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Genitourinary Imaging |
1 Division of Tropical Medicine, Hospital Carlos III, C/Sinesio Delgado 10,
Madrid 28029, Spain.
2 Department of General Surgery, Hospital Universitario 12 de Octubre, Avda.
Córdoba s/n, Madrid 28041, Spain.
3 Department of Radiology, Hospital Carlos III, Madrid 28029, Spain.
Received October 22, 2003; accepted after revision January 6, 2004.
Address correspondence to J. L. Polo
(polosabau{at}yahoo.com).
Case Report
A 21-year-old woman from the Republic of Equatorial Guinea was referred to our institution to complete the diagnostic workup for suspected heart or liver disease. She had delivered a healthy child 3 years earlier. At 10 years old, she began to feel a progressive increase in the size of her abdomen, with occasional mild diffuse pain and, lately, shortness of breath when resting.
At physical examination, she did not appear ill. She was afebrile, her arterial blood pressure was 100/60 mm Hg, and a mild tachypnea was noted. Abdominal examination disclosed a global distention, with dullness on the left half and tympany on the right, regardless of changes in the patient's position. No other physical findings were observed.
Blood cell count showed microcytic anemia and a mild leukopenia. Results of routine biochemistry evaluations, including renal and liver function tests, were normal. CT revealed a large cystic mass occupying most of the abdomen and pelvis (Figs. 1A and 1B). There were no signs of organ infiltration, and normal ovaries were identified.
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Surgical excision of the mass was recommended. After laparotomy, a great cystic retroperitoneal mass was seen, extending from the epigastrium to the pelvis, with lateral displacement of the small and large bowel. The mass did not infiltrate the pancreas, spleen, uterus, or ovaries; all these organs had a normal appearance. Because the mass closely adhered to the cranial extremity of the left kidney, resection of the adrenal gland of that side was performed. The tumor measured 38 x 30 x 30 cm and weighed 10 kg (Fig. 1C). The postoperative period was uneventful.
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Histologic analysis confirmed the diagnosis of a mature teratoma, with abundant fat deposits and different tissues such as cartilage, skin, and hair identified in the cystic wall (Fig. 1D). Because of the close relationship of the mass to the left adrenal gland, we could not rule out this organ as the site of origin.
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Discussion
Teratoma is a germ cell tumor derived from totipotential cells, which comprise several parenchymal cell types originating from more than one germ cell layer, usually all three, and giving rise to different tissues such as skin, muscle, nerve, fat, and tooth structures. Most teratomas are found in the gonads. Nevertheless, many extragonadal sites have been reported, including the mediastinum, retroperitoneum, cranium, sacrococcygeal region, the large bowel, and even the tongue [1].
Mature teratomas are usually benign, although they have the potential for malignant transformation [2]. Most of them are cystic and therefore are classically called dermoid cysts. Radiologic findings such as calcification and predominant fat content are often seen and make a teratoma, in some instances, difficult to distinguish from other lipomatous tumors of the adrenal gland such as myelolipoma or angiomyolipoma [3]. Compared with these other adrenal tumors, teratomas tend to exhibit a more heterogeneous appearance with a mixture of fluid, fat, and sebum forming a fat–fluid level as seen in our patient, although she did not have calcifications.
The adrenal gland as the origin of a teratoma is extremely rare. To our knowledge, this is the fifth case reported in the English language literature [4, 5]. However, in all cases, including ours, there have been serious difficulties clarifying the exact origin of the mass because retroperitoneal teratomas with extension to the paraadrenal region may also occur [6]. Because of the close relationship of the tumor with the adrenal gland observed during surgery and after microscopic examination, origin in this gland cannot be ruled out.
The other unique features of this case, the striking tumor size and long-term evolution, favored the benign nature of the tumor and prevented the preoperative location of the site of origin. Surprisingly, despite its large volume, the mass did not produce significant symptoms in the patient other than abdominal discomfort or lead to malfunction of any organ in the vicinity.
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