at our institution over a 10-year period was conducted. Patients developing local recurrence or lymph node metastasis were identified. The presence or absence of lymphatics in the primary tumours was assessed by immunohistochemical expression of LYVE-1 and podoplanin. Results: 27 patients (mean age 62 years) were included in the study. 15 were female and 12 male. Lymph node metastasis was seen in only two cases (7%); intratumoural lymphatics were identified in the primary tumours of both these cases. Local recurrence occurred in 25.9% of cases despite complete excision and post-operative radiotherapy; the mean time to recurrence was 10.1 months. Conclusion: On the basis of this study, we do not advocate sentinel lymph node biopsy in this group of patients except in those cases in which intratumoural lymphatics can be demonstrated. Close follow up is important especially for high grade leiomyosarcomas, particularly in the first year, as these tumours have a high incidence of local recurrence. Introduction sarcomas, particularly epithelial sarcoma [7-9]. The inci- Leiomyosarcoma of soft tissues is a malignant tumour dence of lymph node metastasis in extremity leiomyo- composed of tumour cells that exhibit smooth muscle sarcomas is clearly important with regard to whether differentiation. Leiomyosarcomas are generally thought SLNB should be carried out for this tumour. In previous to account for 5-10% of soft tissue sarcomas [1-3]. retrospective reviews of the literature, pooling data from These tumours arise most commonly in the retroperito- published reports on regional lymph node involvement, neum but can develop in any location; in one study of Weingrad and Rosenberg [10] and Mazeron and Suit 75 soft tissue leiomyosarcomas, 33% were noted to arise [11] found the incidence in leiomyosarcoma was 10.6% in extremity soft tissues\The behaviour of leiomyosar- and 4% respectively; in the prospective study of Fong et coma of extremity deep soft tissues has not been studied al [5], the incidence was reported to be 2.7%. These stu- dies, however, did not distinguish leiomyosarcoma of independently of those arising in other locations. Regional lymph node metastasis in patients with soft extremity deep soft tissues from those arising in other tissue sarcomas is an infrequent event occurring in 2.6 - locations; this is an important factor as leiomyosarcoma 5% of all patients [4-6]. Sentinel lymph node biopsy occurs more commonly in the retroperitoneum, mesen- (SLNB) has been employed for staging of soft tissue tery, abdominal and pelvic viscera than in extremity soft tissues and lymph node metastasis from sarcomas of * Correspondence: nick.athanasou@ndorms.ox.ac.uk visceral origin occurs less commonly than from sarco- Nuffield Department of Orthopaedics, Rheumatology and Musculoskeltal, mas arising in extremity soft tissues [5]. The recurrence Sciences, University of Oxford, Department of Pathology, Nuffield rate following excision of deep soft tissue extremity Orthopaedic Centre, Oxford, OX3 7LD, UK © 2011 Lamyman et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Lamyman et al. Clinical Sarcoma Research 2011, 1:7 Page 2 of 5 http://www.clinicalsarcomaresearch.com/content/1/1/7 leiomyosarcomas is also unknown; this has not been complete clearance with as wide a margin as possible. assessed independently of recurrence of superficial 21 of the patients (78%) received adjuvant radiotherapy (cutaneous) leiomyosarcomas, which have a favourable following primary excision. Details of patients develop- prognosis, or of retroperitoneal tumours, which have a ing local recurrence are shown in Table 2 and of those poor prognosis. developing lymph node metastasis in Table 3. Lymph Theaim of this studyhas been to determinethe node metastasis occurred in two patients (7%). recurrence rate and incidence of lymph node metastasis A review of the pathology of the primary tumour in of deep soft tissue leiomyosarcomas of the extremities. these two cases showed that both tumours contained intratumoural lymphatics, as assessed by endothelial cell As the presence of lymphatic vessels has been noted in malignant soft tissue tumours that metastasise to lymph expression of the lymphatic markers, podoplanin and nodes [12], we determined whether immunohistochem- LYVE-1. (Figure 3) The remaining tumours, which did ical identification of lymphatics in the primary tumour not metastasise to lymph nodes, were negative for lym- could provide a guide as to whether lymph node metas- phatic markers. In one patient the nodal recurrence was tasis of extremity leiomyosarcoma occurred and thus, extensive, encasing femoral vessels and was not resect- whether a SLNB might be indicated in such cases. able. In the second patient an inguinal and iliac lymph node dissection was performed. In this patient the Patients and methods lymph node metastasis occurred early, before radiother- A search of the pathology database detected all patients apy was instituted. with a histological diagnosis of deep soft tissue leiomyo- Local recurrence occurred in seven patients (25.9%). sarcoma over a 10 year period, between 1998 and 2008. The mean time from surgical excision to recurrence was Only patients diagnosed and treated at the Nuffield 10.1 months (range 3-24 months). There was no inci- Orthopaedic Centre with leiomyosarcoma of the extre- dence of local recurrence or lymph node metastasis in mities were entered into the study. Patients with superfi- patients with low grade leiomyosarcoma. Post-operative cial cutaneous soft tissue leiomyosarcomas or radiotherapy was received by all patients who subse- gynaecological, retroperitoneal, intra-abdominal or quently presented with local recurrence. In six of these intrathoracic primary tumours were excluded. A case seven patients, the tumour had been excised with a notes review was performed. clear margin. In one patient the excision was described Local recurrence and lymph node metastasis was con- as marginal. In all but one case the recurrence was trea- sidered to have occurred only if proven though open ted by further surgical resection. biopsy. The histological diagnosis of leiomyosarcoma was based on morphological and immunohistochemical Discussion criteria detailed in the WHO classification of soft tissue The role of SLNB in the management of soft tissue sar- tumours [1]. Immunohistochemical expression of at coma has yet to be defined [8,9,13]. In our institution it least two smooth muscle antigens (smooth muscle actin, is current practice to undertake SLNB in patients with desmin, h-caldesmon) was seen in all cases. Identifica- epithelioid sarcoma given the relatively high rate of tion of lymphatics was carried out using anti-Lyve -1 lymph node metastasis in these tumours. Previous stu- and anti-podoplanin antibodies as previously described dies have reported that the incidence of lymph node [12]. metastasis in such tumours is between 16.7 and 80% [5,10,11]. A positive SLNB in these cases is followed by Results a formal lymph node dissection. A number of soft tissue 35 patients were identified as eligible for entry into the sarcomas, such as rhabdomyosarcoma, clear cell sar- study. Five patients had to be excluded as either the coma and synovial sarcoma, have also been shown to case notes could not be found or were incomplete. Two have a propensity for regional lymph node metastasis patients were excluded because they died following their and some observers have suggested that SLNB may be biopsy but before definitive surgery, and one patient was of prognostic benefit in these tumours [9]. Previous esti- excluded because metastatic disease was found on pre- mates of the incidence of lymph node metastasis in all sentation. The case notes of the remaining 27 patients patients with leiomyosarcoma have been between 2.7 [510H] were reviewed. Fifteen were female and twelve male and 10.6%. These studies examined the metastatic (Figure 1). The mean age at presentation was 62 years. rate of leiomyosarcomas arising at several different sites The mean follow up was 19.9 months, median 15 collectively and not just that of leiomyosarcomas of months (range 4 to 59 months). The sites of the primary deep soft tissues of the extremities. In the present study tumour are shown in Figure 2. The size, grade and stage we found that the rate of lymph node metastasis in of the tumours are shown in Table 1. In all cases, local extremity deep soft tissue leiomyosarcomas to be 7%. excision of the tumours was performed aiming for Lamyman et al. Clinical Sarcoma Research 2011, 1:7 Page 3 of 5 http://www.clinicalsarcomaresearch.com/content/1/1/7 soft tissue leiomyosarcomas would not appear to justify the extra morbidity (eg extra operating time, potential wound problems) associated with undertaking SLNB. Recent work at our institution has shown that soft tis- sue sarcomas with a high propensity to metastasise to lymph nodes contain intratumoural lymphatics [12]; intratumoural lymphatics were found to be present in all epithelioid sarcomas and a number of other sarcomas including leiomyosarcoma. Lymph node metastasis has been reported in up to 80% of epithelioid sarcomas [5,10,11]. The lower incidence of lymph node metastasis in leiomyosarcomas may reflect the fact that intratu- moural lymphatics are found less commonly in these tumours. It is none the less significant that in our study the two leiomyoarcomas which did metastasise to regio- nal lymph nodes both contained intratumoural lympha- Figure 1 Age and sex distribution of cases. tics. Immunohistochemical demonstration of lymphatic vessels in these primary leiomysarcomas was of prognos- tic significance with regard to the development of lymph In patients with intermediate thickness melanoma, node metastasis, and it could be argued that SLNB is SLNB has become widely accepted as a minimally inva- indicated in primary leiomyosarcomas of the extremities sive method of staging the regional lymph nodes [14,15]. where intratumoural lymphatics are identified. When SLNB is performed in these patients, 20% will be We found a high rate of local recurrence in extremity found to have micrometastasis. However when SLNB is deep soft tissue leiomyosarcoma patients with 25.9% performed in thin melanomas, with a Breslow thickness experiencing recurrence despite adequate resection and less than I mm, the micrometastasis rate falls to 5% adjuvant radiotherapy. Mankin and Hornicek report a [16]. Current AJCC guidelines do not recommend rou- recurrence rate of 10.8% in 65 patients with leiomyosar- tine use of SLNB in this group [17,18], and on this basis coma [19]. Again this study did not differentiate the comparable rate of lymph node metastasis in deep between leiomyosarcoma of the extremities and other Figure 2 Sites of primary tumour with number and approximate percentage of cases. Lamyman et al. Clinical Sarcoma Research 2011, 1:7 Page 4 of 5 http://www.clinicalsarcomaresearch.com/content/1/1/7 Table 1 Size, Grade and Stage of the primary leiomyosarcoma Case Size in maximum Diameter (cm) Grade MSTS Stage 118 2 2b 28 3 2b 35 3 2b 4 Unknown 2 2b 5 7.5 1 lb 6 4.5 2 lb 710 3 2b S7 3 2b 94 2 2b 10 8 3 2b 11 13 3 2b 12 14 3 2b 13 2 2 2b 14 3.8 1 lb 15 12 2 2b 16 25 2 2b 17 12 2 2b 18 3.5 2 2b 19 8.5 3 2b 20 10 3 2b 21 16 3 2b 22 9 1 lb Figure 3 Intratumoural lymphatic vessels in a primary 23 13 1 lb leiomyosarcoma, showing podoplanin expression by lymphatic endothelial cells. 24 9 3 3 25 Unknown 2 2b 26 10 3 2b 27 8 3 2b MSTS - Musculoskeletal Tumour Society Table 2 Patients with local recurrence following primary excision Age Sex Site Max Diam eter (cm) Grade MSTS Time to first Number of recurrences Margins Adjuvant Radio therapy Stage recurrenc e (Months) 55 F Arm 5 3 2b 3 2 Clear Yes 77 F Calf Unknown 2 2b 24 2 Clear Yes 77 M Buttock 8 3 2b 3 4 Clear Yes 67 F Thigh 13 3 2b 9 2 Clear Yes 67 M Buttock 12 2 2b 4 2 Clear Yes 55 F Calf 9 3 2b 5 1 Marginal Yes 76 M Forearm unknown 2 2b 19 1 Clear Yes Table 3 Patients with lymph node metastasis Age Sex Site Max Diameter Grade MSTS Stage Margins Aduvant Radiotherapy Time to detection of lymph node metastasis (cm) (months) 79 F Thigh 8.5 3 2b Clear Yes 21 53 M Thigh 8 3 2b Clear No 3 Lamyman et al. Clinical Sarcoma Research 2011, 1:7 Page 5 of 5 http://www.clinicalsarcomaresearch.com/content/1/1/7 13. Blazer DG, Sabel MS, Sondak VK: Is there a role for sentinel lymph node sites. The findings of the present study indicate that biopsy in the management of sarcoma? Surg Oncol 2003, 12:201-206. deep soft tissue leiomyosarcoma of the extremities, in 14. Morton DL, Cochran AJ, Thompson JF: The rational for sentinel-node contrast to leiomyosarcoma arising at other sites has a biopsy in melanoma. Nat Clin Pract Oncol 2008, 5:510-511. 15. McMasters KM, Reintgen DS, Ross MI, et al: Sentinel lymph node biopsy greater propensity to local recurrence. Such recurrences for melanoma: Controversy despite widespread agreement. J Clin Oncol are difficult to treat and surgical resection of an already 2001, 19:2851-2855. irradiated area remains the only option. 16. Puleo CA, Messina JL, Riker Al, et al: Sentinel node biopsy for thin melanomas: Which patients should be considered. Cancer Control 2005, 12:230-235. Conclusion 17. Balch CM, Soong SJ, Gershenwald JE, et al: Prognostic factors analysis of This study has shown that patients with leiomyosarcoma 17,6000 melanoma patients: validation of the American Joint Committee on Cancer melanoma staging system. J Clin Oncol 2001, 19:3622-3634. of deep soft tissues of the extremities have a rate of 18. Balch CM, Buzzaid AC, Atkins MB, et al: A new American Joint Committee lymph node metastasis of 7% and a local recurrence rate on Cancer staging system for cutaneous melanoma. Cancer 2000, of 25.9% despite adequate excision and post-operative 88:1484-1491. 19. Mankin HJ, Hornicek FJ: Diagnosis, classification, and management of soft radiotherapy. On the basis of this study, we do not tissue sarcoma. Cancer Control 2005, 12:5-21. advocate the use of SLNB to this group of patients except in cases where lymphatics can be demonstrated doi:10.1186/2045-3329-1-7 Cite this article as: Lamyman et al.: Local recurrence and assessment of in the primary tumour. Our findings emphasise the sentinel lymph node biopsy in deep soft tissue leiomyosarcoma of the importance of close follow up, especially for high grade extremities. Clinical Sarcoma Research 2011 1:7. leiomyosarcomas, particularly in the first year post sur- gery, as there is a high incidence of local recurrence. Authors’ contributions HG, PC, MG and DW contributed to the design of the study. HG, MG and PC conducted the study. NA carried out pathological studies and MJL, HPG, MG and NA wrote the paper. All authors have read and approved the final manuscript. Competing interests The authors declare that they have no competing interests. Received: 11 February 2011 Accepted: 1 August 2011 Published: 1 August 2011 References 1. Evans HL, Shipley J: Leiomyosarcoma. In Pathology and genetics of tumours of soft tissue and bone. Edited by: Fletcher DCM, Unni KK, Mertens F. Lyon, IARC Press; 2002:131-134. 2. Weiss SW, Goldblum JR: Leiomyosarcoma. Enzinger and Weiss’s Soft Tissue Tumours. 5 edition. St. Louis Mosby; 2008, 545-564. 3. Hashimoto H, Daimaru Y, Tsuneyoshi M, Enjoji M: Leiomyosarcoma of the external soft tissue. Cancer 1986, 57:2077-2088. 4. Sondak VK, Chang AE: Clinical evaluation and treatment of soft tissue sarcomas. In Enzinger and Weiss’s soft tissue tumours.. Fourth edition. Edited by: Weiss SW, Goldblum JR. St Louis Mosby; 2001:21-44. 5. Fong Y, Coit DG, Woodruff JM, et al: Lymph node metastasis from soft tissue sarcoma in Adults: Analysis of data from a prospective database of 1772 sarcoma patients. Ann Surg 1993, 217:72-77. 6. Behranwala KA, A’Hern R, Al-Muderis O, et al: Prognosis of lymph node metastasis in soft tissue sarcoma. Ann Surgical Oncol 2004, 11:714-719. 7. Gow KW, Rapkin LB, Olson TA, et al: Sentinel lymph node biopsy in the pediatric population. J Pediatr Surg 2008, 43:2193-8. 8. Kayton ML, Delgado R, busam K, et al: Experience with 31 sentinel lymph Submit your next manuscript to BioMed Central node biopsies for sarcomas and carcinomas in pediatric patients. Cancer and take full advantage of: 2008, 1;112:2052-9. 9. Maduekwe UN, Hornicek FJ, Springfield DS, et al: Role of sentinel lymph • Convenient online submission node biopsy in the staging of synovial, epithelioid, and clear cell sarcomas. Ann Surg Oncol 2009, 16:1356063. • Thorough peer review 10. 10 DN, Weingrad DN, Rosenberg SA: Early lymphatic spread of osteogenic • No space constraints or color figure charges and soft-tissue sarcomas. Surgery 1978, 84:231-240. • Immediate publication on acceptance 11. Mazeron JJ, Suit HD: Lymph nodes as sites of metastasis from sarcomas of soft tissue. Cancer 1987, 60:1800-1808. • Inclusion in PubMed, CAS, Scopus and Google Scholar 12. Mahendra G, Kliskey K, et al: Intratumoural lymphatics in benign and • Research which is freely available for redistribution malignant soft tissue tumours. Virchows Archiv Pathol Anat 2008, 453:457-464. Submit your manuscript at www.biomedcentral.com/submit http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Clinical Sarcoma Research Springer Journals

Local recurrence and assessment of sentinel lymph node biopsy in deep soft tissue leiomyosarcoma of the extremities

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Springer Journals
Copyright
Copyright © 2011 by Lamyman et al; licensee BioMed Central Ltd.
Subject
Biomedicine; Cancer Research; Oncology; Surgical Oncology
eISSN
2045-3329
DOI
10.1186/2045-3329-1-7
pmid
22612847
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Abstract

Background: Leiomyosarcoma of deep soft tissues of the extremities is a rare malignant tumour treated primarily by surgery. The incidence of local recurrence and lymph node metastasis is uncertain and it is not known whether a sentinel lymph node biopsy is indicated in these tumours. Methods: A retrospective review of patients treated for extremity deep soft tissue leiomyosarcoma at our institution over a 10-year period was conducted. Patients developing local recurrence or lymph node metastasis were identified. The presence or absence of lymphatics in the primary tumours was assessed by immunohistochemical expression of LYVE-1 and podoplanin. Results: 27 patients (mean age 62 years) were included in the study. 15 were female and 12 male. Lymph node metastasis was seen in only two cases (7%); intratumoural lymphatics were identified in the primary tumours of both these cases. Local recurrence occurred in 25.9% of cases despite complete excision and post-operative radiotherapy; the mean time to recurrence was 10.1 months. Conclusion: On the basis of this study, we do not advocate sentinel lymph node biopsy in this group of patients except in those cases in which intratumoural lymphatics can be demonstrated. Close follow up is important especially for high grade leiomyosarcomas, particularly in the first year, as these tumours have a high incidence of local recurrence. Introduction sarcomas, particularly epithelial sarcoma [7-9]. The inci- Leiomyosarcoma of soft tissues is a malignant tumour dence of lymph node metastasis in extremity leiomyo- composed of tumour cells that exhibit smooth muscle sarcomas is clearly important with regard to whether differentiation. Leiomyosarcomas are generally thought SLNB should be carried out for this tumour. In previous to account for 5-10% of soft tissue sarcomas [1-3]. retrospective reviews of the literature, pooling data from These tumours arise most commonly in the retroperito- published reports on regional lymph node involvement, neum but can develop in any location; in one study of Weingrad and Rosenberg [10] and Mazeron and Suit 75 soft tissue leiomyosarcomas, 33% were noted to arise [11] found the incidence in leiomyosarcoma was 10.6% in extremity soft tissues\The behaviour of leiomyosar- and 4% respectively; in the prospective study of Fong et coma of extremity deep soft tissues has not been studied al [5], the incidence was reported to be 2.7%. These stu- dies, however, did not distinguish leiomyosarcoma of independently of those arising in other locations. Regional lymph node metastasis in patients with soft extremity deep soft tissues from those arising in other tissue sarcomas is an infrequent event occurring in 2.6 - locations; this is an important factor as leiomyosarcoma 5% of all patients [4-6]. Sentinel lymph node biopsy occurs more commonly in the retroperitoneum, mesen- (SLNB) has been employed for staging of soft tissue tery, abdominal and pelvic viscera than in extremity soft tissues and lymph node metastasis from sarcomas of * Correspondence: nick.athanasou@ndorms.ox.ac.uk visceral origin occurs less commonly than from sarco- Nuffield Department of Orthopaedics, Rheumatology and Musculoskeltal, mas arising in extremity soft tissues [5]. The recurrence Sciences, University of Oxford, Department of Pathology, Nuffield rate following excision of deep soft tissue extremity Orthopaedic Centre, Oxford, OX3 7LD, UK © 2011 Lamyman et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Lamyman et al. Clinical Sarcoma Research 2011, 1:7 Page 2 of 5 http://www.clinicalsarcomaresearch.com/content/1/1/7 leiomyosarcomas is also unknown; this has not been complete clearance with as wide a margin as possible. assessed independently of recurrence of superficial 21 of the patients (78%) received adjuvant radiotherapy (cutaneous) leiomyosarcomas, which have a favourable following primary excision. Details of patients develop- prognosis, or of retroperitoneal tumours, which have a ing local recurrence are shown in Table 2 and of those poor prognosis. developing lymph node metastasis in Table 3. Lymph Theaim of this studyhas been to determinethe node metastasis occurred in two patients (7%). recurrence rate and incidence of lymph node metastasis A review of the pathology of the primary tumour in of deep soft tissue leiomyosarcomas of the extremities. these two cases showed that both tumours contained intratumoural lymphatics, as assessed by endothelial cell As the presence of lymphatic vessels has been noted in malignant soft tissue tumours that metastasise to lymph expression of the lymphatic markers, podoplanin and nodes [12], we determined whether immunohistochem- LYVE-1. (Figure 3) The remaining tumours, which did ical identification of lymphatics in the primary tumour not metastasise to lymph nodes, were negative for lym- could provide a guide as to whether lymph node metas- phatic markers. In one patient the nodal recurrence was tasis of extremity leiomyosarcoma occurred and thus, extensive, encasing femoral vessels and was not resect- whether a SLNB might be indicated in such cases. able. In the second patient an inguinal and iliac lymph node dissection was performed. In this patient the Patients and methods lymph node metastasis occurred early, before radiother- A search of the pathology database detected all patients apy was instituted. with a histological diagnosis of deep soft tissue leiomyo- Local recurrence occurred in seven patients (25.9%). sarcoma over a 10 year period, between 1998 and 2008. The mean time from surgical excision to recurrence was Only patients diagnosed and treated at the Nuffield 10.1 months (range 3-24 months). There was no inci- Orthopaedic Centre with leiomyosarcoma of the extre- dence of local recurrence or lymph node metastasis in mities were entered into the study. Patients with superfi- patients with low grade leiomyosarcoma. Post-operative cial cutaneous soft tissue leiomyosarcomas or radiotherapy was received by all patients who subse- gynaecological, retroperitoneal, intra-abdominal or quently presented with local recurrence. In six of these intrathoracic primary tumours were excluded. A case seven patients, the tumour had been excised with a notes review was performed. clear margin. In one patient the excision was described Local recurrence and lymph node metastasis was con- as marginal. In all but one case the recurrence was trea- sidered to have occurred only if proven though open ted by further surgical resection. biopsy. The histological diagnosis of leiomyosarcoma was based on morphological and immunohistochemical Discussion criteria detailed in the WHO classification of soft tissue The role of SLNB in the management of soft tissue sar- tumours [1]. Immunohistochemical expression of at coma has yet to be defined [8,9,13]. In our institution it least two smooth muscle antigens (smooth muscle actin, is current practice to undertake SLNB in patients with desmin, h-caldesmon) was seen in all cases. Identifica- epithelioid sarcoma given the relatively high rate of tion of lymphatics was carried out using anti-Lyve -1 lymph node metastasis in these tumours. Previous stu- and anti-podoplanin antibodies as previously described dies have reported that the incidence of lymph node [12]. metastasis in such tumours is between 16.7 and 80% [5,10,11]. A positive SLNB in these cases is followed by Results a formal lymph node dissection. A number of soft tissue 35 patients were identified as eligible for entry into the sarcomas, such as rhabdomyosarcoma, clear cell sar- study. Five patients had to be excluded as either the coma and synovial sarcoma, have also been shown to case notes could not be found or were incomplete. Two have a propensity for regional lymph node metastasis patients were excluded because they died following their and some observers have suggested that SLNB may be biopsy but before definitive surgery, and one patient was of prognostic benefit in these tumours [9]. Previous esti- excluded because metastatic disease was found on pre- mates of the incidence of lymph node metastasis in all sentation. The case notes of the remaining 27 patients patients with leiomyosarcoma have been between 2.7 [510H] were reviewed. Fifteen were female and twelve male and 10.6%. These studies examined the metastatic (Figure 1). The mean age at presentation was 62 years. rate of leiomyosarcomas arising at several different sites The mean follow up was 19.9 months, median 15 collectively and not just that of leiomyosarcomas of months (range 4 to 59 months). The sites of the primary deep soft tissues of the extremities. In the present study tumour are shown in Figure 2. The size, grade and stage we found that the rate of lymph node metastasis in of the tumours are shown in Table 1. In all cases, local extremity deep soft tissue leiomyosarcomas to be 7%. excision of the tumours was performed aiming for Lamyman et al. Clinical Sarcoma Research 2011, 1:7 Page 3 of 5 http://www.clinicalsarcomaresearch.com/content/1/1/7 soft tissue leiomyosarcomas would not appear to justify the extra morbidity (eg extra operating time, potential wound problems) associated with undertaking SLNB. Recent work at our institution has shown that soft tis- sue sarcomas with a high propensity to metastasise to lymph nodes contain intratumoural lymphatics [12]; intratumoural lymphatics were found to be present in all epithelioid sarcomas and a number of other sarcomas including leiomyosarcoma. Lymph node metastasis has been reported in up to 80% of epithelioid sarcomas [5,10,11]. The lower incidence of lymph node metastasis in leiomyosarcomas may reflect the fact that intratu- moural lymphatics are found less commonly in these tumours. It is none the less significant that in our study the two leiomyoarcomas which did metastasise to regio- nal lymph nodes both contained intratumoural lympha- Figure 1 Age and sex distribution of cases. tics. Immunohistochemical demonstration of lymphatic vessels in these primary leiomysarcomas was of prognos- tic significance with regard to the development of lymph In patients with intermediate thickness melanoma, node metastasis, and it could be argued that SLNB is SLNB has become widely accepted as a minimally inva- indicated in primary leiomyosarcomas of the extremities sive method of staging the regional lymph nodes [14,15]. where intratumoural lymphatics are identified. When SLNB is performed in these patients, 20% will be We found a high rate of local recurrence in extremity found to have micrometastasis. However when SLNB is deep soft tissue leiomyosarcoma patients with 25.9% performed in thin melanomas, with a Breslow thickness experiencing recurrence despite adequate resection and less than I mm, the micrometastasis rate falls to 5% adjuvant radiotherapy. Mankin and Hornicek report a [16]. Current AJCC guidelines do not recommend rou- recurrence rate of 10.8% in 65 patients with leiomyosar- tine use of SLNB in this group [17,18], and on this basis coma [19]. Again this study did not differentiate the comparable rate of lymph node metastasis in deep between leiomyosarcoma of the extremities and other Figure 2 Sites of primary tumour with number and approximate percentage of cases. Lamyman et al. Clinical Sarcoma Research 2011, 1:7 Page 4 of 5 http://www.clinicalsarcomaresearch.com/content/1/1/7 Table 1 Size, Grade and Stage of the primary leiomyosarcoma Case Size in maximum Diameter (cm) Grade MSTS Stage 118 2 2b 28 3 2b 35 3 2b 4 Unknown 2 2b 5 7.5 1 lb 6 4.5 2 lb 710 3 2b S7 3 2b 94 2 2b 10 8 3 2b 11 13 3 2b 12 14 3 2b 13 2 2 2b 14 3.8 1 lb 15 12 2 2b 16 25 2 2b 17 12 2 2b 18 3.5 2 2b 19 8.5 3 2b 20 10 3 2b 21 16 3 2b 22 9 1 lb Figure 3 Intratumoural lymphatic vessels in a primary 23 13 1 lb leiomyosarcoma, showing podoplanin expression by lymphatic endothelial cells. 24 9 3 3 25 Unknown 2 2b 26 10 3 2b 27 8 3 2b MSTS - Musculoskeletal Tumour Society Table 2 Patients with local recurrence following primary excision Age Sex Site Max Diam eter (cm) Grade MSTS Time to first Number of recurrences Margins Adjuvant Radio therapy Stage recurrenc e (Months) 55 F Arm 5 3 2b 3 2 Clear Yes 77 F Calf Unknown 2 2b 24 2 Clear Yes 77 M Buttock 8 3 2b 3 4 Clear Yes 67 F Thigh 13 3 2b 9 2 Clear Yes 67 M Buttock 12 2 2b 4 2 Clear Yes 55 F Calf 9 3 2b 5 1 Marginal Yes 76 M Forearm unknown 2 2b 19 1 Clear Yes Table 3 Patients with lymph node metastasis Age Sex Site Max Diameter Grade MSTS Stage Margins Aduvant Radiotherapy Time to detection of lymph node metastasis (cm) (months) 79 F Thigh 8.5 3 2b Clear Yes 21 53 M Thigh 8 3 2b Clear No 3 Lamyman et al. Clinical Sarcoma Research 2011, 1:7 Page 5 of 5 http://www.clinicalsarcomaresearch.com/content/1/1/7 13. Blazer DG, Sabel MS, Sondak VK: Is there a role for sentinel lymph node sites. The findings of the present study indicate that biopsy in the management of sarcoma? Surg Oncol 2003, 12:201-206. deep soft tissue leiomyosarcoma of the extremities, in 14. Morton DL, Cochran AJ, Thompson JF: The rational for sentinel-node contrast to leiomyosarcoma arising at other sites has a biopsy in melanoma. Nat Clin Pract Oncol 2008, 5:510-511. 15. McMasters KM, Reintgen DS, Ross MI, et al: Sentinel lymph node biopsy greater propensity to local recurrence. Such recurrences for melanoma: Controversy despite widespread agreement. J Clin Oncol are difficult to treat and surgical resection of an already 2001, 19:2851-2855. irradiated area remains the only option. 16. Puleo CA, Messina JL, Riker Al, et al: Sentinel node biopsy for thin melanomas: Which patients should be considered. Cancer Control 2005, 12:230-235. Conclusion 17. Balch CM, Soong SJ, Gershenwald JE, et al: Prognostic factors analysis of This study has shown that patients with leiomyosarcoma 17,6000 melanoma patients: validation of the American Joint Committee on Cancer melanoma staging system. J Clin Oncol 2001, 19:3622-3634. of deep soft tissues of the extremities have a rate of 18. Balch CM, Buzzaid AC, Atkins MB, et al: A new American Joint Committee lymph node metastasis of 7% and a local recurrence rate on Cancer staging system for cutaneous melanoma. Cancer 2000, of 25.9% despite adequate excision and post-operative 88:1484-1491. 19. Mankin HJ, Hornicek FJ: Diagnosis, classification, and management of soft radiotherapy. On the basis of this study, we do not tissue sarcoma. Cancer Control 2005, 12:5-21. advocate the use of SLNB to this group of patients except in cases where lymphatics can be demonstrated doi:10.1186/2045-3329-1-7 Cite this article as: Lamyman et al.: Local recurrence and assessment of in the primary tumour. Our findings emphasise the sentinel lymph node biopsy in deep soft tissue leiomyosarcoma of the importance of close follow up, especially for high grade extremities. Clinical Sarcoma Research 2011 1:7. leiomyosarcomas, particularly in the first year post sur- gery, as there is a high incidence of local recurrence. Authors’ contributions HG, PC, MG and DW contributed to the design of the study. HG, MG and PC conducted the study. NA carried out pathological studies and MJL, HPG, MG and NA wrote the paper. All authors have read and approved the final manuscript. Competing interests The authors declare that they have no competing interests. Received: 11 February 2011 Accepted: 1 August 2011 Published: 1 August 2011 References 1. 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Clinical Sarcoma ResearchSpringer Journals

Published: Aug 1, 2011

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