Esquivel Jose E, Santos Ana B, Hong Anthony, Ruiz Francis
Endocrinology, Hospital San Juan de Dios, San José, CRI.
Medicine, University of Costa Rica, San José, CRI.
Cureus. 2024 Jul 10;16(7):e64231. doi: 10.7759/cureus.64231. eCollection 2024 Jul.
Spontaneous remission of Cushing's disease (CD) is uncommon and often attributed to pituitary tumor apoplexy. We present a case involving a 14-year-old female who exhibited clinical features of Cushing's syndrome. Initial diagnostic tests indicated CD: elevated 24h urinary cortisol (235 µg/24h, n < 90 µg/24h), abnormal 1 mg dexamethasone overnight test (cortisol after 1 mg dex 3.4 µg/dL, n < 1.8 µg/dL), and elevated adrenocorticotropic hormone concentrations (83.5 pg/mL, n 10-60 pg/mL). A pituitary adenoma was suspected, so a nuclear MRI was performed, with findings suggestive of a pituitary microadenoma. The patient was referred for a transsphenoidal resection of the microadenoma. While waiting for surgery, the patient presented to the emergency department with a headache and clinical signs of meningism. A computed axial tomography of the central nervous system was performed, and no structural alterations were found. The symptoms subsided with analgesia. One month later, she presented again to the emergency department with clinical findings of acute adrenal insufficiency (cortisol level of 4.06 µg/dL), and she was noted to have spontaneous biochemical remission associated with the resolution of her symptoms of hypercortisolism. For that reason, spontaneous CD remission induced by pituitary apoplexy (PA) was diagnosed. The patient has been managed conservatively since the diagnosis and remains in clinical and biochemical remission until the present time, after 10 months of follow-up. There are three unique aspects of our case: the early age of onset of symptoms, the spontaneous remission of CD due to PA, which has been rarely reported in the medical literature, and the fact that the patient presented a microadenoma because there are fewer than 10 clinical case reports of PA associated with microadenoma.
库欣病(CD)的自发缓解并不常见,通常归因于垂体瘤卒中。我们报告一例14岁女性患者,其表现出库欣综合征的临床特征。初始诊断检查提示为CD:24小时尿皮质醇升高(235μg/24小时,正常<90μg/24小时),1毫克地塞米松过夜试验异常(1毫克地塞米松后皮质醇为3.4μg/dL,正常<1.8μg/dL),促肾上腺皮质激素浓度升高(83.5pg/mL,正常10 - 60pg/mL)。怀疑有垂体腺瘤,于是进行了核磁共振成像(MRI)检查,结果提示为垂体微腺瘤。该患者被转诊接受微腺瘤经蝶窦切除术。在等待手术期间,患者因头痛和脑膜刺激征的临床体征到急诊科就诊。进行了中枢神经系统计算机断层扫描(CT),未发现结构改变。症状经止痛治疗后缓解。一个月后,她再次因急性肾上腺功能不全的临床表现(皮质醇水平为4.06μg/dL)到急诊科就诊,且发现她出现了与高皮质醇血症症状缓解相关的自发生化缓解。因此,诊断为垂体卒中(PA)诱发的库欣病自发缓解。自诊断以来,该患者一直接受保守治疗,经过10个月的随访,至今仍处于临床和生化缓解状态。我们的病例有三个独特之处:症状出现的年龄早,因垂体卒中导致库欣病自发缓解,这在医学文献中鲜有报道,以及患者存在微腺瘤,因为与微腺瘤相关的垂体卒中临床病例报告少于10例。