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1 POLSKI PRZEGLĄD CHIRURGICZNY 2011, 83, 9, /v x Reconstruction of the distl rdio-ulnr joint with prosthesis of the distl uln in the tretment of recurrent gint cell tumour Dniel Kotrych 2, Andrzej Żyluk 1, Ireneusz Wlszek 1, Andrzej Bohtryrewicz 2 Deprtment of Generl nd Hnd Surgery, Pomernin Medicl University in Szczecin 1 Kierownik: prof. dr h. A. Żyluk Deprtment of Orthopedics nd Trumtology, Pomernin Medicl University in Szczecin 2 Kierownik: prof. dr h. A. Bohtryrewicz We present cse of 35-yer old left-hnded womn with recurrent gint-cell tumour ffecting ¼ of the distl prt of the left uln, with ssocited ulnr nerve involvement. After resection of the tumour nd 1 cm of the ulnr nerve, the distl uln ws reconstructed with n individully designed nd mtched prosthesis, followed y ulnr nerve reconstruction. At 12 months follow-up the ptients ws free of pin, hd excellent recovery of ulnr nerve function, stisfctory wrist rnge of motion nd modertely impired function of the left hnd (DASH score 42). She returned to her originl work in the office. We elieve tht restortion of the ntomy of the distl forerm fter en lock resection of the distl uln is desirle in young, ctive ptients, nd tht the prosthesis we used provides good ntomicl frmework for the recovery of the function of the wrist. Key words: gint-cell tumour, uln, distl rdio-ulnr joint Gint-cell tumor of the one is enign, loclly ggressive neoplsm, with low metsttic potentil nd is ssocited with sustntil skeletl moridity. Histologiclly the tumour is rich in osteoclst-like gint cells nd contins mononucler (stroml) cells tht express RANK lignd, key meditor of osteoclst ctivtion (1). Distl end of the uln is very uncommon locliztion of the lesion, which more frequently ffects distl end of the rdius (2). Mngement of smll lesions is similr to one cysts nd includes curettge of the lesion followed y filling the cve with cncellous one grft or polymethylmetcrylte. There is, however, high rte of recurrence fter most methods of locl tretment. To prevent frequent recurrences, n djuvnt locl tretment with lcohol or phenol is used. In dvnced, inoperle nd recurrent cses with metstses, humn monoclonl ntiody ginst RANK lignds denosum ws successfully tested (1). Gint-cell tumours of the distl rdius or uln re difficult to tret ecuse of their proximity to the wrist, multiple tendons, nerves nd rteries. Tretment of dvnced tumours, involving the whole epiphysis is prticulrly demnding, requiring rdicl en lock resection of the tumour followed y reconstruction of the osseous defect. It is routinely performed in the distl rdius involvement, ut not in the distl uln, where Drrch-type resection my e pplied (3). However, there is evidence tht lck of the ulnr support of the crpus often leds to ulnocrpl instility, unstle rottion round the ulnr xis nd, finlly, functionl impirment of the wrist nd hnd (4, 5). Therefore, restortion of the continuity of the distl uln is desirle, prticulrly in young, ctive persons. To dte, reconstruction of the distl uln fter rdicl resection ws performed y implnt rthroplsty (4, 6). We present cse of recurrent gint-cell tumour ffecting 1/4 of the distl prt of the left uln,

2 Reconstruction of the distl rdio-ulnr joint with prosthesis in recurrent gint cell tumour 519 with ssocited ulnr nerve involvement, in which fter resection of the tumour, the distl uln ws re-estlished with n individully designed nd mtched prosthesis, followed y ulnr nerve reconstruction. Cse report A 35-yer old left-hnded womn ws referred in 2009 to our institution with recurrent gint-cell tumor of the distl uln. She hd pinful, huge tumour t the distl forerm, mostly on the ulnr order (fig. 1). She complined from diminished senstion nd prsthesie loclized in the little nd ulnr hlf of the ring finger. She presented lso typicl ulnr deformtion of these fingers, suggesting ulnr nerve involvement. Six months erlier, the ptient underwent (outside of the Deprtment) curretge of the primry lesion loclized in the hed of the uln, followed y cncellous one grfting of the defect. Histologicl exmintion reveled gint-cell tumor of the distl uln. Present X-ry reveled huge one tumor of the distl uln, much greter thn the primry lesion (fig. 1). Lung CT scnning did not revel ny pthology. A wide resection of the distl uln nd inspection the ulnr nerve ws plnned. Bering in mind the size of the tumor (pproximtely 6 cm length) nd young ge of the ptient, we suggested lso replcement rthroplsty of the distl uln with n individully designed nd mtched endoprothesis. The prothesis (Implntcst GmH, Buxtehude, Germny) ws designed, sed on X-rys nd CT scns of the helthy wrist nd forerm of the ptient, regrding the necessity of resection of the one with n oncologicl mrgin (fig. 2). The prosthesis ws ssumed to restore the.length of the uln nd owing to moile unit the function of the distl rdio-ulnr joint. Pre-opertive ssessment showed sttic 2PD in the ring nd little fingers of 10 mm, touch treshold with lue filment (0.5 g), wrist flexion-extension of 135O (92% of the other side), prontion-supintion 150O (86%), totl grip strength of 16 Kg (59%) nd DASH score of 30 (rnge 0-100). The opertion ws performed under generl nesthesi nd with the tourniquet. Using dorsl ulnr incision reching the hlf of the forerm, the tumor ws pproched, Fig. 1. The view () nd X-ry () of the distl forerm of the ptient efore the second opertion exposed (fig. 3) nd resected with 0.5 cm mrgin of djcent soft tissues. The length of the resected uln ws 8 cm (fig. 4). Ulnr nerve ws found infiltrted t the distl forerm with tumor nd scr tissue nd, therefore, 1 cm of the nerve hd een en lock resected with the tumor, fter filed ttempt of its detchment. Then the distl uln with the rdio-ulnr joint ws re-estlished with the prosthesis. The stem of the rdil module ws inserted in the hole drilled in the distl rdius. The stem of the ulnr module ws preliminry inserted in the distl stump of the ulnr shft, without cementing. Next, oth prts were united y insertion of the rod of the ulnr module into hole in the ll of the rdil module. Protrud- Fig. 2. The scheme () nd view () of the prosthesis of the distl uln nd DRUJ

3 520 D. Kotrych et l, Fig. 3. Intropertive view of the tumour of the distl uln ing of the distl prt of the ulnr rod over the ll ws eliminted y deeper insertion of the stem of the module in the ulnr distl stump (fig. 5). Any necessry djustments were mde efore the definitive fixtion of the ulnr module of the prosthesis. Ulnr mrgin of tringulr firocrtilge ws sutured with the sheth of flexor crpi ulnris tendon over the rdil module. Pssive movements were performed to confirm stisfctory rnge of prontion nd supintion of the forerm (fig. 6). Finlly, the ulnr nerve ws repired end-to-end with the epineurl suture. The forerm ws immoilized for 4 weeks in n ove elow splint, in supintion. Post-opertive course ws uneventful. Active excercises of the wrist were initited t 4 weeks (eecuse of nerve repir) nd continued until chieving stisfctory rnge of motion. The ptient ws followed-up t 12 months nd the X-ry of the wrist nd forerm showed identicl sitution s immeditely post-opertively (fig. 7). She hd no pin t movements of the wrist nd no prsthesie. Post-opertive ssessment showed sttic 2PD in the ring nd little fingers of 7 mm, touch treshold with lue filment (0.5 g), suggesting excellent recovery of the ulnr nerve. Wrist flexionextension of 120O (82% of the other side), prontion-supintion 170O (identicl s in the other wrist, fig. 8), totl grip strength of 15 Kg (57%) nd DASH score of 42 (worse thn pre-opertively). Nine months fter surgery the ptient returned to her originl work in the office. Histopthologicl exmintion confirmed the dignosis of enign gint-cell Fig. 4. Sitution immeditely fter resection of the 8 cm segment of the distl uln Fig. 5. Sitution immeditely fter insertion of the prosthesis Fig. 6. X-ry of the distl forerm immeditely fter the opertion

4 Reconstruction of the distl rdio-ulnr joint with prosthesis in recurrent gint cell tumour Fig. 7. X-ry of the distl forerm t 12 months fter the opertion tumor nd complete resection of the lesion to helty tissue. Discussion Restortion of the ntomy of the distl forerm fter en lock resection of the distl rdius or uln is chllenging (7). To dte, the 521 most common types of surgery for gint-cell tumors of the distl uln included curettge nd pcking, or Drrch-type resection in more dvnced stges (4, 8). Unlike in distl rdius, reconstruction of the distl uln with free vsculrized fiulr grft or structurl llogrft ws not reported. However, ulnr hed prosthesis comined with simple soft tissue repir ws used for filed resection rthroplsties of the distl rdioulnr joint with good results (6). Distl ulnr implnt rthroplsty for recurrent gint-cell tumour of the distl uln ws reported y Roidis et l. (4). An ECentric ulnr hed prosthesis (Wright Medicl Technology, Arlington, TN, USA) ws used to replce 4 cm segment of the distl uln, which ws resected. The stem of the prosthesis ws fixed in the distl stump of the uln with one cement. Two yers fter surgery the ptient ws symptom free, without ny functionl impirment of the upper lim (4). Our result is not so good s tht of Roidis et l. (4), ut our ptient hd more dvnced nd lmost doule-sized tumor, with dditionl ulnr nerve involvement. To our knowledge, this type of prosthesis ws only once used in ptient in the other country. c d Fig. 8. Rnge of motion of the wrist t the finl ssessmnet t 12 months: flexion nd extension (, ) nd rottion (c, d)

5 522 D. Kotrych et l, One yer fter surgery the functionl outcome is in our ptient slightly worse thn sttus efore opertion (higher DASH score). However, the ptient hd resected ig segment of the distl uln for the oncologicl reson, which is lwys ssocited with some impirment of the hnd function, regrdless of the use of implnt rthroplsty. Moreover, the necessity of resection of ulnr nerve might hve negtive effect on finl outcome, regrdless its excellent recovery. Restortion of the power nd cceptle dexterity of the hnd llowed the ptient to return to her previous jo nd ctivities nd, therefore, we elieve tht our tretment option ws correct nd this cse is success. We elieve tht re-estlishment of the ntomy of the distl uln fter resection is desirle in young, ctive ptients, nd tht the prosthesis we used provides good ntomicl frmework, re-estlishing good function of the distl rdio-ulnr joint. However, we do not know how will e ptient s wrist function in the future, since long-term oservtions of such prostheses re lcking. references 1. Henshw TD, Skuiz K, Chwl S et l.: Denosum in ptients with gint-cell tumour of one: n open-lel, phse 2 study. Lncet Oncol Szo RM, Anderson KA, Chen JL: Functionl outcome of en lock excision nd osteorticulr llogrft replcement with the Suve-Kpndji procedure for Cmpncci grde 3 gint-cell tumor of the distl rdius. J Hnd Surg 2006; 31A: Cooney WP, Dmron TA, Sim FH et l.: En lock resection of tumors of the distl end of the uln. J Bone Joint Surg 1997; 79A: Roidis NT, Gougoulis NE, Likou PD et l.: Distl ulnr rthroplsty s definitive tretment of recurrent gint-cell tumor. J Hnd Surg 2007; 32A: Gordon KD, Dunning CD: Kinemtics of ulnr hed rthroplsty. J Hnd Surg 2003; 23B: Vn Schoonhoven J, Fernndez DL, Bowers WH et l.: Slvge of filed resection rthroplsties of the distl rdioulnr joint using new ulnr hed prosthesis. J Hnd Surg 2000; 25A: Cmpell DA, Millner PA, Dreghorn CR: Primry one tumors of the hnd nd wrist. J Hnd Surg 1995; 20B: Hrness NG, Mnkin HJ: Gint-cell tumor of the distl forerm. J Hnd Surg 2004; 29A: Received: r. Adress correspondence: Szczecin, ul. Unii Luelskiej 1