The patient is a 3-yr-old, previously healthy boy who was noted to have fecal incontinence, difficulty and discomfort while sitting, and a burning sensation during urination. Physical examination suggested a pelvic mass. Computed tomography scans confirmed a large sacrococcygeal mass extending into the abdomen as well as metastatic disease in the lungs. Given the sacral involvement and its large size, it was thought that this tumor was unresectable; therefore, surgical biopsy was planned. An abdominal retroperitoneal approach was used because a future re-exploration was considered and was to be performed via a posterior sagittal approach.
Usual monitoring was used. A 22-gauge peripheral IV was in situ, and anesthesia was induced with sodium pentothal, pancuronium, fentanyl, and isoflurane. The trachea was intubated, and esophageal temperature and breath sounds were monitored. Maintenance anesthesia consisted of isoflurane (1%) and N2O (50%) via a semiclosed circle system with supplemental fentanyl and pancuronium. After an abdominal incision, the patient was placed in Trendelenburg’s position, and the intestines were carefully retracted, exposing the retroperitoneum. Dissection revealed the tumor, extending toward the sacrum and retroperitoneal lymphadenopathy. The surgeon reported palpating a large vessel leading up to the mass, and a portion of the mass was felt to be within the lumen of the left internal iliac vein. Subsequent tissue biopsies were taken from an area adjacent to the sacral promontory. Inspection of the area near the large tumor mass in the vein revealed that the palpable mass was now gone. The surgeon informed the anesthesiologist about the possibility of venous embolism. There were no changes in blood pressure (85/52 mm Hg), heart rate (140 bpm), or electrocardiogram (sinus rhythm). Heart and lung sounds were unchanged. There was no heart murmur. Biopsies were sent to the pathologist for frozen section. Approximately 20 min later, the patient suddenly decompensated. The heart rhythm changed to frequent premature atrial contractions, premature ventricular contractions, bradycardia, and pulseless electrical activity. ETCO2 decreased to 19 mm Hg, blood pressure decreased to 58/28 mm Hg, and oxygen saturation became undetectable. A massive tumor embolus was suspected. Fortunately, the surgery was performed in an operating room equipped for pediatric cardiac surgery, and transesophageal echocardiography (TEE) was immediately available. TEE demonstrated a grossly distended right atrium and right ventricle (RV). A large, foreign-body embolus was visualized migrating from the right atrium through the RV outflow tract to the main pulmonary artery. The RV was distended with a septal shift to the left, with compression of the left ventricle and global hypokinesis. The pediatric cardiovascular surgeon and perfusion team were notified for an attempt at tumor thrombus retrieval using cardiopulmonary bypass (CPB).
Resuscitation took place for more than 40 min with cardiopulmonary resuscitation. Intermittent epinephrine and sodium bicarbonate boluses were administered. Pulseless electrical activity persisted until CPB was initiated. After a period of stabilization, tumor emboli were retrieved from the right and left pulmonary artery through a transverse incision in the main pulmonary artery. Small fragments of tumor were removed from the pulmonary artery branches with a Fogarty catheter. Initial arterial blood gas analysis and laboratory test values were pHa 7.31, PaCO2 45 mm Hg, PaO2 216 mm Hg, HCO3 23 mEq/L, O2 saturation 100%, glucose 421 mg/dL, potassium 4.2 mEq/L, hematocrit 30%, ionized Ca 0.65 mmol/L, and lactate 18 mm Hg. After successful tumor emboli removal, a dobutamine infusion was started at 5 μg · kg−1 · min−1. The TEE demonstrated good biventricular contractility with no wall motion abnormalities. Separation from CPB was uneventful. A left subclavian central venous pressure line was placed, and the patient was transported to the pediatric intensive care unit in a hemodynamically stable condition. Upon arrival to the pediatric care unit, the patient was responsive to auditory and tactile stimuli, occasionally opening his eyes, and moving all extremities. Mannitol was administered prophylactically for cerebral edema. The pathology report noted multiple fragments of tumor from the embolus measuring in aggregate 4 × 1 × 0.8 cm. The postoperative diagnosis was Stage IV malignant sacrococcygeal teratoma. The patient was discharged from the hospital 26 days after the initial operation. Neurologically, the patient experienced expressive aphasia and visual peripheral disturbances, which have subsequently resolved.
Microdissemination of tumor is common; however, intraoperative embolization of large tumor fragments to the heart and lungs is extremely rare. Should a massive tumor embolus occur, survival is rare (1–4). The incidence of tumor embolism has been reported to vary between 0.9% to 2.4% in two retrospective autopsy studies during 6- and 15-year intervals (5).
There are a few reports of renal cell carcinoma associated with acute pulmonary tumor embolism and renal tumor extension causing inferior vena cava occlusion (2–4,6). Intraoperative TEE has been reported to guide basket catheter management of renal cell tumor embolism (7). Other cases of tumor embolism have been reported with gastric cancer, causing tumor embolism (8), massive pulmonary emboli associated with a sarcoma causing sudden death (9), and embolism to the left anterior descending coronary artery during pneumonectomy (10).
To our knowledge, this case is the first report of tumor embolism, secondary to a germ cell tumor, diagnosed intraoperatively and successfully treated by CPB and tumor retrieval. This case underscores the usefulness and importance of intraoperative TEE. TEE allowed rapid and precise interrogation of the heart and great vessels, confirmed the diagnosis, and directed the management of the massive tumor embolism. For the patient with a pelvic mass, Doppler or ultrasound studies may be used to eliminate and better delineate vascular tumor involvement. Death secondary to massive tumor embolism may be preventable if detected immediately, and the resources, expertise, and personnel are available for CPB and retrieval of the tumor emboli.
In conclusion, preoperatively, every effort should be made clinically and radiographically to detect the presence of vascular tumor extension. Intraoperative cardiac arrest is often assumed to be related to an anesthetic mishap or surgical vascular injury. Tumor embolism is a rare cause of cardiac arrest that should be considered early in the differential diagnosis in the setting of tumor surgery. Cardiac arrest can best be evaluated with TEE for rapid and precise assessment of the heart and surrounding structures. TEE is useful to visualize tumor emboli as well as emboli migration through the heart.
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