Abdullah Swara H, Hiwa Dilan S, Mustafa Lawen Jamal, Ahmed Farman J, Ahmed Shaho F, Mohammed-Saeed Dana H, Qadir Aso N, Salih Jamal M, Mohammed Hemin S, Kakamad Fahmi H
Department of Medicine, Shar Hospital, Sulaymaniyah, Kurdistan, Iraq.
Scientific Affairs Department, Smart Health Tower, Sulaymaniyah, Kurdistan, Iraq.
Radiol Case Rep. 2025 Jun 26;20(9):4624-4633. doi: 10.1016/j.radcr.2025.06.023. eCollection 2025 Sep.
Bilateral adrenal hemorrhage is a rare and serious complication that might affect patients with concomitant systemic lupus erythematosus (SLE) and antiphospholipid syndrome (APS). However, this study presents a rare case of SLE without APS that developed bilateral adrenal hemorrhage, causing adrenal insufficiency and later complicated by lupus myocarditis, resulting in rapid deterioration and death. A 35-year-old female complained of generalized body aches associated with arthralgia, joint swelling, abdominal pain, and fatigue for 2 weeks duration. The clinical course was complicated by seizure, bilateral adrenal hemorrhage, and resultant adrenal insufficiency in addition to pulmonary embolism. High troponin titers and echocardiographic findings of global wall hypokinesia suggested lupus myocarditis. Despite treatment with multiple medications throughout the patient's course of disease, including hydrocortisone, methylprednisolone, and hydroxychloroquine, the patient's condition deteriorated, and she died. Only a few cases of isolated SLE complicated by adrenal hemorrhage were reported during the literature review, with the majority of other cases associated with APS. Notably, no instances of concurrent myocarditis were reported in the reviewed studies. Adrenal hemorrhage may result from heightened arterial or venous pressure, often linked to elevated adrenocorticotropic hormone levels during stress or hypercoagulability. Anticoagulants can increase this risk, while reperfusion injury after hypotensive episodes also contributes. Patients with an SLE flare can develop bilateral adrenal hemorrhage and cause adrenal insufficiency even in the absence of definitive APS. The development of lupus myocarditis in such patients may result in worsened outcomes and potentially death.
双侧肾上腺出血是一种罕见且严重的并发症，可能影响合并系统性红斑狼疮（SLE）和抗磷脂综合征（APS）的患者。然而，本研究报告了一例罕见的无APS的SLE病例，该病例发生双侧肾上腺出血，导致肾上腺功能不全，随后并发狼疮性心肌炎，导致病情迅速恶化并死亡。一名35岁女性主诉全身疼痛伴关节痛、关节肿胀、腹痛和疲劳，持续2周。临床过程中除了肺栓塞外，还并发癫痫、双侧肾上腺出血及由此导致的肾上腺功能不全。高肌钙蛋白水平和超声心动图显示的全心壁运动减弱提示狼疮性心肌炎。尽管在患者病程中使用了多种药物治疗，包括氢化可的松、甲泼尼龙和羟氯喹，但患者病情仍恶化并死亡。文献综述中仅报道了少数孤立的SLE合并肾上腺出血的病例，大多数其他病例与APS相关。值得注意的是，在综述研究中未报告并发心肌炎的病例。肾上腺出血可能源于动脉或静脉压力升高，这通常与应激或高凝状态下促肾上腺皮质激素水平升高有关。抗凝剂会增加这种风险，低血压发作后的再灌注损伤也有影响。即使没有明确的APS，SLE病情发作的患者也可能发生双侧肾上腺出血并导致肾上腺功能不全。此类患者发生狼疮性心肌炎可能导致预后更差，甚至可能死亡。
