Acute pancreatitis due to pancreatic metastasis of osteosarcoma: a report of two cases

Introduction

Pancreatic metastasis is frequently reported in lung cancer, gastrointestinal malignancies, breast cancer, renal cell carcinoma, melanoma, lymphoma, and soft tissue sarcoma. Osteosarcoma is the most common malignant bone tumor in children and adolescents. The most common sites of metastasis are the lungs (1-3). Metastatic pancreatic tumors are uncommon, with an incidence of 1.6–11% in autopsy studies (4). Acute pancreatitis due to pancreatic metastasis has been reported as metastasis-induced acute pancreatitis (MIAP) (5-8). The mechanism of MIAP may be pancreatic ductal obstruction due to tumor growth, impaired blood flow by the direct tumor invasion, or/and enlarged peripancreatic lymph nodes (7). There have been reports of acute pancreatitis due to pancreatic metastasis of various carcinomas such as small cell lung cancer, hepatocellular carcinoma, and renal cell carcinoma (5-8). Metastasis of osteosarcoma to the pancreas is quite rare, and there have been no reports of MIAP due to pancreatic metastasis of osteosarcoma. Here, we present two cases of MIAP due to pancreatic metastasis of osteosarcoma. We obtained written informed consent from patients for present report when they received surgery.

Case presentation

Case 1

A 47-year-old woman was referred to our hospital due to left femur pain. A diagnosis of osteosarcoma of the distal femur was made with open biopsy. Wide tumor resection and reconstruction with endoprosthesis were performed. The pathological findings at surgery showed high-grade osteosarcoma, and she was treated with adjuvant chemotherapy using doxorubicin and ifosfamide postoperatively. However, 1 year and 4 months after initial treatment, the patient experienced lower back pain, and bone metastases were found in the twelfth thoracic vertebral body. A chest CT scan showed that she also had multiple lung metastases. Chemotherapy using gemcitabine and docetaxel was performed, followed by carbon ion radiotherapy for the twelfth thoracic vertebral body. However, the patient still felt back pain 2 years postoperatively. An abdominal CT showed a 7 cm mass in the pancreatic head (Figure 1). Laboratory examination showed increased level of amylase (340 U/L). Considering that the acute pancreatitis due to pancreatic metastasis, palliative radiotherapy (30 Gy/10 fractions) was performed. Although abdominal pain was improved, she had died of multiple metastasis after 2 months.

Figure 1 The CT showed pancreatic metastasis (arrow) in case 1.

Case 2

A 42-year-old woman presented with left knee pain. After X-ray and MRI were performed in the clinic, she was referred to our hospital. A diagnosis of high-grade osteosarcoma was confirmed by open biopsy at the distal part of the femur. She was treated with three cycles of neo-adjuvant chemotherapy using doxorubicin and ifosfamide, followed by wide tumor resection and distal femur replacement. Although postoperative adjuvant chemotherapy was administered, she developed lung metastases 3 months after the initial treatment. Radiofrequency ablation was performed for the multiple lung metastases. However, she felt abdominal and back pain 6 months after initial treatment. CT scans showed a large mass in the pancreatic tail (Figure 2). The mass increased rapidly over a short duration and complicated the acute pancreatitis. The patient’s levels of amylase were also elevated (200 U/L). Palliative radiotherapy (30 Gy/10 fractions) was performed. After 1 month, the patient died of lung metastases.

Figure 2 The CT showed pancreatic metastasis (arrow) in case 2.

Discussion

We presented two case reports of acute pancreatitis due to pancreatic metastasis in patients with osteosarcoma. There are some case reports concerning pancreatic metastasis of osteosarcoma (3,9-15) (Table 1). Ogose et al. reported 223 patients with osteosarcoma between 1975 and 1998, with two patients (0.8%) developing pancreatic metastases (16). Yoon et al. reported 53 patients with pancreatic metastasis between 1997–2009, with only one patient having primary osteosarcoma (17).

Table 1 The list of the osteosarcoma patients with pancreatic metastasis
Full table

Metastases to the pancreas have various symptoms, such as jaundice, abdominal and back pain, vomiting, and nausea. However, some patients are asymptomatic. Metastases are found during regular follow-up examinations for their primary disease (3,11-13). In the present cases, the patients had severe abdominal pain, elevated levels of amylase, and pancreatic metastases combined with acute pancreatitis as diagnosed with CT scans.

The prognosis after pancreatic metastasis is generally poor, because patients will in most cases develop multiple metastases. In previous reports, data related to patient outcomes was limited due to a quite short follow-up. Although there is no evidence that resection for pancreatic metastasis in osteosarcoma is superior to an alternative, surgical management was reported to be effective in patients with renal cell carcinoma (17). If metastasis can be controlled in patients with osteosarcoma, surgery and/or chemotherapy may be indicated.

Acute pancreatitis due to pancreatic metastasis has been reported as MIAP (18). Most cases of MIAP were diagnosed clinically, because tissue diagnosis may be difficult in MIAP due to poor performance status of the patients (19). MIAP is quite rare, with Stewart et al. reporting only one patient diagnosed with MIAP out of 802 lung cancer patients. They also suggested that treatment for pancreatitis should be first considered, and once the pancreatitis is alleviated, treatment for the metastatic disease can be performed (19). In the present cases, palliative radiotherapy was performed, because the patients developed multiple metastases and no further treatment was planned. In previous reports, all patients with pancreatic metastases developed metastases at other sites, such as the lungs and bone. Therefore, we suggest that follow-up examinations at the abdominal lesion may be necessary for patients with metastatic disease.

Acknowledgments

Funding: None.

Footnote

Provenance and Peer Review: This article was commissioned by the Guest Editors (Kurt R. Weiss and Stella Lee) for the series “Osteosarcoma” published in Annals of Joint. The article has undergone external peer review.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at http://dx.doi.org/10.21037/aoj.2019.09.05). The series “Osteosarcoma” was commissioned by the editorial office without any funding or sponsorship. The authors have no other conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Helsinki Declaration (as revised in 2013). We obtained written informed consent from patients for present report when they received surgery.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

References

1. Ferguson WS, Goorin AM. Current treatment of osteosarcoma. Cancer Invest 2001;19:292-315. [Crossref] [PubMed]
2. Lamplot JD, Denduluri S, Qin J, et al. The Current and Future Therapies for Human Osteosarcoma. Curr Cancer Ther Rev 2013;9:55-77. [PubMed]
3. Toyama H, Asari S, Goto T, et al. A case of pancreatic metastasis of osteosarcoma resected using laparoscopic spleen preserving distal pancreatectomy. Gan To Kagaku Ryoho 2016;43:1988-90. [PubMed]
4. Adsay NV, Andea A, Basturk O, et al. Secondary tumors of the pancreas: an analysis of a surgical and autopsy database and review of the literature. Virchows Arch 2004;444:527-35. [Crossref] [PubMed]
5. Kim KH, Kim CD, Lee SJ, et al. Metastasis-induced acute pancreatitis in a patient with small cell carcinoma of the lung. J Korean Med Sci 1999;14:107-9. [Crossref] [PubMed]
6. Okutur K, Bozkurt M, Korkmaz T, et al. Metastasis-induced acute pancreatitis successfully treated with chemotherapy in a patient with small cell lung cancer. Case Rep Oncol Med 2015;2015:304279 [Crossref] [PubMed]
7. Hung SS, Ardalan B. Metastasis-induced acute pancreatitis. West J Med 1991;155:176-8. [PubMed]
8. Asano T, Iwai K, Hazama K, et al. A case of metastasis-induced acute pancreatitis in renal cell carcinoma. Jpn J Gastroenterol Surg 2009;42:1837-42. [Crossref]
9. Aarvold A, Bann S, Giblin V, et al. Osteosarcoma metastasising to the duodenum and pancreas. J Bone Joint Surg Br 2007;89:542-4. [Crossref] [PubMed]
10. Lasithiotakis K, Petrakis I, Georgiadis G, et al. Pancreatic resection for metastasis to the pancreas from colon and lung cancer, and osteosarcoma. JOP 2010;11:593-6. [PubMed]
11. Akpinar B, Obuch J, Fukami N, et al. Unusual presentation of a pancreatic cyst resulting from osteosarcoma metastasis. World J Gastroenterol 2015;21:8452-7. [Crossref] [PubMed]
12. Araújo JC, Carvalho Junior JA, Monges G, et al. Pancreatic and lymph node metastases from maxillary osteosarcoma diagnosed by endoscopic ultrasound-guided fine needle aspiration. Endoscopy 2012;44 Suppl 2 UCTN:E151-2.
13. Rubin E, Dunham WK, Stanley RJ, et al. Pancreatic metastasis in bone sarcoma: CT demonstration. J Comput Assist Tomogr 1985;9:886-8. [Crossref] [PubMed]
14. Avcu S, Akdeniz H, Arslan H, et al. A case of primary vertebral osteosarcoma metastasizing to pancreas. JOP 2009;10:438-40. [PubMed]
15. Jin P, Wang W, Su H, et al. Osteosarcoma metastasizing to pancreas confirmed by endoscopic ultrasound-guided fine-needle aspiration. Endoscopy 2014;46:E109-10. [Crossref] [PubMed]
16. Ogose A, Morita T, Hotta T, et al. Intra-abdominal metastases in musculoskeletal sarcoma. J Orthop Sci 2000;5:463-9. [Crossref] [PubMed]
17. Yoon WJ, Ryu JK, Kim YT, et al. Clinical features of metastatic tumors of the pancreas in Korea: a single-center study. Gut Liver 2011;5:61-4. [Crossref] [PubMed]
18. Gonlugur U, Mirici A, Karaayvaz M. Pancreatic involvement in small cell lung cancer. Radiol Oncol 2014;48:11-9. [Crossref] [PubMed]
19. Stewart KC, Dickout WJ, Urschel JD. Metastasis-induced acute pancreatitis as the initial manifestation of bronchogenic carcinoma. Chest 1993;104:98-100. [Crossref] [PubMed]