腎上腺淋巴管瘤的手術治療：病例報告與文獻回顧

許哲元、翁瑋駿、劉品均、許兆畬、歐宴泉、童敏哲

童綜合醫院外科部泌尿科

Surgical Management of Adrenal Lymphangioma: A Case Report and Literature Review

Jhe-Yuan Hsu, Wei-Chun Weng, Pin-Chun Liu, Chao-Yu Hsu, Yen-Chuan Ou, Min-Che Tung

Tungs' Taichung MetroHarbor Hospital, Division of Urology, Department of Surgery, Taichung, Taiwan

Introduction

Adrenal lymphangioma is an exceedingly rare, benign cystic lesion of the adrenal gland, typically discovered incidentally during imaging studies for unrelated conditions. These lesions originate from the lymphatic system and are most often asymptomatic, though they may occasionally present with nonspecific symptoms or compressive effects if they reach a significant size. Due to their rarity, adrenal lymphangiomas pose a diagnostic challenge, often requiring a combination of imaging, laboratory evaluations, and, ultimately, histopathological examination for a definitive diagnosis. This case report discusses a 32-year-old female with an incidentally detected right adrenal cyst, identified as an adrenal lymphangioma following surgical resection and histopathological confirmation. This case underscores the importance of careful evaluation and management strategies for incidentally found adrenal lesions, especially in patients with a complex surgical history.

Case presentation

A 32-year-old female with a history of gastroesophageal reflux disorder, sleeve gastrectomy, and fundoplication surgery was admitted due to an incidentally discovered right adrenal tumor. She initially presented with painful urination and frequent urination, leading to a suspicion of pelvic inflammatory disease. An abdominal CT scan revealed the adrenal tumor, prompting further evaluation. Hormonal assays, including renin, aldosterone, cortisol, and adrenocorticotropic hormone, were within normal limits. After discussing surgical options, the patient consented to a robotic-assisted partial adrenalectomy and cyst removal.

During her hospital stay, the patient underwent the planned right robotic-assisted partial adrenalectomy and cyst removal on January 22, 2024, with a postoperative diagnosis of a right adrenal cyst. Pathology confirmed the diagnosis of adrenal lymphangioma. Postoperatively, she experienced mild wound tenderness but no fever. Her recovery included a gradual dietary progression from liquids to a soft diet, and her catheter was removed on January 24, 2024. Vital signs remained stable throughout her stay, and she received supportive care, including intravenous fluids, antibiotics, and analgesics. Sutures were removed on January 29, 2024.The patient was discharged in stable condition with no complications. She was advised to follow up in the outpatient department for continued monitoring and management of her condition.

Discussion

Adrenal lymphangiomas are rare, benign cystic lesions of the adrenal gland, with only a limited number of cases documented in the literature. They are typically discovered incidentally during imaging for unrelated conditions, as in the present case of a 32-year-old female patient. While the exact etiology of adrenal lymphangiomas remains unclear, it is generally believed that these lesions result from the proliferation of lymphatic vessels or lymphatic endothelial cells within the adrenal gland. The differential diagnosis of adrenal cystic lesions can be challenging due to the overlap in imaging features with other adrenal masses, such as pseudocysts, epithelial cysts, and neoplastic cysts. Therefore, while imaging can raise suspicion, a definitive diagnosis of adrenal lymphangioma often requires histopathological confirmation, as was achieved in this case.

The management of adrenal lymphangiomas depends on various factors, including the size of the lesion, presence of symptoms, and hormonal activity. In asymptomatic patients with small, non-functioning cysts, conservative management with periodic imaging may be sufficient. However, for larger cysts, those causing compressive symptoms, or when malignancy cannot be excluded, surgical resection is generally indicated. In this case, although the adrenal cyst was non-functional, the patient opted for surgical intervention after a thorough discussion of the risks and benefits. Robotic-assisted partial adrenalectomy was performed to minimize invasiveness and facilitate a precise excision, and the patient had a favorable postoperative outcome, suggesting that minimally invasive techniques can be both effective and safe for adrenal lymphangioma resection.

The postoperative diagnosis of adrenal lymphangioma highlights the value of histopathological examination in verifying the nature of adrenal lesions, especially for unusual presentations. Adrenal lymphangiomas are benign and carry a good prognosis following complete resection, with a low likelihood of recurrence. Nonetheless, given their rarity and the possibility of misdiagnosis with other adrenal pathologies, this case emphasizes the need for a multidisciplinary approach involving radiologists, endocrinologists, and pathologists. Additionally, long-term follow-up is recommended to monitor for any potential recurrence or complications, particularly in patients with complex surgical histories. This case contributes to the limited literature on adrenal lymphangiomas and reinforces the importance of individualized management strategies for rare adrenal lesions.