Japanese Journal of Clinical Oncology 31:165-167 (2001)
© 2001 Foundation for Promotion of Cancer Research
Intestinal Metastasis Causing Intussusception in a Patient Treated for Osteosarcoma with History of Multiple Metastases: a Case Report
1Department of Pediatrics, 2Section of Medical Oncology, Department of Medicine and 3Department of Pathology, Taipei Veterans General Hospital and School of Medicine, National Yang-Ming University, Taipei, Taiwan
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ABSTRACT |
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 TOP ABSTRACT INTRODUCTIONCASE REPORTDISCUSSIONREFERENCES |
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Intestinal intussusception caused by metastatic tumors is a very rare condition. Preoperative diagnosis is not easy because of the condition’s rarity and because of mild abdominal physical presentation. We report on a patient with osteosarcoma who suffered from abdominal pain and emesis during the period of autologous peripheral blood stem cell transplantation. He had undergone tumor excision and radiotherapy several times prior to autologous peripheral blood stem cell transplantation because of multiple metastases. Intestinal metastasis was suspected initially by computed tomographic scan and sonogram and was proved by surgical resection and pathological findings. Clinicians caring for pediatric patients with osteosarcoma with a history of multiple metastases should consider the possibility of intestinal metastases when equivocal abdominal symptoms develop after intensive chemotherapy.
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INTRODUCTION |
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TOPABSTRACTINTRODUCTIONCASE REPORTDISCUSSIONREFERENCES |
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The treatment of osteosarcoma has improved dramatically over the last two decades. Advances in staging systems, imaging modalities, multi-agent chemotherapy regimens and surgical treatment have improved these patients’ outcome; up to two-thirds will be long-term survivors (1). Historically, the lungs have been the predominant site of metastasis in osteosarcoma. However, as intensive multimodality therapy prolongs these patients’ event-free survival, the pattern of relapse may change (2,3). Intestinal metastasis of osteosarcoma causing acute abdominal symptoms during clinically remitted status has rarely been reported. Here we report one case of osteosarcoma who presented with extrapulmonary disease causing intestinal intussusception during high-dose chemotherapy, followed by autologous peripheral blood stem cell support.
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CASE REPORT |
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TOPABSTRACTINTRODUCTIONCASE REPORTDISCUSSIONREFERENCES |
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A 13-year-old boy was diagnosed with high-grade osteosarcoma over the right distal femur in October 1995. Neoadjuvant chemotherapy plus limb salvage operation and extracorporeal radiation at 200 Gy was performed. Adjuvant chemotherapy composed of high-dose methotrexate, cisplatin, ifosfamide and epirubicin for a total of 25 courses was prescribed from December 1995 to October 1996.
However, bilateral multiple pulmonary metastases were detected in March 1999. Surgical resection for metastatic tumors was performed twice during March and April 1999. Chemotherapy with high-dose methotrexate, cyclophosphamide, ifosfamide and etoposide was administered from May to September 1999.
Subsequently, the patient suffered from multiple intracranial metastatic lesions over the left frontal-parietal and bilateral occipital lobes in October 1999. Whole brain irradiation with 4575 cGy in 12 fractions, with a boost to left frontal-parietal and right occipital lesions to 5955 cGy, was given. The metastatic brain tumor regressed completely after completing the radiation therapy. Owing to multiple metastases of his disease, high-dose chemotherapy with autologous peripheral blood stem cell rescue was performed in February 2000 after his disease was under control (4).
The conditioning regimen consisted of melphalan 140 mg/m2 for 1 day and carboplatin 250 mg/m2/day for 5 days. Unfortunately, the patient developed acute abdominal pain on day –1 and the symptoms quickly subsided after oral medication. Autologous peripheral blood stem cell with mononuclear cell 4.85 x 108 per kilogram of body weight (/kg) (CD 34+ cell 2.20 x 106/kg) was infused on day 0. However, his abdominal pain recurred and was exacerbated on day +1. Intestinal intussusception was suspected by abdominal sonography and hydrostatic reduction was attempted under ultrasonic guidance with pressure up to 100 mmHg, but failed. Abdominal CT scan showed jejunojejunal intussusception with a suspected tumor (Fig. 1a) and enlarged bilateral adrenal glands which were suspected to be metastases.
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The patient soon became febrile and vomited with bile-stained vomitus. Emergent exploratory laparotomy was performed on day +2. During operation, manual reduction of the intussusception was performed, then segmental resection of the jejunum including the tumor with end-to-end anastomosis was done. The tumor was about 2.5 cm in length and the segment of intussusception was hyperemic in appearance without gangrenous change. There was no other metastatic lesion in the patient’s abdomen. The pathology of the intestinal tumor presented a picture of metastatic osteosarcoma (Fig. 1b).
The patient’s WBC was 300/µl when he underwent the operation. After the operation, neutropenic fever with hypotension and shock developed. Despite intensive care and the empirical use of broad-spectrum antibiotics, he died of septic shock on day +4 with negative results of all cultures.
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DISCUSSION |
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TOPABSTRACTINTRODUCTIONCASE REPORTDISCUSSIONREFERENCES |
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Although the incorporation of multidrug chemotherapy into the front-line treatment of patients with non-metastatic osteosarcoma has resulted in an impressive improvement in prognosis, some of these patients ultimately will experience disease relapse, the most common sites of relapse being the lungs and other bones. Less commonly involved and generally seen either preterminally or at postmortem are the heart and visceral abdominal structures and, rarely, the brain (1–3,5–9).
Conventionally, the presence of multiple brain metastases is considered a contraindication to surgery. However, Bindal et al. reported that there was a large improvement in the median survival for patients who underwent removal of all known lesions (10). This patient hesitated to have further surgery when multiple brain metastases developed, although his metastatic brain tumor regressed markedly after whole brain irradiation.
The cause of intussusception differs between pediatric and adult populations. In adults, an underlying cause is present 90% of the time, whereas in children a precipitating lesion is found in only 10% of patients. It is for this reason that pediatric intussusception is initially treated with non-operative reduction and adult intussusception is treated with surgical exploration. The controversy in adult intussusception is whether or not the intestine should be reduced before resection of the bowel. Even if reduction is feasible in the adult, the inciting pathology remains and must be dealt with surgically (11). Eisen et al. (11) recommended that small bowel intussusception in adults should be reduced only in patients for whom a benign diagnosis has been made preoperatively or in patients in whom resection may result in short gut syndrome. Our patient’s age was only 13 years, i.e. he belonged to the pediatric population; however, the clinical manifestation of his intestinal intussusception was similar to those observed in the adult population. Since laparotomy could lead to severe infection during pancytopenia status, hydrostatic reduction was tried on a hope but failed. Subsequent segmental resection of the intussusception was performed. Unfortunately, the patient did not survive the immediate operation.
Metastatic osteosarcoma is very rare as a major cause of intussusception and very few cases have been reported in the literature (12–14). The diagnosis of intussusception in adults is usually delayed and is generally not made until laparotomy because the clinical picture is not typical. The most common presentation of adult intussusception for the patient reported by Eisen et al. (11) was signs and symptoms of intestinal obstruction including abdominal pain and emesis; our pediatric patient had a similar presentation. In contrast, with this patient the classic triad of abdominal mass, intermittent abdominal pain and currant jelly stool commonly observed in childhood intussusception was not observed.
In conclusion, abdominal pain and emesis were the main symptoms of jejunojejunal intussusception in this 13-year-old patient. Intestinal metastasis causing intussusception should be considered in patients with a history of multiple metastatic tumors who develop abdominal pain and emesis during intensive chemotherapy.
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FOOTNOTES |
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+ For reprints and all correspondence: Tzeon-Jye Chiou, Section of Medical Oncology, Department of Medicine, Taipei Veterans General Hospital, 201 Shih-Pai Road, Section 2, Taipei 112, Taiwan. E-mail: tjchiou@vghtpe.gov.tw
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Received October 10, 2000; accepted December 22, 2000.
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