Moradi Golnaz, Zarei Diana, Issaiy Mahbod
Department of Radiology, Sina Hospital, Tehran University of Medical Sciences, Tehran, Iran.
Advanced Diagnostic and Interventional Radiology Research Center (ADIR), Tehran University of Medical Sciences, Tehran, Iran.
Int J Surg Case Rep. 2024 Mar;116:109398. doi: 10.1016/j.ijscr.2024.109398. Epub 2024 Feb 26.
Myelolipoma, a benign tumor characterized by mature fat cells and hematopoietic cells, is predominantly found in the adrenal glands, accounting for 6-16 % of all adrenal tumors. These tumors are often asymptomatic and discovered incidentally during imaging. We present a rare case of concurrent adrenal and extra-adrenal myelolipomas, contributing to the limited research in this area.
A 65-year-old female with a history of Steven-Johnson syndrome presented with epigastric pain, initially diagnosed with emphysematous cholecystitis. Imaging revealed unexpected lesions near the left kidney. During surgery for presumed cholecystitis, significant hemorrhaging occurred following an attempted biopsy of the left adrenal lesion. This complication necessitated a complete adrenalectomy. Pathological examination confirmed the presence of myelolipomas in the left adrenal gland, para-aortic, and left para-iliac regions.
The simultaneous occurrence of adrenal and extra-adrenal myelolipomas is exceptionally rare, posing diagnostic and management challenges. This case highlights the complexity of managing patients with multiple comorbidities and the critical importance of differentiating myelolipomas from other fat-containing retroperitoneal masses. The incidental discovery of these tumors and their potential for significant intraoperative complications, as seen in our case, underscores the need for careful surgical planning and thorough preoperative assessment.
This case emphasizes the diagnostic challenges and management complexities in patients with incidental findings of myelolipoma, particularly when accompanied by significant medical histories. The occurrence of unexpected intraoperative complications highlights the importance of cautious decision-making in surgical interventions. This report provides valuable insights into the unpredictable nature of medical practice and the management of rare pathologies.
肾上腺髓质脂肪瘤是一种以成熟脂肪细胞和造血细胞为特征的良性肿瘤，主要见于肾上腺，占所有肾上腺肿瘤的6% - 16%。这些肿瘤通常无症状，多在影像学检查时偶然发现。我们报告一例罕见的同时发生肾上腺和肾上腺外髓质脂肪瘤的病例，以补充该领域有限的研究。
一名65岁有史蒂文斯 - 约翰逊综合征病史的女性因上腹部疼痛就诊，最初诊断为气肿性胆囊炎。影像学检查发现左肾附近有意外病变。在假定为胆囊炎的手术过程中，对左肾上腺病变进行活检尝试后发生了大量出血。这种并发症需要进行全肾上腺切除术。病理检查证实左肾上腺、主动脉旁和左髂旁区域存在髓质脂肪瘤。
肾上腺和肾上腺外髓质脂肪瘤同时发生极为罕见，带来了诊断和管理方面的挑战。该病例突出了管理患有多种合并症患者的复杂性，以及将髓质脂肪瘤与其他含脂肪的腹膜后肿块区分开来的至关重要性。如我们病例所示，这些肿瘤的偶然发现及其术中出现重大并发症的可能性强调了仔细的手术规划和全面的术前评估的必要性。
本病例强调了偶然发现肾上腺髓质脂肪瘤患者的诊断挑战和管理复杂性，尤其是伴有重要病史时。术中意外并发症的发生凸显了手术干预中谨慎决策的重要性。本报告为医疗实践的不可预测性和罕见病理的管理提供了有价值的见解。
