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Osteosarcoma with delayed metastasis to the stomach
Journal of Pediatric Surgery (2007) 42, 737 – 739
www.elsevier.com/locate/jpedsurg
Osteosarcoma with delayed metastasis to the stomach Vivian E. Stronga, Jaime Shalkowb, Cristina R. Antonescuc, Paul Meyersb, Michael P. La Quagliab,* a
Department of Surgery, Memorial Sloan-Kettering Cancer Center, New York, NY 1002, USA Department of Pediatrics, Memorial Sloan-Kettering Cancer Center, New York, NY 1002, USA c Department of Pathology, Memorial Sloan-Kettering Cancer Center, New York, NY 1002, USA b
Index words: Osteosarcoma; Metastasis; Pediatric
Abstract This case illustrates a primary osteosarcoma in a child that has metastasized to the stomach. Such a presentation has only been documented once before and raises the importance of vigilant and thorough follow-up. In addition, this case questions the relationship of prolonged treatment and subsequent sites of recurrence. D 2007 Elsevier Inc. All rights reserved.
Osteosarcoma is the most common primary bone tumor in children, and the lung is the most frequent metastatic site. We report herein a case of a primary osteosarcoma with an exceptionally unusual course. This patient, with a gastric metastasis, is to our knowledge the first such case occurring in the United States.
1. Case report A previously healthy 17-year-old white adolescent boy sustained a knee injury while playing volleyball in September 1998. The patient sought medical attention several months later when the pain persisted, also complaining of low-grade fevers, night sweats, and a 15-lb weight loss. Radiological workup of the right lower extremity with magnetic resonance imaging and computed
* Corresponding author. Department of Surgery, Memorial SloanKettering Cancer Center, New York, NY 1002, USA. Tel.: +1 212 639 7002; fax: +1 212 717 3373. E-mail address: [email protected] (M.P. La Quaglia). 0022-3468/$ – see front matter D 2007 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2006.12.005
tomographic scan revealed an ill-defined lesion on the right femoral distal metaphysis with extension into the epiphysis and upper diaphysis. He underwent a core needle biopsy and a staging computed tomographic scan and was diagnosed with a high-grade osteosarcoma of the distal right femur and bilateral pulmonary metastases. The patient was treated with several cycles of a multidrug chemotherapy regimen that included doxorubicin, ifosfamide, cisplatin, and high-dose methotrexate between April 1999 and November 2000. In July 1999, he underwent resection of left pulmonary metastases, and in August 1999, a limbpreserving, en bloc resection of the primary tumor with placement of a knee prosthesis. Pathologic examination revealed a high-grade osteosarcoma, osteoblastic type, with a good chemotherapeutic response (grade III, 90% necrosis) and focal cortical bone breakthrough into surrounding soft tissues. Two months later, the patient underwent an elective right thoracotomy with multiple wedge resections of his pulmonary metastases. The pathology report revealed metastatic osteosarcoma with 80% to 90% necrosis, associated with reactive bronchiolitis obliterans. He first relapsed in November 2000 with right lung nodules and underwent a
738
Fig. 1 Intraoperative photograph showing the anterior wall of the gastric antrum with the invaginated lesion. The stomach wall is being retracted with silk stitches, and the mass has been highlighted with purple ink.
second right thoracotomy for resection of recurrent pulmonary metastases. In June 2001, he again relapsed with a metastatic lesion to the left buttock that was resected and resumed chemotherapy with high-dose ifosfamide and etoposide until November 2001. The patient was without further findings until February 2002, when he noted black, tarry stools and vomiting followed by a syncopal episode. His hemoglobin was 5 g/dL, and he was transfused. An upper gastrointestinal endoscopy revealed a 3-cm-diameter lesion of the gastric antrum with an ulcerated surface and a clot attached to it. A biopsy was consistent with osteosarcoma, and he subsequently underwent a resection of the gastric tumor with negative margins. Fig. 1 shows a gross picture of the gastric lesion intraoperatively, and Figs. 2 and 3 show a gross picture and microscopic section, respectively. The gastric tumor showed predominantly
Fig. 2 Macroscopic appearance of the interior of the resected gastric wall, with the well-defined osteogenic sarcoma tumor showing a white-tan cut surface with focal hemorrhagic areas.
V.E. Strong et al.
Fig. 3 Microphotograph of the specimen showing normal gastric mucosa on the right and a highly cellular spindle cell proliferation with focal osteoid production on the left, consistent with metastatic osteogenic sarcoma.
viable, osteoblastic-type osteosarcoma. The patient is currently at home and stable.
2. Discussion Approximately 20% of patients who are diagnosed with osteosarcoma have distant metastases at diagnosis. The lung is the most common site, and metasectomy is associated with improved survival [1]. Other sites of metastases include the bones, liver, and pleural extension from pulmonary lesions [2]. Although other metastatic sites have been reported, they are exceptionally rare. In contrast, primary extraosseous osteosarcoma is more aggressive than osteosarcoma of bone and is more likely to occur in unusual sites including soft tissue and small intestine [3,4]. Intestinal involvement, in particular, has only been reported in patients with extraosseous primary lesions. There is only one report of a metastatic recurrence of osteosarcoma in the stomach. This is a single case report from Germany. This metastasis was presumed to arise from a primary bone osteosarcoma. A history of an eye excision early in infancy for unknown reasons raises the possibility of retinoblastoma. Hereditary and sporadic retinoblastoma is known to predispose to a higher frequency of osteosarcoma as a secondary malignancy and may have affected this patient’s presentation [5]. In our case, we report a male with a definitive primary osteosarcoma of the bone that developed metastatic lesions in the lung, buttock, and most recently gastric antrum. It is of interest that 2 years elapsed from initial presentation to the gastric metastasis, and this was after multiple cycles of chemotherapy. Guiliano et al [6] addressed the question of whether advances in therapy have altered the pattern of recurrences
Osteosarcoma with delayed metastasis to the stomach in patients treated for osteosarcoma. They looked at the clinical course of more than 100 patients with high-grade skeletal osteosarcoma from 1971 to 1982. Fifty-three percent of the patients developed clinical metastases, and 61% of these were to the lung. Ninety-five percent of patients treated by amputation alone developed pulmonary metastases as the site of initial recurrence. In contrast, 45% of patients treated after 1974 with surgery and chemotherapy developed pulmonary metastases, whereas 28% from this group developed extrapulmonary metastases as the initial site of recurrent disease. This article goes on to suggest that patterns of metastatic recurrence may be altered by current therapeutic regimens [6]. The stomach is an extremely unusual site for a metastatic lesion of osteosarcoma. To our knowledge, this report is the second case reported in the literature and the first occurring in the United States. This unusual site of recurrence may be related to multiple courses of therapy. Prolonged treatment
739 of metastatic sarcoma may be associated with uncommon sites of recurrence.
References [1] LaQuaglia MP. Osteosarcoma: specific tumor management and results. Chest Surg Clin N Am 1998;8:77 - 95. [2] Goorin AM, Abelson HT, Frei E. Osteosarcoma: fifteen years later. N Engl J Med 1985;313:1637 - 43. [3] Nojima T, Gebhardt MC, Mankin HJ, et al. Extraosseous osteosarcoma presenting with intestinal hemorrhage: case report and literature review. Hum Pathol 1986;17:85 - 7. [4] Donoso L, Valls R, Montserrat E, et al. CT findings in a case of polypoid-like metastases in the ileum from extraosseous osteosarcoma. Eur J Radiol 1988;8:100 - 1. [5] Overberg-Schmidt US, Baumgarten E, Bassir C, et al. The stomach and pulmonary interlobular space as unusual sites of osteosarcoma. Pediatr Hematol Oncol 1999;16:61 - 6. [6] Giuliano AE, Feig S, Eilber FR. Changing metastatic patterns of osteosarcoma. Cancer 1984;54:2160 - 4.
www.elsevier.com/locate/jpedsurg
Osteosarcoma with delayed metastasis to the stomach Vivian E. Stronga, Jaime Shalkowb, Cristina R. Antonescuc, Paul Meyersb, Michael P. La Quagliab,* a
Department of Surgery, Memorial Sloan-Kettering Cancer Center, New York, NY 1002, USA Department of Pediatrics, Memorial Sloan-Kettering Cancer Center, New York, NY 1002, USA c Department of Pathology, Memorial Sloan-Kettering Cancer Center, New York, NY 1002, USA b
Index words: Osteosarcoma; Metastasis; Pediatric
Abstract This case illustrates a primary osteosarcoma in a child that has metastasized to the stomach. Such a presentation has only been documented once before and raises the importance of vigilant and thorough follow-up. In addition, this case questions the relationship of prolonged treatment and subsequent sites of recurrence. D 2007 Elsevier Inc. All rights reserved.
Osteosarcoma is the most common primary bone tumor in children, and the lung is the most frequent metastatic site. We report herein a case of a primary osteosarcoma with an exceptionally unusual course. This patient, with a gastric metastasis, is to our knowledge the first such case occurring in the United States.
1. Case report A previously healthy 17-year-old white adolescent boy sustained a knee injury while playing volleyball in September 1998. The patient sought medical attention several months later when the pain persisted, also complaining of low-grade fevers, night sweats, and a 15-lb weight loss. Radiological workup of the right lower extremity with magnetic resonance imaging and computed
* Corresponding author. Department of Surgery, Memorial SloanKettering Cancer Center, New York, NY 1002, USA. Tel.: +1 212 639 7002; fax: +1 212 717 3373. E-mail address: [email protected] (M.P. La Quaglia). 0022-3468/$ – see front matter D 2007 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2006.12.005
tomographic scan revealed an ill-defined lesion on the right femoral distal metaphysis with extension into the epiphysis and upper diaphysis. He underwent a core needle biopsy and a staging computed tomographic scan and was diagnosed with a high-grade osteosarcoma of the distal right femur and bilateral pulmonary metastases. The patient was treated with several cycles of a multidrug chemotherapy regimen that included doxorubicin, ifosfamide, cisplatin, and high-dose methotrexate between April 1999 and November 2000. In July 1999, he underwent resection of left pulmonary metastases, and in August 1999, a limbpreserving, en bloc resection of the primary tumor with placement of a knee prosthesis. Pathologic examination revealed a high-grade osteosarcoma, osteoblastic type, with a good chemotherapeutic response (grade III, 90% necrosis) and focal cortical bone breakthrough into surrounding soft tissues. Two months later, the patient underwent an elective right thoracotomy with multiple wedge resections of his pulmonary metastases. The pathology report revealed metastatic osteosarcoma with 80% to 90% necrosis, associated with reactive bronchiolitis obliterans. He first relapsed in November 2000 with right lung nodules and underwent a
738
Fig. 1 Intraoperative photograph showing the anterior wall of the gastric antrum with the invaginated lesion. The stomach wall is being retracted with silk stitches, and the mass has been highlighted with purple ink.
second right thoracotomy for resection of recurrent pulmonary metastases. In June 2001, he again relapsed with a metastatic lesion to the left buttock that was resected and resumed chemotherapy with high-dose ifosfamide and etoposide until November 2001. The patient was without further findings until February 2002, when he noted black, tarry stools and vomiting followed by a syncopal episode. His hemoglobin was 5 g/dL, and he was transfused. An upper gastrointestinal endoscopy revealed a 3-cm-diameter lesion of the gastric antrum with an ulcerated surface and a clot attached to it. A biopsy was consistent with osteosarcoma, and he subsequently underwent a resection of the gastric tumor with negative margins. Fig. 1 shows a gross picture of the gastric lesion intraoperatively, and Figs. 2 and 3 show a gross picture and microscopic section, respectively. The gastric tumor showed predominantly
Fig. 2 Macroscopic appearance of the interior of the resected gastric wall, with the well-defined osteogenic sarcoma tumor showing a white-tan cut surface with focal hemorrhagic areas.
V.E. Strong et al.
Fig. 3 Microphotograph of the specimen showing normal gastric mucosa on the right and a highly cellular spindle cell proliferation with focal osteoid production on the left, consistent with metastatic osteogenic sarcoma.
viable, osteoblastic-type osteosarcoma. The patient is currently at home and stable.
2. Discussion Approximately 20% of patients who are diagnosed with osteosarcoma have distant metastases at diagnosis. The lung is the most common site, and metasectomy is associated with improved survival [1]. Other sites of metastases include the bones, liver, and pleural extension from pulmonary lesions [2]. Although other metastatic sites have been reported, they are exceptionally rare. In contrast, primary extraosseous osteosarcoma is more aggressive than osteosarcoma of bone and is more likely to occur in unusual sites including soft tissue and small intestine [3,4]. Intestinal involvement, in particular, has only been reported in patients with extraosseous primary lesions. There is only one report of a metastatic recurrence of osteosarcoma in the stomach. This is a single case report from Germany. This metastasis was presumed to arise from a primary bone osteosarcoma. A history of an eye excision early in infancy for unknown reasons raises the possibility of retinoblastoma. Hereditary and sporadic retinoblastoma is known to predispose to a higher frequency of osteosarcoma as a secondary malignancy and may have affected this patient’s presentation [5]. In our case, we report a male with a definitive primary osteosarcoma of the bone that developed metastatic lesions in the lung, buttock, and most recently gastric antrum. It is of interest that 2 years elapsed from initial presentation to the gastric metastasis, and this was after multiple cycles of chemotherapy. Guiliano et al [6] addressed the question of whether advances in therapy have altered the pattern of recurrences
Osteosarcoma with delayed metastasis to the stomach in patients treated for osteosarcoma. They looked at the clinical course of more than 100 patients with high-grade skeletal osteosarcoma from 1971 to 1982. Fifty-three percent of the patients developed clinical metastases, and 61% of these were to the lung. Ninety-five percent of patients treated by amputation alone developed pulmonary metastases as the site of initial recurrence. In contrast, 45% of patients treated after 1974 with surgery and chemotherapy developed pulmonary metastases, whereas 28% from this group developed extrapulmonary metastases as the initial site of recurrent disease. This article goes on to suggest that patterns of metastatic recurrence may be altered by current therapeutic regimens [6]. The stomach is an extremely unusual site for a metastatic lesion of osteosarcoma. To our knowledge, this report is the second case reported in the literature and the first occurring in the United States. This unusual site of recurrence may be related to multiple courses of therapy. Prolonged treatment
739 of metastatic sarcoma may be associated with uncommon sites of recurrence.
References [1] LaQuaglia MP. Osteosarcoma: specific tumor management and results. Chest Surg Clin N Am 1998;8:77 - 95. [2] Goorin AM, Abelson HT, Frei E. Osteosarcoma: fifteen years later. N Engl J Med 1985;313:1637 - 43. [3] Nojima T, Gebhardt MC, Mankin HJ, et al. Extraosseous osteosarcoma presenting with intestinal hemorrhage: case report and literature review. Hum Pathol 1986;17:85 - 7. [4] Donoso L, Valls R, Montserrat E, et al. CT findings in a case of polypoid-like metastases in the ileum from extraosseous osteosarcoma. Eur J Radiol 1988;8:100 - 1. [5] Overberg-Schmidt US, Baumgarten E, Bassir C, et al. The stomach and pulmonary interlobular space as unusual sites of osteosarcoma. Pediatr Hematol Oncol 1999;16:61 - 6. [6] Giuliano AE, Feig S, Eilber FR. Changing metastatic patterns of osteosarcoma. Cancer 1984;54:2160 - 4.