Reconstruction of the Shoulder Joint with a Custom-Made

Reconstruction of the Shoulder Joint with a Custom-Made Ceramic Implant After a Total Scapulectomy

A Case Report

Yoshihiro Araki, MD, Akihiko Yoshida, MD, PhD, Yoshikazu Tanzawa, MD, PhD, Makoto Endo, MD, PhD,Eisuke Kobayashi, MD, PhD, and Akira Kawai, MD, PhD

Investigation performed at the National Cancer Center Hospital, Tokyo, Japan

AbstractCase: We describe a 22-year-old woman who underwent total scapulectomy and shoulder joint reconstruction with use ofa custom-made ceramic implant composed of hydroxyapatite and beta-tricalcium phosphate (b-TCP) for a recurrentatypical perineurioma that had arisen from the scapula.

Conclusion: To our knowledge, there have been no previous reports of shoulder joint reconstruction with use of a custom-made ceramic implant after a total scapulectomy. The patient showed excellent function of the new shoulder joint andgood range of motion without pain or dislocation at 18 months postsurgery. This new method of reconstructing theshoulder joint after a total scapulectomy appears useful and promising.

Atotal scapulectomy (Malawer type-III resection) isperformed for carefully selected patients with malig-nant tumors involving the scapula1,2. Although various

methods of reconstruction after total scapulectomy have beenreported, no optimal procedure has emerged because of thedifficulty in reconstructing the complex shape of the scapularbone and retaining a wide range of shoulder joint motion, aswell as the rarity of such cases3-15. However, in the fields ofdental and craniofacial surgery, custom-made artificial boneimplants prepared by computer-aided design (CAD) andcomputer-aided manufacturing (CAM) systems recently havebeen used to reconstruct large bone defects after injury or tu-mor resection16-18.

We describe a 22-year-old woman who underwent totalscapulectomy and shoulder joint reconstruction because of arecurrent atypical perineurioma, a low-grade malignant bonetumor, that had arisen from the scapular bone. The custom-made ceramic implant was manufactured using a CAD/CAMsystem.

The patient was informed that data concerning the casewould be submitted for publication, and she provided consent.

Case Report

The patient had begun to feel left shoulder pain at the age of16 years, and had been referred to our institution with

findings of bone-destructive tumors in the left scapula. An

open biopsy had been performed, and a histologic diagnosis ofatypical perineurioma of the bone had been made. Primarysurgery involving curettage and bone-grafting with use of ar-tificial bone had been performed when she was 17 years old.Eighteen months after the primary operation, local recurrencewas detected by follow-up magnetic resonance imaging (MRI).The recurrent tumor was slow-growing, and local pain grad-ually increased.

Radiography revealed osteolytic bone destruction andscalloping of the affected scapula (Fig. 1-A). Computed to-mography (CT) showed that the lesion extended from thescapular body and spine to the coracoid process and glenoid,with focal cortical destruction and bone expansion (Figs. 1-Band 1-C). MRI demonstrated both intraosseous and extraoss-eous extension of the tumor, appearing as an isointense signalon T1-weighted images (Fig. 2-A) and as an isointense-to-hyperintense signal on T2-weighted images (Fig. 2-B) relativeto the surrounding muscles. The tumor was enhanced intenselyand homogeneously after intravenous administration ofgadolinium-diethylenetriamine pentaacetic acid (Fig. 2-C).Positron emission tomography (PET) showed high fluo-rodeoxyglucose uptake within the tumor, with a maximumstandardized uptake value of 9.7 (Fig. 2-D).

The patient was fully informed about the surgical recon-struction method and the cost of the custom-made ceramic im-plant, which would be covered by health insurance. She provided

Disclosure: The authors indicated that no external funding was received for any aspect of this work. The Disclosure of Potential Conflicts of Interestforms are provided with the online version of the article (http://links.lww.com/JBJSCC/A660).

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JBJS Case Connect 2018;8:e12 d http://dx.doi.org/10.2106/JBJS.CC.17.00061

http://links.lww.com/JBJSCC/A660

Fig. 1

Figs. 1-A, 1-B, and 1-C Imaging. Fig. 1-A Anteroposterior radiograph revealing osteolytic bone destruction and scalloping of the affected scapula. Axial (Fig.

1-B) and coronal (Fig. 1-C) CT showing that the lesion extended from the scapular body and spine to the coracoid process and glenoid, with focal cortical

destruction and bone expansion.

Fig. 2

Figs. 2-A through 2-D MRI demonstrating both intraosseous and extraosseous extension of the tumor, appearing as an isointense signal on the

T1-weighted image (Fig. 2-A) and as an isointense-to-hyperintense signal on the T2-weighted image (Fig. 2-B) relative to the surrounding muscles. Fig.

2-C The fat-saturated T2-weighted image shows that the tumor was enhanced intensely and homogeneously after intravenous administration of

gadolinium-diethylenetriamine pentaacetic acid. Fig. 2-D PET showed high fluorodeoxyglucose uptake within the tumor, with a maximum standardized

uptake value of 9.7.

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VOLUME 8 d NUMBER 1 d FEBRUARY 28, 2018RECONSTRUCTION OF A SHOULDER JOINT WITH A CUSTOM-MADE

CERAMIC IMPLANT AFTER TOTAL SCAPULECTOMY

consent to undergo a Malawer type-IIIA total scapulectomy andreconstruction of the shoulder joint with a custom-made ceramicimplant (Ceratite; Ngk Spark Plug) that wasmanufactured using aCAD/CAM system (Figs. 3 and 4). CT images of the lesion wereacquired and modified into a 3-dimensional (3D) reconstructionmodel; this was transferred to a CAD program, allowing acustom-made ceramic implant to be designed. The shape of theimplant was modified with reference to the 3D model (Fig. 3).The acromion and the coracoid process were omitted to simplifythe anatomic structure and to prevent fracture due to thinness orpoor strength, but the shape of the glenoid surface was retainedbecause it was anticipated that this would provide awider range ofmotion after the scapulectomy15. With regard to the acromion inparticular, we considered that if the thickness was increased toprovide strength, pain or a rotator cuff tear would likely occur

because of collision between the acromion and the humeral head,leading to a disturbance of excursion. We planned to create a newconnection between the inferior aspect of the distal part of theclavicle and the lateral upper aspect of the implant, which wasintended to replace the original acromioclavicular joint. There-fore, the implant was designed to be suspended at the distal end ofthe clavicle and to acquire an osseous connection to providestability in addition to the reattachment of soft tissue. Althoughtime was needed to design the implant, the production processusing a CAD/CAM system was not complicated because the jointsurface design was matched to the original one, and the otherparts were simplified considerably. The CAM software generated aset of tool paths for manufacture of the implant on a computer-numerical-control milling machine to acquire the exact 3Dshape. The custom-made ceramic implant was milled from a

Fig. 3

Figs. 3-A and 3-B Implant. Fig. 3-A The 3D plaster model was made by a 3D printer. Fig. 3-B The custom-made ceramic implant was manufactured by the

CAD/CAM system with reference to the 3D plaster model.

Fig. 4

Figs. 4-A through4-E The custom-made ceramic implant wasmade fromhydroxyapatite andb-TCP. The shape of the implant wasmodifiedwith reference to

the 3D model.

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hydroxyapatite and beta-tricalcium phosphate (b-TCP) block;the process took approximately 1 month to complete.

The patient was 22 years old at the time of surgery. First, atotal scapulectomywas performed, and the extraosseous tumorsinfiltrating the surrounding muscles, especially the subscapu-laris, supraspinatus, and infraspinatus, were resected with awide surgical margin. The suprascapular nerve had beendamaged because of infiltration by the lesion. The axillary nerveand its vessels were identified and preserved. A circumferentialcapsulotomy was completed, and then the scapula was removedwith the lesion (Fig. 5-A). All of the preservedmuscles, tendons,and capsules were marked for later reattachment. After thescapulectomy, reconstruction was performed with the custom-made ceramic implant. The implant was adapted to the inferiorsurface of the distal aspect of the clavicle as designed, and it wascarefully placed in the correct anatomic position on the chestwall. It was then suspended through suture holes that weremade at the distal end of the clavicle, which were aligned withthe suture holes in the implant. The remaining capsule of theglenohumeral joint was then reattached, and all of the sur-rounding residual muscles and tendons were reattached usingsutures that were passed through the small peripheral holes inthe implant (Fig. 5-B). The rhomboid major and minor on themedial side; the teres major and minor on the lateral side; theresidual infraspinatus on the posterior side; the residual su-praspinatus on the upper side; and the pectoralis minor, theshort head of the biceps brachii, the coracobrachialis, the ser-ratus anterior, and the residual subscapularis on the anteriorside were reattached to cover the implant. The deltoid andtrapezius were then reattached through the vacant suture holesin the implant or by direct suturing to the clavicle; when this wasnot possible, we sutured the muscles surrounding the implant,thus completing the reconstruction. Pathologic examination ofthe resected specimen revealed recurrence of the atypical peri-neurioma of the bone (Fig. 6).

A shoulder abduction orthosis was applied for 6 weekspostsurgery. At the 18-month follow-up, the patient dem-onstrated excellent shoulder function and a comparativelygood range of motion. She was able to perform active

Fig. 5

Figs. 5-A and 5-B Intraoperative photographs. Fig. 5-A A total scapulectomy was performed, and the extraosseous tumors infiltrating the surrounding

muscles, especially the subscapularis, supraspinatus, and infraspinatus, were resected with a wide surgical margin. A circumferential capsulotomy was

completed, and then the scapula was removed with the lesion. Fig. 5-B The implant was carefully placed in the correct anatomic position on the chest wall

andsuspended through thedistal endof the clavicle; the remaining capsuleof the glenohumeral jointwas then reattached, followedby reattachment of all of

the surrounding residual muscles using sutures that were passed through small peripheral holes in the implant.

Fig. 6

The tumor was composed of hypercellular infiltrative tissue showing stori-

form and whorled growth. The tumor cells were spindle to epithelioid, and

exhibited nuclear hyperchromasia andmild pleomorphism; however, mitotic

activity was very low, and necrosiswasnot observed (hematoxylin and eosin,

·200). Immunohistochemically, the tumor cells were positive for epithelial

membrane antigen, glucose transporter 1 (GLUT1), and claudin 1, but

negative for cytokeratin, S100 protein, actin, desmin, progesterone recep-

tor, somatostatin receptor 2a,mucin 4 (MUC4), and cluster of differentiation

(CD34). Electron microscopy demonstrated elongated cytoplasmic pro-

cesses and pinocytotic vesicles, consistent with perineurial differentiation;

however, there was no evidence of complex interdigitating processes or

desmosomes. The diagnosis of atypical perineurioma was made.

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shoulder abduction to 60�, active flexion to 80�, passive ab-duction to 100�, and passive flexion to 120� without pain ordislocation of the glenohumeral joint. The widely resectedsubscapularis, supraspinatus, and infraspinatus muscles didnot work normally, but all of the other reattached muscles,especially the deltoid, worked well. Additionally, the patientwas able to retract the implant using the reattached trapezius,rhomboid major, and rhomboid minor, and could also pro-

tract the implant using the reattached serratus anterior andpectoralis minor muscles. The International Society of LimbSalvage (ISOLS) functional score was 23 (76.7%) of 30points, including 5 of 5 points for pain, 4 of 5 points forfunction, 4 of 5 points for emotional acceptance, 3 of 5 pointsfor hand positioning, 5 of 5 points for manual dexterity, and2 of 5 points for lifting ability. Postoperative radiographyshowed good positioning of the humeral head against the

Fig. 7

Figs. 7-A through 7-E Imaging. Fig. 7-A The postoperative radiograph showed good positioning of the humeral head and the implant. Coronal (Fig. 7-B) and

axial (Fig. 7-C) CT showed smooth conformity of the reconstructed glenohumeral joint. At the final follow-up, T1-weighted (Fig. 7-D) and enhanced fat-

saturated T2-weighted (Fig. 7-E) MRI demonstrated no evidence of recurrence or degenerative change of the humeral head.

TABLE I Comparison of the Various Reconstruction Methods After Total Scapulectomy in Terms of Functional Outcomes and Complications*

Reconstruction MethodNo. ofPatients

Follow-up(months)

ISOLSScore (%)

Flexion(degrees)

Abduction(degrees) Complications

Humeral suspension

Nakamura et al., 19994 10 48 64.6 NA NA NA

Hayashi et al., 20113 4 51 57.7 12.5 11.3 None

Total shoulder arthroplasty

Pritsch et al., 20079 16 90 78.5 40 40 Dislocation (1), wounddehiscence (1)

Tang et al., 201110 10 36 76.7 51 40 Dislocation (1), superficialinfection (1)

Allograft/autograft reconstruction

Zhang et al., 200915 3 26 79 43 55 Chronic pain (1)

Capanna et al., 201514 6 66 66.7 60 62 Osteosynthesis failure (2),fracture (2)

Custom-made ceramic implant

Present case 1 18 76.7 80 60 None

*ISOLS = International Society of Limb Salvage, and NA = not available.

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implant (Fig. 7-A). Although osseous fusion between theimplant and the distal aspect of the clavicle had not beenclearly recognized, the glenohumeral rhythm motion of theimplant during abduction of the reconstructed shoulder jointwas reproducibly observable using fluoroscopy. CT (Figs. 7-Band 7-C) and MRI (Figs. 7-D and 7-E) showed smoothconformity of the reconstructed glenohumeral joint and nodegenerative change in the humeral head. There was no localrecurrence of the lesion (Fig. 7).

Discussion

Unresolved problems after total scapulectomy includeshoulder joint dysfunction and poor cosmetic out-

comes. Although various reconstruction techniques aftertotal scapulectomy have been reported, no optimal ap-proach has emerged3-15 (Table I). We have described re-construction of the shoulder joint using a custom-madeceramic implant after a Malawer type-IIIA total scapulec-tomy for recurrent atypical perineurioma, a low-grademalignant bone tumor19-22.

The humeral suspension method has been reported tobe satisfactory in terms of safety and reliability. However, ityields only functionally fair results, with a low range ofmotion and an unsatisfactory appearance3,4. In contrast, totalshoulder replacement has been reported to yield functionallyexcellent results and a better range of motion, but it requireshumeral head replacement, even if the head has no lesion; italso has a high risk of complications, including infection,dislocation, and implant breakage9-13. In comparison with theestablished form of metal implant, a ceramic implant thatpossesses the original glenoid surface, which can be custom-made using a CAD/CAM system, retains its joint congruityand can prevent humeral head dislocation, degenerativechanges in the head surface, and implant breakage. Althougha few reported cases of allograft reconstruction or autologousrecycled bone graft reconstruction using liquid nitrogen orradiation treatment have shown functionally better resultsthan humeral suspension, and the appearance has been sat-isfactory, there was also a high rate of complications, such asfracture of the grafted bone and implant fixation failure14,15.These complications may occur when the anatomic structureis thin or if absorption occurs after surgery. In planning forour patient, we built a 3D model of the shoulder joint using a3D printer (Fig. 3-A). Our analysis revealed that the body ofthe affected scapula was very fragile; therefore, its use forautologous recycled bone graft reconstruction would carry ahigh risk of breakage. The custom-made ceramic implantthat we used was made from hydroxyapatite and b-TCP;preoperatively, it was confirmed to have sufficient strengthby load testing, and was able to withstand a potential bendingstress of 1,700 kgf/cm2 and a compressive stress of

6,000 kgf/cm2. These strengths are similar to or greater thanthose of cortical bone in the human body23.

Our patient showed excellent shoulder function and acomparatively good range of motion at 18 months after sur-gery. We believed that customization of a ceramic hydroxyap-atite and b-TCP implant using the CAD/CAM system wouldyield a good cosmetic appearance, sufficient strength, goodmatching with the bone defect, and fairly good function, asdescribed in the previous reports in the field of dental andcraniofacial surgery16-18. The osseous fusion between the im-plant and the distal aspect of the clavicle had not been clearlyrecognized; however, this new connection contributed to thesmooth abduction motion of the new shoulder joint, replacingthe original acromioclavicular joint, and did not place excessivestress on the sternoclavicular joint and the clavicle itself. Incomparison with a metal implant, an additional potential meritof the ceramic implant is easier detection of any tumor re-currence by CTor MRI because of fewer image artifacts. On theother hand, one possible disadvantage of the ceramic implant isthe period of approximately 1 month that was required for itspreparation. Additionally, a longer follow-up period will benecessary before any definite conclusion can be reached aboutthe value of this method.

To our knowledge, there has been no previous report ofscapular reconstruction using a custom-made ceramic implantfollowing a total scapulectomy. We believe that this newmethod offers a promising option for reconstruction of theshoulder joint after total scapulectomy. n

Yoshihiro Araki, MD1,2

Akihiko Yoshida, MD, PhD1

Yoshikazu Tanzawa, MD, PhD1,2

Makoto Endo, MD, PhD1

Eisuke Kobayashi, MD, PhD1

Akira Kawai, MD, PhD1

1Divisions of Musculoskeletal Oncology (Y.A., Y.T., M.E., E.K., and A.K.)and Pathology and Clinical Laboratories (A.Y.), National Cancer CenterHospital, Tokyo, Japan

2Department of Orthopaedic Surgery, Graduate School of MedicalSciences, Kanazawa University, Kanazawa, Japan

E-mail address for Y. Araki: [email protected] address for A. Yoshida: [email protected] address for Y. Tanzawa: [email protected] address for M. Endo: [email protected] address for E. Kobayashi: [email protected] address for A. Kawai: [email protected]

ORCID iD for A. Kawai: 0000-0003-2116-586X

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mailto:[email protected]






http://orcid.org/0000-0003-2116-586X

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Reconstruction of the Shoulder Joint with a Custom-Made