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Tenosynovial giant cell tumor in the foot and ankle
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Tenosynovial giant cell tumor in the foot and ankle Hüseyin Bilgehan Çevika,* , Sibel Kayahanb , Engin Eceviza , Seyit Ali Gümüştaşa a b
_ University of Health Sciences, Kartal Dr. Lütfi Kırdar Research and Training Hospital, Department of Orthopaedics and Traumatology, Istanbul, Turkey _ University of Health Sciences, Kartal Dr. Lütfi Kırdar Research and Training Hospital, Department of Pathology, Istanbul Turkey
A R T I C L E I N F O
A B S T R A C T
Article history: Received 5 February 2019 Received in revised form 28 February 2019 Accepted 28 August 2019 Available online xxx
Background: Tenosynovial giant cell tumor (TSGCT) originates from the synovial cells of the tendon sheath and is the most common soft tissue tumor of the foot and ankle. Due to the lack of clinical data about TSGCT in the foot and ankle, this study was performed with the aim of investigating the clinical characteristics, and surgical outcomes that might predict the likelihood of recurrence. Methods: Clinical data, obtained from the pathology records and the clinic files, along with the tumor subtype, local recurrence, and patient functional status among 26 cases of TSGCT were evaluated with the mean 73 months follow-up period. Results: There were 26 patients including 16 males and 10 females with a mean age of 40 years, who underwent surgery. There were 15 localised TSGCT and 11 diffuse TSGCT. The diffuse TSGCT was more likely to be in the hindfoot dorsum (54,5%, 6/11). The localised TSGCT was mostly located in the forefoot (80%, 12/15). The recurrence rate in the diffuse TSGCT was 27,3% (3/11). In the localised TSGCT, recurrence was seen in 6,6% of patients (1/15). The mean AOFAS score was 79. Conclusion: Diffuse TSGCT is more likely to occur in the hindfoot and localised TSGCT is more common in the forefoot. Excision with clear margins is an effective treatment for TSGCT, with good oncological and clinical outcomes. But the orthopaedic surgeons should consider the equilibrium between surgical margins and the functional status of the patient. © 2019 European Foot and Ankle Society. Published by Elsevier Ltd. All rights reserved.
Keywords: Tenosynovial giant cell tumor Foot and ankle Giant cell tumor of the tendon sheath Recurrence
1. Introduction Tenosynovial giant cell tumor (TSGCT) or giant cell tumor of the tendon sheath originates from the synovial cells of the tendon sheath. TSGCT is the most common soft tissue tumor of the foot and ankle [1]. Inflammatory and neoplastic components are both involved in the etiopathogenesis of TSGCT [2,3]. The biological behavior of TSGCT can be classified as localized (L-) or diffuse (D-) TSGCT based on growth pattern. According to localization, they may be classified as intra- or extra-articular. D-TSGCT is also known as villonodular synovitis because of the typical villous pattern histologically. L-TSGCT is less aggressive, usually as a single nodular lesion, and D-TSGCT is more aggressive, and frequently recurs after excision (14–55%) [4]. The gold standard treatment of TSGCT is surgical excision with clear margins [4–6]. Several risk factors have been identified for recurrence, including diffuse type [7], incomplete excision of the lesion [8], radiological
* Corresponding author at: Kartal Dr. Lütfi Kırdar Training and Research Hospital, _ Şemsi Denizer Street, E-5 Highway, Cevizli Region, Kartal, Istanbul, Turkey. E-mail address: [email protected] (H.B. Çevik).
bone erosion, size more than 2 cm [9] and tendon or neurovascular involvement [10]. Due to the lack of clinical data about TSGCT in the foot and ankle, this study was performed with the aim of investigating the clinical characteristics, and operative outcomes from a series of cases treated at a single institution. 2. Materials and methods Cases were identified by searching through the hospital charts, the pathology records, and the clinic files of the Department of Orthopedics from 2006 to 2016. The study was performed in a single tertiary care hospital. A retrospective clinical file review was performed following Institutional Review Board approval. A total of 31 eligible patients were identified. Due to incomplete clinical data, 3 patients were excluded and a further 2 patients who did not attend the final evaluation. Thus, the study included 26 patients surgically treated at our institution with a confirmed histological diagnosis of TSGCT of the foot and ankle. The records of 26 patients identified with TSGCT, were examined in respect of preoperative variables including gender, age at the time of diagnosis, history of previous surgical procedures (primary versus recurrent), and presence and duration of
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symptoms (from onset to date of surgery). Intraoperative variables consisted of the largest dimensions of the tumor and the affected tendon. Postoperative variables included tumor subtype, local recurrence, and patient functional status at the final clinical evaluation. The patients were contacted by telephone to attend a final evaluation, when all patients were evaluated for local recurrence with MRI. The mean follow-up period was 73 months (range, 24–175 months). Pathology reports were also evaluated. Before excision, biopsy was performed on 8 patients, all of which were core-needle biopsy and were examined by a musculoskeletal pathologist. Finally all pathological specimens were re-checked by the attending musculoskeletal pathologist. The American Academy of Foot and Ankle Surgeon (AOFAS) [11] ankle–hindfoot, midfoot or metatarsophalangeal–interphalangeal scores were used to assess pain, function, and alignment according to the lesion area at the final follow-up visit. The AOFAS is an approved tool to assess outcomes after foot and ankle surgery and is scored out of 100. The average score for the normal population is 50 [11]. 3. Results The clinical data are summarized in Table 1. Over the course of 10 years, 26 patients with TSGCT of the foot and ankle were diagnosed and surgically treated at our institution. 24 cases were extra-articular TSGCT, comprising 15 with L-TSGCT and 9 with D-TSGCT (Fig. 1). There were also 2 intra-articular D-TSGCT (Fig. 1). The most common site was the ankle (over extensor hallucis longus tendon). There was no significant relationship between plantar or dorsal TSGCT and gender, age, size of tumor, and whether the tumor was L- or D- type.
The L-TSGCT group included 12 males and 3 females with a mean age of 26 years (range, 8–63 years). L-TSGCT was seen to occur most in the forefoot with (12/15) followed by the hindfoot (2/15) and midfoot (1/15). The D-TSGCT group included 4 males and 7 females with a mean age of 40 years (range, 17–62 years). D-TSGCT occurred most in the hindfoot dorsum in 6/11 (54.5%) cases, then in the midfoot dorsum in 2 cases (18.2%), the forefoot dorsum in 2 (18.2%), and in the forefoot plantar surface in 1 (9.1%). A palpable mass was the most common presenting symptom (91%) followed by pain (9%). The mean time from onset of symptoms to surgical excision was 26 months (range, 3–60) months. According to the pathology reports, the mean greatest dimension of tumors was 3.65 cm. The tendon sheath most commonly involved was the extensor hallucis longus tendon sheath at the ankle level in 5 cases. In 2 cases (Cases 11 and 15) the toe had to be amputated. Case15 was amputated at the level of the metatarsophalangeal joint and Case-11 at the level of the proximal interphalangeal joint. Neither of these cases were not candidates for reconstruction because of severe cortical erosion in the toes. In 1 case (Case-5) intra-articular D-TSGCT was determined over the medial dorsal inter-cuneiform ligament (Fig. 2). The tumor had destroyed the cortex of the medial cuneiform, so the joint was unstable after excision (Fig. 3). Arthrodesis with 2 screws was performed on the medial inter-cuneiform joint. The resection status was obtained from patient files and pathology reports. In 1 patient (case-7) in the D-TSGCT group and 1 (case-21) in the L-TSGCT group, there was seen to have been incomplete resection and the tumor recurred. The incomplete resection rate was 6.6% in the D-TSGCT group and 9% in the L-TSGCT group.
Table 1 Clinical details of the 26 patients with TSGCT of the foot and ankle. Case no.
Sex Age Size (cm)
1 2 3
F F F
61 23 29
4
F
26
5 6 7 8
M F M M
26 25 41 63
9 10 11 12 13 14 15 16 17 18 19 20 21 22
F F F F M M M F F F F F F M
43 33 47 52 11 25 62 15 8 39 35 29 8 17
23
F
28
24
F
10
25 26
F F
46 30
4 31 3,5 3 2 2,5 1,5 1,5 2,5 1,5 1,5 3 2,5 2 4 2,5 1,5 532 2,5 1,5 0,8 5,5 4 4 4 3 1,5 532 2,7 2 1,3 1 1X0,5 3 2,8 1,8 543 2 1,8 1,5 633 1,5 1 1 543 4,5 3 2 4,5 3 2 4,4 3,5 2 4,5 4 2,5 3,5 1,5 0,7 3 3X1 3 21
Preoperative duration
Preoperative investigation
Preoperative diagnosis
Subtype Follow up (months)
Recurrence [follow up after recurrence] (months)
6 months 9 months 2 years
XR, MRI XR, MRI, Bx XR, MRI
Ganglion TSGCT Hemangioma
D L L
175 161 160
No No No
1 year
XR, MRI
Ganglion
L
159
No
1 3 2 5
year years years years
XR, XR, XR, XR,
MRI MRI MRI, Bx MRI
TSGCT Schwannoma TSGCT Hemangioma
D L D L
122 118 98 98
No No Yes (60) No
1 3 2 6 3 3 5 1 4 6 5 1 2 2
year years years months months years years year years months years year years years
XR, XR, XR, XR, XR, XR, XR, XR, XR, XR, XR, XR, XR, XR,
MRI, MRI, MRI, MRI MRI MRI, MRI, MRI MRI, MRI MRI, MRI MRI, MRI,
TSGCT TSGCT None Ganglion Ganglion TSGCT TSGCT None Hemangioma TSGCT Schwannoma None TSGCT None
D D D L L L D L L L D D L D
84 77 76 53 51 51 47 40 39 38 34 33 33 30
No Yes (40) No No No No Yes (47) No No No No No Yes (24) No
Bx Bx Bx
Bx Bx Bx Bx Bx Bx
5 years
XR, MRI, Bx
None
L
29
No
3 years
XR, MRI
Schwannoma
L
28
No
1 year 6 months
XR, MRI XR, MRI, Bx
Hemangioma TSGCT
D L
44 24
No No
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surgery, although no significant lesion was determined on clinical examination or MRI. There was seen to be recurrence at the 36month follow-up examination. The patient was then treated with surgical excision and no recurrence was seen in the following 60 months until the final follow-up examination. In Case-10, recurrence was seen on follow-up MRI at 12 months after excision. This was treated with re-excision and no recurrence was seen during the following 40 months until the final evaluation. Case-15 underwent primary excision surgery at another center. One year after the initial surgery, swelling of the toe recurred, and the patient was admitted to our institution 24 months after this complaint. Cortical bone erosion and severe deformation of the toe were the indications for amputation over the metatarsophalangeal joint. In the L-TSGCT group, recurrence was seen in 1 of 15 (6.6%) patients (Case-21). The tumor was re-excised and there was no involvement of extensor tendon or the ankle capsule. The re-excision was performed with wide surgical margins and no recurrence occurred during the following 24 months until the final evaluation. 4. Discussion
Fig. 1. This drawing shows the distrubition of TSGCT of the foot and ankle, N = 26.
The mean overall AOFAS score was 79 (range, 69–91), as 77 in the D-TSGCT group and 80 in the L-TSGCT group. The results of all the patients were evaluated as very good according to the AOFAS score, including patients with recurrence after re-excision. When it is considered that the average AOFAS score of the normal population is 50, this shows the consistency of good functional outcomes. The recurrence rate in the D-TSGCT group was 27.3% (3/11). Case-7 had complaints of pain at 24 months after the initial
TSGCT, formerly known as giant cell tumor of tendon sheath and pigmented villonodular synovitis, frequently presents as a solid, slow-growing, multi-lobular, non-tender mass, located adjacent to the tendon sheath or periarticular soft tissues of small joints. It is a benign soft tissue tumor. Both L-TSGCT and D-TSGCT are the most common soft-tissue tumors of the foot and ankle [1]. However it is difficult to determine the actual incidence of foot and ankle involvement of TSGCT [12,13]. In respect of gender distribution, previous papers have reported a slight female predominance [14–16]. In the current study, the female to male ratio was 2.7:1. TSGCT of the foot and ankle can be seen at any age [17–19], and in this study, the patients with TSGCT of the foot and ankle were in the wide age range of 8–63 years (mean, 32 years). Although it has been previously reported that TSGCT occurrence is rare in children [19], 6 patients (23.1%) in the current study were aged less than 18 years. A noticeable mass is the most common presenting complaint of TSGCT, followed by pain [26]. Likewise, similar results were obtained in this study, as 92.3% of the patients had a noticeable mass as a presenting symptom. The other 2 patients (7.7%) had pain and a mass located on the plantar aspect of the third toe and third metatarsal bone. Pain may be associated with weightbearing.
Fig. 2. MRI of TSGCT over the medial dorsal inter-cuneiform ligament (Case-5). (A) T1 axial image; (B) T1 coronal image; (C) T2 sagittal image.
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Fig. 3. (A) Exposure of the mass; (B) The TSGCT had destroyed the cortex of the medial cuneiform.
In orthopaedic oncology, one of the most important clinical findings is the location of the mass. Of the 26 patients in this study, the forefoot was involved in 15, the midfoot in 8, the hindfoot in 2 and the ankle in 1. TSGCT was seen to most commonly develop in the forefoot (15/26 patients), especially on the dorsal surface (8/13 patients). This was consistent with literature, as most cases of TSGCT have been reported to be in the forefoot [13,18,20]. L-TSGCTs are commonly found around the dorsal surface of the forefoot (especially adjacent to the interphalangeal joints) [18,20]. Of the 15 forefoot tumors in the current study, 12 were L-TSGCT, 8 of which were located on the dorsal surface and 4 on the plantar surface. Of the 15 forefoot tumors, 3 were D-TSGCT, 2 of which were dorsal and 1 was on the plantar surface. D-TSGCTs are commonly found around the subtalar joint, often in association with the ankle joint [13,21,22]. In this study, most of the midfoot tumor sub-types were D-TSGCT (7/8 patients) and D-TSGCT was mostly located in the midfoot (7/11 patients). The anatomic sites of the lesions are summarized in Fig. 1. Radiographs may show cortical bone erosions, cysts or soft tissue calcifications [23,24]. Magnetic resonance imaging is the optimal modality for preoperative assessment of tumor size, extent and invasion of adjacent tissue and joint, and also provides information about recurrence [13,16]. All patients had radiographs (X-Ray) and MRI before surgical excision in this study group. Fraser et al. stated that when classic MRI findings are seen, additional diagnostic tools may not be necessary, but if MRI findings are uncertain, then additional investigation such as biopsy may be required to rule out malignant pathological processes, especially synovial sarcoma [13]. In the current series, 13 patients underwent biopsy in addition to X-ray and MRI (50%, 13/26). Eventually
the diagnosis was pathologically confirmed by the attending musculoskeletal pathologist after surgical excision of the tumor. The information about pre-excision/biopsy diagnosis of tumors was taken from preoperative patient charts. Of 26 patients, 10 had a correct diagnosis, 5 did not have a specific preoperative diagnosis but benign mass was recorded on the patient charts. The remaining 11 patients were diagnosed with benign soft tissue tumors such as hemangiomas, schwannomas, and ganglions. The 38.5% rate of correct preoperative diagnosis, based on the clinical findings and imaging studies was average in this study as diagnosis of TSGCT is known to be challenging [25]. The gold standard treatment of TSGCT is surgical excision [4–6]. However, inadequate tumor clearance may increase recurrence, and additional surgeries may increase operative morbidity. When literature is examined it can be seen that primary surgical excision of L-TSGCT in particular has resulted in low recurrence rates and good clinical outcomes [12,15]. The reported rates of local recurrence L-TSGCT in the foot and ankle have ranged from 0% to 33% after excision [13,18,20,22]. In the current study, the recurrence rate was determined as 6.7% (1/15). Palmerini et al. stated that complete resection remains the mainstay of treatment [8], but they also recommended that there should always be equilibrium between the quality of surgical margins and function preservation [8]. If more aggressive resection is applied, the outcome may negatively affect quality of life, whereas incomplete resection may lead to recurrence. Aggressive resections may be preferred in cases where recurrence has developed. Nevertheless, it should be remembered that this is a benign condition. In this study, 2 patients had recurrence because of incomplete resection. It can also be recommended that
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orthopaedic surgeons consider the equilibrium between surgical margins and the functional status of the patient. Rochwerger et al. reported cortical bone erosion and arthritic joint changes in 7 of 8 patients (88%) and these cases were managed with arthrodesis in 6 patients and amputation in 1 [29]. Nevertheless, overall good functional outcomes were reported and no recurrence at the time of final follow-up [29]. In the current series, toe amputation was necessary in 2 patients in the D-TSGCT group because of cortical bone erosion and severe soft tissue deformation. One was amputated at the level of the metatarsophalangeal joint and the other at the level of the proximal interphalangeal joint. In addition, medial cuneiform arthrodesis was performed to one patient in the current series because of joint instability. Incomplete excision, male gender, previous recurrence, tumor cellularity and mitotic activity, tumor size (>20 mm) and cortical bone erosions have been associated with recurrence [7,8,13]. The overall recurrence rate was 15.4% (4/26) in the current series. There was no statistically significant difference in recurrence between this series and the findings of previous studies. The cases with recurrence were followed up for a minimum of 24 months and no further recurrence developed (Table 1). Bruns et al. stated that the foot had the lowest recurrence rate of TSGCT in comparison with other large joints, but this has not been confirmed in large cohort studies [8,27]. Recurrence rates of foot and ankle D-TSGCT have been shown to range from 25% to 40% after open excision [22,28]. In this series, the recurrence rate of D-TSGCT was 27.3% (3/11). Sung and Ko stated that complete resection was not possible at the time of primary surgery in 1 of 4 patients with recurrence [28]. There are some previous reports in literature which have described objective clinical outcome scores for patients with foot and ankle TSGCTs [28,30,31]. The authors of those studies stated that, if the AOFAS scores were more than 55 points, the functional outcomes may be good and excellent. From the results of the current study, the mean AOFAS score of 79 after TSGCT excision can be considered relatively favorably. There were several limitations to this study, primarily the retrospective design and the low number of patients. Retrospective analyses are limited to the data obtained from hospital patient charts, which may differ from the actual characteristics of the studied disease. However, the small sample size inhibits interpretation of specific outcomes in foot and ankle TSGCTs. Although TSGCT is the most common benign tumor of the foot and ankle, they are uncommon tumors that represent a treatment challenge for orthopaedic surgeons. L-TSGCT is more likely to occur in the forefoot and D-TSGCT is more common in the hindfoot. Careful operative intervention still remains the primary treatment for both D- and L- TSGCTs, and this is effective treatment, with good oncological and functional outcomes. Although clinical significance is an unclear issue, negative surgical margins can be considered to decrease the likelihood of recurrence. Declarations of interest None. References [1] Chou LB, Ho YY, Malawer MM. Tumors of the foot and ankle: experience with 153 cases. Foot Ankle Int 2009;30:836. [2] West RB, Rubin BP, Miller MA, Subramanian S, Kaygusuz G, Montgomery K, et al. A landscape effect in tenosynovial giant-cell tumor from activation of CSF1 expression by a translocation in a minority of tumor cells. Proceedings of the National Academy of Sciences 2006;103:. p. 690.
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[3] Cupp JS, Miller MA, Montgomery KD, Nielsen TO, O’connell JX, Huntsman D, et al. Translocation and expression of CSF1 in pigmented villonodular synovitis, tenosynovial giant cell tumor, rheumatoid arthritis and other reactive synovitides. Am J Surg Pathol 2007;31:970. [4] Verspoor FG, Van Der Geest IC, Vegt E, Veth RP, Van Der Graaf WT, Schreuder HB. Pigmented villonodular synovitis: current concepts about diagnosis and management. Future Oncol 2013;9:1515. [5] Ehrenstein V, Andersen SL, Qazi I, Sankar N, Pedersen AB, Sikorski R, et al. Tenosynovial giant cell tumor: incidence, prevalence, patient characteristics, and recurrence. A registry-based cohort study in Denmark. J Rheumatol 2017;160816. [6] Mastboom MJ, Verspoor FG, Verschoor AJ, Uittenbogaard D, Nemeth B, Mastboom WJ, et al. Higher incidence rates than previously known in tenosynovial giant cell tumors: a nationwide study in the Netherlands. Acta Orthop 2017;88:688. [7] Ottaviani S, Ayral X, Dougados M, Gossec L. 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JBJS 2004;86:902. [12] Monaghan H, Salter D, Al-Nafussi A. Giant cell tumour of tendon sheath (localised nodular tenosynovitis): clinicopathological features of 71 cases. J Clin Pathol 2001;54:404. [13] Fraser EJ, Sullivan M, Maclean F, Nesbitt A. Tenosynovial giant-cell tumors of the foot and ankle: a critical analysis review. JBJS Rev 2017;5:. [14] Suresh S, Zaki H. Giant cell tumor of tendon sheath: case series and review of literature. J Hand Microsurg 2010;2:67. [15] Adams EL, Yoder EM, Kasdan ML. Giant cell tumor of the tendon sheath: experience with 65 cases. Eplasty 2012;12:. [16] Wang C, Song RR, Kuang PD, Wang LH, Zhang MM. Giant cell tumor of the tendon sheath: magnetic resonance imaging findings in 38 patients. Oncol Lett 2017;13:4459. [17] Darwish F, Haddad W. Giant cell tumour of tendon sheath: experience with 52 cases. Singapore Med J 2008;49:879. [18] Zhang Y, Huang J, Ma X, Wang X, Zhang C, Chen L. 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Giant cell tumor of tendon sheath: spectrum of radiologic findings. Skeletal Radiol 1992;21:219. ski A. Diagnostic difficulties in pigmented villonodular [25] Panasiuk M, Iwan synovitis. Presentation of cases. Ortop Traumatol Rehabil 2008;10:613. [26] NdSA Somerhausen, Fletcher CD. Diffuse-type giant cell tumor: clinicopathologic and immunohistochemical analysis of 50 cases with extraarticular disease. Am J Surg Pathol 2000;24:479. [27] Bruns J, Ewerbeck V, Dominkus M, Windhager R, Hassenpflug J, Windhagen H, et al. Pigmented villo-nodular synovitis and giant-cell tumor of tendon sheaths: a binational retrospective study. Arch Orthop Trauma Surg 2013;133:1047. [28] Sung K-S, Ko KR. Surgical outcomes after excision of pigmented villonodular synovitis localized to the ankle and hindfoot without adjuvant therapy. J Foot Ankle Surg 2015;54:160. [29] Rochwerger A, Groulier P, Curvale G, Launay F. Pigmented villonodular synovitis of the foot and ankle: a report of eight cases. 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Tenosynovial giant cell tumor in the foot and ankle Hüseyin Bilgehan Çevika,* , Sibel Kayahanb , Engin Eceviza , Seyit Ali Gümüştaşa a b
_ University of Health Sciences, Kartal Dr. Lütfi Kırdar Research and Training Hospital, Department of Orthopaedics and Traumatology, Istanbul, Turkey _ University of Health Sciences, Kartal Dr. Lütfi Kırdar Research and Training Hospital, Department of Pathology, Istanbul Turkey
A R T I C L E I N F O
A B S T R A C T
Article history: Received 5 February 2019 Received in revised form 28 February 2019 Accepted 28 August 2019 Available online xxx
Background: Tenosynovial giant cell tumor (TSGCT) originates from the synovial cells of the tendon sheath and is the most common soft tissue tumor of the foot and ankle. Due to the lack of clinical data about TSGCT in the foot and ankle, this study was performed with the aim of investigating the clinical characteristics, and surgical outcomes that might predict the likelihood of recurrence. Methods: Clinical data, obtained from the pathology records and the clinic files, along with the tumor subtype, local recurrence, and patient functional status among 26 cases of TSGCT were evaluated with the mean 73 months follow-up period. Results: There were 26 patients including 16 males and 10 females with a mean age of 40 years, who underwent surgery. There were 15 localised TSGCT and 11 diffuse TSGCT. The diffuse TSGCT was more likely to be in the hindfoot dorsum (54,5%, 6/11). The localised TSGCT was mostly located in the forefoot (80%, 12/15). The recurrence rate in the diffuse TSGCT was 27,3% (3/11). In the localised TSGCT, recurrence was seen in 6,6% of patients (1/15). The mean AOFAS score was 79. Conclusion: Diffuse TSGCT is more likely to occur in the hindfoot and localised TSGCT is more common in the forefoot. Excision with clear margins is an effective treatment for TSGCT, with good oncological and clinical outcomes. But the orthopaedic surgeons should consider the equilibrium between surgical margins and the functional status of the patient. © 2019 European Foot and Ankle Society. Published by Elsevier Ltd. All rights reserved.
Keywords: Tenosynovial giant cell tumor Foot and ankle Giant cell tumor of the tendon sheath Recurrence
1. Introduction Tenosynovial giant cell tumor (TSGCT) or giant cell tumor of the tendon sheath originates from the synovial cells of the tendon sheath. TSGCT is the most common soft tissue tumor of the foot and ankle [1]. Inflammatory and neoplastic components are both involved in the etiopathogenesis of TSGCT [2,3]. The biological behavior of TSGCT can be classified as localized (L-) or diffuse (D-) TSGCT based on growth pattern. According to localization, they may be classified as intra- or extra-articular. D-TSGCT is also known as villonodular synovitis because of the typical villous pattern histologically. L-TSGCT is less aggressive, usually as a single nodular lesion, and D-TSGCT is more aggressive, and frequently recurs after excision (14–55%) [4]. The gold standard treatment of TSGCT is surgical excision with clear margins [4–6]. Several risk factors have been identified for recurrence, including diffuse type [7], incomplete excision of the lesion [8], radiological
* Corresponding author at: Kartal Dr. Lütfi Kırdar Training and Research Hospital, _ Şemsi Denizer Street, E-5 Highway, Cevizli Region, Kartal, Istanbul, Turkey. E-mail address: [email protected] (H.B. Çevik).
bone erosion, size more than 2 cm [9] and tendon or neurovascular involvement [10]. Due to the lack of clinical data about TSGCT in the foot and ankle, this study was performed with the aim of investigating the clinical characteristics, and operative outcomes from a series of cases treated at a single institution. 2. Materials and methods Cases were identified by searching through the hospital charts, the pathology records, and the clinic files of the Department of Orthopedics from 2006 to 2016. The study was performed in a single tertiary care hospital. A retrospective clinical file review was performed following Institutional Review Board approval. A total of 31 eligible patients were identified. Due to incomplete clinical data, 3 patients were excluded and a further 2 patients who did not attend the final evaluation. Thus, the study included 26 patients surgically treated at our institution with a confirmed histological diagnosis of TSGCT of the foot and ankle. The records of 26 patients identified with TSGCT, were examined in respect of preoperative variables including gender, age at the time of diagnosis, history of previous surgical procedures (primary versus recurrent), and presence and duration of
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symptoms (from onset to date of surgery). Intraoperative variables consisted of the largest dimensions of the tumor and the affected tendon. Postoperative variables included tumor subtype, local recurrence, and patient functional status at the final clinical evaluation. The patients were contacted by telephone to attend a final evaluation, when all patients were evaluated for local recurrence with MRI. The mean follow-up period was 73 months (range, 24–175 months). Pathology reports were also evaluated. Before excision, biopsy was performed on 8 patients, all of which were core-needle biopsy and were examined by a musculoskeletal pathologist. Finally all pathological specimens were re-checked by the attending musculoskeletal pathologist. The American Academy of Foot and Ankle Surgeon (AOFAS) [11] ankle–hindfoot, midfoot or metatarsophalangeal–interphalangeal scores were used to assess pain, function, and alignment according to the lesion area at the final follow-up visit. The AOFAS is an approved tool to assess outcomes after foot and ankle surgery and is scored out of 100. The average score for the normal population is 50 [11]. 3. Results The clinical data are summarized in Table 1. Over the course of 10 years, 26 patients with TSGCT of the foot and ankle were diagnosed and surgically treated at our institution. 24 cases were extra-articular TSGCT, comprising 15 with L-TSGCT and 9 with D-TSGCT (Fig. 1). There were also 2 intra-articular D-TSGCT (Fig. 1). The most common site was the ankle (over extensor hallucis longus tendon). There was no significant relationship between plantar or dorsal TSGCT and gender, age, size of tumor, and whether the tumor was L- or D- type.
The L-TSGCT group included 12 males and 3 females with a mean age of 26 years (range, 8–63 years). L-TSGCT was seen to occur most in the forefoot with (12/15) followed by the hindfoot (2/15) and midfoot (1/15). The D-TSGCT group included 4 males and 7 females with a mean age of 40 years (range, 17–62 years). D-TSGCT occurred most in the hindfoot dorsum in 6/11 (54.5%) cases, then in the midfoot dorsum in 2 cases (18.2%), the forefoot dorsum in 2 (18.2%), and in the forefoot plantar surface in 1 (9.1%). A palpable mass was the most common presenting symptom (91%) followed by pain (9%). The mean time from onset of symptoms to surgical excision was 26 months (range, 3–60) months. According to the pathology reports, the mean greatest dimension of tumors was 3.65 cm. The tendon sheath most commonly involved was the extensor hallucis longus tendon sheath at the ankle level in 5 cases. In 2 cases (Cases 11 and 15) the toe had to be amputated. Case15 was amputated at the level of the metatarsophalangeal joint and Case-11 at the level of the proximal interphalangeal joint. Neither of these cases were not candidates for reconstruction because of severe cortical erosion in the toes. In 1 case (Case-5) intra-articular D-TSGCT was determined over the medial dorsal inter-cuneiform ligament (Fig. 2). The tumor had destroyed the cortex of the medial cuneiform, so the joint was unstable after excision (Fig. 3). Arthrodesis with 2 screws was performed on the medial inter-cuneiform joint. The resection status was obtained from patient files and pathology reports. In 1 patient (case-7) in the D-TSGCT group and 1 (case-21) in the L-TSGCT group, there was seen to have been incomplete resection and the tumor recurred. The incomplete resection rate was 6.6% in the D-TSGCT group and 9% in the L-TSGCT group.
Table 1 Clinical details of the 26 patients with TSGCT of the foot and ankle. Case no.
Sex Age Size (cm)
1 2 3
F F F
61 23 29
4
F
26
5 6 7 8
M F M M
26 25 41 63
9 10 11 12 13 14 15 16 17 18 19 20 21 22
F F F F M M M F F F F F F M
43 33 47 52 11 25 62 15 8 39 35 29 8 17
23
F
28
24
F
10
25 26
F F
46 30
4 31 3,5 3 2 2,5 1,5 1,5 2,5 1,5 1,5 3 2,5 2 4 2,5 1,5 532 2,5 1,5 0,8 5,5 4 4 4 3 1,5 532 2,7 2 1,3 1 1X0,5 3 2,8 1,8 543 2 1,8 1,5 633 1,5 1 1 543 4,5 3 2 4,5 3 2 4,4 3,5 2 4,5 4 2,5 3,5 1,5 0,7 3 3X1 3 21
Preoperative duration
Preoperative investigation
Preoperative diagnosis
Subtype Follow up (months)
Recurrence [follow up after recurrence] (months)
6 months 9 months 2 years
XR, MRI XR, MRI, Bx XR, MRI
Ganglion TSGCT Hemangioma
D L L
175 161 160
No No No
1 year
XR, MRI
Ganglion
L
159
No
1 3 2 5
year years years years
XR, XR, XR, XR,
MRI MRI MRI, Bx MRI
TSGCT Schwannoma TSGCT Hemangioma
D L D L
122 118 98 98
No No Yes (60) No
1 3 2 6 3 3 5 1 4 6 5 1 2 2
year years years months months years years year years months years year years years
XR, XR, XR, XR, XR, XR, XR, XR, XR, XR, XR, XR, XR, XR,
MRI, MRI, MRI, MRI MRI MRI, MRI, MRI MRI, MRI MRI, MRI MRI, MRI,
TSGCT TSGCT None Ganglion Ganglion TSGCT TSGCT None Hemangioma TSGCT Schwannoma None TSGCT None
D D D L L L D L L L D D L D
84 77 76 53 51 51 47 40 39 38 34 33 33 30
No Yes (40) No No No No Yes (47) No No No No No Yes (24) No
Bx Bx Bx
Bx Bx Bx Bx Bx Bx
5 years
XR, MRI, Bx
None
L
29
No
3 years
XR, MRI
Schwannoma
L
28
No
1 year 6 months
XR, MRI XR, MRI, Bx
Hemangioma TSGCT
D L
44 24
No No
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surgery, although no significant lesion was determined on clinical examination or MRI. There was seen to be recurrence at the 36month follow-up examination. The patient was then treated with surgical excision and no recurrence was seen in the following 60 months until the final follow-up examination. In Case-10, recurrence was seen on follow-up MRI at 12 months after excision. This was treated with re-excision and no recurrence was seen during the following 40 months until the final evaluation. Case-15 underwent primary excision surgery at another center. One year after the initial surgery, swelling of the toe recurred, and the patient was admitted to our institution 24 months after this complaint. Cortical bone erosion and severe deformation of the toe were the indications for amputation over the metatarsophalangeal joint. In the L-TSGCT group, recurrence was seen in 1 of 15 (6.6%) patients (Case-21). The tumor was re-excised and there was no involvement of extensor tendon or the ankle capsule. The re-excision was performed with wide surgical margins and no recurrence occurred during the following 24 months until the final evaluation. 4. Discussion
Fig. 1. This drawing shows the distrubition of TSGCT of the foot and ankle, N = 26.
The mean overall AOFAS score was 79 (range, 69–91), as 77 in the D-TSGCT group and 80 in the L-TSGCT group. The results of all the patients were evaluated as very good according to the AOFAS score, including patients with recurrence after re-excision. When it is considered that the average AOFAS score of the normal population is 50, this shows the consistency of good functional outcomes. The recurrence rate in the D-TSGCT group was 27.3% (3/11). Case-7 had complaints of pain at 24 months after the initial
TSGCT, formerly known as giant cell tumor of tendon sheath and pigmented villonodular synovitis, frequently presents as a solid, slow-growing, multi-lobular, non-tender mass, located adjacent to the tendon sheath or periarticular soft tissues of small joints. It is a benign soft tissue tumor. Both L-TSGCT and D-TSGCT are the most common soft-tissue tumors of the foot and ankle [1]. However it is difficult to determine the actual incidence of foot and ankle involvement of TSGCT [12,13]. In respect of gender distribution, previous papers have reported a slight female predominance [14–16]. In the current study, the female to male ratio was 2.7:1. TSGCT of the foot and ankle can be seen at any age [17–19], and in this study, the patients with TSGCT of the foot and ankle were in the wide age range of 8–63 years (mean, 32 years). Although it has been previously reported that TSGCT occurrence is rare in children [19], 6 patients (23.1%) in the current study were aged less than 18 years. A noticeable mass is the most common presenting complaint of TSGCT, followed by pain [26]. Likewise, similar results were obtained in this study, as 92.3% of the patients had a noticeable mass as a presenting symptom. The other 2 patients (7.7%) had pain and a mass located on the plantar aspect of the third toe and third metatarsal bone. Pain may be associated with weightbearing.
Fig. 2. MRI of TSGCT over the medial dorsal inter-cuneiform ligament (Case-5). (A) T1 axial image; (B) T1 coronal image; (C) T2 sagittal image.
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Fig. 3. (A) Exposure of the mass; (B) The TSGCT had destroyed the cortex of the medial cuneiform.
In orthopaedic oncology, one of the most important clinical findings is the location of the mass. Of the 26 patients in this study, the forefoot was involved in 15, the midfoot in 8, the hindfoot in 2 and the ankle in 1. TSGCT was seen to most commonly develop in the forefoot (15/26 patients), especially on the dorsal surface (8/13 patients). This was consistent with literature, as most cases of TSGCT have been reported to be in the forefoot [13,18,20]. L-TSGCTs are commonly found around the dorsal surface of the forefoot (especially adjacent to the interphalangeal joints) [18,20]. Of the 15 forefoot tumors in the current study, 12 were L-TSGCT, 8 of which were located on the dorsal surface and 4 on the plantar surface. Of the 15 forefoot tumors, 3 were D-TSGCT, 2 of which were dorsal and 1 was on the plantar surface. D-TSGCTs are commonly found around the subtalar joint, often in association with the ankle joint [13,21,22]. In this study, most of the midfoot tumor sub-types were D-TSGCT (7/8 patients) and D-TSGCT was mostly located in the midfoot (7/11 patients). The anatomic sites of the lesions are summarized in Fig. 1. Radiographs may show cortical bone erosions, cysts or soft tissue calcifications [23,24]. Magnetic resonance imaging is the optimal modality for preoperative assessment of tumor size, extent and invasion of adjacent tissue and joint, and also provides information about recurrence [13,16]. All patients had radiographs (X-Ray) and MRI before surgical excision in this study group. Fraser et al. stated that when classic MRI findings are seen, additional diagnostic tools may not be necessary, but if MRI findings are uncertain, then additional investigation such as biopsy may be required to rule out malignant pathological processes, especially synovial sarcoma [13]. In the current series, 13 patients underwent biopsy in addition to X-ray and MRI (50%, 13/26). Eventually
the diagnosis was pathologically confirmed by the attending musculoskeletal pathologist after surgical excision of the tumor. The information about pre-excision/biopsy diagnosis of tumors was taken from preoperative patient charts. Of 26 patients, 10 had a correct diagnosis, 5 did not have a specific preoperative diagnosis but benign mass was recorded on the patient charts. The remaining 11 patients were diagnosed with benign soft tissue tumors such as hemangiomas, schwannomas, and ganglions. The 38.5% rate of correct preoperative diagnosis, based on the clinical findings and imaging studies was average in this study as diagnosis of TSGCT is known to be challenging [25]. The gold standard treatment of TSGCT is surgical excision [4–6]. However, inadequate tumor clearance may increase recurrence, and additional surgeries may increase operative morbidity. When literature is examined it can be seen that primary surgical excision of L-TSGCT in particular has resulted in low recurrence rates and good clinical outcomes [12,15]. The reported rates of local recurrence L-TSGCT in the foot and ankle have ranged from 0% to 33% after excision [13,18,20,22]. In the current study, the recurrence rate was determined as 6.7% (1/15). Palmerini et al. stated that complete resection remains the mainstay of treatment [8], but they also recommended that there should always be equilibrium between the quality of surgical margins and function preservation [8]. If more aggressive resection is applied, the outcome may negatively affect quality of life, whereas incomplete resection may lead to recurrence. Aggressive resections may be preferred in cases where recurrence has developed. Nevertheless, it should be remembered that this is a benign condition. In this study, 2 patients had recurrence because of incomplete resection. It can also be recommended that
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orthopaedic surgeons consider the equilibrium between surgical margins and the functional status of the patient. Rochwerger et al. reported cortical bone erosion and arthritic joint changes in 7 of 8 patients (88%) and these cases were managed with arthrodesis in 6 patients and amputation in 1 [29]. Nevertheless, overall good functional outcomes were reported and no recurrence at the time of final follow-up [29]. In the current series, toe amputation was necessary in 2 patients in the D-TSGCT group because of cortical bone erosion and severe soft tissue deformation. One was amputated at the level of the metatarsophalangeal joint and the other at the level of the proximal interphalangeal joint. In addition, medial cuneiform arthrodesis was performed to one patient in the current series because of joint instability. Incomplete excision, male gender, previous recurrence, tumor cellularity and mitotic activity, tumor size (>20 mm) and cortical bone erosions have been associated with recurrence [7,8,13]. The overall recurrence rate was 15.4% (4/26) in the current series. There was no statistically significant difference in recurrence between this series and the findings of previous studies. The cases with recurrence were followed up for a minimum of 24 months and no further recurrence developed (Table 1). Bruns et al. stated that the foot had the lowest recurrence rate of TSGCT in comparison with other large joints, but this has not been confirmed in large cohort studies [8,27]. Recurrence rates of foot and ankle D-TSGCT have been shown to range from 25% to 40% after open excision [22,28]. In this series, the recurrence rate of D-TSGCT was 27.3% (3/11). Sung and Ko stated that complete resection was not possible at the time of primary surgery in 1 of 4 patients with recurrence [28]. There are some previous reports in literature which have described objective clinical outcome scores for patients with foot and ankle TSGCTs [28,30,31]. The authors of those studies stated that, if the AOFAS scores were more than 55 points, the functional outcomes may be good and excellent. From the results of the current study, the mean AOFAS score of 79 after TSGCT excision can be considered relatively favorably. There were several limitations to this study, primarily the retrospective design and the low number of patients. Retrospective analyses are limited to the data obtained from hospital patient charts, which may differ from the actual characteristics of the studied disease. However, the small sample size inhibits interpretation of specific outcomes in foot and ankle TSGCTs. Although TSGCT is the most common benign tumor of the foot and ankle, they are uncommon tumors that represent a treatment challenge for orthopaedic surgeons. L-TSGCT is more likely to occur in the forefoot and D-TSGCT is more common in the hindfoot. Careful operative intervention still remains the primary treatment for both D- and L- TSGCTs, and this is effective treatment, with good oncological and functional outcomes. Although clinical significance is an unclear issue, negative surgical margins can be considered to decrease the likelihood of recurrence. Declarations of interest None. References [1] Chou LB, Ho YY, Malawer MM. Tumors of the foot and ankle: experience with 153 cases. Foot Ankle Int 2009;30:836. [2] West RB, Rubin BP, Miller MA, Subramanian S, Kaygusuz G, Montgomery K, et al. A landscape effect in tenosynovial giant-cell tumor from activation of CSF1 expression by a translocation in a minority of tumor cells. Proceedings of the National Academy of Sciences 2006;103:. p. 690.
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