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CASE REPORT
Ahead of print publication  

Two cases of total bone endoprosthetic reconstructions as limb salvage following resection of rare malignancies of proximal femur and humerus


1 Ex-Prof and HoD, Department of Orthopaedics, AFMC, Pune, Maharashtra, India
2 Assistant Professor, Department of Orthopaedics, Base Hospital, Lucknow, Uttar Pradesh, India
3 Assistant Professor, Department of Orthopaedics, Military Hospital, Secunderabad, Telangana, India

Date of Submission13-May-2020
Date of Decision04-Jul-2020
Date of Acceptance06-Jul-2020
Date of Web Publication23-Sep-2020

Correspondence Address:
BM Naveen,
Department of Orthopaedics, Base Hospital, Lucknow - 226 002, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jmms.jmms_54_20

  Abstract 


The femur and humerus are the most common long bones affected by both primary and malignant tumors. The choice of limb salvage or amputation always remains a dilemma to achieve an optimum outcome. Two such patients of malignant tumors of the long bone, one in the proximal femur and the other at the humeral shaft, were managed at our center with limb salvage. The first patient is a 36-year-old male with a rare malignant nerve sheath tumor of proximal femur treated at a peripheral hospital with curettage and stabilization by an intramedullary nail for the lesion in the proximal femur, presuming it to be a benign lesion. He was subsequently managed by total femur resection followed by an endoprosthetic reconstruction, which resulted in an excellent outcome with no local recurrence or metastasis at 5-year follow-up. Similarly, the other case, a 42-year-old male with primary chondrosarcoma of humeral shaft of 2 years' duration, was successfully treated with total humeral excision followed by an endoprosthetic reconstruction. He also achieved an excellent functional outcome and resumed his job 1 year postoperatively. At 5 years' follow-up, there was no local recurrence or distal metastasis in this case too. These two cases of malignant lesions of femur and humerus were unique in many ways and gave a lot of insights into the diagnosis and management of rare malignancies of long bones.

Keywords: Endoprosthesis, femur, humerus, reconstruction, salvage, tumor



How to cite this URL:
Sharma Y, Naveen B M, Phillip VM. Two cases of total bone endoprosthetic reconstructions as limb salvage following resection of rare malignancies of proximal femur and humerus. J Mar Med Soc [Epub ahead of print] [cited 2020 Nov 24]. Available from: https://www.marinemedicalsociety.in/preprintarticle.asp?id=295894




  Introduction Top


Limb salvage surgery for malignant long bone tumors is an effective surgical option in recent years with advancements in chemotherapy and better implant designs. Most commonly involved long bones are femur and humerus. The femur is the most common long bone affected by both benign and malignant lesions.[1] Conventionally, all malignant tumors were treated by amputation. With the advent of better implants, improved chemotherapeutic regimens and refinements in surgical techniques, the concept of limb salvage has evolved. Sometimes, radical excision is required to achieve tumor clearance resulting in complete removal of involved bones.[1] Various modalities of reconstruction include autografts, allografts, allograft-prosthetic composite, rotationplasty, and total femoral or humeral endoprosthesis. Limb salvage in the form of endoprosthetic reconstruction results in better functional outcomes and similar long time survival in comparison to amputation.[2],[3],[4] Here, we present two interesting cases, one each of total femur reconstruction and a total humerus reconstruction using an endoprosthesis following excision of rare malignant tumors of the proximal femur and middle-third humerus.


  Case Report Top


Case summary 1

A 36-year-old male presented to a peripheral hospital with complaints of acute onset pain in left hip of 20 days' duration followed by a limp. It was progressive and localized to anterior and lateral aspects of the hip. The examination was noncontributory except for tenderness over the hip region. Radiographs showed a destructive expansile lesion in the trochanteric region, which extended onto the neck and subtrochanteric region [Figure 1]a. Magnetic resonance imaging revealed 28 mm × 78 mm sharply defined lesion with internal septations suggestive of monostotic fibrous dysplasia.
Figure 1: (a) Initial anteroposterior radiograph of the pelvis with both hips showing the lesion in the left proximal femur (blue arrow). (b) Anteroposterior and lateral radiograph of the left femur after curettage and prophylactic nailing

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He underwent extended curettage, autologous bone grafting, and prophylactic nailing of the femur [Figure 1]b at a peripheral hospital. Later on, the histopathological examination revealed a high-grade spindle cell sarcoma with no malignant osteoid. Immunohistochemistry suggested either a malignant fibrohistiocytic tumor or pleomorphic liposarcoma. Hence, the patient was transferred to a tertiary care center where in HPE blocks were reviewed, and the diagnosis of high-grade spindle cell sarcoma was confirmed. Initial bone scan, contrast-enhanced computed tomography (CECT) scan femur, and CECT chest did not reveal any metastasis. However, fluorodeoxyglucose/positron emission tomography (FDG/PET) revealed increased uptake in the soft tissue of proximal femur and metastasis to the left external iliac lymph nodes. Based on these clinicoradiological findings, 4 cycles of chemotherapy consisting of adriamycin, ifosamide, and dacarbazine was given in consultation with the oncology team. Post chemotherapy, CECT chest showed few densities in both the lung fields and bone scan showed evidence of hot spots in the proximal femur with no other skeletal metastasis. FDG/PET revealed only focal FDG uptake in the soft tissue of proximal femur, which decreased on repeat scan at 2 months.

Finally, the patient was taken up for limb salvage surgery with total femur endoprosthetic reconstruction. Care was taken to include previous surgical scars in the incision [Figure 2]a. The femur was dissected out in total, along with previous scar and nail [Figure 2]b. The stability and movements were checked with trial total femur endoprosthesis with bipolar hip and rotating hinge knee. Finally, GMRS (Global Modular Replacement System, stryker) implants consisting of 70 mm proximal femur, 90 mm connecting piece, 210 mm stem extension piece, and 65 mm distal femur were inserted [Figure 2]c. Hip joint reduced, and its stability assessed. The edges of the remnants of the capsule were closed around the head of the prosthesis with purse string sutures using mersilene tape. Hip abductors were reattached to holes provided in the greater trochanteric region of the endoprosthesis from above and vastus lateralis was reattached to the same from below [Figure 2]d. Hip extensors were attached to posterior aspect of these muscle sleeves. Rectus femoris was reattached to soft tissue sheath in front of abductors. Other soft tissues were approximated over the implant, and closure was done in layers. The total femur specimen dissected out was sent for histopathological examination.
Figure 2: (a) Marking of the planned incision before surgery. (b) Intraoperative image showing the resection of the entire femur with the adjacent involved soft tissue and skin involving previous operation tracts (blue arrow). (c) Intraoperative image showing the total femur prosthesis in situ. (d) Intraoperative image showing an approximation of hamstrings, rectus and abductors over the prosthesis

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Postoperatively, there were no distal neurovascular deficits. Postoperative radiograph showed implant in good alignment [Figure 3]a. Thromboprophylaxis with low molecular weight heparin continued for 3 weeks. Hip and Knee ROM exercises were started immediately while ambulation started at 4 weeks with support. Histopathology confirmed malignant peripheral nerve sheath tumor with rhabdomyoblastic differentiation. The cut margins were tumor-free and the patient underwent radiotherapy postoperatively. At 3 months follow-up, knee range of motion was 0–50° which improved subsequently with aggressive physiotherapy [Figure 3]b. At 5-year follow-up, the individual is independent; mobile unsupported and has got good hip and knee range of motion.
Figure 3: (a) Immediate postoperative radiograph showing the prosthesis in acceptable position and alignment (anteroposterior view). (b) Immediate postoperative picture of the patient showing assisted ambulation

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Case summary 2

A 42-year-old male, an army veteran with right-hand dominance presented to an outpatient department of a tertiary care teaching hospital with complaints of insidious onset, gradually progressing pain over the right arm of 2-year duration. He also noticed diffuse swelling in the middle third of arm for the last 1 year. The individual took on and off symptomatic treatment from a local hospital for the entire duration. However, as the pain and swelling worsened, he sought medical care at our center for definitive management. There was no recent or remote history of trauma, fever, breathlessness, or any other symptoms. General physical examination was unremarkable. Local examination of the right arm revealed a 15 cm × 10 cm × 20 cm swelling encircling the entire arm with dilated superficial veins. The swelling was diffuse, tender, immobile, and firm in consistency. The skin was stretched, shiny and was freely mobile from the underlying swelling. The shoulder range of motion and elbow flexion were globally restricted with no distal neurovascular deficits.

Anteroposterior and lateral radiographs of the right arm showed a permeative lesion in the middle third of humeral diaphysis extending into soft tissues with poorly defined margins. Fluffy calcification, erosion, and thickening of cortex with no periosteal new bone formation was also noted [Figure 4]a. CT of right arm showed an expansile 12 cm × 13.5 cm × 22.3 cm lobulated hypodense mass over the middle third of humeral shaft with internal calcifications and cortical thickening. Magnetic resonance imaging revealed a large well defined lobulated mass lesion epicentered over the mid humeral diaphysis extending to about 20 cm, involving soft tissue of all compartments [Figure 4]b. Proximal marrow involved till the anatomical neck of the humerus just 3.1 cm away from the glenohumeral joint, and the distal involvement was till the supracondylar level of humerus, 2.2 cm away from the elbow joint. Neurovascular bundles were stretched and displaced with no obvious infiltration. Technetium 99 bone scan showed an increase in tracer concentration in the middle third shaft of the humerus and its adjoining soft tissue and a small focus in the proximal humerus [Figure 4]c. There was no other hot focus seen elsewhere in the body. PET CT scan reported as weakly metabolic and necrotic mixed soft-tissue mass in arm involving all compartments with no other FDG avid distant lesion. USG KUB, CXR PA VIEW, NCCT CHEST were all normal. A provisional diagnosis of a malignant lesion was made based on the clinicoradilogical findings and the imaging. Later on, the patient was subjected for tru-cut biopsy, which revealed a periostesteal chondrosarcoma.
Figure 4: (a) Anteroposterior and lateral radiograph of the arm with both shoulder and elbow joint showing the lesion in the mid diaphysis of humerus. (b) Magnetic resonance imaging of the arm with both shoulder and elbow joint showing the lesion in the mid diaphysis of humerus and extent of involvement of the marrow (blue arrow). (c) Technetium 99 bone scan of the whole body showing the increased tracer uptake in the lesion at the middle third of arm and the proximal humerus (blue arrow)

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The tumor was a slow-growing, malignant lesion involving almost the entire humerus with sparing of adjacent shoulder and elbow joints and the neurovascular bundle. There were no metastasis, and all other relevant investigations were also normal. The most important technical aspect that decides overall survival is the attainment of a wide surgical margin regardless of the procedure.[5] Salvage of the native shoulder and elbow joints were not possible due to the extent of marrow involvement, both proximally and distally. Hence, definitive treatment was discussed in the tumor board of the hospital, and approval taken for radical resection of the tumor followed by a total humerus endoprosthetic reconstruction. Neoadjuvant chemotherapy was given as per the oncosurgical protocol of the hospital. Subsequently, the patient underwent en masse radical excision of the tumor along with the entire humerus [Figure 5]a. Care was taken to excise the biopsy track during the incision [Figure 5]b. All neurovascular structures isolated and protected throughout the surgery. The entire humerus and the involved soft tissues along with adequate tumor margins were excised in total, and the reconstruction done with RESTOR ADLER Modular Total Humerus Prosthesis (Titanium) [Figure 5]c. Remnants of the shoulder capsule were secured to the prosthetic head using a composite mesh. Rotator cuff, tendons of latissimus dorsi, pectoralis major, teres major, and deltoid were reattached to the mesh wrapped prosthesis on their respective locations and the elbow joint reconstructed in a similar fashion. Immediate postoperative period was uneventful and early rehabilitation of the extremity was encouraged. Postoperative radiograph revealed a well-aligned total humerus endoprosthesis with congruous shoulder and elbow joints [Figure 6]a. Adjuvant radiotherapy was given postoperatively.
Figure 5: (a) Intraoperative image showing the resection of the entire humerus with the adjacent involved soft tissue and skin. (b) Intraoperative image showing the resected specimen of the entire humerus with involved skin and soft tissues, including the previous biopsy tract (blue arrow). (c) Restor Adler total femoral prosthesis with elbow joint components

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Figure 6: (a) Immediate postoperative radiograph showing the prosthesis in acceptable position and alignment (anteroposterior view). (b) Postoperative clinical picture showing well-healed surgical scar with postradiotherapy lymphedema and scarring at 3 months

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At the end of 3 months, he had a good recovery of function with a reasonable range of movements at both shoulder and elbow. The biopsy of the specimen revealed a grade 2, moderately differentiated, low-grade conventional chondrosacoma with TNM Staging of T2N0M0. All the bone and soft tissue margins were tumor-free. At 1-year follow-up, he developed lymphedema of the right upper limb following radiotherapy [Figure 6]b. However, he had glenohumeral abduction of 30°, flexion of 90°, internal rotation of 45° and external rotation of 10°. He had good grip strength with a mild flexor weakness of the index finger. Most importantly, he resumed his job as a clerk at the central railway's department at 1 year. At 5-year follow-up, he is active with minimal functional restrictions, and there is no local recurrence or evidence of any metastasis.


  Discussion Top


Extensive malignant femoral lesions sometimes may require hindquarter amputations while humeral lesions a forequarter amputation.[6],[7] With a better understanding of the tumor morphology, imaging, and surgical techniques coupled with new innovations in prosthetic designs, limb salvage surgery has become a safe and feasible option in extensive tumors of these long bones.[8],[9],[10],[11],[12],[13],[14] With improvements in chemotherapy, Limb salvage has replaced amputation as the optimum treatment for malignant bone tumors of extremities, without adversely affecting survival.[15],[16] It is more cost-effective than amputation and is usually preferred by the patients as it provides improved functional outcomes.[2],[3] Although limb salvage offers better functional outcomes in the early postoperative period, it should not compromise survival rates.[17]

The total femoral replacement may not be required in all cases, but this procedure is necessary in cases with skip metastases or with extensive diaphyseal involvement. There are only a few survival studies on total femoral replacements in malignancy, and certain small series have documented a high incidence of complications related to prosthesis and malignancy itself. Ahlmann et al.[18] reported 100% survival of the implant at 10 years in seven patients he followed up. Ward et al.[19] reviewed 21 patients with a mean follow-up of 31 months. They used both fixed-length custom implants and modular prostheses (Howmedica, Rutherford, New Jersey, and Link America, Denville, New Jersey). The functional results reported as per the authors were good in seven, fair in nine and poor in three. Morris et al.[20] reviewed seven patients who had total resection of the femur with replacement by the Kotz Modular Femur-Tibia Reconstruction system (HowmedicaOsteonics) and described the results as excellent or good, although the follow-up was only of 23 months on an average. In our case, after the completion of neoadjuvant chemotherapy, we decided in favor of limb salvage due to its advantages of better gait and the preservation of one's own limb. The important aspect was to find a balance between adequate tumor removal and preservation of remaining soft tissues to provide stability and function. It was achieved by en bloc resection of the entire femur with the nail in situ followed by reconstruction of the femur using total femur endoprosthesis, which contained a bipolar hip joint and rotating hinge knee joint. The main cause of failure reported in total femoral replacements is the instability of the hip. The other causes being dislocation, local recurrence, and infection. Bickels et al.[21] reported only one dislocation in 57 patients. As per Ward et al.[22] the margin of resection, poor response to chemotherapy, the intravascular extension of the tumor accounted for local recurrence. As always, infection is a major problem and may be related to inadequate soft-tissue cover and the immunosuppressive effect of chemotherapy and radiotherapy. We did not have any of these complications in our case. The patient has been followed up for >5 years now. He has no recurrence and is comfortably ambulant unassisted.

The common presentation of progressive pain in the hip and limp with an osteolytic lesion with well-defined margins usually suggests a benign lesion. Such cases have been commonly managed with curettage and prophylactic nailing to prevent impending pathological fractures. In this case, the clinical presentation and imaging modalities suggested a benign lesion with a diagnosis of monostotic fibrous dysplasia with other differentials as chondromyxoid fibroma, GCT, ABC. However, in hindsight, a rare diagnosis of high-grade spindle cell sarcoma was provided by histopathological examination, which is a bit uncommon and not routinely reported in the literature. The final diagnosis of the specimen by histopathology was malignant peripheral nerve sheath tumor with rhabdomyoblastic differentiation, which is rare, and its occurrence in the proximal femur was very unusual. This benign presentation of a high-grade malignant nerve sheath tumor with rhabdomyoblastic differentiation in the proximal femur is unique and makes this case report interesting. There is also a definite paucity of publication of such instances/cases in the literature. Total femur replacement is not done routinely due to very few indications and can only be done when these limited indications present at a center where the expertise for carrying out such a procedure exists and appropriate implants are available. Most importantly, the patient should be willing for the total femoral replacement, which is an expensive and infrequent surgery. This index case had all of the above-mentioned rarities but still resulted in a satisfactory outcome, which was heartening for both the patient and the surgical team.

Total humerus reconstruction with its adjacent joints remains a challenge following the tumor resection. It gives a patient feeling of his own normal-looking extremity with the satisfactory elbow and hand function with no compromise on tumor clearance or recurrence rates.[23],[24],[25],[26] This also alleviates the problems of union reported sometimes with allograft reconstruction when the patient is subjected to radiotherapy or chemotherapy postoperatively. In addition, it gives immediate stability, which helps in early and faster rehabilitation. Reconstruction of shoulder joint function is highly demanding in extensive resections. All intrinsic stabilizers, along with rotator cuff tendons, become incompetent, resulting in loss of active motion like abduction and elevation of the extremity.[23],[27] Tendons are generally attached to the holes or surface of the prosthesis, but their integration with the prosthesis interface is generally inadequate. Hence a composite mesh was used in our case for an additional anchorage of the tendons to the prosthesis to increase the stability. When the patient tries to abduct or elevate the extremity, the humeral head migrates superiorly due to the unopposed contraction of the deltoid as the stabilization of cuff tendons is suboptimal. This change in biomechanics results in reduced active antigravity movements due to the loss of fulcrum. Hence, long and intensive shoulder rehabilitation is required to achieve a useful active range of motion. Many times people compensate by using their scapulothoracic motion for activities of daily living. Subacromial impingement due to this superior migration of head is generally well tolerated; however, it requires intervention if becomes too symptomatic. However, patients will generally be satisfied despite this shortcoming as their motion at elbow and hand restores their function to an acceptable level. In our case, the individual was satisfied with the outcome as he resumed his occupation and started earning his livelihood.

There are very little data on total humeral endoprosthetic reconstruction for such primary tumors of the humerus. Natarajan et al. reported well to excellent cumulative survival rates of 90.9% and 77.9% at 1 and 5 years, respectively.[23] The average MSTS score was around 80% in their series of 11 patients with malignant tumors of the humerus who underwent radical excision and reconstruction with custom-made total humerus endoprosthesis. They also described minimal active shoulder motion but relatively good elbow and hand function, which were evident in our case too. Funovics et al. reported a final MSTS score of 70% and 3 cases of deep infection in a series of 11 cases reconstructed with total humeral replacement following tumor excision.[24] To augment the anchorage of rotator cuff tendons on the humeral head, synthetic nylon or Prolene mesh can be wrapped around the humeral head, which results in better range of motion and also reduces chances of dislocation.[28] We used a composite mesh which added to the stability of the joint and reduced the chances of dislocation. Presently, at >5 years' follow-up, the individual does not have any history of dislocation and is able to ride a two-wheeler. Refinements in soft tissue anchoring techniques on metal surfaces have led to improved functional outcomes.

Though chondrosarcomas are the second most common malignancy of bone,[29] there is a lot of doubt and dilemma on the reliability of pathologic and radiographic grading.[30] it poses a lot of diagnostic and therapeutic challenges in the diagnosis and treatment of these lesions. Grade 1 chondrosarcomas are considered as low-grade tumors, whereas grade 2 and 3 tumors, a high grade. However, there exists a high disparity in the biologic behavior of these tumors between the grades. Moreover, biopsy is unreliable in cases of intramedullary chondrosarcomas, hence radiographic characteristics of cortical thickening, expansion, destruction, and soft-tissue invasion are considered to be an indication of aggressive, high-grade variety.[30],[31] In addition, there is a lot of debate on adequate surgical margins for chondrosarcomas, but majority of the studies indicate wide resection for grades 2 and 3, which are high-grade tumors.[32] Hence, keeping these all factors in mind, we went in a for a total humerus resection followed by endoprosthetic reconstruction in our case, which resulted in a satisfactory outcome. This case study gave a lot of insight about the unpredictability of these tumors in its presentation, biologic behavior, grading and treatment. So, with a good assessment of history, clinical signs and radiologic correlation, a proper diagnosis can be made, and limb salvage becomes a possibility in select cases to get an excellent functional outcome without compromising the long-time survival.


  Conclusion Top


The first case study presents custom mega prosthesis (GMRS) as a reliable method of reconstruction following resection of extensive malignant tumors of the femur. It also reiterates that irrespective of the clinical and radiological presentation, which may suggest a benign lesion, a histopathological diagnosis must always be obtained before any intervention. The second case report highlights the wide variations of tumor behavior and total humeral endoprosthetic replacement as an effective and safe surgical option following a total excision of the humerus if adequate tumor clearance can be achieved. It restores a useful and acceptable upper extremity function with fewer complications. The advantages of these procedures include early functional recovery, relatively low complication rate, and a high level of emotional acceptance. However, the success of this limb salvage procedure depends on careful patient selection, meticulous surgical technique, and good prosthetic design when performed at a specialist center.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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