PMY Tang, NSY Chao

Abstract

Aim: Osteosarcoma typically metastasizes to the lung and the brain via haematogenous spread. Intestinal spread is uncommon. Data on the management protocol for metastatic lesion in the small bowel causing ileo-ileal intussusception in children is lacking. Methods: We reported a case of a 17-year-old boy with known past history of pulmonary, brain and soft tissue metastatic osteosarcoma, presenting with intestinal obstruction. His clinical presentation and management pathway was discussed. Results: A 17-year-old boy with left femur osteosarcoma five years ago presented with vomiting and abdominal pain for one day. He had known past history of pulmonary, brain and right left soft tissue osteosarcoma metastasis. On admission, he remained haemodynamically stable with a full GCS score. Abdominal X-ray showed prominent small bowel loops. Computed tomography abdomen showed ileo-ileal intussusception with suspicious intestinal metastasis. Emergency laparotomy showed ileo-ileal intussusception, small bowel resection and anastomosis were performed. Histology findings of the resected specimen confirmed metastatic osteosarcoma into small bowel. Post op recovery was uneventful and patient was able to tolerate full oral feeding on day 9 post operatively. Conclusion: In patient with metastatic osteosarcoma, one of the potential causes of intestinal obstruction could be ileo-ileal intussusception with pathological lead point. In comparsion with the more common peritoneal metastasis presenting with carcinomatosis in the adult population, isolated intra-luminal intestinal metastasis is a more readily surgically treatable condition. High index of suspicion and early surgical intervention were essential in children with metastatic osteosarcoma.

Introduction

Osteosarcoma is a bone tumour commonly affecting the long bone. It typically metastasizes to the lung and brain. With the advances in adjuvant therapy and longer survival, unusual site of metastasis such as intestinal metastasis had been reported.¹ Herewith we reported a case of a 17-year-old boy with intestinal metastatic osteosarcoma presenting as ileo-ileal intussusception.

Case History

A 17-year-old boy with known history of left femur osteosarcoma five years ago presented to the surgical department with acute abdominal pain and undigested food vomiting for one day. He had known history of left lower lobe lung metastasis with lobectomy, recurrent left occipital lobe, right parietal and right frontal lobe brain metastasis with repeated excision, spinal cord metastasis with spinal cord decompression and right thigh soft tissue metastasis with local excision.

On clinical examination, he was haemodynamically stable with a GCS of 15/15. He was oriented and alert, and was able to walk with stick. He had low grade fever of 37.8^oC. Physical examination showed soft, non-distended abdomen with mild diffuse tenderness at left upper quadrant. Abdominal X-ray showed dilated small bowel loops (Figure 1). Given his clinical history of known multi-sites tumor metastases, clinical diagnosis of suspected peritoneal or intestinal metastasis was made. Urgent CT abdomen showed 8cm ileo-ileal intussusception with suspicious calcified intra-luminial mass acting as the lead point (Figure 2). Emergency laparotomy showed ileo-ileal intussusception, with a 2.5 cm intra-luminal calcified ulcerated polyp like mass acting as the pathological lead point (Figures 3-5). No other lesion was palpable in the small and large bowel, no other lesion was seen on the stomach, liver and the gallbladder. 30 cm small bowel including the intussusception was resected, primary small bowel to small bowel anastomosis was made. The patient had uneventful post op recovery and was able to tolerate full oral feeding on day 9 post operatively.

Figure 1 AXR showing prominent small bowel loops.

Figure 2 CT showing the ileo-ileal intussusception.

Figure 3 Intra-operative finding of ileo-ileal intusussception.

Figure 4 Resected specimen showing the 30 cm small bowel.

Figure 5 Resected ileum showing the intra-luminal ulcerated mass.

The histological report of the resected specimen came back to be metastatic osteosarcoma.

Classical intussusception in children typically occurs between the age of 6-month-old to 4-year-old, with the clinical triad of colicky abdominal pain, vomiting and red currant jelly stool. When intussusception occurs out of the expected spectrum of age, or when it was not located at the typical ileo-caecal location, surgical exploration is usually warranted.

While laparoscopic reduction of the intussusception was a potentially viable option, in the context of suspected intestinal metastasis, it had been reported that the lack of tactile bowel palpation in laparoscopic intervention may results in missed metachronous intestinal metastasis, furthermore, intussusception with a lead point usually would require bowel resection and anastomosis.²

Conclusion

Reviewing the literature, four paediatric small bowel metastatic osteosarcoma causing small bowel intussusception had been reported.³ It was reported that the small bowel intussusception usually occurred 1.5 to 5 years after the initial osteosarcoma diagnosis.⁴ The presence of gastro-intestinal metastasis would often indicate aggressive disease with poor prognosis.⁵ Keeping with the goal of palliative management in our patient, conservative non-operative management had been briefly discussed upon admission. However, with the CT confirmation of a pathological lead point causing a surgically reversible cause of intestinal obstruction, it became clear that surgical exploration was indicated.

Declaration of Interest

All authors have disclosed no conflicts of interest.

References

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