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Symptomatic Giant Adrenal Myelolipoma Associated with Cholelithiasis: Two Case Reports

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Symptomatic Giant Adrenal Myelolipoma Associated with Cholelithiasis: Two Case Reports Case Report Symptomatic giant adrenal myelolipoma associated with cholelithiasis: Two case reports Shahina Bano, Sachchida Nand Yadav1, Vikas Chaudhary2, Umesh Chandra Garga1 Department of Radiodiagnosis, Govind Ballabh Pant Hospital and Maulana Azad Medical College, 1Department of Radiodiagnosis, Dr. Ram Manohar Lohia Hospital and Postgraduate Institute of Medical Education & Research, New Delhi, 2Department of Radiodiagnosis, Employees' State Insurance Corporation Model Hospital, Gurgaon, Haryana, India Abstract In this article, we have discussed about two cases of adrenal myelolipoma and aim to discuss the role of imaging in their diagnosis and their management. Different imaging techniques such as ultrasound, computed tomography and magnetic resonance imaging were used to aid in diagnosis in each of the cases. The findings have been highlighted here. In each of the cases, the diagnosis could be confirmed by imaging, and there was cholelithiasis seen associated with unilateral adrenal myelolipoma. Adrenal myelolipomas are rare, benign, non-functional tumors of adrenal gland. Most tumors are unilateral and small; bilateral, giant myelolipomas are extremely rare. The association of adrenal myelolipoma with gallstones is uncommon. To our knowledge only two cases of such an association have been reported in the literature. However, the possibility does exist and steps should be taken to ensure a complete diagnosis. Also, it is important to understand the key points which help us in diagnosing adrenal myelolipomas by imaging. Key Words: Adrenal myelolipoma, computed tomography, magnetic resonance imaging, magnetic resonance cholangiopancreatography, ultrasound Address for correspondence: Dr. Shahina Bano, Room No: 603, New Resident Doctor’s Hostel, Govind Ballabh Pant Hospital, New Delhi ‑ 110 002, India. E‑mail: [email protected] Received: 07.11.2010, Accepted: 13.12.2010 INTRODUCTION known to occur at various sites, including lung, mediastinum, liver, spleen, mesentery, pelvis, presacral region, retroperitoneum Adrenal myelolipomas are benign, biochemically inactive and thoracic spine.[2] The association of myelolipomas with neoplasms composed of mature adipose and hematopoietic obesity, hypertension, and diabetes mellitus is well known;[3‑5] tissues in varying proportion. They were first described by Gierke however, their association with gallstones is uncommon. To our in 1905, and were named as myelolipoma in 1929 by Oberling.[1] knowledge only two cases of such an association are present in Since they are usually asymptomatic and diagnosed incidentally literature.[6,7] We herein report two cases of giant symptomatic at autopsy or on imaging studies, they are often classified as adrenal myelolipoma associated with cholelithiasis. We further ‘incidentalomas’. Although myelolipomas are commonly found discuss the role of imaging in the diagnosis of myelolipomas, in adrenal glands, rare extra‑adrenal myelolipomas are also various important but rare complications associated with them, and their management alternatives. Access this article online CASE REPORTS Quick Response Code: Website: www.urologyannals.com Case 1 A 65‑year‑old obese female with remote past history of DOI: cholecystectomy (for cholelithiasis), presented with complaint 10.4103/0974-7796.91621 of nausea, mild jaundice and persistent dull aching pain in whole upper abdomen of few weeks duration. There was Urology Annals | Jan - Apr 2012 | Vol 4 | Issue 1 55 Bano, et al.: Adrenal myelolipoma associated with cholelithiasis no significant past or present medical history. Per abdomen atypia. The diagnosis of adrenal myelolipoma was confirmed. examination revealed a well defined, smooth mass in left The postoperative recovery was uneventful. hypochondrium. On laboratory analysis, liver function test revealed increase in total bilirubin (3.24 mg/dl), conjugated Case 2 bilirubin (2.17 mg/dl), and alkaline phosphatase (224 U/L). A 33‑year‑old obese female presented with vague right Renal function test was unremarkable. Routine blood abdominal pain for last three months. She had no other examination revealed mildly decreased hemoglobin (8 g/dL). significant medical history. On laboratory analysis, hemogram, Endocrine evaluation of the adrenal mass revealed normal biochemical and endocrine function tests were within normal 24 hours urinary metanephrine (0.9 mg/24 hours) and limits. Liver and renal function tests were also normal. Levels plasma cortisol (<5 µg/dl). Furthermore, the levels of two of tumor markers such as carcinoembryonic‑antigen and tumor markers, carcinoembryonic antigen (1.25 ng/ml) and alpha‑fetoprotein were unremarkable. Physical examination alpha‑fetoprotein (1.00 ng/ml) were also within normal range. revealed a right hypochondrial lump. The patient was sent to the radiology department for evaluation of obstructive jaundice and left hypochondrial lump. Ultrasound examination of abdomen showed a huge, well encapsulated, solid echogenic mass lesion between the liver and Ultrasound examination revealed a large well encapsulated, right kidney [Figure 2a]. Normally distended gallbladder with heterogeneous left suprarenal mass lesion [Figure 1a]. In addition, multiple intraluminal calculi was also noted. CBD and IHBR non‑visualization of gall bladder (status post‑cholecystectomy were normal. Plain and contrast CT abdomen [Figure 2b] for cholelithiasis), with dilatation of common bile duct (CBD revealed a well circumscribed 83.9 (AP) × 72.2 (TR) × 104.4 20.8 mm diameter), increased width and prominence of (SI) mm sized right suprarenal mass predominantly of fat central intrahepatic biliary radicals (IBHR) in absence of any density (‑50 to ‑90 HU) with peripheral small enhancing soft intraluminal filling defect were also noted. The plain and contrast tissue component on the postero‑medial aspect of the mass. A computed tomography image [Figure 1b] of the abdomen punctate focal calcified area was also seen adjacent to the soft revealed a well circumscribed, heterogeneous enhancing mass of tissue component. Furthermore, multiple small focal calcific 91.4 (AP) × 67.3 (TR) × 101.3 (SI) mm in the left supra‑renal lesions (calcified granulomas) randomly distributed within the region causing inferior displacement of the left kidney. The lesion splenic parenchyma were seen. Suprarenal nature of the mass was predominantly of fat density (‑30 to ‑90 HU) with few was confirmed by coronal and sagittal CT images [Figure 2c], enhancing soft tissue components and irregular septations. Few which also showed postero‑inferior displacement of right kidney. tiny flecks of calcifications were also visible in the lateral wall Left adrenal gland and both kidneys appeared normal. MR of the mass. The coronal and sagittal CT images [Figure 1c] abdomen revealed predominantly hyperintense mass on T1‑and confirmed the adrenal origin of the mass. The function of T2‑weighted image [Figure 2d]. T2W‑fat‑suppression image both kidneys appeared normal. Abdominal magnetic resonance [Figure 2e] showed loss of signal from large fatty portion of imaging revealed a heterogeneous, high intensity mass on T1 the mass, but persistence of increased signal intensity in areas and T2‑weighted images [Figure 1d], with hypointense septae. occupied by hematopoietic tissue and intratumoral hemorrhage. Linear hyperintense areas posteriorly on T2WI represented Double echo chemical shift gradient echo imaging [Figure 1f] intratumoral bleed. Fat‑saturated T2‑ weighted gradient echo demonstrated few focal areas of signal loss in out‑of‑phase image sequences [Figure 1e] showed loss of signal intensity in the fatty as compared to in‑phase image, consistent with the presence of part of the mass, while the hematopoietic tissue and hemorrhage microscopic (intracellular) fat, while the major portion of the demonstrated persistent areas of increased signal intensities. mass demonstrated high signal intensity consistent with the Magnetic resonance cholangiopancreatography (MRCP) presence of macroscopic (extracellular) fat. confirmed the ultrasonographic finding of dilated CBD and central IHBR; T2‑haste coronal sequence demonstrated partial The imaging finding and hormonal evaluation highly suggested benign stricture at ampullary end of CBD as a cause of biliary the diagnosis of a nonfunctioning right adrenal myelolipoma. dilation [Figure 1f]. Right adrenalectomy and cholecystectomy was performed in the same setting to prevent future risk of spontaneous The imaging findings were highly suggestive of left adrenal hemorrhage and gall stone associated complications. myelolipoma, with CBD and central IHBR dilatation due to Histopathology confirmed the diagnosis. Post‑operative benign stricture at ampullary end of CBD. Right adrenal gland period was uneventful. was normal. The patient underwent surgical resection of large adrenal mass and endoscopic stenting of the biliary stricture. DISCUSSION Histopathology of resected left adrenal mass showed mature fat cells admixed with hematopoietic tissues without cellular Adrenal myelolipomas are slow growing, uncommon benign 56 Urology Annals | Jan - Apr 2012 | Vol 4 | Issue 1 Bano, et al.: Adrenal myelolipoma associated with cholelithiasis Figure 1b: On axial plain and contrast-enhanced computed tomography images through upper abdomen, the mass appears to be well circumscribed and predominantly of fat density (‑30 to ‑90 HU). Heterogeneous enhancement pattern is due to presence of enhancing soft tissue component (myeloid tissue) and septations. Few tiny flecks of calcifications
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