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Journal of Bone and Soft Tissue Tumors (JBST) is the official Journal of The Indian Musculo Skeletal Oncology Society
Biphasic synovial sarcoma – A ten year experience with molecular profile and clinical outcome
Vol 4 | Issue 2 | July-Dec 2018 | Page 14-19 | Kala Gnanasekaran Kiruthiga, Anne Jennifer Prabhu, Rekha Pai, Sramana Mukhopadhyay, Reka.K, V.T.K.Titus, Selvamani Backianathan.
Authors: Kala Gnanasekaran Kiruthiga [1], Anne Jennifer Prabhu [1], Rekha Pai [1], Sramana Mukhopadhyay [1], Reka.K [2], V.T.K.Titus [3], Selvamani Backianathan [4].
[1] Department of Pathology, Christian Medical College, Vellore, Tamil Nadu, India.
[2] Department of Biostatistics, Christian Medical College, Vellore, Tamil Nadu, India.
[3] Department of Orthopaedics, Christian Medical College, Vellore, Tamil Nadu, India.
[4] Department of Radiotherapy, Christian Medical College, Vellore, Tamil Nadu, India.
Address of Correspondence
Dr. Anne Jennifer Prabhu,
Address: Department of Pathology, ASHA Building, Christian Medical College, Vellore, Tamil Nadu, India.
E-mail: annejennifer91@gmail.com
Abstract
Context: Synovial sarcoma is one of the commonly encountered spindle cell sarcomas of soft tissue. However, Biphasic synovial sarcoma (BSS) is a rare subtype of synovial sarcoma with limited literature on clinical profile, molecular characteristics and survival outcome.
Aims: We propose to describe the immuno – morphology, clinical features, molecular profile and outcome of patients with BSS.
Settings and Design: This retrospective study included 13 cases of BSS, 3.2% of all synovial sarcomas diagnosed over 10 years in our institute. The clinico-pathological features were studied in detail and immunohistochemistry for TLE-1, EMA, CD34, CD99 and S100 was done. Real time PCR and DNA sequencing for the common translocations (SYT-SSX1/ SYT-SSX2) were performed.
Statistical analysis used: Statistical Package for Social Services (SPSS) software Version 21.0 (Armonk, NY: IBM Corp).
Results: BSS was most commonly seen in young, the most common site being soft tissue of extremities (92.3%). 53.8% of patients presented at Enneking stage IIB. The FNCLCC grade varied between 2(46.2%) and 3(53.8%). All cases were positive for EMA and TLE-1; negative for CD34 and S100. Ten of the eleven (90.9%) patients tested had SYT-SSX1 translocation. Over a period of 8 to 46 months, 53.8% cases were alive and well with no evidence of disease; three had (30%) recurred, one (10%) had lung metastasis and one (10%) died.
Conclusions: BSS is most common in extremities. The immunohistochemical profile matches that of monophasic synovial sarcoma. FNCLCC grade is 2 to 3; however the grade does not correlate with clinical outcome. Most cases show SYT-SSX1 translocation. 53.8% cases were alive and well after a mean follow up of 20 months.
Keywords: Biphasic, follow-up, immunoprofile, SYT-SSX1, synovial sarcoma.
Keyword: Biphasic synovial sarcoma has the same immunoprofile as the monophasic subtype. All patients who tested positive had SYT-SSX1 translocation. More than 50% of patients were alive and well after 20 months.
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Osteosarcoma of Extragnathic Skull Bones-clinicopathological Profile of Eight Cases
Vol 4 | Issue 2 | July-Dec 2018 | Page 11-13 | Mahfooz Basha Mohamed, Jayasree Kattoor, Kusumakumary Parukuttyamma, Geetha Narayanan, Anitha Mathews, Thara Somanathan.
Authors: Mahfooz Basha Mohamed [1], Jayasree Kattoor [2], Kusumakumary Parukuttyamma [3], Geetha Narayanan [4], Anitha Mathews [2], Thara Somanathan [2].
[1] Department of Laboratory Medicine, GKNM Hospital, Coimbatore 641006, Tamil Nadu, India.
[2] Department of Pathology, Regional Cancer Centre, Trivandrum 695011, Kerala, India.
[3] Department of Paediatric Oncology, Regional Cancer Centre, Trivandrum 695011, Kerala, India.
[4] Department of Medical Oncology, Regional Cancer Centre, Trivandrum 695011, Kerala.
Address of Correspondence
Dr. JayasreeKattoor,
Department of Pathology, Regional Cancer Centre, Trivandrum-695011, Kerala, India.
Email: jayasreeramdas@gmail.com
Abstract
Osteosarcoma is the most common primary malignant tumor of bone, usually arising from the metaphysis of the long bones around the knee joint. In 6-13% cases they are located in the head and neck region, of which maxilla and mandible are the most common sites. Osteosarcoma involving the extra-gnathic craniofacial bones account for less than 2% cases. We report eight such cases of osteosarcoma involving this unusual location in the last three years (2011 -2014) and present their clinicopathological profile. Seven patients were under 15 years of age and one patient was 37 years old. Out of the eight cases, four were males and four were females. The location of the tumor included occipital bone, parietal bone, external auditory canal, nasal bone and mastoid. Two patients presented as multicenteric disease with multiple lesions in the skull and elsewhere. Two patients succumbed to the disease while five patients are on follow up. One patient was lost to follow up.A complete en-bloc dissection of the tumor with free margins is a challenge for the operating surgeons. Radiologically they can simulate non-neoplastic lesions or benign tumors as well.These tumors pose a unique therapeutic challenge owing to their unusual location and require a multidisciplinary team approach for management of the patient.
Keywords: Extragnathic, skull, bone, osteosarcoma.
References
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2. Oda D, Bavisotto L M, Schmidt R A. et al., Head and neck osteosarcoma at the University of Washington. Head Neck. 1997; 19(6):513–523.
3. Santhosh Kumar N, Elizabeth Mathew Iype, Shaji Thomas, JayasreeK,SivaramanGanesan.Osteogenic sarcoma of mastoid bone. Journal of Case Reports 2014; 4(2):334-337.
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8. Jasnau S, Meyer U, Potratz J, et al. Craniofacial osteosarcoma: experience of the cooperative German-Austrian-Swiss osteosarcoma study group. Oral Oncol 2008; 44(March (3)):286–94.
9. VijayaKamble, KajalMitra, ChetanaRatnaparkhi, AkshayKapila. Primary Osteogenic Sarcoma of Zygomatic Arch: A Case Report, with World Literature Review. Journal of Evolution of Medical and Dental Sciences 2014; Vol. 3, Issue 36, August 18; Page: 9494-9499, DOI: 10.14260/jemds/2014/3226.
10. Hadley C, Gressot LV, Patel AJ, Wang LL, Flores RJ, Whitehead WE, et al. Osteosarcoma of the cranial vault and skull base in pediatric patients. J NeurosurgPediatr 2014; 13: 380-5.
11. Brad W. Neville, Douglas D. Damm, Angela C. Chi, Carl M. Allen.Oral and Maxillofacial Pathology 4th Edition,Elsevier Health Sciences;2015.Chapter 14:p.614.
12. Horvai A, Unni KK. Premalignant conditions of bone. Journal of Orthopaedic Science. 2006; 11(4):412-423.
13. Gon S, Kundu T, Ghosh BN. Synchronous multifocal osteosarcoma with small cell histological variant: A double rarity. Clin Cancer Investig J 2016; 5:533-6.
14. Currall VA, Dixon JH. Synchronous multifocal osteosarcoma: Case report and literature review. Sarcoma 2006; 2006:53901.
15. Mathkour M, Garces J, Beard B, et al. Primary high-grade osteosarcoma of the clivus: a case report and literature review. World Neurosurg 2016; 89:730.e9–13.
16. Meel R, Thulkar S, Sharma MC, Jagadesan P, Mohanti BK, Sharma SC, et al: Childhood osteosarcoma of greater wing of sphenoid: case report and review of literature. J PediatrHematolOncol 34:e59–e62, 2012.
17. Thiele OC, Freier K, Bacon C, et al. Interdisciplinary combined treatment of craniofacial osteosarcoma with neoadjuvant and adjuvant chemotherapy and excision of the tumour: a retrospective study. Br J Oral MaxillofacSurg 2008;46:533-6.
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Spectrum of Bone and Soft Tissue Tumors in A Tertiary Cancer Institute in Eastern India
Vol 4 | Issue 2 | July-Dec 2018 | Page 7-10 | Ashwini Natekar, Gaurav Gupta, Subhasis Basu.
Authors: Ashwini Natekar [1], Gaurav Gupta [1], Subhasis Basu [1].
[1] Dept of Pathology, Chittaranjan national cancer institute, Kolkata, India
Address of Correspondence
Dr. Ashwini Natekar,
Dept of Pathology, Chittaranjan national cancer institute, Kolkata, India
Abstract
Introduction: Bone and soft tissue tumours are uncommon tumours that can affect any age group. Soft tissue tumors are said to be heterogeneous group of mesenchymal malignancies. Primary bone sarcomas are rare tumors, comprising approximately 0.2% of all cancers. Their true incidence is difficult to estimate because of their rarity.[1]The outcome of the disease depends on the age and time at diagnosis.
Material and Methods: A study was carried out in department of pathology in tertiary cancer institute in East India from December 2015 to September 2017. A total of 60 cases were included in the study. Clinical and radiological details of patients were noted along with gross specimen findings and microscopic examination of H &E stained slides. IHC was also carried out for confirmatory diagnosis.
Aims and Objecctives: 1- To study the histological spectrum of bone and soft tissue tumors in a tertiary cancer institute in Eastern India
2- To study gender distribution and site distributionof bone and soft tissue tumors
Results: Out of the total 60 cases studied 24 cases were of bone and cartilaginous tumours and 36 cases included were of soft tissue tumors. Benign tumors comprised of 08 cases (13.3%) and malignant tumors accounted to 52 cases (86.7%). Age of the patients ranged from 10 to 80 years. Male female ratio was 1.4:1.
Conclusion: Multimodal therapies of treatment is practiced for bone and soft tissue tumors. Early detection and treatment is essential for diagnosis of these malignant tumors. Molecular studies are most important in diagnosing, classifying and also prognosticating bone and soft tissue tumors.
Keywords: bone tumors, soft tissue tumors, histology.
References
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9. Anant Ramaswamy, Bharat Rekhi,1 Sameer Bakhshi,2 Sachin Hingmire,3 and Manish Agarwal4Indian data on bone and soft tissue sarcomas: A summary of published study resultsSouth Asian Journal of Cancer.
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11. Askin FB, Rosai J, Sibley RK, Dehner LP, Mc Alister WH. Malignant small cell tumour of the thoracopulmonary region in childhood: a distinctive clinicopathologic entity of uncertain histogenesis. Cancer. 1979;43:2438–2451. doi: 10.1002/1097-0142(197906)43:6<2438::AID-CNCR2820430640>3.0.CO;2-9. [PubMed] [Cross Ref]
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18. AF Mavrogenis,1 G Bianchi,2 NA Stavropoulos,1 PJ Papagelopoulos,1 and P Ruggieri2 Clinicopathological features, diagnosis and treatment of clear cell sarcoma/melanoma of soft parts Hippokratia. 2013 Oct-Dec; 17(4): 298–302.
19. Meis Kindblom JM. Clear cell sarcoma of tendons and aponeuroses: A historical perspective and tribute to the man behind the entity. Adv Anat Pathol. 2006;13:286–92. [PubMed]
20. O. Hocar,1 A. Le Cesne,2 S. Berissi,1 P. Terrier,2 S. Bonvalot,2 D. Vanel,3 A. Auperin,4 C. Le Pechoux,2 B. Bui,5 J. M. Coindre,5 and C. Robert1Clear Cell Sarcoma (Malignant Melanoma) of Soft Parts: A Clinicopathologic Study of 52 Cases:Dermatology Research and Practice
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Guest Editorial – Asia Pacific Musculoskeletal Tumor Society Conference 2018
Vol 4 | Issue 2 | July-Dec 2018 | page: 2-3 | Ajay Puri.
Author: Ajay Puri [1].
[1] Orthopedic Oncology Services, Department of Surgical Oncology, Tata Memorial Hospital, Mumbai.
Address of Correspondence
Dr. Ajay Puri,
Department of Orthopaedic Oncology, Room No: 26, Tata Memorial Hospital, Borges Road, Parel, Mumbai – 400 012. India.
Email: docpuri@gmail.com
Asia Pacific Musculoskeletal Tumor Society Conference 2018
For advances in musculo skeletal oncology to occur we need a host of clinical specialties along with nursing, rehabilitation and support staff interacting on a continuous basis. Sarcomas are rare and each individual and institution can only benefit from having access to pooled information and experience. For sarcoma care to evolve, ideas to surface and multi institute or multi-disciplinary collaborations to develop in the fields of basic research, patient care, biomaterials and prosthesis, there is a need for a common platform where all of us involved in the treatment of sarcomas can interact. Our socio economic milieu in the Asia Pacific region is unique. We have a spectrum ranging from resource rich societies with extensive state supported health care to resource constrained nations where patients have inconsistent access to health care and funding for health care is limited. Solutions and protocols applicable to the Western world need not necessarily be the most suitable in our scenario. Our large numbers coupled by the sheer native ingenuity necessary to offer quality health care in a resource challenged population can help us develop solutions that can be adapted everywhere.
The Asia Pacific Musculoskeletal Tumor Society (APMSTS) was conceptualized during discussions at the 7th International Symposium of Limb Salvage Surgeons (ISOLS) meeting in 1993 and its first meeting was held in 1995 in Japan under the stewardship of Professor Yoshio Ogihara. Mirroring a similar initiative, the Indian Musculo Skeletal Oncology Society (IMSOS) was established in 2013 to “promote scientific, evidence based, comprehensive multidisciplinary management of bone and soft tissue sarcomas and encourage basic and clinical research”. This year Jaipur, India plays host to the 12th APMSTS meeting in October. This is a joint meeting co-hosted with IMSOS. The theme of the conference is “Education – Collaboration – Innovation”. This epitomizes our desire to share and disseminate knowledge, the spirit of collaboration necessary to find answers to common problems and the need to derive innovative solutions best suited to our socio economic milieu.
The conference logo depicts the feathers of the peacock in the Indian national colours adorning a group of people with interlinked arms who represent the collaboration and camaraderie between individuals and countries that form the Asia Pacific Musculoskeletal Society. The logo represents the beauty and spirituality of India while signifying the desire to showcase skills from the Asia Pacific region. The peacock, the national bird of India symbolizes many of the qualities that we as “healers” must inculcate. In Hinduism, the peacock is associated with Saraswati the goddess of wisdom and learning. In Buddhism, the peacock is a symbol of purity and the ‘eyes’ in the peacock’s tail represent a symbol of watchfulness. The figure of the peacock is painted in various Islamic religious buildings while in Christianity the peacock was known as the symbol of resurrection and renewal. Just as the peacock elegantly unfolds its vibrant colours during its ritual dance, so too this conference is an occasion for us to demonstrate the best from all participating delegates.
We have more than 300 delegates from 28 countries participating in this meeting. Deviating from the accepted norm of didactic lectures the meeting focusses more on interactive sessions that will promote healthy discussion. There are case based panel discussions and sessions where senior musculoskeletal oncologists share their diverse experiences so that all attendees can benefit from the collective experience of others in order to enable them to make better decisions when managing similar cases. While providing adequate sessions for free papers there are two “show piece” orations. The IMSOS oration “Lessons learned from the European-American Osteosarcoma Study EURAMOS1” will be delivered by Prof. Stefan Bielack while the inaugural APMSTS “Prof. Yoshio Ogihara Oration” will be delivered by Dr. Suresh Nathan. The conference is preceded by multidisciplinary workshops that encompass all specialties involved in sarcoma care, offering delegates an opportunity to interact with faculty in smaller groups.
This unique joint meeting also offers the ideal platform to unveil a brief monograph “IMSOS guidelines for musculoskeletal sarcomas”, IMSOS’s endeavour to standardise the management of musculoskeletal sarcomas in India.
All our efforts are ultimately focussed on improving health care for our patients. A meeting such as this would be incomplete unless we involved them too. The IMSOS sarcoma support initiative will simultaneously conduct a parallel session that helps patients share their experience and presents an occasion where we can felicitate support personnel who selflessly give their time to enable us to optimise overall patient care.
We are sure the ambience and hospitality of Jaipur combined with an exciting and informative scientific programme will make this joint APMSTS – IMSOS conference a memorable and enriching event for all delegates.
It is our pleasure and privilege to welcome all attendees to Jaipur to partake of the experience that is “Incredible India”!
Prof. Ajay Puri
President – Indian Musculo Skeletal Oncology Society
President – Asia Pacific Musculoskeletal Tumor Society
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Denosumab Therapy Related Changes in Giant cell Tumor (Osteoclastoma) of Bone : A New Osteosarcoma Mimicker
Vol 4 | Issue 2 | July-Dec 2018 | Page 4-6 | Pradnya Manglekar, Sujit Joshi, Yogesh Panchwagh.
Authors: Pradnya Manglekar [1], Sujit Joshi [1], Yogesh Panchwagh [1].
[1] Dept. of Pathology Deenanath Mangeshkar Hospital and Research Centre, Pune
[2] Orthopaedic Oncology Clinic, Pune, India.
Address of Correspondence
Dr. Sujit Joshi,
Dept. of Pathology Deenanath Mangeshkar Hospital and Research Centre, Pune
Email: sujitjoshi30@gmail.com
Abstract
Objectives: Giant cell tumor (Osteoclastoma)of bone is locally aggressive osteolytic tumor. Denosumab–A monoclonal antibody against RANKL is recently being used to treat this tumor. We discuss histopathological changes occurring in giant cell tumor of bone after Denosumab treatment.
Method: A retrospective study of 12 cases from January 2014 to March 2018. All patients included were diagnosed as giant cell tumor (osteoclastoma) of bone on needle or open biopsy. Subsequently these patients received Denosumab therapy followed by surgical resection (extended curettage or wide excision). Histomorphological features after Denosumab therapy were evaluated in these specimens and compared with morphological features of prior biopsy samples.
Results: Needle or open biopsy samples studied prior to receiving Denosumab therapy showed typical morphological features of osteoclastoma i.e. presence of uniformly distributed osteoclastic cells and sheets of mononuclear stromal cells. No atypical mitoses or matrix production noted. Post Denosumab therapy resection specimens showed marked reduction in number of osteoclastic giant cells. There was predominance of mononuclear stromal cells along with abundant, irregular new bone (osteoid) formation with osteoid matrix deposition. Occasional mitotic activity was seen. Few foci of necrosis were noted.
Conclusion: Denosumab treatment causes significant giant cell depletion accompanied by abundant new bone formation separated by cellular stromal proliferation. These features bear very little resemblance to their pre-treatment counterparts and can mimic morphological features of osteosarcoma and other bone forming tumors. Hence, one should be aware of these changes so that a misdiagnosis of osteosarcoma can be avoided.
Keywords: Giant cell tumor (osteoclastoma), Denosumab, RANKL.
References
1. Kumar V, Abbas AK, Aster JC. Robbins and Cotran Pathologic Basis of Disease.9th edition. Saunders Elsevier: Philadelphia; 2015. Chapter 26, Bones, Joints and Soft tissue tumors; p1179 – 1226.
2. Rosai J. Rosai and Ackerman’s Surgical Pathology. 10thedition. Elsevier Mosby: St.Louis, Missouri; 2011. Chapter 24, Bone and joints; p2013-2104.
3. Branstetter DG, Nelson SD, Manivel JC, Blay JY et al. Denosumab induces Tumor Reduction and Bone formation in Patients with Giant-Cell Tumor of Bone. Clin Cancer Res; 18(16); 4415-24.2012 AACR.
4. Singh AS, Chawla NS, Chawla SP. Giant-cell tumor of bone: treatment options and role of denosumab. Biologics: Targets and Therapy 2015;9, 69-74.
5. Huang L, Teng XY, Cheng YY, Lee KM, Kumta SM. Expression of preosteoblast markers and Cbfa-1 and Osterix gene transcripts instromal tumour cells of giant cell tumour of bone. Bone 2004 Mar;34 (3):p393–401.
6. James IE, Dodds RA, Olivera DL, Nuttall ME, Gowen M. Human osteoclastoma-derived stromal cells: correlation of the ability to form mineralized nodules in vitro with formation of bone in vivo. J Bone Miner Res 1996 Oct;11 (10):1453–60.
7. Wojcik J, Rosenberg AE, Bredella MA, et al. Denosumab-treated giant cell tumor of bone exhibits morphologic overlap with malignant giant cell tumor of bone. Am J SurgPathol 2016 Jan; 40(1): 72-80.
8. Rekhi B, Verma V, Gulia A, et al. Clinicopathological features of a series of 27 cases of post-denosumab treated giant cell tumors ofbones: a single institutional experience at a tertiary cancer referralcentre, India. PatholOncol Res 2017 Jan; 23 (1): 157-64.
9. Chang-Che Wu, Pin-Pen Hsieh. Denosumab-Treated Giant Cell Tumor of the Bone Mimicking Low-Grade Central Osteosarcoma.Journal of Pathology and Translational Medicine 2018; 52: 133-135.
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JBST- Special APMSTS 2018 Issue
Vol 4 | Issue 2 | July-Dec 2018 | page:1 | Dr. Yogesh Panchwagh, Dr. Ashish Gulia, Dr. Ashok Shyam.
Author: Yogesh Panchwagh [1], Ashish Gulia [2], Ashok Shyam [1,3].
[1] Orthopaedic Oncology Clinic, Pune, India.
[2] Orthopedic Oncology Services, Department of Surgical Oncology, Tata Memorial Hospital, Mumbai.
[3] Indian Orthopaedic Research Group, Thane, India,
[4] 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
JBST- Special APMSTS 2018 Issue
It gives us immense pleasure in presenting the special IMSOS (Indian Musculoskeletal Oncology Society) issue of Journal of bone and soft tissue tumors on the occasion of the 12th Asia Pacific Musculoskeletal Tumor Society meeting, Jaipur, India. It’s only the second time, the first being in 2002, that APMSTS conference is being held in India. It is being co-hosted by the IMSOS, a relatively new body comprising of mostly the young but enthusiastic clinicians and paramedical staff involved in sarcoma care in India. Under the able presidentship of Dr Ajay Puri, a veteran in the field, the 12th APMSTS is all set to give the delegates a memorable experience. It’s but natural that Dr Ajay Puri, President – APMSTS 2018 and IMSOS has penned the guest editorial for this issue.
JBST (Journal of bone and soft tissue tumors) is proud to be associated with IMSOS as its official journal. All the members of IMSOS have contributed wholeheartedly to this issue echoing the theme of APMSTS 2018: “Education – Collaboration – Innovation”. The issue comprises of original articles on the data from institutes located across India on varied topics ranging from demographics of sarcoma in eastern India to osteosarcoma of facial bones from South India to recent topics like post denosumab changes in the histopathology of Giant cell tumors from western India. Even the case reports, which are rare clinical presentations of pathologies like osseous hydatid cyst, chordoma, clear cell chondrosarcoma, sacral inclusion cyst, hold a high educational value.
We also take this opportunity to invite all delegates to submit to Journal of Bone and Soft tissue tumors, which is one of the few journals that publish bone tumor research. We have a great editorial board and an online submission and review system. JBST has completed four years now and we are committed to make JBST the best resource for publishing bone tumor research in the world. This can be only achieved by co-operation and collaboration from all of you
We are happy to present this issue at the 12th APMSTS at Jaipur. Hope you will enjoy it as much as the vibrant, colourful and warm Indian hospitality.
Dr. Yogesh Panchwagh
Dr. Ashish Gulia
Dr. Ashok Shyam
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