Benavente Kevin, Kozai Landon, Silangcruz Krixie, Banerjee Dipanjan
Department of the Internal Medicine Residency Program, University of Hawaii, 1356 Lusitania Street #510, Honolulu, HI 96813, USA.
Department of the Cardiology Fellowship Program, University of Hawaii, 1301 Punchbowl Street, Pauahi 3rd Floor, Queen's Heart, Honolulu, HI 96813, USA.
Eur Heart J Case Rep. 2023 Aug 3;7(8):ytad373. doi: 10.1093/ehjcr/ytad373. eCollection 2023 Aug.
Orthotopic heart transplant (OHT) recipients are at increased risk for varicella zoster reactivation, and severe complications may arise due to their immunosuppressive regimens. Managing immunosuppression in acute infection is difficult, and specific guideline recommendations or evidence from the literature are lacking. However, patient care must involve weighing the risk of transplant rejection with the consequences of worsening infection.
An OHT patient with a history of multiple episodes of acute rejection, latent varicella zoster virus (VZV) infection, and recent completion of anti-viral prophylaxis presented with unilateral facial droop and pain, abducens nerve palsy, crusting facial rash, and ear swelling. Imaging revealed necrotizing otitis externa, with associated otitis media, and petrous apicitis concerning for Gradenigo syndrome. A VZV-positive viral panel confirmed our suspicion for Ramsay Hunt syndrome (RHS). The patient's mentation continued to decline, and subsequent lumbar puncture also revealed VZV meningoencephalitis. The patient's mycophenolate mofetil (MMF) was suspended, with continuation of tacrolimus, and initiation of intravenous acyclovir. The patient demonstrated gradual resolution of his infection, without developing any signs of acute rejection.
Varicella zoster virus reactivation is common in OHT patients, particularly when viral prophylaxis is discontinued; however, cardiologists should be aware of the rarer manifestations that can manifest in these immunocompromised patients. This is the first documented case of simultaneous RHS, Gradenigo syndrome, and VZV meningoencephalitis in any patient, regardless of transplant status. We demonstrate that even in patients at very high risk of rejection, MMF can be safely discontinued and host immunity maintained with temporary tacrolimus monotherapy.
原位心脏移植（OHT）受者发生水痘带状疱疹病毒再激活的风险增加，并且由于其免疫抑制方案可能会出现严重并发症。在急性感染时管理免疫抑制很困难，并且缺乏具体的指南建议或文献证据。然而，患者护理必须权衡移植排斥的风险与感染恶化的后果。
一名有多次急性排斥发作史、潜伏性水痘带状疱疹病毒（VZV）感染且近期完成抗病毒预防的OHT患者，出现单侧面部下垂和疼痛、展神经麻痹、面部皮疹结痂以及耳部肿胀。影像学检查显示为坏死性外耳道炎，并伴有中耳炎，岩尖炎提示Gradenigo综合征。VZV阳性的病毒检测结果证实了我们对拉姆齐·亨特综合征（RHS）的怀疑。患者的意识状态持续下降，随后的腰椎穿刺也显示出VZV脑膜脑炎。患者的霉酚酸酯（MMF）被停用，继续使用他克莫司，并开始静脉注射阿昔洛韦。患者的感染逐渐得到缓解，且未出现任何急性排斥的迹象。
水痘带状疱疹病毒再激活在OHT患者中很常见，尤其是在抗病毒预防停药时；然而，心脏病专家应意识到这些免疫功能低下患者可能出现的罕见表现。这是首例记录在案的任何患者（无论移植状态如何）同时发生RHS、Gradenigo综合征和VZV脑膜脑炎的病例。我们证明，即使在排斥风险非常高的患者中，MMF也可以安全停用，并通过临时单一使用他克莫司维持宿主免疫力。
