李蕎青^1,2、丁慧恭¹、林佑俊³、曹智惟¹、查岱龍¹、于大雄¹、吳勝堂¹

¹三軍總醫院外科部泌尿外科  ²國軍高雄總醫院外科部 ³三軍總醫院病理部

Chiao-Ching, Li^1,2, Hui-Gung, Ting¹, Chin-Yun, Lin³,

Chih-Wei, Tsao¹, Tai-Lung, Cha¹, Dah-Shyong, Yu¹, Sheng-Tang, Wu¹

1. Division of Urology, Department of Surgery, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan

2. Department of Surgery, Kaohsiung Armed Forces General Hospital, Kaohsiung, Taiwan

3. Department of Pathology, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan

Spontaneous adrenal hemorrhage(AH) is an unusual condition and it has been reported in 0.3% to 1.8% of undetected cases in autopsy research. The reasons are associated with adrenal masses, sepsis, anticoagulation, pregnancy, and hematologic diseases. In our case, the AH is presented as flank pain and Castleman disease(CD) is diagnosed after laparoscopic adrenalectomy. CD is a rare disease of lymph nodes and related tissues of lymphoproliferative disorder and it is common seen at chest and abdomen. CD originated from adrenal gland is rare report and is suggested for follow-up.

Medical record is from the 50-year-old man who had past history of hypertension without regular medication. Initially, he came to our emergency room due to severe left flank pain. It was not like knocking tenderness, but dullness. The vital sign was stable. The laboratory examinations were no pyuria, hematuria, anemia. Subsequently, the heterogeneously hyperdensity image at left suprarenal region was revealed from computed tomography(CT) scan of abdomen. He was admitted because of impression of adrenal hemorrhage, left. After conservative treatment included bed rest for 1 week during hospitalization, the patient’s condition got recovery. He discharged and was under outpatient department follow-up. The resolution of hematoma was showed from magnetic resonance imaging(MRI) 6 months later. Therefore, laparoscopic adrenalectomy was performed after hormone study was completed.    

The laparoscopic adrenalectomy, left is performed smoothly. The patient discharges 5 days after operation. The gross specimen is pink in color and 6x5x4cm in size. The surprised findings in immunohistochemical stain are that is positive in the stains of CD3, CD20 and CD10. They mean the tissue had T cells, B cells and  centroblasts. Furthermore, it has highlight mantle zone cells in stain of Bcl-2. Therefore, Castleman disease is proved via pathology findings.

The castleman disease presents as spontaneous adrenal hemorrhage is uncommon. The treatment plan for adrenal hemorrhage is maintenance of stable vital sign first and surgical intervention after resolution of hematoma. The importance prior laparoscopic adrenalectomy is hormone study to prevent the functional adrenal tumor of pheochromocytoma. Castleman disease is classified to localized and systemic disease. The further blood or image should be surveyed if unspecific manifestations, such as fever, night sweats, weight loss, loss of appetite, and weakness.