Journal of Bone and Soft Tissue Tumors – Unique Status

Vol 2 | Issue 2 | May – Apr 2016 | page:1-2 | Dr Yogesh Panchwagh & Dr Ashok Shyam.


Author: Dr Yogesh Panchwagh [1] & Dr Ashok Shyam [1,2].

[1]Orthopaedic Oncology Clinic, Pune, India.
[2] Indian Orthopaedic Research Group, Thane, India
[3] Sancheti Institute for Orthopaedics &Rehabilitation, Pune, India.

Address of Correspondence
Dr. Yogesh Panchwagh.
Orthopaedic Oncology Clinic, 101, Vasant plot 29, Bharat Kunj Society -2, Erandwana, Pune – 38, India.
Email: drpanchwagh@gmail.com


Journal of Bone and Soft Tissue Tumor (JBJST) enjoys a special status in being one of the unique journals exclusively dedicated to bone and soft tissue tumors. The clinical expertise and technological development has been very rapid in this field and JBST already had a demand among the clinicians involved in taking care of musculoskeletal Oncology. JBST has successfully filled this vacuum that existed and has received great support from musculoskeletal tumor surgeons and clinicians. The platform provided by JBST has been used worldwide to access articles and also to submit original research. There are other factors that further add to its uniqueness. One of the most unique point is that this is a journal that is initiated by clinicians. JBST was conceived and initiated by people who were directly involved in care of musculoskeletal tumor patients. They perceived the need of such a journal and were instrumental in moulding it in its current shape. Another unique point of JBST is that it is not a pure research journal but also a tool to educate the young trainees and practitioners. JBST has a dedicated symposium in each issue which provides a comprehensive overview of the subject along with recent updates. This is very helpful for students, trainees and practitioners of the subject. These symposium articles are written by carefully solicited authors that have years of practical experience which add to the flavour of the article. The authors are requested to add the practical tips and cases to these symposium reviews to make it much more clinically relevant rather than simply publish a theoretical review of literature. Special attention is given to students in JBST and a students corner is published in every issue which is a brief overview article on a single bone tumor. This is specially created with keeping students in mind and is co-created with help of a trainee or a student. This has received excellent response as far as student readership is concerned and we thank Dr Ashish Gulia for initiating and supporting this initiative. Oncomedia is another unique part of JBST where recent conferences, upcoming conferences and new updates regarding bone and soft tissue tumors are listed. Videos and other academic materials are also included in this section. It’s a very unique source of information to our readers and is presented in a very reader friendly manner.This particular issue is also unique as it features and is dedicated to one of the most prestigious orthopaedic oncology unit in India from Tata Memorial Hospital (TMH) in Mumbai. The guest editorial is been contributed by the TMH team and it traces the journey of the hospital from its inception till today. JBST plans to continue this feature where such units, who have contributed significantly to growth of musculoskeletal oncology will be featured. We intend to include this a regular feature in JBST. Another feature that we really wish to include is academic interview, featuring individual personalities in the field of musculoskeletal oncology. Other journals from orthopaedic research group like trauma international and International Journal of Paediatric Orthopaedics have been regularly publishing such academic interviews. Hopefully JBJST will start this feature by next year. The JBST team is always looking for making the journal better with the aim to provide the best content to its reader which is presented in the most accessible and easy format. Being focussed on the subject of bone and soft tissue tumours and created and run by focussed and expert team has helped the journal achieve a unique status already and with help of the entire fraternity it is sure to grow further. If you have any suggestions for the editorial team, please feel free to write to us.
Dr Yogesh PachwaghDr Ashok Shyam


How to Cite this article: Panchwagh Y, Shyam AK. Journal of Bone and Soft Tissue Tumors – Unique Status. Journal of Bone and Soft Tissue Tumors May-Aug 2016; 2(2):1-2.


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Osteofibrous Dysplasia – an update

Volume 2 | Issue 2 | May-Aug 2016 | Page 23-25 | Pankaj Panda1,  Ashish Gulia1


Authors: Pankaj Panda[1],  Ashish Gulia[1] 

[1]Orthopedic Oncology Services, Department of Surgical Oncology, Tata Memorial Hospital, Mumbai

Address of Correspondence
Dr Ashish Gulia, MS (Ortho), Mch (Surgical Oncology)
Associate Professor, Orthopedic Oncology, Dept. of Surgical Oncology, Tata Memorial Hospital, Mumbai – 400012, India.
Email: aashishgulia@gmail.com


Abstract

Introduction: Osteofibrous dysplasia (OFD) is a rare, benign, self-limiting, fibro-osseous lesion occurring in long bones especially of lower limbs. Patients typically presents with painless swelling with or without anterior bowing of tibia .The diagnosis can be confirmed by peculiar radiological feature of well defined intracortical lytic lesion with variable degree of osteolysis and osteosclerosis. Admantinoma is close differential of this lesion . Most cases regress spontaneously by puberty , surgical intervention is required only for progressive lesions or in case of pathological fracture .
Keywords: Osteofibrous Dysplasia , Management


Introduction
Osteofibrous dysplasia (OFD) is a rare, benign, self-limiting, fibro-osseous lesion occurring in long bones especially of lower limbs. It is also called as Kempson-Campanacci lesion or cortical fibrous dysplasia. The prominence of the osteoblasts led Kempson in 1966 to describe the entity as ossifying fibroma of the long bones [2]. In 1976, Campanacci, gave the term “osteofibrous dysplasia of the tibia and fibula” in reference to its histological features, developmental origin and anatomic location [1].
Etiopathogenesis – Exact etiology is not known. A few of the cases are known to have occurred in families. It has also been reported that OFD may act as a precursor of adamantinoma which is supported by occurrence of OFD like adamantinomas. The evidence for this is limited and most of the cases are considered to be arising spontaneously.
Incidence – OFD is a rare benign self-limiting tumor, which accounts for about 0.2% of all primary bone tumors [3]. These lesions are mainly seen in the first two decades of life. It is very uncommon after skeletal maturity with any gender predilection [4].
Site – It is invariably a disorder of the tibia and fibula. The lesion usually has its epicenter in anterior cortex of tibia. Tibial mid-diaphysis and proximal metaphysis are affected the most. Ipsilateral or contra lateral fibula may be involved. Even though most of the lesions are confined to a limited portion of the bone a few may grow rapidly and involve almost entire bone. Isolated fibular involvement is rare. Forearm bones (radius and ulna) are other uncommon sites of affection [5].
Clinical featuresThe typical presentation is painless swelling with or without anterior bowing of the tibia. Pain is only present in about one third of the cases and is usually due to pathological fracture. About a third of the cases are detected incidentally [6].

Radiological Features:
Radiograph – The lesion appears as a well defined intracortical lytic lesion, with variable degree of osteolysis and osteosclerosis located in the anterior cortex of the tibia. These lesions may present as a single focus or multiple elongated foci interspersed with reactive bone. The overlying cortical shell presents itself in a wavy pattern giving it a “saw tooth appearance”. Most of the lesions are associated with anterior tibial bowing and buttress type of benign periosteal reaction. Aggressive lesions may involve entire diaphysis and metaphysic and may have associated pathological fractures [7, 8].
Computed Tomography – It is helpful in assessing exact extent of the lesion, cortical involvement, periosteal reaction and pathological fractures and acts as an adjuvant to MRI in the overall assessment of the lesion.
Magnetic resonance imaging – MRI helps in delineating the cortical based lesion and to assess its medullary or soft tissue extension. The lesion demonstrates mixed signals on T1 and high intensity lesions on T2 weighted images. MRI is helpful in surgical planning and differentiating OFD from adamantinoma [9].
Pathology:
On gross examination, a typical specimen appears as a whitish or yellowish solid lesion with surrounding gritty bony architecture. The cortex may be expanded and thinned out deficient at places with intact periosteum. Lesions may show medullary extension, which is usually demarcated by a sclerotic rim [10].
Microscopically, OFD demonstrates a zonal architecture with loose fibrous tissue containing spicules of woven bone in the centre which is lined by a layer of lamellar bone lined by prominent osteoblasts at the periphery. This shows a progressive maturation of the bone trabeculae from a central zone of delicate trabecular bone in a vascular fibrous stroma, to an outer zone of lamellar bone. The fibrous component in most cases contains cells which react positively for pan-cytokeratin. Desmosomes, tonofilaments, and microfilaments are seen on electron microscopy [2, 11].
Differential Diagnosis:
Several tumor and tumor like lesions can mimic Osteofibrous dysplasisas on radiographs [12]. The differential diagnoses are that of a cortical, lytic, expansile lesion. Adamantinoma is the most closest differential diagnosis as both lesions are very similar clinico- radiologically and even on histopathology. Adamantinomas are more aggressive lesions and may lead to local and distant recurrences. These commonly involve the medullary cavity, but there is usually cortical infiltration, break and soft tissue component. Other differentials include Fibrous dysplasia, Nonossifying fibroma, Aneurysmal bone cyst, Chondromyxoid fibroma, Langerhans cell histiocytosis, Osteomyelitis and Hemangioendothelioma [13]. A thorough clinico-pathological correlation substantiated with characteristic radiological findings is very essential for a definitive diagnosis of OFD.
Treatment:
According to the case series on OFDs from the Rizzoli Institute in Milan and the Mayo Clinic, these lesions, owing to their benign nature, seldom progress during childhood and undergo spontaneous regression at puberty, thus can be carefully observed with serial plain radiographs and clinical evaluation at regular intervals. If associated with significant or progressive bowing then conservative treatment in the form of bracing may be helpful to minimize deformity and prevent pathological fracture [5, 14].
Surgical intervention is mainly required in extensive cases with progressive deformity or for pathologic fracture. Extraperiosteal “shark-bite” excision is the most widely considered surgical option for OFDs. The resultant defects may be reconstructed with auto or allo-strut grafts. Other surgical interventions may include curettage bone grafting and internal fixation after correction of deformity. [9].
Prognosis – OFD has a very good prognosis. Most of the lesion even though they grow in first decade of life get stablised during the second decade and heal by spontaneous resolution. Deformities may persist for a longer time and may remodel slowly. Aggressive lesions may have severe deformity or pathological fracture, which usually heal well with surgical intervention. Excisions are mostly curative. A few lesions may progress to OFD like adamantinoma or adamantinoma and require aggressive treatment accordingly [3, 6].


References

1. Campanacci M, Olmi R. Ossifying fibroma of the long bones. A light and electron microscopic study. Arch Pathol. 1966 Sep;82(3):218-33
2. Kempson RL.Ossifying fibroma of the long bones. A light and electron microscopic study. Arch Pathol. 1966 Sep;82(3):218-33.
3. Most MJ, Sim FH, Inwards CY. Osteofibrous dysplasia and adamantinoma. J Am Acad Orthop Surg 2010;18:358-66.
4. Hahn SB, Kim SH, Cho NH, Choi CJ, Kim BS, Kang HJ. Treatment of osteofibrous dysplasia and associated lesions. Yonsei Med J. 2007 Jun 30;48(3):502-10.
5. Park YK, Unni KK, McLeod RA, Pritchard DJ. Osteofibrous dysplasia: clinicopathologic study of 80 cases. Hum Pathol. 1993 Dec;24(12):1339-47.
6. Gleason BC, Liegl-Atzwanger B, Kozakewich HP, Connolly S, Gebhardt MC, Fletcher JA, Perez-Atayde AR. Osteofibrous dysplasia and adamantinoma in children and adolescents: a clinicopathologic reappraisal. Am J Surg Pathol. 2008 Mar;32(3):363-76..
7. Levine SM, Lambiase RE, Petchprapa CN. Cortical lesions of the tibia: characteristic appearances at conventional radiography. RadioGraphics 2003;23:157-77.
8. Greenspan A. Malignant bone tumors II. In: Orthopedic imaging: a practical approach. 5th ed. Philadelphia, USA: Lippincott Williams & Wilkins; 2011. p. 754.
9. Khanna M, Delaney D, Tirabosco R, Saifuddin A. Osteofibrous dysplasia, osteofibrous dysplasia-like adamantinoma and adamantinoma: correlation of radiological imaging features with surgical histology and assessment of the use of radiology in contributing to needle biopsy diagnosis. Skeletal Radiol 2008;37:1077-1084
10. Fitzpatrick KA, Taljanovic MS, Speer DP, Graham AR, Jacobson JA, Barnes GR, HunterTB.Imaging findings of fibrous dysplasia with histopathologic and intraoperative correlation. AJR Am J Roentgenol. 2004 Jun;182(6):1389-98.
11. Kahn L. Adamantinoma, osteofibrous dysplasia and differentiated adamantinoma. Skeletal Radiol 2003;32:245-58.
12. Levine SM, Lambiase RE, Petchprapa CN. Cortical lesions of the tibia:characteristic appearances at conventional radiography. RadioGraphics 2003;23:157-77.
13. Izquierdo FM, Ramos LR, Sánchez-Herráez S, Hernández T, de Alava E, Hazelbag HM. Dedifferentiated classic adamantinoma of the tibia: a report of a case with eventual complete revertant mesenchymal phenotype. Am J Surg Pathol. 2010 Sep;34(9):1388-92.
14. Campanacci M, Laus M. Osteofibrous dysplasia of the tibia and fibula. J Bone Jt Surg Am 1981;69(A):367-75.


How to Cite this article:1. Panda P, Gulia A. Osteofibrous Dysplasia – an update. Journal of  Bone and Soft Tissue Tumors May- Aug 2016;2(2):23-25 .

Dr. Paramanandam V

Dr. Paramanandam V

Dr. Anuradha A Daptardar

Dr. Anuradha A Daptardar

Dr. Ashish Gulia

Dr. Ashish Gulia


(Abstract    Full Text HTML)      (Download PDF)


 


Rehabilitation following Limb-Salvage Surgery in Sarcoma

Volume 2 | Issue 2 | May-Aug 2016 | Page 19-22 |  Vincent S  Paramanandam1, Anuradha A Daptardar1, Ashish Gulia2


Authors:  Vincent S  Paramanandam[1], Anuradha A Daptardar[1], Ashish Gulia[2]

[1]Physiotherapy Department, Tata Memorial Hospital, Mumbai
[2]Orthopedic Oncology Services, Department of Surgical Oncology, Tata Memorial Hospital, Mumbai.

Address of Correspondence
Dr. Paramanandam V
Technichal Officer C, Physiotherapy Department, Tata Memorial Hospital, Mumbai
Email: vinsu24@gmail.com


Abstract

Introduction: Limb salvage after tumor resection has become a norm in today’s era. There are number of biological and non biological reconstruction options available for the reconstruction of these bone defects. The success story of these surgical procedures is mainly based on their excellent functional outcome. Post surgical rehabilitation plays an important role in achieving optimal functional outcome and good quality of life. The rehabilitation protocol following limb salvage surgery is complex and it differs with type of reconstruction procedure. Present articles discusses in detail the various rehabilitation protocols required to achieve above goals.

Keywords:


Introduction
Until 1970, amputation was the primary surgical treatment offered to bone and soft tissue sarcomas. However, from that time the treatment options have evolved dramatically and now approximately 90% of these cases undergo Limb Salvage Surgery (LSS)[1]. LSS has become the main line of treatment option for bone and soft tissue sarcomas along with adjuvant and/or neo adjuvant treatment modalities (Chemotherapy/ radiotherapy). The overall survival rate has been estimated as 55%-65%, based on the age of diagnosis, and it is considered to be comparable to that of amputation.
LSS is considered to be less invasive, provides better function and quality of life than amputation [2]. Moreover, it has been proposed that patients’ acceptability of LSS is high in view of the fact that it restores the body image better than amputation[3]. Nevertheless, LSS, unlike amputation, is associated with more peri-operative complications, prolonged hospital stay and requires repeated surgeries due to various reasons such as infection and prosthetic failure. LSS demands high surgical skills, whereas, amputation is a simple surgical procedure. Additionally, recent progress in prosthetic limbs, for example microprocessor based joints and endo-skeletal prosthetic reconstructions, have improved the functional outcome and cosmetic outlook following amputation [4].
A systematic review conducted by Bekkering et al [5] reported that the quality of life outcome from current available evidence is inconclusive in supporting LSS or amputation. Another recent systematic review and meta-analysis concluded that both surgical procedures provides similar functional recovery and quality of life [6].Despite the fact that early physical rehabilitation is the key to achieve good functional outcome and quality of life after LSS, rehabilitation techniques following LSS is largely neither tested nor documented in detail [7]. Lack of adequate early rehabilitation measures following LSS could be one of the rationales for conflicting interests reported by various studies examining the quality of life in LSS vs amputation.Hence, we have attempted to summarise basic principles and site specific considerations one must utilise to develop individual case specific rehabilitation protocol.

Common rehabilitation principles in LSS
In a recent paper, Shehadeh et al. [7] attempted to standardize the rehabilitation protocol for LSS following high grade bone and soft tissue sarcomas. They reported that following a standardized rehabilitation protocol produced improved functional outcome in group of 59 patients with LSS. Their conclusion, however, is based on small observational study with heterogeneous population who received different type of LSS for different anatomical sites. Following set protocol in LSS, unlike general orthopaedic procedure, will be counterproductive. In general orthopaedic procedures, more or less,
specific anatomical structures are involved with minimal damage to the bone, joint and
soft tissue structures. In contrast, in LSS
following sarcomas, these structures are
extensively resected and may not be
identical between two individuals
undergoing similar procedures for a particular site. For example, resection length for distal end of femur osteogenic sarcoma may depend on the extent of disease in two individuals [4].
Following are common rehabilitation prospectives that need to be considered to formulate comprehensive rehabilitation protocol for LSS.
Bone and joint reconstructions: Stability and mobility following LSS largely depends on the bone and joint structure loss and the type of reconstruction. For example, megaprosthetic distal femur replacement with cementing will allow the patient to be ambulated full weight bearing (FWB), whereas, if it is a bone graft , like in most of biological reconstructions, weight bearing needs to be delayed till the osteosynthesis is confirmed by radiographical evaluation.
Neuromuscular loss: Oncology resection demands large resections, which will also include a part of uninvolved soft tissue cover as surgical margin. Large resection may require additional rotational or free flaps for soft tissue coverage. In addition, nearby neuro-vascular bundle may need to be excised or repaired, hence, complete evaluation of neuro-motor loss would be necessary to plan the dynamic strength training and external support requirements.
Skin involvement: Donor sites of free flaps often receive split thickness skin graft which may hinder the early mobilisation of nearby joint. Moreover, scar development following open biopsy and LSS may need special attention from the rehabilitation team to prevent any future functional loss.
External supports: Temporary or permanent external support in the form of static or dynamic splinting may be required to provide support to the limb. To exemplify, prophylactic use of abduction brace along with derotation boot to prevent hip dislocation following proximal femur replacement and dynamic cock-up splint for radial nerve palsy needs to be the integral part of rehabilitation service.
Oncology treatments: Deranged blood count often hinders with the intensity of rehabilitation; hence, it prolongs the overall rehabilitation(8). Radiation induced fibrosis could cause severe restrictions in the joint range of motion. Thus, rehabilitation professionals must plan around the chemotherapy cycles and add prophylactic measures to prevent any impending radiation induced joint and soft tissue dysfunction.
Multidisciplinary approach: Limb salvage surgery is complex and demands close concordance in treatment specific outcomes between various health professional working in the rehabilitation team. This team may comprise team of surgeons, medical oncologists, radiation therapists, nurses, physiotherapists, occupational therapists, prosthetics orthotics and medical social workers. Prehabilitation, rehabilitation even before starting the primary cancer therapy and surgery, such as crutch muscles strengthening, would be greatly beneficial in post-treatment functional outcome. Although in the field of LSS evidence of prehabilitation is lacking, there are considerable evidence to show beneficial effects in overall rehabilitation following cancer therapies [9–11].
Rehabilitation prescriptions and follow-up: Rehabilitation protocol for LSS must be tailor made considering the general principles and site specific modification, hence specific and also progressive. However, some negative effects of adjuvant therapies, such as the deranged blood counts and infection, may alter the course of rehabilitation process. Thus, frequent follow-up and close monitoring may be required during adjuvant therapy till they are functionally independent.
Rehabilitation consideration for specific sites
Site specific rehabilitation principles following LSS have been presented below for few common sites.
Mega prosthetic replacement for distal femoral resection:
Distal femur is the commonest site for primary high grade sarcoma and giant cell tumors. Overall strengthening other than the affected site, in all possibility, should begin preoperatively. Limb elevation and ankle toe movement should be encouraged from post operative day one to prevent deep vein thrombosis. Cemented and semiconstrained(allows rotations and flexion/extension) knee joint endoprosthetic replacement permits early joint mobilization and FWB walking. Unlike other centres [4,7], in our centre knee joint mobilisation starts from day one with the help of continuous passive motion units (Figure1) and active assisted methods within the pain tolerance level unless tight suturing. Close communication between the surgical team and the rehabilitation team helps in personalising the rehabilitation protocol as per the patients’ requirements. With adequate pain relief through appropriate medical management, active exercises could be started from day one to three. Full weight bearing walking could be started from day one initially with walker and later without any support if patient could effectively extend the knee “locking the knee”. Prior to ambulation, one leg standing and spot marching must be encouraged with appropriate support. After acute inflammation subsides slow progressive muscle strengthening exercises must be encouraged with the goal of achieving 900knee flexion, complete knee extension and muscle strength equivalent to the contra lateral lower limb by end of three months. Active passive motion devices, such as the one shown in Figure 2, may help in joint mobilisation and strengthening. Summary of rehabilitation protocol is tabulated in Table 1.

Figure 1: Continuous passive motion

Figure 1: Continuous passive motion

Figure 2: Active passive motion unit

Figure 2: Active passive motion unit

Table 1: Rehabilitation protocol following LSS

Table 1: Rehabilitation protocol following LSS

Mega prosthetic replacement for proximal tibial resection:
Proximal tibia and knee joint is the second most common site for primary high grade sarcoma and giant cell tumors. It is indeed a challenging location for rehabilitation in view of the fact that the extensor mechanism have to be reconstructed in these cases [4]. In most of the cases a gastrocnemius flap is done to provide a dynamic anchorage and direct anchorage to prosthesis provide a static attachment to help reattaching extensor mechanism to proximal tibial prosthesis. Protecting the extensor mechanism reconstruction till it attaches through fibrosis along with the surrounding soft tissue structures are crucial to prevent quadriceps lag. Hence, knee bending and quadriceps strengthening will be delayed for six weeks until then the knee is protected with the help of long knee brace. Re-attached gastrocnemius flap could lead to protective muscle spasm of plantar flexors and if not mobilised early it may create plantar flexors contracture. Thus, achieving/maintaining dorsi flexion of the ankle joint in the early post operative period is crucial for appropriate weight bearing.

Table 2: Rehabilitation protocol following Mega prosthetic replacement for proximal tibial resection

Table 2: Rehabilitation protocol following Mega prosthetic replacement for proximal tibial resection

Mobilisation of knee joint and quadriceps strengthening could be started after six weeks; however, therapist must consider that immobilisation of the knee joint in long knee brace leads to severe restriction of patella mobility. Unless adequate patella mobility is achieved, especially the medial lateral movement, knee flexion exercises could prevent smooth gliding of patella over the femoral condyle. This will increase strain on the reconstructed patella ligament. Therefore, our institute follows a unique mobilisation protocol following proximal tibia and knee replacement which is depicted in the Table 2. In few cases quadriceps lag could be evident due to patella tendon overstretch/avulsion, this could be due to improper patella mobilisation or forceful knee bending. Figure 3 a, b and c depicts the patella tendon overstretch.

Figure 3: a) Patella position following proximal tiabia and knee joint replacement b) Patellar tendon stretch after 3months c) Patella mal position (over ride) on patient.

Figure 3: a) Patella position following proximal tiabia and knee joint replacement b) Patellar tendon stretch after 3months c) Patella mal position (over ride) on patient.

Mega prosthetic replacement for proximal and total femoral resection:
Resection of proximal femur and prosthetic replacement may be done for proximal femur tumor or as a part of total femoral resection and reconstruction. Partial or complete loss of joint capsule and dynamic stabilisers of hip joint during tumor resection may leave the hip joint vulnerable to dislocation. This may get potentiated with certain combination movements, if these joint movements are allowed beyond a certain limit. This restriction largely depends upon the surgical approach. Postero-lateral approach being most common in the LSS of this site, hip rotations, especially internal rotation and, flexion more than 600 and adduction of the hip joint needs to be prevented up to 6 weeks [4,7]. These movement restrictions could be achieved using hip abduction pillow/brace and de-rotation splint. Before the patient gets discharged from the hospital, training them for bed transfer, supine to standing, standing to supine and sitting in a chair/commode becomes paramount important in the early phase of
rehabilitation. Knee joint mobilisation must be started early either by the edge of bed with hip joint well supported or in side lying with pillows between legs. Any restriction of knee joint range would adversely affect the overall function since hip joint function of the ipsilateral leg has already been compromised. Early FWB ambulation could be started from post operative day one initially with walker, then with walking stick. Later most of them would be trained to walk without any walking aid. Total femoral resections may require more intense rehabilitation with additional emphasis on knee strengthening as discussed earlier (Table 2). Patients may life long need to use walking aids in view of the fact that large motor loss in these cases.
Mega prosthetic replacement for proximal humeral resections:
Proximal humerus and the shoulder girdle are the third common place for primary bone sarcomas [4]. Despite endoprosthetic replacements for functional shoulder girdle structures, such as reverse glenoid prosthesis, are available, lack of muscular structures post excision and damage to the axillary nerve often prevent their use. Frequently, the proximal end of the humerus is replaced with the endoprosthesis and suspended by the remaining muscles and soft tissue structures by suturing around the proximal end of the prosthesis. The objective of the procedure is to achieve a stable shoulder to facilitate good elbow and hand function.
To prevent the weight of the endoprosthesis and the limb acting on the newly formed pseudo joint, shoulder sling and elbow pouch are provided for 4 – 6 weeks. Early post operative rehabilitation consists of elbow and hand range of motion (ROM) and strengthening exercises within pain limit. Again these exercises must be performed in supine position only to avoid undue stress on the shoulder. After six weeks, shoulder joint limited ROM exercise in the form of pendular movements and vigorous strengthening of shoulder girdle, elbow and hand complex should be commenced. Additionally, postural correction must be included in the rehabilitation program.

Biological reconstructions
Wherever feasible, biological reconstructions are preferred over endoprosthetic implants in order to provide a stable and permanent solution for reconstruction of defects after tumor resection. However, rehabilitation following biological reconstructions needs careful considerations regarding weight bearing and joint mobilization. Utmost importance to surgical notes and communication with operative surgeon is of prime importance. Early joint mobilisation is the key to prevent joint stiffness and functional loss; nevertheless, often protective functional braces may be required to prevent damage. For example, curettage and bone grafting of the lower end of femur close to the joint demands hinge knee brace to avoid varus and valgus stress. Strengthening exercises also could be started early with functional knee brace (Fig. 4).

Figure 4: Hinge knee brace following bone graft.

Figure 4: Hinge knee brace following bone graft.

Patients are taught to walk non-weight bearing with brace generally from post operative day one with the help of axillary crutches up to 8 weeks. Once, osteosynthesis is confirmed through radiological evaluation, progressive weight bearing walking could be started. FWB walking and complete joint range and strength are expected to be achieved by the end of 3 months to 4 months.


conclusion  

Although limb salvage surgery for primary malignant tumours have achieved commendable advancement in surgical techniques and endo-prosthetic design and manufacturing, without optimal and timely peri and post-operative physical rehabilitation, achieving the desired quality of life outcome may not be feasible. This paper has highlighted few important rehabilitation principles and we have summarised rehabilitation protocol for specific area. Most of the oncology resection and reconstruction vary from one individual to another even in one particular site and needs tailor made rehabilitation protocol; nevertheless, this summary will be a guide for necessary foundation to design individual rehabilitation program.


References

1. Chopra BK. Health related quality of life studies in Indian patients after limb salvage surgery: Need of the hour. Med J Armed Forces India. 2013 Jul;69(3):209–10.
2. Ottaviani G, Robert RS, Huh WW, Palla S, Jaffe N. Sociooccupational and physical outcomes more than 20 years after the diagnosis of osteosarcoma in children and adolescents. Cancer. 2013;119(20):3727–36.
3. Frieden RA, Ryniker D, Kenan S, Lewis MM. Assessment of patient function after limb-sparing surgery. Arch Phys Med Rehabil. 1993 Jan;74(1):38–43 6p.
4. Oren R, Zagury A, Katzir O, Kollender Y, Meller I. Principles of rehabilitation after limb-sparing surgery for cancer. In: Musculoskeletal Cancer Surgery [Internet]. Springer; 2001 [cited 2014 Sep 3]. p. 583–93. Available from: http://link.springer.com/chapter/10.1007/0-306-48407-2_36
5. Bekkering WP, Vliet Vlieland TPM, Fiocco M, Koopman HM, Schoones JW, Nelissen RGHH, et al. Quality of life, functional ability and physical activity after different surgical interventions for bone cancer of the leg: A systematic review. SurgOncol. 2012 Jun;21(2):e39–47.
6. Mei J, Zhu X-Z, Wang Z-Y, Cai X-S. Functional outcomes and quality of life in patients with osteosarcoma treated with amputation versus limb-salvage surgery: a systematic review and meta-analysis. Arch Orthop Trauma Surg. 2014 Nov;134(11):1507–16.
7. Shehadeh A, Dahleh ME, Salem A, Sarhan Y, Sultan I, Henshaw RM, et al. Standardization of rehabilitation after limb salvage surgery for sarcomas improves patients’ outcome. HematolOncol Stem Cell Ther. 2013 Sep;6(3–4):105–11.
8. Schmitz KH, Courneya KS, Matthews C, Demark-Wahnefried W, Galvão DA, Pinto BM, et al. American College of Sports Medicine roundtable on exercise guidelines for cancer survivors. Med Sci Sports Exerc. 2010 Jul;42(7):1409–26.
9. Singh F, Newton RU, Galvão DA, Spry N, Baker MK. A systematic review of pre-surgical exercise intervention studies with cancer patients. SurgOncol. 2013 Jun;22(2):92–104.
10. Silver JK. Cancer Prehabilitation and its Role in Improving Health Outcomes and Reducing Health Care Costs. SeminOncolNurs. 2015 Feb;31(1):13–30.
11. Silver JK. Cancer prehabilitation and its role in improving health outcomes and reducing health care costs. SeminOncolNurs. 2015 Feb;31(1):13–30.


How to Cite this article:

Dr. Paramanandam V

Dr. Paramanandam V

Dr. Anuradha A Daptardar

Dr. Anuradha A Daptardar

Dr. Ashish Gulia

Dr. Ashish Gulia


(Abstract    Full Text HTML)      (Download PDF)


 


Limb salvage with megaprosthesis in extremity osteosarcoma –a case-based approach

Volume 2 | Issue 2 | May-Aug 2016 | Page 13-18 | Gurpal Singh1, Mark Edward Puhaindran1


Authors: Gurpal Singh[1], Mark Edward Puhaindran[1]

[1] Division of Musculoskeletal Oncology, University Orthopaedics, Hand and Reconstructive Microsurgery Cluster, National University Health System, Singapore.

Address of Correspondence
Dr. Gurpal Singh
Division of Musculoskeletal Oncology, University Orthopaedics, Hand & Reconstructive Microsurgery Cluster, National University Health System, 1E Kent Ridge Road, Singapore 119228.
Email: gurpal_singh@nuhs.edu.sg


Abstract

Introduction: Musculoskeletal oncology is an evolving field in orthopaedic surgery and surgical management for patients with osteosarcoma has changed fundamentally over the past three decades. Osteosarcoma is a rare tumour, but it is the most common type of primary bone cancer with a biomodal peak. Primary cases tend to occur within the first two decades of life and secondary osteosarcomas affect older patients. Common causes of secondary osteosarcoma include Paget’s disease of bone and radiation exposure. Due to advances in chemotherapeutic regimes, imaging modalities, surgical techniques, material and prosthesis designs, amputation is no longer considered as treatment of choice for most patients. Limb salvage surgery including endoprosthetic reconstructions is also constantly evolving, addressing surgical challenges such as margin control, reconstructive procedures and soft tissue management.
In this review, we focus our discussion on the management of patients with osteosarcoma undergoing megaprosthetic reconstruction. Based on three real cases (osteosarcoma of the distal femur, pelvic osteosarcoma involving the hip joint and osteosarcoma of the proximal tibia), this paper aims to highlight surgical challenges that sometimes need to be overcome in this very challenging field.

Keywords: Limb salvage surgery; megaprosthetic reconstruction; osteosarcoma; musculoskeletal oncology.


Introduction
Musculoskeletal oncology is a rapidly evolving field in orthopaedic surgery and surgical management for patients with osteosarcoma has progressed significantly over the past three decades. Due to advances in neoadjuvant treatment, imaging modalities, surgical techniques and material and prosthesis designs, amputation is no longer considered as standard of care in most cases. Literature is currently reporting limb salvage surgeries in 85% to 95% without compromising oncological principles when compared to amputation [1–5]. Main objectives of limb salvage surgery include maximized functional outcome, satisfactory wound coverage for adjuvant therapy and optimized aesthetic outcome without compromising oncologic principles [6]. The reconstructive options for limb salvage surgery can be thought of as biological (autograft, allograft etc.) or endoprosthetic. Sometimes, a combination of both may be necessary.
When considering treatment options for patients with osteosarcoma, several factors need to be considered, including the response to neoadjuvant chemotherapy, evidence of periprosthetic fracture, anatomic site, involvement of soft tissue, joint and neurovascular structures as well as the evidence of metastases at diagnosis. Adequate margin control during surgery is crucial. Surgery represents only one component of the multidisciplinary treatment protocol of osteosarcoma patients. Since the 1970s, the role of neoadjuvant therapy has evolved and intense multi-agent chemotherapy has improved the prognosis significantly by eradicating accompanying micrometastases and also reducing the reactive zone around the tumour. The main drawback of chemotherapy prior to surgery includes immunosuppression. Therefore, a multidisciplinary approach between medical oncologists and the surgical team is essential in order to optimise treatment for the patient. After completion of neoadjuvant chemotherapy, re-staging is performed to assess response to chemotherapy and verify resectability and evaluate margins prior to proceeding with definite surgery. Limb salvage surgery for osteosarcoma is typically described as consisting of three parts, starting with en bloc resection of the tumour. Based on the exact location, intra- or extra-articular resection may be considered. In skeletally immature patients, the possibility of physis preservation should be considered. Thereafter, the bone defect is reconstructed. The third part of the surgery consists of soft tissue coverage and functional re-establishment. Treatment protocols for osteosarcoma further include the administration of adjuvant chemotherapy and subsequent long-term surveillance.
After limb salvage surgery, overall survival rates of 60% to 70% at 5 years have been reported in literature [1–4, 9, 15]. A recent systematic review and meta-analysis by Stokke et al. evaluating the quality of life among paediatric, adolescent and young adult bone tumour survivors suggested that quality of time improves over time. According to their results, female patients and patients at older age are more likely to have a poor quality of life. Interestingly, there was no difference in outcomes between patients who underwent limb salvage surgery versus amputation for local control [16].
Failure rates in patients undergoing reconstruction with megaprostheses are generally higher compared to patients treated with conventional arthroplasties [7]. However, a direct comparison of study findings is difficult due to different settings and definitions used. Failure rates between 40% to 73% at 5 to 15 years have been reported from factors due to the underlying disease contributing to unfavourable early and late surgical outcome, complications can be specifically related to endoprosthetic reconstruction including aseptic loosening, periprosthetic fracture, infection, implant failure, dissociation of modular components and wear [1, 2, 5, 8, 9]. Implant survival rates for fixed-hinge prostheses have been reported to be approximately 70%, whereas rotating hinge prostheses survival between almost 80 to 100% have been reported [1, 7, 10–13]. A classification system proposed by Henderson et al. for megaprosthesis failure indicates 5 causes of failure, including soft tissue failure (Type 1), aseptic loosening (Type 2), structural fracture (Type 3), infection (Type 4) and local tumour recurrence (Type 5) [14].

Three case examples of patients with osteosarcoma
Case 1 – Osteosarcoma of the distal femur
A 16 year old male patient presented with right knee pain at the lateral aspect of the knee and night pain for about one month. There was no known trauma to the right knee. Clinical findings revealed tenderness over the medial and lateral aspect of the knee and a decreased range of motion 10-110°. Plain radiograph, magnetic resonance imaging, computed tomography and bone scan for local and distal staging were performed, showing an aggressive right distal femoral tumour, with extraosseous soft tissue extension abutting the quadriceps and gastrocnemius muscles, iliotibial band, and medial and lateral gutters of the knee joint (Figure 1a). No fracture or neurovascular involvement was detected. There was no evidence of distant osseous or pulmonary metastases. An open biopsy confirmed the diagnosis of a high-grade conventional osteosarcoma.

Figure 1A: Anteroposterior radiograph of the right knee demonstrating a gross cortical breach at the lateral aspect of the distal metaphysis.

Figure 1A: Anteroposterior radiograph of the right knee demonstrating a gross cortical breach at the lateral aspect of the distal metaphysis.

After completion of neoadjuvant chemotherapy, the patient developed an extensive local fungal infection on the lateral aspect of the thigh, surrounding the biopsy incision site. Systemic antifungal therapy was given for two weeks, directed by a dermatologist and surgery was delayed by two weeks. Given the small window of opportunity between neoadjuvant and adjuvant chemotherapy and to minimise further delay in treatment, the surgical team in the outlined case decided to proceed with tumour resection. In view of the recent tinea corporis with lesions on the lateral aspect of the right thigh, the decision was made to proceed with an anteromedial approach with the excision of the biopsy tract laterally. Local tumour recurrence as a result of spread of malignant cells during biopsy has been reported [17, 18]. Consequently, the biopsy tract is considered as contaminated and conventionally excised during tumour removal. However, more recent studies have shown preliminary evidence for the safety of limb salvage surgery without biopsy tract excision following a diagnostic core needle biopsy and fine-needle aspiration, respectively [19, 20].
In the outlined case, a medial dissection via interval between vastus medialis and rectus femoris was performed for en bloc resection of the tumour. Femoral vessels were identified and traced into the popliteal fossa via adductor canal. The femur osteotomy was made at pre-templated 150 mm from the joint line. The sciatic and peroneal nerve were identified and preserved. Histopathological analysis of frozen sections and tumour specimen showed negative margins. After change of instruments, distal femur reconstruction was continued. Femur and tibia were prepared and a rotating-hinge Global Modular Reconstructive System (GMRS) (Stryker Inc, Rutherford, NJ, USA) knee prosthesis was used for reconstruction (Figure 1b). The vastus medialis was mobilised and brought down to cover the defect medially. The extensor mechanism was reconstructed over the prosthesis. No post-operative complications occurred, all wounds healed satisfactorily. Six months after surgery, the patient is walking without any support.

Figure 1B: Anteroposterior radiograph showing the defect reconstructed with a rotating-hinge Global Modular Reconstructive System (GMRS) prosthesis.

Figure 1B: Anteroposterior radiograph showing the defect reconstructed with a rotating-hinge Global Modular Reconstructive System (GMRS) prosthesis.

This case highlights the necessity of non-conventional surgical approaches when necessary. Although the main tumour burden was on the lateral aspect of the knee joint, given the location of the previous treated fungal infection, the decision for a medial approach was made. Extra- versus intraarticular resection needs to be discussed in the appropriate context. Preliminary evidence is hinting towards an overestimation of neoplasm seeding in the biopsy tract, especially when the biopsy is performed by the operating surgeon himself. The point to emphasize in this case is the close communication between medical oncologist and the surgeon and balancing the risks of delaying adjuvant chemotherapy versus an “unconventional” surgical approach. This patient underwent an intraarticular resection of the distal femur. In cases where the knee joint is found to be contaminated or being directly invaded by the tumour, an extraarticular resection of the entire joint with en-bloc removal is recommended. Usually, this approach is associated with poorer functional results.

Case 2 – Pelvic osteosarcoma involving the hip joint
A 24 year old female patient presented with a left neck of femur fracture with underlying osteosarcoma. Prognostic and treatment implications of pathological fractures at presentation have been widely debated. In the past, pathological fractures have been considered as indication for amputation due to the risks of local recurrence from contamination caused by the fracture hematoma. However, a recent systematic review and meta-analysis by Salunke et al. is demonstrating similar rates of local recurrence in patients with limb salvage surgeries compared to amputation, provided carefully selection of patients has been performed [21]. Our own 18-year experience of high-grade osteosarcoma with a pathological fracture at initial presentation showed no differences in the survival and recurrence rates of patients with pathological fractures compared to those with no fractures. Moreover, we did not see any difference in survival between amputated and salvaged patients with fractures, provided margin control attempts are aggressive.

Figure 2A: Anteroposterior radiograph of the pelvis 6 years after pelvic osteosarcoma removal and reconstruction of the proximal femur.

Figure 2A: Anteroposterior radiograph of the pelvis 6 years after pelvic osteosarcoma removal and reconstruction of the proximal femur.

In the outlined case, the patient was treated according to protocol with neoadjuvant chemotherapy. Given the complexity of the case, but still aiming for limb salvage, the decision was made for using endoprosthetic and biologic reconstructive options, namely the combination of a hemipelvectomy with an extra-articular proximal femur resection followed by proximal femur replacement and acetabular column plating/deep frozen bone (Figure 2a). Biologic reconstruction options such as allografts, autografts and re-implantation of sterilized tumour bone replace the defect with a biologic construct, providing the theoretical advantage of incorporation of the biological graft. However, retrieval studies have shown that grafts become necrotic and may act as a spacer rather than a stable replacement. Typical failures include graft-fracture, non-union between the graft and host-bone and increased infection-rates. Drawbacks also include the dependence of graft incorporation for rehabilitation. A reliable tissue bank and associated logistics are sine qua non, and harvesting the autograft or preparing the allograft and subsequently increasing surgical time need to be taken into account. Since a long-term study showed that less than 50% of allografts lasted less than 10 years [22], this technique is mostly preserved for special occasions only, such as osteosarcomas in skeletally immature patients without involving the growth plates. Combined usage of endoprosthetic and biologic reconstruction is described by the allograft prosthetic composite. The concept behind includes restoration of the bone stock by allograft and using endoprosthetic components to restore articulating surfaces, theoretically offering a higher durability in the long-term when compared to pure biologic reconstruction. In the outlined case, the patient’s own pre-treated tumour bone was used for part of the recontruction. Sterilization of the bone can be performed in various ways including pasteurizing, autoclaving, radiotherapy, and freezing. Similarly to the use of allografts, non-union, infection and fracture belong to the main complications in biologic reconstruction with autografts. As described in the outlined case, liquid nitrogen was used for pre-treatment of the bone. Cryosurgery in combination with autograft-prosthesis composites shows promising results with excellent functional outcomes, low complication rates and improved union rates, especially when used with the pedicle-freezing technique [23–25].
After completion of adjuvant chemotherapy and being in remission, the patient in the outlined case developed a hematogenous methicillin susceptible staphylocococcus aureus (MSSA) infection of the endoprosthesis six years postoperatively. Infection is still the most common complication in megaprosthetic reconstruction and in general, it is more likely to happen when adequate wound coverage is missing. In primary megaprosthesis infection rates between 2% to 20% have been reported, with the numbers increasing to more than 40% in revision cases [1, 2, 5, 10, 14, 26–31]. The classic approach in periprosthetic infection is a two-stage exchange revision surgery, including the removal of original endoprosthetic components, thorough debridement, administration of intravenous antibiotics, and implantation of a spacer followed by the re-implantation of implants.
In the outlined case, this approach was not practical. A consensus with the multidisciplinary musculoskeletal oncology team and the patient was made for management with ultrasound-guided drainage of the left hip joint and suppressive antibiotics with follow-ups by our colleagues for infectious disease. The patient was discharged with peripherally inserted central catheter line in situ. She continued to develop a collection in the left hip and anterior thigh. A formal arthrotomy with extensive debridement and removal of all necrotic tissue was performed. A large bore chest drain was left in situ, exited through quadriceps anteriorly, plus two additional drains subcutaneously. An urostomy pouch was applied and the patient was kept on long term drainage, allowing the sinus track to the left lateral thigh to mature. In the following, kept with suppressive antibiotics (ciprofloxacin and rifampicin), the patient was under close follow-up with a regular change of the urostomy bag. Erythema and tenderness regressed and drainage successively decreased (Figure 2b). The chest drain was removed two months after surgery. On examination, 29 months after debridement the left thigh is non-tender and showed no erythema. Scars to the left tight (sinus track) are well healed and she had no fever, chills or rigor and denied any pain. She is walking with a slight limp and was returned to work recently. She remains on long-term suppressive antibiotics as guided by the infectious disease physician.

Figure 2B: Photograph of the left thigh, 23 months after debridement showing externally healed sinus track. Mild erythema was noted surrounding the sinus track secondary to occlusive dressing. Previous dressing was dry and intact.

Figure 2B: Photograph of the left thigh, 23 months after debridement showing externally healed sinus track. Mild erythema was noted surrounding the sinus track secondary to occlusive dressing. Previous dressing was dry and intact.

This case highlights how limb salvage surgery has pushed the boundaries over the last decades. Combining different reconstructive options may be considered for complex cases. This case also illustrates the option of a relatively conservative management strategy for periprosthetic infection in a compliant patient, achieving a satisfactory result.

Case 3 – Osteosarcoma of the proximal tibia
A 16 year old female patient was diagnosed with non metastatic osteosarcoma of the left proximal tibia (stage IIb). She underwent left tibial osteosarcoma resection, reconstruction with megaprosthesis, extensor mechanism reconstruction, medial gastrocnemius flap and split skin graft (Figure 3a and 3b).

Figure 3A: Intraoperative photograph of the osteosarcoma of the left proximal tibia after en-block removal.

Figure 3A: Intraoperative photograph of the osteosarcoma of the left proximal tibia after en-block removal.

Figure 3B: Anteroposterior radiograph showing megaprosthetic reconstruction after proximal tibia osteosarcoma excision.

Figure 3B: Anteroposterior radiograph showing megaprosthetic reconstruction after proximal tibia osteosarcoma excision.

Proximal tibia resection presents unique local anatomical features which need to be addressed during limb salvage surgery. Neurovascular structures in the popliteal fossa as well as the peroneal nerve between the biceps femoris and the lateral head of the gastrocnemius muscle need to be preserved as much as possible. Displacement of these structures due to a large tumour mass are likely, therefore careful preparation in this area is required. In most cases, biological borders such as perivascular fat or the popliteus muscle are separating the tumour from the neurovascular bundle. Intraoperatively, overstretching these structures due to overdistraction of the femur and tibia (after resecting the tumour) needs to be avoided in order to prevent neuropraxia and endothelial injury. Involvement of tissue adjacent to the tibiofibular joint is usually of concern and this joint is typically included in the resection. Once the patellar tendon has been divided, reconstruction of the extensor mechanism is obligatory. Various techniques to attach the tendon to the endoprostetic implant have been described in a systematic review by Ek et al. including direct fixation using screw/washer or sutures, synthetic soft tissue augments such as tubes, sutures, cerclage wires and non-absorbable tapes and biological augmentation such as graft from the biceps or satorius tendons or gastrocnemius flaps with or without synthetic materials [32]. The authors report a trend towards improved outcomes with biologic reconstructive options. However, there is no clear evidence for a single technique. Extensor lag is of major concern in all surgeries requiring reconstruction of the extensor mechanism. Therefore, postoperative management in the outlined case included immobilization with a long-leg cast and a long-leg brace respectively for six weeks to allow healing. Obtaining full extension and allowing the extensor mechanism to heal was given priority compared to knee flexion in view of its impact on ambulation and there was a strong emphasis on extensor mechanism strengthening during rehabilitation.
The close proximity of the proximal tibia to the skin makes the wound more susceptible to infectious complications. Keeping thick flaps during preparation at the beginning of the surgery minimize necrosis of the skin. Typically, a primary medial gastrocnemius flap is used to improve wound coverage with the advantage of offering a larger muscular material and also bypassing a longer distance when compared to the lateral gastrocnemius [33, 34]. This technique has been first described by Dubousset et al. [35]. Skin grafting provides an additional supportive procedure while relieving tension during skin closure and therefore preventing skin flap necrosis. Currently, split-thickness skin graft is considered as gold standard in any major skin loss. Typically, the graft is meshed in order for expansion while keeping the morbidity to the donor side as low as possible. Postoperative management in patients with skin grafts includes a close observation of the donor side for scarring, pain and signs suggestive of infection.
This case illustrates a conventional proximal tibia resection, highlighting surgical considerations with regards to anatomical characteristics and postoperative management.

Conclusion
Medical management, surgical techniques and prosthetic design have improved significantly during the last three decades. Amputation is no longer considered as standard of care in patients with osteosarcoma in most cases. However, limb salvage with megaprosthesis remains challenging. Complication rates are high, with infection occurring most commonly. A good interaction within a multidisciplinary team in a preferably high-volume centre is required for optimal management. Given the unique features of these patients, “unconventional” approaches and combinations of reconstructive options may be considered sometimes, provided oncologic principles are not compromised.


References

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How to Cite this article: Singh G, Puhaindran ME. ELimb salvage with megaprosthesis in extremity osteosarcoma –a case-based approach. Journal of  Bone and Soft Tissue Tumors May- Aug 2016;2(2):13-18 .

Dr. Gurpal Singh

Dr. Gurpal Singh

Dr. Mark Edward Puhaindran

Dr. Mark Edward Puhaindran

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Is limb salvage surgery a contra indication in pathological fractures secondary to osteosarcoma? Do we know the answer?

Volume 2 | Issue 2 | May-Aug 2016 | Page 10-12 | Zeeshan Khan1, Shakir Hussain1, Simon Carter1


Authors: Zeeshan Khan[1], Shakir Hussain[1], Simon Carter[1]

[1] The Bone and soft tissue tumour unit,
The Royal Orthopaedic Hospital, Birmingham, UK. B31 2AP.

Address of Correspondence
Dr. Zeeshan Khan
Bone tumour unit, The royal orthopaedic hospital, Birmingham, UK. B31 2AP.
Email: zeek1978@yahoo.co.uk


Abstract

Introduction: Osteosarcoma is the commonest primary bone tumour with a bimodal age distribution. The survivorship of patients with osteosarcoma has improved with advances in chemotherapy making limb salvage surgery the commonest surgical procedure. Pathological fractures associated with osteosarcoma, however are rare and suggests the aggressiveness of the tumour. These patients are considered as a special group due to the variable outcomes reported in the literature due to some special characteristics, prompting the discussion between limb salvage surgery versus ablative surgery.
This article reviews the reasons why this group of patients are considered challenging and also the various outcomes reported in the literature.
Keywords: Pathological fracture, osteosarcoma, outcomes


Introduction
Primary bone and soft tissue sarcomas are rare tumours. Osteosarcoma is the commonest primary bone tumour with a bimodal age distribution and with a reported incidence of 2-3 per million population per year [1, 2]. With advances in chemotherapy, the survivorship of patients with osteosarcoma has improved significantly with various studies revealing similar results with limb salvage surgery when compared with amputation [5]. Contra indications to limb salvage surgery may include involvement of the neurovascular bundle, joint involvement, progression of disease whilst on treatment, patient choice, infection and a pathological fracture (Figure 1).
A pathological fractureassociated with osteosarcoma at presentation or during treatment is even rarer with a reported incidence of 5-10% [3, 4].A pathological fracture can be the mode of presentation for osteosarcomas in certain cases whereas it can occur during treatment in others. This is generally considered to be an aggressive biological behaviour of the disease which in turn, historically, has been considered as a poor prognostic factor in the outcome of this special group of patients [6].

Figure 1: Plain radiograph of a skeletally mature patent with a pathological fracture secondary to osteosarcoma with involvement of the joint both clinically and radiologically.

Figure 1: Plain radiograph of a skeletally mature patent with a pathological fracture secondary to osteosarcoma with involvement of the joint both clinically and radiologically.

Why is this group of patients special?
This select group of patients poses a challenge to the treating orthopaedic surgeon about the modality of surgical procedure. There has been a debate over the years whether these patients should have ablative or limb salvage surgery and if there is a difference in the outcome of both with varying results reported by different authors [8].
What makes these patients special is the associated hematoma with the pathological fracture which is considered to have tumour cells which spreads locally in the tissues [7]. The extent of spread also depends on the anatomic location of fracture and whether it is intra or extra capsular. The disruption of local microvasculature is also considered to be a risk factor for development of metastasis [7]. Understandably, extra articular resection for intra articular extension of tumours is a more challenging procedure particularly when limb salvage surgery is attempted with the reported outcomes of extra articular resections in limb salvage surgery considered to be compromised as well [9]. The local contamination of soft tissues with the tumour cells is also considered to be a risk factor for local recurrence. This prompted the thought that early and aggressive surgery in the form of ablative surgery will halt the progression and spread of disease any further. The presence of a pathological fracture in osteosarcoma, therefore, has been considered as a poor prognostic factor by some authors but not by all [8, 10, 11].

Initial treatment& work up:
Perhaps the most important step in the management of this select group of patients is the early recognition of the aggressiveness of the lesion and prompt referral to a specialist unit. Failure of recognition of these fractures as being pathological can lead to inappropriate treatment and potentially worse outcomes (Figure 2) [14]. The rest of the management in a multidisciplinary team setting involves a detailed history, examination of the involved limb and joints for any effusion, local and systemic staging, biopsy and neoadjuvant chemotherapy after confirmation of diagnosis of osteosarcoma.
It is also important to note that one of the most challenging issues with this group of patients is pain management and immobilisation during the pre-operative period whilst they wait for surgery and have neoadjuvant chemotherapy.

Figure 2: Plain radiograph of an inappropriately managed pathological fracture secondary to osteosarcoma making limb salvage very challenging.

Figure 2: Plain radiograph of an inappropriately managed pathological fracture secondary to osteosarcoma making limb salvage very challenging.

Immobilisation
Immobilisation for pain relief can be challenging as this depends on the location of the fracture and may involve a plaster cast, simple sling, skin traction or in some cases external fixators [15]. Significant attention should be paid to the placement of the schanz pins if an external fixator is used due to the risk of tumour spread into non-involved compartments and risk of infection which would compromise limb sparing surgery.

Prognostic factors
A pathological fracture is independently considered a poor prognostic factor in osteosarcoma but was not considered one in cases of chondrosarcoma and Ewing’s sarcoma [8, 10]. A poor response to chemotherapy and local recurrence are also considered to be poor prognostic factors for survivorship [8, 10-13]. It is however, important to note that the efficacy of chemotherapy and healing of fracturesin these special cases are considered as supportive factors for limb salvage surgery [20].
Fracture consolidation
It has been noted that these fractures heal whilst patients are on chemotherapy and in most of the cases these patients have had significant post chemotherapy necrosis(Figure 3) [8]. On the contrary some fractures might happen whilst patients are on chemo which depicts the aggressive nature of the disease.

Figure 3A: Radiograph showing a pathological fracture secondary to osteosarcoma.

Figure 3A: Radiograph showing a pathological fracture secondary to osteosarcoma.

Figure 3B: Radiograph showing post chemotherapy consolidation in the pathological fracture.

Figure 3B: Radiograph showing post chemotherapy consolidation in the pathological fracture.

 

What is the verdict?
Limb salvage surgery should be attempted, if possible, in these patients after neoadjuvant treatment but if clear surgical margins cannot be obtained during surgery or limb salvage will result in a poor functioning limb, then ablative surgery should be considered, particularly in the paediatric population where they can adapt to prosthetics earlier than adults [16]. It is however, also important to note that after wide resection of tumour, limb salvage is still a viable option with reconstruction performed with either arthrodesis or rotationplasty where appropriate.
Scully et al, suggested that a pathological was a poor prognostic factor but it is important to note that this study was performed over a 30 year period where some patients in their series had not received any chemortherapy and there have been advances in this field over the study time period [8]. Similarly Finn et al, suggested early amputation due to the risk of local and distant tumour spread [14]. In another study, the 5 year survival in patients with pathological fractures secondary to osteosarcoma was lower than those without a fracture [18]. On the contrary Bacci et al, and Abudu et al, showed that there was no difference in the survivorship of these patients when they were treated with neoadjuvant chemotherapy [7, 11]. In a recent meta-analysis comparing limb salvage with ablative surgery for pathological fractures in high grade osteosarcomas, no significant difference between local recurrence and 5 year survival was noted [19]. Adjuvant radiotherapy in these patients has not been shown to reduce the risk of local recurrence and in fact might increase the risk of these patients undergoing further surgical procedures compromising there outcomes [7].

Future direction
All the studies performed on this select group of patients are retrospective and contain a small number of patients over a prolonged period of time. The results are further effected by variables including the heterogeneity of the patient and fracture characteristics and also the advances in chemotherapy over a period of time. Improvements in surgical techniques have also resulted in improved outcomes. Most of these variables are un avoidable due to the rarity of these cases but in order to come to a definite conclusion, a multi central randomised trial will eradicate all these bias and should guide treatment.


References

1. 1.Bielack S, Carrle D, Jost L. ESMO guidelines working group osteosarcoma: ESMO clinical recommendations for diagnosis, treatment and follow up. Annals of Oncology.2008; 19, supplement 2: 94-96.
2.Widhe B, Widhe T. Initial symptoms and clinical features in osteosarcoma and Ewing sarcoma. J Bone Joint Surg Am. 2000; 82:667-74.
3. Jaffe N, Spears R, Eftekhari F, Robertson R, Cangir A, Takaue Y, Carrasco H, Wallace S,
Ayala A,Raymond K, et al. Pathologic fracture in osteosarcoma. Impact of chemotherapy on primarytumorand survival. Cancer. 1987; 59:701-09.
4. Mulder JO, Schutte HE, Kroon HM, Taconis WK. Radiologic atlas of bone tumors. Amsterdam:Elsevier Science. 1993. Intraosseous osteosarcoma: conventional type: 51-5.
5. Simon MA. Current concepts review. Limb salvage in osteosarcoma. J Bone Joint Surg Am.1988; 70:307-10.
6. Coley BL, Pool JL. Factors influencing the prognosis in osteogenic sarcoma. Ann Surg. 1940; 112:1114-28.
7. Abudu A, Sferopoulos NK, Tillman RM, Carter SR, Grimer RJ. The surgical treatment and outcome of pathological fractures in localised osteosarcoma. J Bone Joint Surg Br. 1996; 78:694-8.
8. Scully SP1, Ghert MA, Zurakowski D, Thompson RC, Gebhardt MC. Pathologic fracture in osteosarcoma: prognostic importance and treatment implications. J Bone Joint Surg Am. 2002 Jan;84-A (1):49-57.
9. Hardes J1, Henrichs MP, Gosheger G, Gebert C, Höll S, Dieckmann R, Hauschild G, Streitbürger A. Endoprosthetic replacement after extra-articular resection of bone and soft-tissue tumours around the knee. Bone Joint J. 2013 Oct; 95-B (10):1425-31.
10. Bramer JAM, Abudu AA, Grimer RJ, Carter SR, Tillman RM. Do pathological fractures influence survival and local recurrence rate in bony sarcomas?. Eur J Cancer. 2007 Sep; 43(13):1944-51.
11. Bacci G1, Ferrari S, Longhi A, Donati D, Manfrini M, Giacomini S, Briccoli A, Forni C, Galletti S.Nonmetastatic osteosarcoma of the extremity with pathologic fracture at presentation: local andsystemic control by amputation or limb salvage after preoperative chemotherapy. ActaOrthop Scand. 2003 Aug; 74(4):449-54.
12. Meyers PA, Heller G, Healey J, Huvos A, Lane J, Marcove R, ApplewhiteA, Vlamis V, Rosen G. Chemotherapy for nonmetastatic osteogenic sarcoma: the Memorial Sloan-Kettering experience. J ClinOncol. 1992; 10:5-15.
13. Glasser DB, Lane JM, Huvos AG, Marcove RC, Rosen G. Survival, prognosis, and therapeutic response in osteogenic sarcoma. The Memorial Hospital experience. Cancer.
1992; 69:698-708.
14. Mankin HJ, Mankin CJ, Simon MA. The hazards of the biopsy, revisited: Members of the Musculoskeletal Tumor Society. J Bone Joint Surg [Am] 1996; 78-A: 656–663.
15. Chandrasekar CR, Grimer RJ, Carter SR, et al. Pathological fracture of the proximal femur in osteosarcoma: need for early radical surgery? ISRN Oncol 2012; 2012:512389.
16. Hosalkar HS, Dormans JP. Limb sparing surgery for pediatric musculoskeletal tumors. Pediatr Blood Cancer 2004; 42:295–310.
17. Finn HA, Simon MA. Limb-salvage surgery in the treatment of osteosarcoma in skeletally immature individuals. ClinOrthopRelat Res 1991; 262:108–118.
18. Ferguson PC, McLaughlin CE, Griffin AM, et al. Clinical and functional outcomesof patients with a pathologic fracture in high-grade osteosarcoma. J SurgOncol2010; 102:120–124.
19. Yin K, Liao Q, Zhong D, Ding J, Niu B, Long Q, Ding D. Meta-analysis of limb salvage versusamputation for treating high-grade and localized osteosarcoma in patients with
pathological fracture. ExpTher Med. 2012 Nov; 4(5):889-894.
20. Scully SP, Temple HT, O’Keefe RJ, et al. The surgical treatment of patients with osteosarcoma who sustain a pathological fracture. ClinOrthop. 1996; 324:227-232.


How to Cite this article:Khan Z, Hussain S, Carter S. Is limb salvage surgery a contra indication in pathological fractures secondary to osteosarcoma? Do we know the answer? Journal of  Bone and Soft Tissue Tumors May- Aug 2016;2(2):10-12 .

Dr. Zeeshan Khan

Dr. Zeeshan Khan


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Tata Memorial Centre – The Journey so Far!!!

Volume 2 | Issue 2 | May-Aug 2016 | Page 3-4 | Ashish Gulia1, Ajay Puri1, Rajendra.A. Badwe1


Authors: Ashish Gulia[1], Ajay Puri[1], Rajendra.A. Badwe[1]

[1]Orthopedic Oncology Services, Department of Surgical Oncology, Tata Memorial Hospital, Mumbai.

Address of Correspondence
Dr. Ashish Gulia
Associate Professor, Orthopedic oncology, Department of Surgical Oncology, Tata Memorial Hospital, Mumbai.
Email: aashishgulia@gmail.com


Tata Memorial centre, considered as “Mecca” of Oncology care in the country and sub continent, stands today at its Dodranscentennial Anniversary. Institution started as Tata Memorial Hospital, which was inaugurated on 28th February 1941 by His Excellency Sir Roger Lumley, then Governor of Mumbai. Declaring the centre open, he estimated with extraordinary prescience “…the hospital will become the spearhead of attack on cancer in this country, providing not only a center where specialized treatment can be given, but also one from which knowledge of new methods of treatment and diagnosis will go out to doctors and hospitals throughout the country…”
The past 75 years stand as glowing testimony to Sir Lumley’s prophecy. A recent peer group review of the hospital has accredited it as the premier comprehensive center in India, in addition recognized it as potentially and prominently featuring in the top five cancer centers globally.
The advent of TMH in chapters of the history of medicine in India can best be defined as a quantum leap. It owed its genesis to the philanthropic sentiments of the illustrious Tata family. Later on, in 1961 was passed under the guardianship of Department of Atomic Energy which remarkably aided and upheld the meteoric rise of the sterling institution. In the year 1966 Tata Memorial Hospital merged with Cancer Research Institute (the pioneer research institute), the conjoined institutes were collectively called Tata Memorial Centre (TMC). TMC exemplifies private philanthropy boosted by Government support with a mandate for Service, Education & Research in Cancer. Commencing its journey as a torchbearer, the first dedicated cancer specialty hospital in Asia, TMC today continues to maintain its excellence of being the largest cancer institute in Asia. A fledgling 80 bedded hospital spread over 15,000 sq meters with an annual budget of half a million (INR), has escalated to a lofty institution with 700+ beds, covering over 75,000 sq meters, utilizing an annual budget of 23,00 million (INR). TMC indomitably bears the responsibility of cancer care burden not only of the large Indian populace but also several parts of Asia, Middle East and Africa, catering to an annual footfall of 65,000 new cases and 450,000 follow up cases.[1]
Research and education have unarguably been the essential arms of a comprehensive cancer care centre. TMC since origin has embraced the holistic model of delivering cancer care by augmenting and developing its research and education facilities as it sought to provide treatment which was affordable, innovative and particularly relevant to the needs of the country. The research wing of TMC originally established in 1952 as CRI (Cancer Research Institute) was subsequently revamped as ACTREC (Advanced Centre for Treatment Research and Education in Cancer) in 2002. ACTREC is a state of art research and training centre which has over the years focussed on the integration of basic and clinical research including evolving inspiring pathways for translational research. The academic unit of TMC imparts training in specialty and super specialty courses in oncology and is affiliated to the acclaimed Homi Bhabha National Institute, Mumbai.[1]
The noble mission of the institute was to provide comprehensive cancer care to one and all. A practice which has been resolutely followed since its inception is, that every patient who walks in is attended to and treated irrespective of their ability to pay. Over 60% of TMC’s patients receive free or highly subsidized treatment. The essence of the center however, lies in its undeviating constancy towards extending compassionate care and assuaging suffering in times of extreme distress.
Patients and caregivers often refer to the hospital as a temple which bespeaks diligence and dedication of the individuals who work there. As millions stream in seeking hope, this Mecca continually labors to ensure means of cure, care and cope. Seventy five glorious years of Tata Memorial Centre have borne witness to its commitment to excellence, expertise and eminence in cancer treatment, care, research, education and awareness. As a tribute to this iconic institution we mark a yearlong celebration of its Platinum Jubilee (Dodranscentennial Anniversary) 2016.[1]
The appointment of a full time orthopaedic surgeon on the staff of Tata Memorial Centre in 1999 resulted in the establishment of a specialist orthopaedic oncology service [2]. Over the years this service has now grown to be recognised as one of the leading musculoskeletal oncology units globally. Like all other services at the centre this too functions as a “Disease Management Group”, where specialists from all disciplines involved in sarcoma care (radiology, pathology, medical oncology, radiation oncology, etc.) come together to provide multidisciplinary integrated care under one umbrella.
Today the orthopaedic oncology service caters to about 2500 new musculoskeletal oncology cases per year while performing about 700 major and 1200 minor surgical procedures each year.
The service has collaborated with industry to develop cost effective mechanical reconstructive options suited to the local socio economic milieu. The development of an indigenous prosthesis (TMH – NICE / RESTOR) which was recognised by the Golden Peacock award for health innovation is a proud feather in the cap of the institute. Innovative biological reconstruction techniques published in reputed peer reviewed journals are now practised in other centres globally too.
Both clinical and basic research also plays a part in the units’ contribution to furthering insights in sarcoma care. The service has to its credit the only randomised trial ever conducted for surveillance in sarcomas and is part of a global consortium that has published one of the largest series of patients detailing the genomic profile of sarcomas. [3,4]
Imparting training and sharing of knowledge and information has always been part of the culture of this institute and a host of young orthopaedic oncologists practising all across the country are a proud testimony in keeping with this tradition. There is a constant stream of international trainees and visitors and the service has helped in initiating and developing the limb salvage program in many resource challenged areas like Nepal, Myanmar, Palestine and Nigeria. [5]
The TMC “Evidence Based Guidelines” for the management of bone and soft tissue sarcomas is a useful handbook that helps disseminate the concept of scientific and rational treatment for these challenging tumors. TMC staff and alumni have been at the forefront in establishing the Indian Musculo Skeletal Oncology Society (IMSOS), an organisation that seeks to provide a common forum for interaction and mutual collaboration between different specialists and institutes involved in the treatment of sarcomas.
As the institute seeks to expand its footprint over the next decade by adding infrastructure and staff, both in Mumbai and centres across the country the orthopaedic oncology service too will need to evolve. Challenges of adopting new technology will have to be met.

Yet, offering the right balance between these enhanced options while maintaining the principles of rational , cost effective medical care will necessitate “the wisdom of Solomon”.
Just as it has for the past 75 years, the institute and every individual connected with it will always strive with utmost passion to fulfil its motto of “Service – Education – Research” in a ceaseless endeavour to offer the best possible care to the patients that repose their trust in this institute.


References

1. Tata Memorial Centre. 2016. Tata Memorial Centre Platinum Jubilee – Celebrating 75 Glorious Years. Available at: http://tmcplatinumjubilee.org/tmc-platinum-jubilee.php
2. Puri A – The “ODYSSEY”: “Orthopaedic Oncology” – My journey thus far! Journal of Bone and Soft Tissue Tumors May-Aug 2015; 1(1):4-6
3. Puri A, Gulia A, Hawaldar R, Ranganathan P, Badwe RA. Does Intensity of Surveillance Affect Survival After Surgery for Sarcomas? Results of a Randomized Noninferiority Trial. ClinOrthopRelat Res. 2014 May;472(5):1568-75.
4. Ballinger ML, Goode DL, Ray-Coquard I, James PA, Mitchell G, Niedermayr E, Puri A, Schiffman JD, Dite GS, Cipponi A, Maki RG, Brohl AS, MyklebostO,Stratford EW, Lorenz S, Ahn SM, Ahn JH, Kim JE, Shanley S, Beshay V, Randall RL, Judson I, Seddon B, Campbell IG, Young MA, Sarin R, Blay JY, O’DonoghueSI,Thomas DM; International Sarcoma Kindred Study. Monogenic and polygenic determinants of sarcoma risk: an international genetic study. Lancet Oncol. 2016 Aug 4.
5. Gulia A, Wilding C, Suman MB, Puri A. OrthOncoCon-2014: Continuing education in musculoskeletal oncology. Indian J Cancer. 2015 Apr-Jun;52(2):184-5.


How to Cite this article: Gulia A, Puri A, Badve, RA. Tata Memorial Centre – The Journey so Far!!!. Journal of  Bone and Soft Tissue Tumors May- Aug 2016;2(2):3-4 .

Dr. Ashish Gulia

Dr. Ashish Gulia

Dr. Ajay Puri

Dr. Ajay Puri

Dr. Rajendra.A. Badwe

Dr. Rajendra.A. Badwe


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