Oweidat Majd, Abu Rumilah Anas, Maraqa Mohammed, Natsheh Motaz
College of Medicine, Hebron University, Hebron, West Bank, Palestine.
Division of Endocrinology, Department of Internal Medicine, Al-Ahli Hospital, Hebron, West Bank, Palestine.
AACE Endocrinol Diabetes. 2025 Apr 10;12(1):24-29. doi: 10.1016/j.aed.2025.03.007. eCollection 2025 May-Jun.
BACKGROUND/OBJECTIVE: Adrenal hemorrhage (AH) and adrenal insufficiency (AI) are rare, life-threatening conditions, typically linked to trauma, sepsis, or anticoagulation. Burkitt's lymphoma (BL), an aggressive B-cell non-Hodgkin lymphoma, rarely involves the adrenal glands and has not been reported to present with AH and AI. This report describes a unique case of AH and AI presenting as manifestations of BL, highlighting the importance of considering malignancy in unexplained AH and AI.
A 16-year-old boy presented with acute right flank pain and muscle spasms. Imaging revealed a right suprarenal hematoma with active contrast extravasation, managed with adrenal artery embolization. Two weeks later, he developed recurrent abdominal pain, vomiting, and distention. Computed tomography showed bilateral adrenal enlargement with necrosis and hemorrhage. Laboratory test results revealed normocytic anemia, increased lactate dehydrogenase level, and hypoglycemia. AI was confirmed (serum cortisol level, 1.5 μg/dL; adrenocorticotropic hormone level, 82 pg/mL), prompting hydrocortisone replacement. Positron emission tomography/computed tomography revealed hypermetabolic lesions in bilateral adrenal glands, lymph nodes, pleura, peritoneum, and bone marrow. Neck lymph node biopsy confirmed BL, showing a "starry sky" pattern, CD20 positivity, and Ki-67 nearing 100%. Intensive chemotherapy led to significant clinical improvement.
This case highlights the diagnostic challenge of AH and AI without typical risk factors. BL, although rarely involving the adrenal glands, should be considered in unexplained AH.
This report highlights the importance of considering malignancy in unexplained AH and AI, even without conventional risk factors.
背景/目的：肾上腺出血（AH）和肾上腺功能不全（AI）是罕见的、危及生命的病症，通常与创伤、脓毒症或抗凝有关。伯基特淋巴瘤（BL）是一种侵袭性B细胞非霍奇金淋巴瘤，很少累及肾上腺，且尚无报告称其会表现为AH和AI。本报告描述了一例以BL表现形式出现的AH和AI的独特病例，强调了在不明原因的AH和AI中考虑恶性肿瘤的重要性。
一名16岁男孩出现急性右侧胁腹疼痛和肌肉痉挛。影像学检查显示右肾上腺血肿伴有造影剂外渗，通过肾上腺动脉栓塞进行治疗。两周后，他出现反复腹痛、呕吐和腹胀。计算机断层扫描显示双侧肾上腺肿大伴坏死和出血。实验室检查结果显示正细胞性贫血、乳酸脱氢酶水平升高和低血糖。确诊为AI（血清皮质醇水平为1.5μg/dL；促肾上腺皮质激素水平为82pg/mL），遂开始氢化可的松替代治疗。正电子发射断层扫描/计算机断层扫描显示双侧肾上腺、淋巴结、胸膜、腹膜和骨髓有高代谢病变。颈部淋巴结活检确诊为BL，呈现“星空”样图案，CD20阳性，Ki-67接近100%。强化化疗使临床症状显著改善。
本病例突出了无典型危险因素的AH和AI的诊断挑战。BL虽然很少累及肾上腺，但在不明原因的AH中应予以考虑。
本报告强调了在不明原因的AH和AI中考虑恶性肿瘤的重要性，即使没有传统危险因素。
