Acute lower limb ischemia in pheochromocytoma surgery: a rare but serious complication (a case report)

¹Kaoutar El Fakhr, Pediatric Anesthesia and Intensive Care, Mother and Child Hospital Abderrahim Harouchi, University Hospital Center Ibn Rochd, Casablanca, Morocco

^&Corresponding author
Kaoutar El Fakhr, Pediatric Anesthesia and Intensive Care, Mother and Child Hospital Abderrahim Harouchi, University Hospital Center Ibn Rochd, Casablanca, Morocco

Introduction    

Pheochromocytoma is a rare adrenal medullary tumor arising from chromaffin cells. It is an uncommon condition in children [1]. The placement of an arterial catheter is a crucial step in the anesthesia management of pheochromocytoma to control blood pressure levels. Although complications from arterial catheter placement are rare, they can be severe and lead to limb amputation. We report a case of subtotal arterial thrombosis following femoral arterial catheterization.

Patient and observation     

Patient information: an 11-year-old female patient, with no known medical history, presented with profuse sweating and intense headaches. Diagnostic imaging confirmed bilateral pheochromocytoma, and the patient was scheduled for left adrenalectomy.

Clinical findings: during the pre-anesthetic consultation, the clinical examination revealed good general health, blood pressure (BP) of 170/90 mmHg, normal cardiopulmonary auscultation, and no predictive signs of difficult intubation (Mallampati II). Cardiologic evaluation was performed and found to be normal. The patient was started on alpha-blockers, beta-blockers, and a calcium channel inhibitor.

Diagnosis and management: at the operating room admission, anesthesia induction was performed with fentanyl (2μg/kg), etomidate (0.3mg/kg), and rocuronium (0.6mg/kg). Intubation was easy with a 5.5 CH cuffed endotracheal tube. Anesthesia was maintained with fentanyl, isoflurane, midazolam, and rocuronium. A right central venous catheter and a right femoral arterial catheter were placed without incident for invasive blood pressure monitoring. Hemodynamic management was achieved with nicardipine (1mg/ml in an electric syringe). Blood pressure remained stable between 90/60 and 110/70 mmHg throughout the procedure. The surgery lasted 10 hours without hemodynamic instability during tumor mobilization and resection. At the end of the surgery, the limb was cold with diminished femoral and popliteal pulses (Figure 1). The arterial catheter was removed, and a Doppler ultrasound confirmed a subtotal thrombosis of the common femoral artery (CFA) and total occlusion of the deep femoral artery (DFA).

Therapeutic interventions: the patient was started on unfractionated heparin (500 IU/kg/day) at 6 hours post-op, and a thrombophilia workup was initiated. She was then taken back to the operating room for embolectomy using a Fogarty catheter. Immediate postoperative evolution showed restored femoral and popliteal pulses, but the anterior tibial pulse remained absent (Figure 2). A CT angiogram confirmed subtotal occlusion of the distal portion of the right dorsal pedis artery, with weak opacification of the plantar arch. The patient remained on unfractionated heparin (600 IU/kg/day) with a target activated clotting time (ACT) of 1.5-2.5 times the baseline, and motor physiotherapy was initiated with good evolution. By the second postoperative day, all pulses were palpable. The patient was transferred to the pediatric visceral surgery department on postoperative day 2. Two months later, she was readmitted for right adrenalectomy. Preoperative assessment revealed a conscious patient in good general health, receiving aspirin, calcium channel blockers, alpha-blockers, and beta-blockers. The thrombophilia workup showed: protein C: 103%; protein S: 42%; antithrombin III: 100%. The anesthesia protocol was similar to the first surgery, but without femoral arterial catheterization. Nicardipine infusion was stopped once blood pressure normalized. However, intraoperative hemodynamic instability during tumor resection required norepinephrine (0.3 γ/kg/min). The patient was successfully weaned in intensive care with a favorable clinical outcome (Figure 3).

Patient´s perspective: the patient did not fully grasp the risks. She relied on her parents and initially felt distress over unusual pain, difficulty moving her leg, and cold sensations postoperatively. Medical staff and family played a crucial role in explaining the situation. Over time, she noticed gradual recovery and accepted medical care more readily as she saw progress. Ultimately, she overcame the experience with a positive outcome.

Parental consent: the patient's parents have given their consent for the writing and publication of her case.

Discussion     

Pheochromocytoma is a rare tumor that develops from chromaffin cells. It is a rare and severe condition in children. While Menard's triad is commonly observed in adults, clinical signs in children are often subtle [1]. Imaging helps guide the diagnosis, which is confirmed by measuring urinary or plasma catecholamine levels [2]. Early onset and bilateral involvement contribute to its severity. Its management is multidisciplinary, and surgical treatment consists of tumor excision. Anesthesia for pheochromocytoma remains challenging due to the uncontrolled secretion of catecholamines. Surgical management requires a multidisciplinary approach to reduce perioperative morbidity and mortality.

The placement of an arterial catheter is mandatory for perioperative blood pressure management. Although a simple procedure, it is not without risks. Complications are rare but can be severe. The causes of ischemia are varied, and idiopathic cases have been reported [3,4]. Treatment can be medical, surgical, or interventional radiology-based [5,6]. Literature reports cases of ischemia occurring in the context of pheochromocytoma. The excessive synthesis of catecholamines promotes thrombotic events [7,8]. Despite significant advancements in management, morbidity and mortality rates remain high. Early revascularization (within six hours of diagnosis) improves prognosis [9].

Conclusion     

Pheochromocytoma is a rare but potentially severe tumor. Beyond its typical clinical presentation, it can cause severe ischemic complications.

Competing interests     

The authors declare no competing interests.

Authors' contributions     

All the authors have read and agreed to the final manuscript.

Figures     

Figure 1: clinical appearance of lower limb ischemia

Figure 2: limb revascularization after embolectomy

Figure 3: limb evolution two month postoperatively

References