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Sentinel node biopsy as an adjunct to limb salvage surgery for epithelioid sarcoma of the hand Seal, Alex; Tse, Raymond; Wehrli, Bret; Hammond, Alex; Temple, Claire L Jun 29, 2005

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ralWorld Journal of Surgical OncologyssBioMed CentOpen AcceCase reportSentinel node biopsy as an adjunct to limb salvage surgery for epithelioid sarcoma of the handAlex Seal1, Raymond Tse2, Bret Wehrli3, Alex Hammond4 and Claire LF Temple*2Address: 1University of British Columbia, Vancouver, British Columbia, Canada, 2Division of Plastic Surgery, University of Western Ontario, London, Ontario, Canada, 3Department of Pathology, University of Western Ontario, London, Ontario, Canada and 4Department of Radiation Oncology, London Region Cancer Centre, London, Ontario, CanadaEmail: Alex Seal - aseal@interchange.ubc.ca; Raymond Tse - ray.tse@utoronto.ca; Bret Wehrli - Bret.Wehrli@lhsc.on.ca; Alex Hammond - alex.hammond@lrcc.on.ca; Claire LF Temple* - ctemple4@uwo.ca* Corresponding author    AbstractBackground: Epithelioid sarcomas of the hand are rare, high-grade tumors with a propensity forregional lymphatic spread approaching 40%.Case presentation: A 54-year-old male with an epithelioid sarcoma of the palm was treated withneoadjuvant radiation, wide excision, and two-stage reconstruction. Sentinel lymph node biopsywas used to stage the patient's axilla. Sentinel node biopsy results were negative. The patient hasremained free of local, regional and distant disease for the follow-up time of 16 months.Conclusion: The rarity of this tumor makes definitive conclusions difficult but SLN biopsy appearsto be a useful adjunct in the treatment of these sarcomas.BackgroundEpithelioid sarcoma is a rare, high-grade, soft tissue sar-coma. These tumors typically present on the extremities,in males who are 20 to 30 years of age. Overall 5 and 10-year survival rates are 70% and 42% respectively [1]. Epi-thelioid sarcoma is among a group of sarcomas with apropensity for regional lymphatic spread, with lymphnode metastasis rates reported between 17%-80% [2-7].Due to the risk of regional spread, sentinel lymph nodebiopsy (SLN) may be useful in the management of thistumor.The success of SLN biopsy is based on the principle thatsentinel nodes has been shown to reflect the histology ofthe entire lymphatic basin [8,9]. This approach is cur-rently the least invasive and most accurate nodal stagingprocedure for breast cancer and melanoma [10,11]. How-ever, SLN biopsy has not been thoroughly investigated forsarcoma.We report a case of SLN biopsy in conjunction with limbsalvage surgery and complex soft tissue, neurovascular,and staged tendon reconstruction for the management ofan epithelioid sarcoma of the hand.Case presentationPublished: 29 June 2005World Journal of Surgical Oncology 2005, 3:41 doi:10.1186/1477-7819-3-41Received: 22 April 2005Accepted: 29 June 2005This article is available from: http://www.wjso.com/content/3/1/41© 2005 Seal 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.Page 1 of 8(page number not for citation purposes)the primary tumor drains to one or a few lymph nodes inthe regional basin. Histopathological analysis of theseAn otherwise healthy 54-year-old right-handed laborerpresented with an eight-year history of a slowly enlargingWorld Journal of Surgical Oncology 2005, 3:41 http://www.wjso.com/content/3/1/41"callus" in the palm of his right hand. He underwent exci-sion at an outside institution. The operative note sug-gested that the tumor had the appearance of a sebaceouscyst with a large amount of surrounding tissue reaction.The neurovascular bundles were identified and preservedto the 4th web space. An epithelioid sarcoma was identi-fied upon histological analysis. Review of surgical pathol-ogy at our institution confirmed this diagnosis (Figure 1aand 1b). The excision was incomplete with tumor extend-ing to several margins.On examination, the patient had a transverse scar justproximal to the 4th web space, with no palpable tumor.Neurovascular exam was normal, with normal range ofmotion of the associated digits. There were several small,palpable ipsilateral axillary nodes.MRI of the right hand showed an ill-defined signal changewithin the palmar subcutaneous fat just deep to the surgi-cal incision and distal to the 4th and 5th metacarpal-phalangeal joints, consistent with post-surgical changesand scarring. No discrete soft tissue mass was seen to sug-gest gross tumor. Edema signal was seen in the distalaspect of the lumbrical muscle between the ring and smallflexor tendons. The interosseous muscles appeared unin-volved. No osseous or articular abnormalities werePreoperative computerized tomography (CT) of the chestwas negative. The axilla was reported as having benignappearing lymph nodes, fatty in nature with no evidenceof necrosis.The patient received preoperative radiation of 50 Gy in 25fractions. CT simulation was used for planning the grosstumor volume (GTV) to ascertain the depth to be treatedusing electrons. A customized lead cutout was designed toavoid treating the full width of the hand and to avoid nor-mal tissues ulnarly, radially and at depth. Bolus wasplaced over the palm to ensure adequate superficial skinand scar dose whilst ensuring the dose at depth coveredthe tumor and previous operative bed. A daily dose of 2Gy was delivered to a total of 50 Gy over a five weekperiod. Moderate erythema of the palm occurred whichhealed well post-treatment. There was no significantedema post-irradiation. Dysethesias were reported on theradial aspect of the small finger; however, two point dis-crimination remained normal.Surgery was performed 6 weeks following completion ofradiotherapy. Preoperative lymphoscintography (figure2) identified a single "hot" ipsilateral axillary lymphnode, which was successfully removed. Vital blue dye wasnot used, as it was felt that the blue stained tissues wouldEpithelioid sarcoma a) Conglomerate of tumor nodules with central necrosis mimicking a necrotizing granulomatous process at low magnificati n (hematoxylin and sin ×4) b) Epithelioid lls with abundant eosi ophilic cy oplasm and prominent nucle ratypia, appreciated at high magnificati n (hematoxylin and eosi  ×40), distinguishes epithe oid sarco a from its benign mimicsFigure 1Epithelioid sarcoma a) Conglomerate of tumor nodules with central necrosis mimicking a necrotizing granulomatous process at low magnification (hematoxylin and eosin ×4) b) Epithelioid cells with abundant eosinophilic cytoplasm and prominent nuclear atypia, appreciated at high magnification (hematoxylin and eosin ×40), distinguishes epithelioid sarcoma from its benign mimics. Note the presence of a mitotic figure.Page 2 of 8(page number not for citation purposes)identified. worsen visibility for the wide excision.World Journal of Surgical Oncology 2005, 3:41 http://www.wjso.com/content/3/1/41Wide, en bloc excision was carried out including palmarskin, subcutaneous fat, palmar fascia, flexor digitorumsuperficialis and profundus tendons (including their asso-ciated lumbricals) to the small and ring finger, and theneurovascular bundles to the 3rd and 4th web space. Theexcision plane was carried down to the level of the fasciaof the interossei, along the volar shafts of the metacarpals(Figure 3).Reconstruction included the placement of silicone rods tothe small and ring fingers for the first part of a 2-stageflexor tendon reconstruction (Figure 4). Sural nerve graftswere placed to the small, ring and long finger. The smalland ring fingers were revascularized from the superficialpalmar arch with a Y-shaped vein graft harvested from thevolar forearm.The wound was then covered with a contralateral freeradial forearm flap anastomosed to the radial artery andvena comitantes. The flap was innervated by neuror-rhaphy of the lateral antebrachial cutaneous nerve in theflap to the palmar cutaneous branch of the median nerve.A small skin graft was used to cover the proximal pedicleto avoid compression.Surgical pathology of the en bloc excision was negative forresidual malignancy. Metastatic tumor was not identifiedwithin the sentinel lymph node following examination ofmultiple tissue levels of the node using both standardhematoxylin-eosin staining and immunohistochemicalstaining with antibodies against multiple cytokeratins andCD34, immunomarkers (frequently positive in epithe-lioid sarcoma).Preoperative lymphoscintigraphy identifies uptake of the radiolabelled tracer in a single axillary lymph nodeFigur  2Preoperative lymphoscintigraphy identifies uptake of the radiolabelled tracer in a single axillary lymph node.Page 3 of 8(page number not for citation purposes)World Journal of Surgical Oncology 2005, 3:41 http://www.wjso.com/content/3/1/41The appearance of the hand is shown following wide excision of skin, palmar fascia, flexor tendons, lumbricals, and neurovascu-lar bundlesFigure 3The appearance of the hand is shown following wide excision of skin, palmar fascia, flexor tendons, lumbricals, and neurovascu-lar bundles. The small and ring fingers are postured in extension due to the absence of flexor tendons. The resection specimen is shown above.Page 4 of 8(page number not for citation purposes)World Journal of Surgical Oncology 2005, 3:41 http://www.wjso.com/content/3/1/41At six months post-procedure, the radial forearm flap anddonor site were well healed (Figure 5). The second-stagetendon reconstruction was undertaken by exchanging thesilicone rods with extensor digitorum longus grafts fromhis 3rd and 4th toes for restoration of active finger flexion.At 16 months post treatment, the patient remains free oflocal, regional and distant disease. He has regainedacceptable hand function, with small and ring finger indi-vidual joint range of motion of 90 degrees at the metacar-pal-phalangeal joints, 30 degrees at the proximalinterphalyngeal joints, and 30 degrees at the distal inter-phalangeal joints (Figure 6). His moving two-point dis-DiscussionSoft tissue sarcomas generally have a low incidence ofregional lymph node metastasis (3–10%) [2] and regionallymph node recurrence (4–10%) [12,13]. Standard treat-ment includes wide local excision with pre-or postopera-tive radiotherapy. Limb salvage surgery providesacceptable local control comparable to amputation, withno difference in survival. [13-20] Multivariate analysis hasdemonstrated that the presence of metastasis at presenta-tion is the single most important risk factor for local recur-rence. [4] This likely reflects the more aggressive biologicpotential of tumors that metastasize early and are morelikely to fail local treatment.Sural nerve grafts, reversed Y-vein graft for digital revascularization of the small and ring fingers, and first-stage tendon recon-struction with silicone od insert on is seen hereFigure 4Sural nerve grafts, reversed Y-vein graft for digital revascularization of the small and ring fingers, and first-stage tendon recon-struction with silicone rod insertion is seen here. The construct was then covered with a contralateral free radial forearm flap.Page 5 of 8(page number not for citation purposes)crimination ranges from 5 to 7 mm.World Journal of Surgical Oncology 2005, 3:41 http://www.wjso.com/content/3/1/41Treatment of sarcomas of the hand is particularly chal-lenging due to the concentrated and intricate anatomy,which makes sparing of critical structures difficult. Fur-thermore, the majority of these tumors are extra-compart-mental, violating multiple tissue planes. Microsurgicalskill for complex vascular and neural repair is an integralpart of the overall planning of these cases, since withoutsophisticated reconstruction, limb salvage for hand sarco-mas is unlikely to be useful. Free flaps are commonlyrequired to restore function as well as to facilitate primaryhealing. [14-18]The propensity of epithelioid sarcoma for regional spreadsupports the role of minimally invasive regional nodestaging procedures for prognosis and treatment. SLNbiopsy has dramatically changed the management ofmelanoma and breast cancer. It has been investigated inthe mapping of other tumors including penile [21], lung[22], colon [23,24], upper GI tumors [25], gynecologiccancer [26,27], thyroid cancer [28,29], and squamous cellcarcinoma of the head and neck [7,30,31].Given the success of the technique in other malignancies,it seems reasonable to apply SLN biopsy to soft tissue sar-comas of the extremity [8]. The role of SLN biopsy has notbeen extensively investigated in the treatment of sarcoma.In fact, there is only a single published report on its use ina child with rhabdomyosarcoma [9]. With the improvedsurvival advantage of radical lymphadenectomy for clini-important. Furthermore, patients with a negative sentinellymph node biopsy for micrometastasis would be sparedthe morbidity of formal lymphadenectomy.Identification of the sentinel node in sarcomas is morechallenging than in breast and melanoma patients.Although successful identification of sentinel nodesexceeds 95% when using both a vital blue dye and anuclear tracer [32,33], we avoided blue dye because itstain tissues and obscure planes. For resection in the hand,it is paramount to maintain precise visibility and a dye-free and bloodless field. Furthermore, when neoadjuvantradiotherapy is used, radiation-associated scarring of lym-phatics could alter the accuracy of lymphatic mapping.Therefore, despite SLN biopsy, close follow-up of regionalnodal basins is required. This includes assessment withclinical examination as well as with imaging such as high-resolution ultrasound. Our particular patient requiredserial chest computed-tomography scans for follow-up ofunrelated, non-specific lung nodules. This provided a con-current, detailed, and serial assessment of the benignappearance of his operated axillary bed.ConclusionWe report a case of epithelioid sarcoma of the hand suc-cessfully managed with a multi-disciplinary approachincluding neoadjuvant radiation, sentinel node biopsyand wide surgical excision. The rarity of this tumor makesdefinitive conclusions difficult but SLN biopsy appears tobe a useful adjunct in the treatment of these sarcomas.Competing interestsIn the past five years we have not received reimburse-ments, fees, funding, or salary from an organization thatmay in any way gain or lose financially from the publica-tion of this manuscript, either now or in the future. Nosuch an organization financed this manuscript (includingthe article-processing charge). We do not hold any stocks or shares in an organizationthat may in any way gain or lose financially from the pub-lication of this manuscript, either now or in the future. We do not hold nor are applying for any patents relatingto the content of the manuscript. We have not receivedreimbursements, fees, funding, or salary from an organi-zation that holds or has applied for patents relating to thecontent of the manuscript. We have no other financial competing interests.We do not have any non-financial competing interests(political, personal, religious, academic, intellectual, com-Prior to second-stage tendon grafting, the patient had a well-healed radial forearm flap on his right palmFigure 5Prior to second-stage tendon grafting, the patient had a well-healed radial forearm flap on his right palm. The donor site on the left forearm was satisfactory.Page 6 of 8(page number not for citation purposes)cally evident lymph node metastases from sarcoma, [4]accurate early detection of micrometastases may bemercial or any other) to declare in relation to thismanuscript.World Journal of Surgical Oncology 2005, 3:41 http://www.wjso.com/content/3/1/41Authors' contributionsCT: Primary surgeon; report conception, writing, prepara-tion and revision of manuscript, response to reviewers'questions, submission of manuscript, photographsRT: First assistant surgeon; literature review, chart review,data collection, writing and preparation of manuscriptAS: Second assistant surgeon; literature review, chartreview, data collection, writing and preparation ofmanuscriptAH: Radiation oncologist; chart review, writing and prep-BW: Pathologist; writing, preparation and revision ofmanuscript, histologic slide review, photographsAcknowledgementsPatient consent was obtained for publication of this case report.References1. 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