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Hand Surgery, Vol. 20, No. 1 (2015) 191–195 © World Scientific Publishing Company DOI: 10.1142/S0218810415300028

CURRENT MANAGEMENT OF HAND ENCHONDROMA: A REVIEW Chris Tang, Marcus Chan, Margaret Fok and Boris Fung Department of Orthopaedics and Traumatology Queen Mary Hospital, Hong Kong Received 1 August 2014; Revised 24 August 2014; Accepted 25 August 2014; Published 20 January 2015 ABSTRACT Enchondromas are benign bone tumours originating from cartilages. It is mainly discovered incidentally in radiographs or due to symptoms like pathological fracture or pain. Conservative treatment through regular check-up and surgical excision using curettage are the two major treatment methods for enchondromas. This review concludes that small localized asymptomatic lesions can be treated conservatively while most expanding or symptomatic lesions should be treated with simple curettage. Adjuvant treatments like high-speed burring or alcohol instillation are not recommended. Keywords: Enchondroma; Treatment; Hand Surgery.

\outcome", \treatment" and \recurrence". The inclusion criteria were (1) publication in English and (2) papers concerning the treatment of hand enchondroma. Recurrence rate, complications were reviewed. References in review papers were screened for potentially relevant studies not yet identified.

INTRODUCTION Enchondroma is the most common hand tumour (47.1% of all hand tumours).1 It involves a persistent cartilaginous island that arised from physis.2 Common symptoms of presentation include pain, swelling, deformity and pathological fracture at the site of tumour. The proximal phalanges (48.9%) and the little fingers (34.3%) were the most common sites involved.3 Malignant transformation of enchondromas to chondrosarcoma is rare.2 Treatment methods include conservative regular followup or surgical excision by means of curettage. Currently, no standard protocol for treating enchondromas of hand has been established. This review aims at giving readers a holistic view on the treatment strategies of hand enchondroma and suggesting a treatment approach under the support of current literatures.

RESULTS Contemporary treatment of hand enchondroma involve observation, curettage alone and curettage with augmentation (bone graft or cement injection).

Observation A number of cases of enchondroma discovered incidentally are actually asymptomatic while other patients may observe deformity without experiencing any pain. Meanwhile, invasive surgical treatment of curettage and bone graft may result in prolonged immobilization period. The formation of adhesions together with immobilization may also result in certain degree

MATERIALS AND METHODS Electronic databases of PubMed and Google Scholar was searched with the keywords \hand enchondroma", \curettage",

Correspondence to: Dr. Chris Tang, 5/F, Professorial Block, Queen Mary Hospital, 102 Pokfulam Road, Hong Kong. E-mail: [email protected] 191

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of loss in motion. In a retrospective review of 73 enchondroma patients, Müller et al.4 also suggested that regular radiological follow-up of asymptomatic enchondromas is a better option due to their low malignant potential (solitary enchondromas, 2–3%) and the high complications rate of surgical treatment (23%, include upper limb, lower limb and pelvis). For such reasons, conservative treatment with regular check-up should be considered in small localized asymptomatic lesions. The relatively more invasive surgical procedures also mean that conservative treatment may be a better option for elderly patients and patients with poor immune function.

strength of the bone. The most common method is to harvest autogenous bone graft from the iliac crest or other sources. An alternative method is to use allografts from suitable donors. Bauer et al. compared the surgical outcomes of 12 enchondroma patients treated using allografts with 16 patients treated using autografts. The bone grafts incorporated and remodeled in all patients and no recurrence and complications are found in patients treated with allografts.7 Jewusiak et al. also obtained satisfactory outcome in 23 patients treated with freeze-dried allografts. Bone healing was achieved without any recurrence.8

Surgical Treatment: Curettage

Curettage with Augmentation by Cement Injection

The excision of enchondromas is done by simple curettage. A cortical window was first made to allow clearance of all tumour materials using a hand curette. Some surgeons will also do extra adjuvant treatment like high-speed burring or alcohol instillation to reduce the recurrence rate. After excision, the traditional approach of reconstructing the tumour cavity involves insertion of an autograft of cancellous bone harvested from the iliac crest or an allograft. Other augmentation methods include cement injection using Plaster of Paris, calcium phosphate cement (CPC) or polymethylmethacrylate (PMMA). Other surgeons adopt simple curettage without augmentation. The cortical window is also replaced in most cases.

Simple Curettage without Augmentation Due to the fact that bone regeneration occur spontaneously even without augmentation, there has been doubt whether bone grafting or cement injection is necessary after curettage. Schaller and Baer reported that no significant difference in bone density and functional results was found between 16 patients without bone graft and eight patients with additional bone graft.5 Morii et al. compared bone formation period of patients treated with simple curettage with those treated with hydroxyapatite injection. No significant difference was found.6

Curettage with Augmentation by Bone Graft Bone graft is traditionally placed in the tumour cavity to promote regeneration of bone tissues and restore mechanical

As an alternative to bone graft, different types of cement are also injected into the tumour cavity for augmentation. The in situ curing property of the cement allows immediate mechanical stability. Early mobilization can therefore be achieved to reduce joint stiffness. Common types of cement include Plaster of Paris, CPC, hydroxyapatite and PMMA. Cement injection has a low complications rate and recurrence rate overall. Gaasbeek et al. also achieved excellent function according to the Musckuloskeletal Tumour Society Score and no recurrence using plaster filling.9 Satisfactory results have also been yielded for construction using CPC. Kim and Kim reported no complications and 93–99% mean arc of motion for joints in 10 patients treated with CPC injection.10 Yasuda et al. obtained similar result in 10 patients but one case of malunion was reported for a patient undergoing early curettage before fracture healing.11 Similar good results are also reported for hydroxyapatite12 and PMMA.13 However, it was noted that 54% of patients treated using PMMA had decreased flexion range without functional limitations.13

Adjuvant Treatment Aiming at reducing the recurrence rate of enchondromas, some surgeons adopt adjuvant treatments like extensive curettage using high-speed burring, alcohol instillation of tumour cavity or even laser sterilization. Cha et al. compared the surgical outcome of alcohol instillation and high-speed burring and found no significant difference.14 Giles et al. treated eight patients with curettage followed by CO2 laser sterilization of tumour margins. No complications or recurrence was found.15

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Current Management of Hand Enchondroma

Treatment Timing Large enchondroma may accompany with pathological fractures. The usual practice is to allow the fracture to heal first before curettage is performed. This ensures mechanical stability for curettage and augmentation. However, the drawback is that the immobilization period of patients will be long and definitive diagnoses will be delayed. Some surgeon, therefore, prefer early curettage before the fracture is healed, which allows early mobilization and reduce joint stiffness. A 0% complications rate was obtained in a study on eight patients treated with early curettage and stabilization using injectable calcium sulfate cement.16 Another review comparing early and late treatment of enchondromas noted a significantly higher complications rate for early treatment (67%) compared to delayed treatment (10%).17 Yasuda et al. also reported a case of patient having malunion after early curettage and fixation using injectable calcium phosphate bone cement.11

Recurrence Various recurrence rates were reported in different publications with different treatments. In the retrospective review on 102 patients by Sassoon et al., a 6% recurrence rate was noted.2 Gaulke and Supplena reported a 14% recurrence rate in a longterm follow-up (mean, nine years) of 21 patients and all three recurrences were discovered over 10 years after operation.18

DISCUSSION Some cases of enchondroma are presented as incidental findings in radiographs with no obvious symptoms or pain. Some research has suggested that the majority of these tumours are latent (Latent lesions are defined as having well-demarcated borders according to the Enneking staging system19) and the risk of pathological fracture is low.20 Conservative treatment should be considered as the major treatment approach for these patients due to the low malignant potential (solitary enchondromas, 1.5–3%) and slow growth of enchondromas.4,20 Small localized asymptomatic enchondromas which are classified as latent in the Enneking staging system are especially suitable for conservative treatment. Surgical treatment may cause prolonged out-of-work period and certain degree of joint stiffness due to postoperative adhesion. Biopsies should be taken in suspected cases or uncertain diagnosis. A regular six-month radiographic follow-up should be adopted to monitor the

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growth and expansion of the tumour. The six-month period is an arbitrary interval suggested by the author. Clinical data on the spontaneous resolution rate and the natural history of enchondromas, however, are still largely absent. This is leading to the lack of evidence to support conservative treatment in clinical practices. More long-term follow-up studies are necessary in this field. Despite having satisfactory outcomes in some studies, early curettage of enchondroma before fracture healing is still not recommended in most cases.16 Other reviews showed that early curettage can lead to much higher complications rate (67%) compared to delayed treatment (10%) and cases of malunion were reported.11,17 Such high-risk surgery lacks adequate evidence to back up its necessity. It is also more technically demanding for surgeons due to the instability of the fracture location if it is located in the juxta-articular region. Corrective osteotomy to treat malunion is also difficult after cement injection. Studies showed that simple curettage without augmentation can achieve similar bone strength at similar recovery time compared to augmentation methods like bone grafting and cement injection. Augmentation using autograft inevitably create an extra surgical wound and increases the risk of infection. Augmentation using cement also does not improve the surgical outcome. It is therefore recommended that simple curettage should be prioritized in treatment of enchondromas. Cement augmentation should only be reserved for large tumour cavities to ensure mechanical stability of bones. Additional adjuvant treatments like high-speed burring, alcohol instillation and laser sterilization are not recommended for treating enchondroma. Although some satisfying results are yielded, similar outcomes are also achieved without such additional measures.9,13–15,21,22 Added with the damage to surrounding bone tissues, such prevention methods may be more suitable for low grade chondrosarcomas. The current literatures were summarized in Table 1. Based on the current evidence, the authors proposed a treatment algorithm (Fig. 1). Despite years of research on treatment methods of enchondroma, current literatures still have quite a number of obvious short-comings which may reduce the accuracy of research data. Firstly, the follow-up period is too short. Most studies have follow-up period of less than five years. However, a long-term study has found three out of 21 patients have recurrence over 10 years after curettage.18 The actual recurrence rate may therefore be higher than reported.

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Table 1

Study

Summary of Treatment of Enchondroma.

Patient Complications Recurrence Number Rate (%) Rate (%)

Follow-Up

Remarks

Simple curettage without augmentation Goto et al.21 At least six months Hasselgren et al.22 Six years

23 28

0 0

0 0

Curettage and augmentation using bone graft Bauer et al.7 NA Jewusiak et al.8 NA Figl and Leixnering23 47 months

12 23 27

0 0 7

0 0 0

Cortico-cancellous allograft Freeze-dried cancellous-bone allograft Autogenous spongy bone graft

Curettage and augmentation using cement injection Gaasbeek et al.9 53 months 19 Kim and Kim10 19 months 10 Yasuda et al.11 41 months 10

0 0 10

0 NA NA

Joosten et al.12 Bickels et al.13

Plaster filling Calcium phosphate bone cement Calcium phosphate bone cement, early curettage is performed in the only case of complications Hydroxyapatite cement Cemented internal fixation using PMMA, high speed drill burring after curettage

NA More than two years

8 13

0 0

NA 0

Adjuvant treatment Cha et al.14

40.8 months

29 33

0 0

0 0

High-speed burring Alcohol instillation

Giles et al.15

35.4 months

8

0

0

CO2 laser sterilization

19 months NA

8 6

0 67

0 NA

Lateral approach, reconstruction using calcium sulfate cement

Early curettage Lin et al.16 Ablove et al.17

Enchondroma suspected by X-ray features (well-defined, centrally placed osteolyc lesion at the juncon of metaphysis & diaphysis; somemes the bone is slightly expanded) presentaon

pathological fracture

incidental finding

wait ll fracture healed

X-ray features of inadequate bone support: - polycentric (diffuse & lobulated) - central lesion with significant corcal thinning - giant lesions (significant corcal expansion)

Yes

No Need for ssue diagnosis

cureage +/- augmentaon with bone gra or cement injecon

Yes

No

biopsy

regular follow up with radiograph every 6 months

Fig. 1

Treatment algorithm of enchondroma in hand.

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Current Management of Hand Enchondroma

Secondly, the sample size was too small. Most studies enrolled less than 30 patients and it was less than 10 in some studies. This leads to the lack of representation and reliability of the research data. In summary, small localized asymptomatic enchondromas that show no sign of impending growth can be treated with regular six month radiographical check-up. Expanding or symptomatic enchondromas should be treated with simple curettage without augmentation. Cement augmentation can be considered for particularly large tumour cavities for mechanical stability. Adjuvant treatments are not recommended due to the lack of sufficient clinical evidence that support its necessity.

References 1. Simon MJ, Pogoda P, Hovelborn F, Krause M, Zustin J, Amling M, Barvencik F, Incidence, histopathologic analysis and distribution of tumours of the hand, BMC Musculoskelet Disord 15:182, 2014. 2. Sassoon AA, Fitz-Gibbon PD, Harmsen WS, Moran SL, Enchondromas of the hand: Factors affecting recurrence, healing, motion, and malignant transformation, J Hand Surg Am 37(6):1229–1234, 2012. 3. Gaulke R, The distribution of solitary enchondromata at the hand, J Hand Surg Br 27(5):444–445, 2002. 4. Muller PE, Durr HR, Wegener B, Pellengahr C, Maier M, Jansson V, Solitary enchondromas: Is radiographic follow-up sufficient in patients with asymptomatic lesions? Acta Orthop Belg 69(2):112–118, 2003. 5. Schaller P, Baer W, Operative treatment of enchondromas of the hand: Is cancellous bone grafting necessary? Scand J Plast Reconstr Surg Hand Surg 43(5):279–285, 2009. 6. Morii T, Mochizuki K, Tajima T, Satomi K, Treatment outcome of enchondroma by simple curettage without augmentation, J Orthop Sci 15(1):112–117, 2010. 7. Bauer RD, Lewis MM, Posner MA, Treatment of enchondromas of the hand with allograft bone, J Hand Surg Am 13(6):908–916, 1988. 8. Jewusiak EM, Spence KF, Sell KW, Solitary benign enchondroma of the long bones of the hand, J Bone Joint Surg Am 53(8):1587–1590, 1971. 9. Gaasbeek RD, Rijnberg WJ, van Loon CJ, Meyers H, Feith R, No local recurrence of enchondroma after curettage and plaster filling, Arch Orthop Trauma Surg 125(1):42–45, 2005. 10. Kim JK, Kim NK, Curettage and calcium phosphate bone cement injection for the treatment of enchondroma of the finger, Hand Surg 17(1):65–70, 2012.

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11. Yasuda M, Masada K, Takeuchi E, Treatment of enchondroma of the hand with injectable calcium phosphate bone cement, J Hand Surg Am 31(1):98–102, 2006. 12. Joosten U, Joist A, Frebel T, Walter M, Langer M, The use of an in situ curing hydroxyapatite cement as an alternative to bone graft following removal of enchondroma of the hand, J Hand Surg Br 25(3):288–291, 2000. 13. Bickels J, Wittig JC, Kollender Y, Kellar-Graney K, Mansour KL, Meller I, Malawer MM, Enchondromas of the hand: Treatment with curettage and cemented internal fixation, J Hand Surg Am 27(5):870–875, 2002. 14. Cha SM, Shin HD, Kim KC, Park IY, Extensive curettage using a highspeed burr versus dehydrated alcohol instillation for the treatment of enchondroma of the hand, J Hand Surg Eur Vol 2013 Dec 24 [Epub ahead of print]. 15. Giles DW, Miller SJ, Rayan GM, Adjunctive treatment of enchondromas with CO2 laser, Lasers Surg Med 24(3):187–193, 1999. 16. Lin SY, Huang PJ, Huang HT, Chen CH, Cheng YM, Fu YC, An alternative technique for the management of phalangeal enchondromas with pathologic fractures, J Hand Surg Am 38(1):104–109, 2013. 17. Ablove RH, Moy OJ, Peimer CA, Wheeler DR, Early versus delayed treatment of enchondroma, Am J Orthop (Belle Mead NJ) 29(10):771– 772, 2000. 18. Gaulke R, Suppelna G, Solitary enchondroma at the hand. Long-term follow-up study after operative treatment, J Hand Surg Br 29(1):64–66, 2004. 19. Enneking WF, Spanier SS, Goodman MA, A system for the surgical staging of musculoskeletal sarcoma, Clin Orthop Relat Res 153:106– 120, 1980. 20. Bauer HC, Brosjo O, Kreicbergs A, Lindholm J, Low risk of recurrence of enchondroma and low-grade chondrosarcoma in extremities. 80 patients followed for 2–25 years, Acta Orthop Scand 66(3):283–288, 1995. 21. Goto T, Yokokura S, Kawano H, Yamamoto A, Matsuda K, Nakamura K, Simple curettage without bone grafting for enchondromata of the hand: With special reference to replacement of the cortical window, J Hand Surg Br 27(5):446–451, 2002. 22. Hasselgren G, Forssblad P, Tornvall A, Bone grafting unnecessary in the treatment of enchondromas in the hand, J Hand Surg Am 16(1):139– 142, 1991. 23. Figl M, Leixnering M, Retrospective review of outcome after surgical treatment of enchondromas in the hand, Arch Orthop Trauma Surg 129(6):729–734, 2009.

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Current management of hand enchondroma: a review.

Enchondromas are benign bone tumours originating from cartilages. It is mainly discovered incidentally in radiographs or due to symptoms like patholog...
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