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Osteoclastic giant cell tumor of the pancreas with synchronous jejunal gastrointestinal stromal tumor: A case report
Journal Pre-proof Osteoclastic giant cell tumor of the pancreas with synchronous jejunal gastrointestinal stromal tumor: A case report Waad Farhat, Houssem Ammar, Abdelkader Mizouni, Fathia harrabi, Amal Bouazzi, Eya hammami, Rahul Gupta, Mohamed amine said, Mohamed Ben Mabrouk, Ali Ben Ali PII:
S2049-0801(19)30176-1
DOI:
https://doi.org/10.1016/j.amsu.2019.11.008
Reference:
AMSU 1479
To appear in:
Annals of Medicine and Surgery
Received Date: 25 July 2019 Revised Date:
14 November 2019
Accepted Date: 19 November 2019
Please cite this article as: Farhat W, Ammar H, Mizouni A, harrabi F, Bouazzi A, hammami E, Gupta R, said Ma, Ben Mabrouk M, Ben Ali A, Osteoclastic giant cell tumor of the pancreas with synchronous jejunal gastrointestinal stromal tumor: A case report, Annals of Medicine and Surgery, https:// doi.org/10.1016/j.amsu.2019.11.008. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd.
Title page
Title: Osteoclastic giant cell tumor of the pancreas with synchronous jejunal gastrointestinal stromal tumor: A case report Running title: Anaplastic pancreatic carcinoma with jejunal GIST
Authors’ names and degrees: Waad Farhat1, Houssem Ammar1, Abdelkader Mizouni1 ,Fathia harrabi1 , Amal Bouazzi1, Eya hammami2, Rahul Gupta3, Mohamed amine said1,Mohamed Ben Mabrouk1, Ali Ben Ali1
1
Department of Gastrointestinal Surgery, University of Sousse, Sahloul Hospital, Sousse,
Tunisia. 2
Department of Gastroenterology, University of Sousse, Sahloul Hospital, Sousse, Tunisia
3
Department of Gastrointestinal Surgery, Synergy Institute of Medical Sciences, Dehradun,
India. Corresponding author’s details: Waad farhat Department of digestive Surgery, Hopital Sahloul, Sousse, Tunisia Address: Route ceinture cité Sahloul, 4054 ,Sousse Email id: [email protected] Mobile No: 0021658133172
Financial support: None Informed consent: The patient provided informed written consent prior to submission of this manuscript. Conflict of interest: The authors declare no conflict of interest.
Patient consent: Informed consent was taken from the patients for this study.
Ethical approval: All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional research committee.
Title: Osteoclastic giant cell tumor of the pancreas with synchronous jejunal gastrointestinal stromal tumor: A case report
Abstract Osteoclastic giant cell tumor of the pancreas is a rare aggressive tumor, counting for 2-7% of all pancreatic cancers. Surgery is considered the most appropriate treatment. We report a case of a 84-year-old man with incidentally detected 11cm tumor in the pancreatic tail and another 6 cm tumor located in the jejunum on abdominal computed tomography. The patient underwent distal pancreatectomy with splenectomy along with segmental resection of the tumor bearing part of the jejunum. On histological examination, osteoclast-like giant cells with some areas of metaplastic bone were observed which confirmed the diagnosis of osteoclastic tumor of the pancreas. The jejunal tumor was strongly c-kit positive on immunohistochemistry which confirmed the diagnosis of GIST. On the last follow up at 2 years after surgery, there is no evidence of recurrence or distant metastasis. Pancreatic OGCT has a better prognosis after resection than pancreatic adenocarcinoma. Its co- existence jejunal GIST, as seen in the index case, has not been reported in the English literature till date.
Abbreviations OGCT = Osteoclastic giant cell tumor, GIST=Stromal gastrointestinal tumor, CT= computed tomography, FNA= fine needle aspiration, EMA= epithelial membrane antigen, Key words OGCT of the pancreas; jejunal GIST
Introduction Osteoclastic giant cell tumor (OGCT) of the pancreas is a rare aggressive tumor, which accounts for 2-7% of all pancreatic cancers [1]. In 2010, the World Health Organization classified these tumors as variants of pancreatic ductal adenocarcinoma under the heading “undifferentiated carcinoma with osteoclastic giant cells,” and they have been identified as fundamentally epithelial-derived tumors with mesenchymal differentiation [1-2]. Here, we report a case of OGCT of the pancreas associated with jejunal gastrointestinal stromal tumor (GIST). This is the first case report of the co-existence of these two tumors in the English literature till date. This case has been reported in line with the SCARE criteria [3].
Case report A 84 -years –old lady with presented with epigastric pain and vomiting for 3 months. The pain was dull aching in nature, paroxysmal, unrelated to food and without radiation. There was no history of associated hematemesis, or weight loss. She had past history of myocardial infarction 3 years back for which she was taking oral antiplatelet therapy, Physical examination revealed an epigastric intra-abdominal mass measuring around 4 x 4 cm, firm in consistency with rounded borders. Routine blood investigations and tumor markers (serum CA 19-9, CEA) were within normal range. Upper gastrointestinal endoscopy was unremarkable. Abdominal ultrasound (US) showed a 10 × 7 cm well-defined, round-like, mixed cystic and solid mass in the body of the pancreas. On contrast enhanced computed
tomography of the abdomen (CT), a well-defined solid-cystic mass of 11 x 7 x 6 cm was found involving the body and tail of the pancreas (Figure 1). The cystic lesion had multiple septae which showed contrast enhancement with solid areas without involvement of the surrounding structures such as bile duct, splenic vessels, stomach and transverse colon. The main pancreatic duct could not be separately identified from the tumor. Additionally, there was a 6 cm tumor in close relation with the proximal jejunum (Figure 1). Based on the imaging findings, a provisional diagnosis of malignant/ borderline malignant pancreatic tumor such as solid pseudopapillary tumor or neuroendocrine tumor or GIST along with benign jejunal tumor was made. As the lesions were resectable on CT and patient was symptomatic, further investigations like endoscopic ultrasound (EUS) or magnetic resonance imaging (MRI) were not performed and the patient was planned for surgical resection. On laparotomy, the pancreatic tumor was found to be involving the body and tail of the pancreas for which distal pancreatectomy with splenectomy was performed. Another tumor was arising from the proximal jejunum for which segmental resection with end to end anastomosis was performed. The operative time was 90 minutes and estimated blood loss was 50 ml. Postoperative course of the patient was uneventful. The hospital stay was 4 days. Macroscopically, it was 11-centimeter well-defined tumor involving the body and tail of the pancreas. On cut section, areas of necrosis could be observed. On histological examination, the tumor cells were fusiform, epithelioid or polygonal in appearance with atypical nuclei. Osteoclast-like giant cells were observed within the stroma, consisting the major component of the tumor. (Figure 2). Some areas of metaplastic bone were also noticed. Eight lymph nodes were examined and all were tumor-free. All the resection margins were free of tumor. The jejunal tumor showed spindle cells with ovoid shaped nuclei and were arranged in fasciculate pattern suggestive of gastrointestinal stromal tumor (GIST) (Figure 2). On immunochemistry, intestinal GIST was positive for cKIT and DOG1 while the pancreatic
OGCT was negative for these markers and strongly expressed CD68, a specific marker of the macrophage/monocyte/histiocyte phagocytic activities suggesting no similarities between these two tumors on histology and immunohistochemistry. On the last follow up at 2 years after surgery, there is no evidence of recurrence or distant metastasis on CT abdomen and pelvis. Discussion Osteoclastic giant cell tumor (OGCT) is a rare, distinctive, malignant pancreatic tumor that is typically large, frequently has a pancreatic ductal adenocarcinoma component, and has 3 classical cell types: osteoclastic, pleomorphic, and mixed. Pancreatic OGCT is more frequent in women with mean age of 58 years [2]. The symptoms are mostly nonspecific. Characteristically, OGCs are large at presentation and show focal hemorrhage and necrosis, but seem slow to give rise to metastases [1-4]. Radiologically, they appear as solid-cystic tumors with delayed enhancement and difficult to differentiate from other pancreatic tumors. Reid et al. have reported a series of 15 patients with OGCT, diagnosed via endoscopic ultrasound or CT guided fine needle aspiration (FNA), demonstrating the efficacy of the technique in this setting [2]. Lesions that appear to be surgically resectable should undergo operative intervention. Histological examination reveals numerous osteoclast-like giant cells set in a sarcomatous stroma, the appearances being similar to those seen in giant cell tumors of bone. Fusiform and pleomorphic cells express the cytokeratin, the epithelial membrane antigen (EMA) and the vimentin antigens. The osteoclast-like giant cells express the CD68 and rarely the vimentin [2-4]. CD68 is highly expressed in macrophages, monocytes and histiocytes. It is also called as phagocyte marker. But the exact origin of these osteoclastic cells in pancreatic tumor is not known. They are distinct from pleomorphic giant cell
carcinomas of the pancreas and have a slightly better prognosis after resection than ductal adenocarcinoma [2-5]. GIST are the most common mesenchymal tumors of the gastrointestinal tract and usually show cKIT and DOG1 positivity on immunohistochemistry. The existence of pancreatic OGCT and jejunal GIST, as seen in the index case, has not been reported in the English literature till date. However, there are some reported cases of the co-existence of GIST having osteoclastic –like giant cells associated with other abdominal tumors [6-7]. Therefore, these findings lead us to consider the hypothesis of common etiology, making the synchronous occurrence of GIST and other abdominal malignancy not just a coincidence. The prognosis in these patients is predominantly determined by the other malignancy and not influenced by GIST [5]. Conclusion Pancreatic OGCT are rare tumors, their association with jejunal GIST is even rarer. This is the first case to be reported in literature associating these two tumors. Its prognosis is better than pancreatic adenocarcinoma and its evolution is generally favorable after total surgical resection.
Provenance and peer review Not commissioned, externally peer reviewed.
References
1.
Osaka H, Yashiro M, Nishino H, et al. A case of osteoclast-type giant cell tumor of the pancreas with high-frequency microsatellite instability. Pancreas. 2004;29:23941.
2. Muraki T, Reid MD, Basturk O, et al. Undifferentiated carcinoma with osteoclastic giant cells of the pancreas: clinicopathological analysis of 38 cases highlights a more protracted clinical course than currently appreciated. Am J Surg Pathol. 2016;40:1203-1216. 3. Agha RA, Borrelli MR, Farwana R, Koshy K, Fowler A, Orgill DP, For the SCARE Group. The SCARE 2018 Statement: Updating Consensus Surgical CAse REport (SCARE) Guidelines. International Journal of Surgery 2018;60:132-136
4. Reid MD, Muraki T, HooKim K, et al. Cytologic features and clinical implications of undifferentiated carcinoma with osteoclastic giant cells of the pancreas: an analysis of 15 cases. Cancer Cytopathol. 2017;125:563-75. 5. Vassos N, Agaimy A, Hohenberger W, Croner RS. Coexistence of gastrointestinal stromal tumors (GIST) and malignant neoplasms of different origin: prognostic implications. Int J Surg. 2014;12:371-77. 6. Leung KM, Wong S, Chow TC, et al. A malignant gastrointestinal stromal tumor with osteoclast-like giant cells. Arch Pathol Lab Med. 2002;126:972-974. 7. Insabato L, Di Vizio D, Ciancia G, et al. Malignant gastrointestinal leiomyosarcoma and gastrointestinal stromal tumor with prominent osteoclast-like giant cells. Arch Pathol Lab Med. 2004;128:440-3.
Figure legends Figure 1 – CT abdomen: (a) axial section showing the pancreatic tumor, (b) coronal section showing the pancreatic tumor (red arrow) and the jejunal GIST (yellow arrow). Figure 2 – Microscopic examination: (a) pancreatic tumor showing a predominant giant osteoclastic like cells component (HEx100), (b) foci of osteoid bone was present within the pancreatic tumor (HEx400), (c) jejunal gastrointestinal stromal tumor made of spindle cells with ovoid shaped nuclei arranged in fascicles (HEx200).
Highlights: •
Osteoclastic giant cell tumor of the pancreas is a rare aggressive tumor, counting 2-7% of all pancreatic cancers.
•
Surgery is considered the most appropriate treatment .
•
Pancreatic OGCT has a better prognosis, after resection than pancreatic adenocarcinoma.
•
Its co- existence jejunal GIST, as seen in the index case, has not been reported in the English literature till date.
Annals of Medicine and Surgery The following information is required for submission. Please note that failure to respond to these questions/statements will mean your submission will be returned. If you have nothing to declare in any of these categories then this should be stated. Please state any conflicts of interest All authors must disclose any financial and personal relationships with other people or organisations that could inappropriately influence (bias) their work. Examples of potential conflicts of interest include employment, consultancies, stock ownership, honoraria, paid expert testimony, patent applications/registrations, and grants or other funding.
The authors declare that they have no conflict of interest.
Please state any sources of funding for your research All sources of funding should be declared as an acknowledgement at the end of the text. Authors should declare the role of study sponsors, if any, in the collection, analysis and interpretation of data; in the writing of the manuscript; and in the decision to submit the manuscript for publication. If the study sponsors had no such involvement, the authors should so state.
This study has not received any funding.
Ethical Approval Research studies involving patients require ethical approval. Please state whether approval has been given, name the relevant ethics committee and the state the reference number for their judgement.
The study was approved by Ethics Committee
Consent Studies on patients or volunteers require ethics committee approval and fully informed written consent which should be documented in the paper. Authors must obtain written and signed consent to publish a case report from the patient (or, where applicable, the patient's guardian or next of kin) prior to submission. We ask Authors to confirm as part of the submission process that such consent has been obtained, and the manuscript must include a statement to this effect in a consent section at the end of the manuscript, as follows: "Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request”. Patients have a right to privacy. Patients’ and volunteers' names, initials, or hospital numbers should not be used. Images of patients or volunteers should not be used unless the information is essential for scientific purposes and explicit permission has been given as part of the consent. If such consent is made subject to any conditions, the Editor in Chief must be made aware of all such conditions. Even where consent has been given, identifying details should be omitted if they are not essential. If identifying characteristics are altered to protect anonymity, such as in genetic pedigrees, authors should provide assurance that alterations do not distort scientific meaning and editors should so note.
Written informed consent was obtained from the patient.
Author contribution Please specify the contribution of each author to the paper, eg study concept or design, data collection, data analysis or interpretation, writing the paper, others, who have contributed in othe ways should be listed as contributors.
Study concept or design – MBM, HA, Data collection – HA, WF, RG Data interpretation – MBM, AB, EH Literature review – WF, SL, EH,ABA Drafting of the paper – HA, MBM, SL Editing of the paper – MBM, WF,AM
Registration of Research Studies In accordance with the Declaration of Helsinki 2013, all research involving human participants has to be registered in a publicly accessible database. Please enter the name of the registry and the unique identifying number (UIN) of your study. You can register any type of research at http://www.researchregistry.com to obtain your UIN if you have not already registered. This is mandatory for human studies only. Trials and certain observational research can also be registered elsewhere such as: ClinicalTrials.gov or ISRCTN or numerous other registries.
1.
Name of the registry:
2.
Unique Identifying number or registration ID:
3.
Hyperlink to the registration (must be publicly accessible):
As this was a case report and not a clinical trial, this study does not require registration.
Guarantor
The Guarantor is the one or more people who accept full responsibility for the work and/or the conduct of the study, had access to the data, and controlled the decision to publish
Waad farhat
S2049-0801(19)30176-1
DOI:
https://doi.org/10.1016/j.amsu.2019.11.008
Reference:
AMSU 1479
To appear in:
Annals of Medicine and Surgery
Received Date: 25 July 2019 Revised Date:
14 November 2019
Accepted Date: 19 November 2019
Please cite this article as: Farhat W, Ammar H, Mizouni A, harrabi F, Bouazzi A, hammami E, Gupta R, said Ma, Ben Mabrouk M, Ben Ali A, Osteoclastic giant cell tumor of the pancreas with synchronous jejunal gastrointestinal stromal tumor: A case report, Annals of Medicine and Surgery, https:// doi.org/10.1016/j.amsu.2019.11.008. This is a PDF file of an article that has undergone enhancements after acceptance, such as the addition of a cover page and metadata, and formatting for readability, but it is not yet the definitive version of record. This version will undergo additional copyediting, typesetting and review before it is published in its final form, but we are providing this version to give early visibility of the article. Please note that, during the production process, errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain. © 2019 Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd.
Title page
Title: Osteoclastic giant cell tumor of the pancreas with synchronous jejunal gastrointestinal stromal tumor: A case report Running title: Anaplastic pancreatic carcinoma with jejunal GIST
Authors’ names and degrees: Waad Farhat1, Houssem Ammar1, Abdelkader Mizouni1 ,Fathia harrabi1 , Amal Bouazzi1, Eya hammami2, Rahul Gupta3, Mohamed amine said1,Mohamed Ben Mabrouk1, Ali Ben Ali1
1
Department of Gastrointestinal Surgery, University of Sousse, Sahloul Hospital, Sousse,
Tunisia. 2
Department of Gastroenterology, University of Sousse, Sahloul Hospital, Sousse, Tunisia
3
Department of Gastrointestinal Surgery, Synergy Institute of Medical Sciences, Dehradun,
India. Corresponding author’s details: Waad farhat Department of digestive Surgery, Hopital Sahloul, Sousse, Tunisia Address: Route ceinture cité Sahloul, 4054 ,Sousse Email id: [email protected] Mobile No: 0021658133172
Financial support: None Informed consent: The patient provided informed written consent prior to submission of this manuscript. Conflict of interest: The authors declare no conflict of interest.
Patient consent: Informed consent was taken from the patients for this study.
Ethical approval: All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional research committee.
Title: Osteoclastic giant cell tumor of the pancreas with synchronous jejunal gastrointestinal stromal tumor: A case report
Abstract Osteoclastic giant cell tumor of the pancreas is a rare aggressive tumor, counting for 2-7% of all pancreatic cancers. Surgery is considered the most appropriate treatment. We report a case of a 84-year-old man with incidentally detected 11cm tumor in the pancreatic tail and another 6 cm tumor located in the jejunum on abdominal computed tomography. The patient underwent distal pancreatectomy with splenectomy along with segmental resection of the tumor bearing part of the jejunum. On histological examination, osteoclast-like giant cells with some areas of metaplastic bone were observed which confirmed the diagnosis of osteoclastic tumor of the pancreas. The jejunal tumor was strongly c-kit positive on immunohistochemistry which confirmed the diagnosis of GIST. On the last follow up at 2 years after surgery, there is no evidence of recurrence or distant metastasis. Pancreatic OGCT has a better prognosis after resection than pancreatic adenocarcinoma. Its co- existence jejunal GIST, as seen in the index case, has not been reported in the English literature till date.
Abbreviations OGCT = Osteoclastic giant cell tumor, GIST=Stromal gastrointestinal tumor, CT= computed tomography, FNA= fine needle aspiration, EMA= epithelial membrane antigen, Key words OGCT of the pancreas; jejunal GIST
Introduction Osteoclastic giant cell tumor (OGCT) of the pancreas is a rare aggressive tumor, which accounts for 2-7% of all pancreatic cancers [1]. In 2010, the World Health Organization classified these tumors as variants of pancreatic ductal adenocarcinoma under the heading “undifferentiated carcinoma with osteoclastic giant cells,” and they have been identified as fundamentally epithelial-derived tumors with mesenchymal differentiation [1-2]. Here, we report a case of OGCT of the pancreas associated with jejunal gastrointestinal stromal tumor (GIST). This is the first case report of the co-existence of these two tumors in the English literature till date. This case has been reported in line with the SCARE criteria [3].
Case report A 84 -years –old lady with presented with epigastric pain and vomiting for 3 months. The pain was dull aching in nature, paroxysmal, unrelated to food and without radiation. There was no history of associated hematemesis, or weight loss. She had past history of myocardial infarction 3 years back for which she was taking oral antiplatelet therapy, Physical examination revealed an epigastric intra-abdominal mass measuring around 4 x 4 cm, firm in consistency with rounded borders. Routine blood investigations and tumor markers (serum CA 19-9, CEA) were within normal range. Upper gastrointestinal endoscopy was unremarkable. Abdominal ultrasound (US) showed a 10 × 7 cm well-defined, round-like, mixed cystic and solid mass in the body of the pancreas. On contrast enhanced computed
tomography of the abdomen (CT), a well-defined solid-cystic mass of 11 x 7 x 6 cm was found involving the body and tail of the pancreas (Figure 1). The cystic lesion had multiple septae which showed contrast enhancement with solid areas without involvement of the surrounding structures such as bile duct, splenic vessels, stomach and transverse colon. The main pancreatic duct could not be separately identified from the tumor. Additionally, there was a 6 cm tumor in close relation with the proximal jejunum (Figure 1). Based on the imaging findings, a provisional diagnosis of malignant/ borderline malignant pancreatic tumor such as solid pseudopapillary tumor or neuroendocrine tumor or GIST along with benign jejunal tumor was made. As the lesions were resectable on CT and patient was symptomatic, further investigations like endoscopic ultrasound (EUS) or magnetic resonance imaging (MRI) were not performed and the patient was planned for surgical resection. On laparotomy, the pancreatic tumor was found to be involving the body and tail of the pancreas for which distal pancreatectomy with splenectomy was performed. Another tumor was arising from the proximal jejunum for which segmental resection with end to end anastomosis was performed. The operative time was 90 minutes and estimated blood loss was 50 ml. Postoperative course of the patient was uneventful. The hospital stay was 4 days. Macroscopically, it was 11-centimeter well-defined tumor involving the body and tail of the pancreas. On cut section, areas of necrosis could be observed. On histological examination, the tumor cells were fusiform, epithelioid or polygonal in appearance with atypical nuclei. Osteoclast-like giant cells were observed within the stroma, consisting the major component of the tumor. (Figure 2). Some areas of metaplastic bone were also noticed. Eight lymph nodes were examined and all were tumor-free. All the resection margins were free of tumor. The jejunal tumor showed spindle cells with ovoid shaped nuclei and were arranged in fasciculate pattern suggestive of gastrointestinal stromal tumor (GIST) (Figure 2). On immunochemistry, intestinal GIST was positive for cKIT and DOG1 while the pancreatic
OGCT was negative for these markers and strongly expressed CD68, a specific marker of the macrophage/monocyte/histiocyte phagocytic activities suggesting no similarities between these two tumors on histology and immunohistochemistry. On the last follow up at 2 years after surgery, there is no evidence of recurrence or distant metastasis on CT abdomen and pelvis. Discussion Osteoclastic giant cell tumor (OGCT) is a rare, distinctive, malignant pancreatic tumor that is typically large, frequently has a pancreatic ductal adenocarcinoma component, and has 3 classical cell types: osteoclastic, pleomorphic, and mixed. Pancreatic OGCT is more frequent in women with mean age of 58 years [2]. The symptoms are mostly nonspecific. Characteristically, OGCs are large at presentation and show focal hemorrhage and necrosis, but seem slow to give rise to metastases [1-4]. Radiologically, they appear as solid-cystic tumors with delayed enhancement and difficult to differentiate from other pancreatic tumors. Reid et al. have reported a series of 15 patients with OGCT, diagnosed via endoscopic ultrasound or CT guided fine needle aspiration (FNA), demonstrating the efficacy of the technique in this setting [2]. Lesions that appear to be surgically resectable should undergo operative intervention. Histological examination reveals numerous osteoclast-like giant cells set in a sarcomatous stroma, the appearances being similar to those seen in giant cell tumors of bone. Fusiform and pleomorphic cells express the cytokeratin, the epithelial membrane antigen (EMA) and the vimentin antigens. The osteoclast-like giant cells express the CD68 and rarely the vimentin [2-4]. CD68 is highly expressed in macrophages, monocytes and histiocytes. It is also called as phagocyte marker. But the exact origin of these osteoclastic cells in pancreatic tumor is not known. They are distinct from pleomorphic giant cell
carcinomas of the pancreas and have a slightly better prognosis after resection than ductal adenocarcinoma [2-5]. GIST are the most common mesenchymal tumors of the gastrointestinal tract and usually show cKIT and DOG1 positivity on immunohistochemistry. The existence of pancreatic OGCT and jejunal GIST, as seen in the index case, has not been reported in the English literature till date. However, there are some reported cases of the co-existence of GIST having osteoclastic –like giant cells associated with other abdominal tumors [6-7]. Therefore, these findings lead us to consider the hypothesis of common etiology, making the synchronous occurrence of GIST and other abdominal malignancy not just a coincidence. The prognosis in these patients is predominantly determined by the other malignancy and not influenced by GIST [5]. Conclusion Pancreatic OGCT are rare tumors, their association with jejunal GIST is even rarer. This is the first case to be reported in literature associating these two tumors. Its prognosis is better than pancreatic adenocarcinoma and its evolution is generally favorable after total surgical resection.
Provenance and peer review Not commissioned, externally peer reviewed.
References
1.
Osaka H, Yashiro M, Nishino H, et al. A case of osteoclast-type giant cell tumor of the pancreas with high-frequency microsatellite instability. Pancreas. 2004;29:23941.
2. Muraki T, Reid MD, Basturk O, et al. Undifferentiated carcinoma with osteoclastic giant cells of the pancreas: clinicopathological analysis of 38 cases highlights a more protracted clinical course than currently appreciated. Am J Surg Pathol. 2016;40:1203-1216. 3. Agha RA, Borrelli MR, Farwana R, Koshy K, Fowler A, Orgill DP, For the SCARE Group. The SCARE 2018 Statement: Updating Consensus Surgical CAse REport (SCARE) Guidelines. International Journal of Surgery 2018;60:132-136
4. Reid MD, Muraki T, HooKim K, et al. Cytologic features and clinical implications of undifferentiated carcinoma with osteoclastic giant cells of the pancreas: an analysis of 15 cases. Cancer Cytopathol. 2017;125:563-75. 5. Vassos N, Agaimy A, Hohenberger W, Croner RS. Coexistence of gastrointestinal stromal tumors (GIST) and malignant neoplasms of different origin: prognostic implications. Int J Surg. 2014;12:371-77. 6. Leung KM, Wong S, Chow TC, et al. A malignant gastrointestinal stromal tumor with osteoclast-like giant cells. Arch Pathol Lab Med. 2002;126:972-974. 7. Insabato L, Di Vizio D, Ciancia G, et al. Malignant gastrointestinal leiomyosarcoma and gastrointestinal stromal tumor with prominent osteoclast-like giant cells. Arch Pathol Lab Med. 2004;128:440-3.
Figure legends Figure 1 – CT abdomen: (a) axial section showing the pancreatic tumor, (b) coronal section showing the pancreatic tumor (red arrow) and the jejunal GIST (yellow arrow). Figure 2 – Microscopic examination: (a) pancreatic tumor showing a predominant giant osteoclastic like cells component (HEx100), (b) foci of osteoid bone was present within the pancreatic tumor (HEx400), (c) jejunal gastrointestinal stromal tumor made of spindle cells with ovoid shaped nuclei arranged in fascicles (HEx200).
Highlights: •
Osteoclastic giant cell tumor of the pancreas is a rare aggressive tumor, counting 2-7% of all pancreatic cancers.
•
Surgery is considered the most appropriate treatment .
•
Pancreatic OGCT has a better prognosis, after resection than pancreatic adenocarcinoma.
•
Its co- existence jejunal GIST, as seen in the index case, has not been reported in the English literature till date.
Annals of Medicine and Surgery The following information is required for submission. Please note that failure to respond to these questions/statements will mean your submission will be returned. If you have nothing to declare in any of these categories then this should be stated. Please state any conflicts of interest All authors must disclose any financial and personal relationships with other people or organisations that could inappropriately influence (bias) their work. Examples of potential conflicts of interest include employment, consultancies, stock ownership, honoraria, paid expert testimony, patent applications/registrations, and grants or other funding.
The authors declare that they have no conflict of interest.
Please state any sources of funding for your research All sources of funding should be declared as an acknowledgement at the end of the text. Authors should declare the role of study sponsors, if any, in the collection, analysis and interpretation of data; in the writing of the manuscript; and in the decision to submit the manuscript for publication. If the study sponsors had no such involvement, the authors should so state.
This study has not received any funding.
Ethical Approval Research studies involving patients require ethical approval. Please state whether approval has been given, name the relevant ethics committee and the state the reference number for their judgement.
The study was approved by Ethics Committee
Consent Studies on patients or volunteers require ethics committee approval and fully informed written consent which should be documented in the paper. Authors must obtain written and signed consent to publish a case report from the patient (or, where applicable, the patient's guardian or next of kin) prior to submission. We ask Authors to confirm as part of the submission process that such consent has been obtained, and the manuscript must include a statement to this effect in a consent section at the end of the manuscript, as follows: "Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request”. Patients have a right to privacy. Patients’ and volunteers' names, initials, or hospital numbers should not be used. Images of patients or volunteers should not be used unless the information is essential for scientific purposes and explicit permission has been given as part of the consent. If such consent is made subject to any conditions, the Editor in Chief must be made aware of all such conditions. Even where consent has been given, identifying details should be omitted if they are not essential. If identifying characteristics are altered to protect anonymity, such as in genetic pedigrees, authors should provide assurance that alterations do not distort scientific meaning and editors should so note.
Written informed consent was obtained from the patient.
Author contribution Please specify the contribution of each author to the paper, eg study concept or design, data collection, data analysis or interpretation, writing the paper, others, who have contributed in othe ways should be listed as contributors.
Study concept or design – MBM, HA, Data collection – HA, WF, RG Data interpretation – MBM, AB, EH Literature review – WF, SL, EH,ABA Drafting of the paper – HA, MBM, SL Editing of the paper – MBM, WF,AM
Registration of Research Studies In accordance with the Declaration of Helsinki 2013, all research involving human participants has to be registered in a publicly accessible database. Please enter the name of the registry and the unique identifying number (UIN) of your study. You can register any type of research at http://www.researchregistry.com to obtain your UIN if you have not already registered. This is mandatory for human studies only. Trials and certain observational research can also be registered elsewhere such as: ClinicalTrials.gov or ISRCTN or numerous other registries.
1.
Name of the registry:
2.
Unique Identifying number or registration ID:
3.
Hyperlink to the registration (must be publicly accessible):
As this was a case report and not a clinical trial, this study does not require registration.
Guarantor
The Guarantor is the one or more people who accept full responsibility for the work and/or the conduct of the study, had access to the data, and controlled the decision to publish
Waad farhat