SCIENTIFIC ARTICLE

Recurrence of a Giant Cell Tumor of the Hand After 42

Years: Case Report

Keith Jackson, MD, Charles Key, MD, Michelle Fontaine, MD, Richard Pope, MD

Giant cell tumors of bone in the hand are rare. We present a case of a recurrent giant celltumor in the metacarpal 42 years after intralesional excision and autogenous bone grafting.The possibility of recurrent disease should be considered in the evaluation of any patientpresenting with new onset of pain at the site of a previously addressed giant cell tumor.Management of these recurrent lesions should include wide excision with digit salvagingprocedures or ray amputation owing to the high rates of treatment failures seen with marginalexcision. (J Hand Surg 2012;37A:783–786. Published by Elsevier Inc. on behalf of theAmerican Society for Surgery of the Hand.)

Key words Giant cell, recurrent, tumor.

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GIANT CELL TUMORS of bone represent 5% ofbone neoplasms and are most commonly foundas a solitary lesion in the metaphysis of long

bones. Although classified as benign, these tumors canbe locally aggressive, and they metastasize to the lungsin approximately 3% of patients.1 Giant cell tumors ofbone infrequently involve the hand. These lesions havea higher risk of local recurrence after intralesional treat-ment than those presenting in other areas of the body.2,3

In most cases, recurrence is within 3 years of surgicaltreatment, but longer periods of quiescence have beenreported.4,5 The purpose of this report is to present apatient with recurrent giant cell tumor of bone treated42 years after curettage and bone grafting of a meta-carpal lesion.

CASE REPORTA 69-year-old, right-handed man was referred to theoutpatient orthopedic clinic complaining of ulnar-sided

From the Dwight David Eisenhower Army Medical Center, Fort Gordon, GA.

Received for publication May 27, 2011; accepted in revised form December 6, 2011.

No benefits in any form have been received or will be received related directly or indirectly to thesubject of this article.

DISCLAIMER: The authors are employees of the U.S. Government. This work was prepared as part oftheirofficialduties.Theviewsexpressedinthisarticlearethoseoftheauthorsanddonotreflecttheofficial policy or position of the Department of the Army, the Department of Defense, or the UnitedStates government.

Corresponding author: CPT Keith Jackson, MD, Dwight David Eisenhower Army Medical Center,300 East Hospital Road, Fort Gordon, GA; e-mail: lynn.jackson9@gmail.com.

0363-5023/12/37A04-0023$36.00/0

Tdoi:10.1016/j.jhsa.2011.12.005

ight hand pain for the past 3 months since striking theand on a wall. He was initially evaluated 1 day afternjury by his primary care physician. Radiographs athat time (Fig. 1) revealed a lytic lesion involving theistal portion of the fourth metacarpal but neitherhe treating physician nor staff radiologist initiallyoted the lesion, and the patient was prescribed aonsteroidal anti-inflammatory medication. Threeonths later, the patient reported that the pain had

radually increased in intensity since his initial visit.The patient recalled having had similar symptoms 43

ears earlier in the same location. At that time, accord-ng to the operative report, he was treated at an outsidenstitution with an intralesional biopsy and iliac crestone grafting for a giant cell tumor involving the fourthetacarpal head. After this procedure, he was able to

eturn to full military duty for approximately 6 monthsntil he developed similar pain in the same location. Aepeat intralesional excision, performed 9 months afterhe index procedure, revealed a recurrence of the fourthetacarpal giant cell tumor. After this second interven-

ion, the patient was able to complete a 20-year careern the military with no activity restrictions.

On presentation to our clinic, the patient deniedevers, chills, or other symptoms related to systemicnfection. His past medical and surgical history wasoncontributory outside of the 2 previously describedrocedures. Examination of the hand revealed a longi-udinal incision in the space between the fourth and fifthetacarpals that was well healed and nonerythematous.

here was diffuse swelling and tenderness along the

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784 RECURRENCE OF GIANT CELL TUMOR

fourth metacarpal. The patient had full motion at themetacarpophalangeal and interphalangeal joints. Hehad normal sensation to light touch in all distributionsof the hand. The finger was well perfused.

Radiographs demonstrated an expansile lytic lesionof the distal fourth metacarpal measuring 3.4 � 2.3 cmwith marked bony destruction (Fig. 2). T2-weightedmagnetic resonance imaging (MRI) revealed a largehyperintense heterogeneous lesion involving the distalportion of the fourth metacarpal with invasion of thesurrounding soft tissues (Fig. 3). A chest radiographshowed no evidence of pulmonary disease. A bone scanshowed a solitary focal increase in radiotracer uptake inthe right fourth metacarpal without pulmonary involve-ment. Given the history of a previously treated giant celltumor of bone at this location, combined with the de-struction of the metacarpal articular surface and inva-sion of the adjacent soft tissue seen on MRI, a fourthray resection was recommended.

Intraoperatively, we found a large, contained, soft-tissue lesion that replaced the bony portion of the distalfourth metacarpal. We obtained aerobic and anaerobiccultures that would ultimately yield no bacterial growth.

FIGURE 1: Posteroanterior x-ray of the right hand demonstratinga contained lytic lesion of the fourth metacarpal head at initialpresentation.

Final pathologic examination revealed sheets of mono-

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nuclear cells with abundant multinucleated giant cells(Fig. 4). No atypical mitotic figures were seen. Thetumor extended into the surrounding soft tissues fo-cally. Postoperatively, the patient recovered unevent-fully and at 1-year follow-up had returned to normalactivities with no residual pain. Hand and chest radio-graphs at that time revealed no evidence of local recur-rence or pulmonary metastases.

DISCUSSIONUp to 2% of giant cell tumors of bone presentingthroughout the body involve the hand.3 These lesionsmost commonly occur in an eccentric location of theepiphyseal region, generally affect individuals betweenthe ages of 20 and 40, and have a slight female predom-inance.1 Microscopic review reveals mononuclear spindleand ovoid-shaped cells associated with multinucleated gi-ant cells and macrophages.6–9 Surgical treatment formetacarpal-based tumors ranges from intralesionalcurettage with bone grafting or filling with polym-ethylmethacrylate cement to ray amputation.1–4,10,11

Involvement of the bones of the hand has beenassociated with higher rates of local recurrence and

FIGURE 2: Posteroanterior x-ray of the right hand demonstratingan expansile lytic lesion of the fourth metacarpal head 3 monthsafter the initial presentation.

pulmonary metastasis than similar lesions presenting in

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other areas of the body.3,12 To this end, Athanasian andassociates2,3 reported recurrence rates in 79% of pa-tients treated with intralesional excision and 36%treated with marginal excision, wide excision, amputa-tion, or ray resection for tumors involving the phalangesor metacarpals. Athanasian et al2 reported local recur-rence in 3 of 4 recurrent tumors treated with curettagewith or without adjuvant treatment. In this same caseseries, 4 of 9 lesions recurred after marginal excision,wide excision, and amputation, whereas neither of 2patients treated with ray resection had documented re-current disease. Averill et al12 reported success in 77%

FIGURE 3: T2-weighted coronal A, sagittal B, and axial Cmetacarpal with expansion into the surrounding soft tissues.

FIGURE 4: Hematoxylin and eosin stain of a tissue sampledemonstrating mononuclear and mononucleated cells intimatelyassociated with residual bone (right fourth metacarpal).

of recurrent lesions treated with wide excision or am-

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putation. The results of these 2 experiences suggest thatrecurrent giant cell tumors of bone involving the handare better treated with resection or amputation.

Athanasian et al2,3 would suggest that our patientwould be at high risk for local recurrence after 2 intra-lesional procedures, but the 42-year period between hissecond surgical intervention and recurrence is notewor-thy. Although recurrence is generally noted within theinitial 3 years in all other areas of the body,4,8,9 lesionsinvolving the bones of the hand typically recur within12 months of treatment.2,12 Despite this predilection forearly recurrence, multiple cases of late treatment fail-ures have been reported in areas outside the hand. In areview of 4 cases, Scully and colleagues5 described laterecurrence of giant cell tumors in areas where previ-ously identified lesions had been surgically managed.The quiescent period in these 4 patients ranged from 19to 30 years, and at the time was the latest-reportedrecurrence of giant cell tumor.

In this case, we chose ray resection because of the 2previous treatment failures combined with destructionof the metacarpal head and the extent of soft tissueinvasion seen on MRI. Few studies have evaluatedtreatment options in the management of recurrent giantcell tumors of bone involving the hand.

The soft tissue invasion seen on MRI also had a rolein our decision. In a review of giant cell tumors of bonewith extraosseous extension (Campanacci grade III)occurring throughout the body, Prosser et al13 reported

es demonstrating a hyperintense lesion of the distal fourth

imag

a 29% recurrence rate after curettage alone. When ex-

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786 RECURRENCE OF GIANT CELL TUMOR

amining tumors involving only the bones of the hand,Athanasian et al2 reported a 100% recurrence rate in 5patients treated with curettage with or without bonegrafting when extraosseous extension was noted preop-eratively. To this end, the authors recommended digitsalvaging procedures after giant cell tumor resectiononly when adequate soft tissue margins could be ob-tained.

In light of these findings, management strategies forperiarticular metacarpal lesions with soft tissue involve-ment are wide excision with structural bone grafting orray resection. Although wide resection and reconstruc-tion with metacarpophalangeal joint arthroplasty hasproven to be a viable treatment option in select patientpopulations,14 in this case attempts at salvage of thedigit would likely have compromised long-term func-tion and risked further recurrence given the amount ofthe surrounding soft tissue needed to be excised toobtain a tumor-free margin.

The 42-year latency between clinical presentationsand the aggressive nature of tumor recurrence representinteresting features of this case. Campanacci et al15

provided a possible mechanistic explanation for theperiod of quiescence seen in these recurrent cases. Re-tained tumor cells maintain their phenotype and canbecome active after a specific inciting event. Althoughthis theory could help explain the prolonged period ofclinical dormancy as well as the aggressive nature inwhich the tumor recurred within the 3 months betweenoutside presentation (Fig. 1) and orthopedic consulta-tion (Fig. 2), additional research is needed on potentialtriggers, tumor characteristics, and ultimate treatmentrecommendations of these late recurrent lesions. Atpresent, we recommend consideration of wide excisionor ray amputation of giant cell tumors of bone involvingthe metacarpal that recur or demonstrate extraosseousextension owing to the high rate of recurrence withintralesional procedures. The decision to reconstruct or

resect the involved ray should be based on the sur-

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geon’s ability to obtain a tumor-free margin and thepatient’s functional demands.

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