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CASE REPORT |
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| Year : 2002 | Volume
: 18
| Issue : 2 | Page : 148-150 |
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Primary benign para-adrenal teratoma in an adult
JN Chakraborty, NK Mondol, P Mahapatra, B Mukherjee
Department of Urology, Kothari Medical Centre and Research Institute, Kolkata, India
Correspondence Address:
B Mukherjee
Department of Urology, Kothari Medical Centre & Research Institute, 8/3, Alipore Road, Kolkata - 700 027
India
 Source of Support: None, Conflict of Interest: None  | Check |
 Abstract | | |
A 38-year-old male presented with right hypochondralgia and backache for two rears. Plain X-ray KUB showed a radio-opaque calculus in the left renal area. Urinalysis and culture were negative. IVU (after a normal serum urea and creatinine report) showed the characteristic tooth-like calcification to be situated above and medial to the upper pole of left kidney which itself is deformed and pushed downwards. CT scan with contrast showed a mass of approx. 6.5 x 6.5 cm with central hypodense area and surrounding dense wall adherent to the adjacent retroperitoneum and major vessels. A solid calcified element (dermoid plug) was seen projecting into the cavity. On exploration, the para-adrenal mass was found to be adherent to the aorta and adjacent structures. The offensive cheesy content was first removed, followed by gradual and careful separation of the wall in parts, thus avoiding injury to the major vessels. The wall contained several abortive tooth structures, hair shafts and skin-like lining. Histopathology showed several ectodermal structures (tooth, hair follicles, sweat and sebaceous glands etc), but no immature element. The patient recovered uneventfully and is doing well at present.
Keywords: Retroperitoneal Teratoma; Dermoid Cyst
How to cite this article:
Chakraborty J N, Mondol N K, Mahapatra P, Mukherjee B. Primary benign para-adrenal teratoma in an adult. Indian J Urol 2002;18:148-50 |
| Introduction | |  |
The vast majority of benign (mature) teratomas are cystic and are better known in clinical parlance as dermoid cysts. Primary benign retroperitoneal teratoma is a rare tumour in the adult population. In this case, we described a case of primary benign cystic teratoma located in the vicinity of the upper pole of left kidney (Paraadrenal).
| Case Report | | |
A 38-year-old male presented with pain in the upper abdomen and back since two years. The pain was dull aching in nature and used to aggravate on walking or jolting. There had been a few episodes of burning micturition. An X-ray KUB showed a radio opaque shadow in the left renal area at the junction of Ll-L2 vertebra which was assumed to be a left renal calculus by his physician and advised operation. But the patient refused operation and after two years, the persistent pain led him to our institute, when an IVU (after a normal serum urea and creatinine report) showed the characteristic tooth-like calcification to be situated above and medial to the left kidney, which itself was pushed downwards and laterally [Figure - 1]. The excretory function was normal on both sides. Urinalysis and culture was negative. USG revealed a heterogenous retroperitoneal (a complex mass of anechoic and echogenic components) situated in between the aorta and upper pole of left kidney. Axial and coronal CT scan with contrast showed an irregular, thick walled, rounded retroperitoneal mass (6.5 x 6.5 cm approx.) behind and inseparable from the tail of pancreas with a deformed, deviated left kidney and evidence of apparent invasion into the adjacent retroperitoneum [Figure - 2]. The mass had a central hypodense area (fatlike) with multiple large calcified shadows as well as air pockets. The first provisional diagnosis was infected dermoid cyst although the possibility of a cystic pancreatic neoplasm was kept in mind.
Abdomen was explored by a roof-top incision - the left colon and splenic flexure mobilised to the right. A retroperitoneal cystic bulge was seen at the para-aortic region medial to the upper pole of left kidney [Figure - 3]. After initial check aspiration showing very offensive, cheesy material, excision of the cyst started, but it was found to be adherent to the lateral wall of aorta, coeliac axis and other adjacent structures. Hence the cyst was removed keeping the right lateral wall intact and the inner lining was excised very carefully. As the wall was adherent to the contiguous retroperitoneal and adjacent structures (and also keeping in mind its proximity to major vessels), the thick wall was removed carefully part by part. The wall was very thick and contained abortive tooth structures, hair and thicked epithelium mimicking epidermis [Figure - 4]. Histopathological study also revealed sebaceous and sweat glands, hair shafts, cartilage, tooth structures and stratified squamous epithelium. No immature element was detected. A drainage tube was left in the retroperitoneal cavity which was removed on the 10 th postoperative day. The patient recovered uneventfully and is presently doing well.
| Discussion | | |
Primary retroperitoneal teratomas are rare in adults. Approximately 3% of all teratomas occur at the extragonadal sites (retroperitoneum, floor of mouth, mediastinum, thyroid, urinary bladder and kidney). Retroperitoneal teratomas represent only 1% of all teratomas. [1] They are believed to originate from totipotent germ cells. Though solid teratomas may contain tissues from all three germ layers (ectoderm, endoderm, mesoderm), cystic teratomas usually contain only ectodermal elements. They may have both mature and immature elements. Malignant transformation in a cystic teratoma may occur in a foci of immature element and the usual type is a squamous cell carcinoma, though other types of malignancy may also be seen. Para-adrenal teratomas usually present with hypochondralgia during exercise. [2] Physical examination is usually normal. Plain antero-posterior radiograph of abdomen can show irregular, multiple tooth-like calcification within the left or right upper quadrant, [3] which may be misinterpreted as renal calculus unless confirmed by other imaging studies. A benign cystic teratoma in juxta-pancreatic location may also present as an inflammatory or neoplastic pancreatic cyst [5] and in that location, Bacteriodes fragilis infection of the cyst is also reported. [5] Transabdominal ultrasonography usually shows a complex mass of anechoic and echogenic components characteristic of a dermoid cyst. [4] CT scan and MRI findings are characteristic and diagnostic, showing central hypodense area (representing fat or sebaceous material), the surrounding well-demarcated wall and irregular, toothlike calcifications in it. [3],[10] The solid element arising from the wall (the dermoid plug), when demonstrated, is very characteristic of cystic teratomas.[2],[9]
Laparoscopy may be helpful in detecting the exact location of the cyst, but if it fails, intraoperative ultrasound is often beneficial in confirming the presence and exact location of a cystic retroperitoneal teratoma. [8] The margins of a benign cystic teratoma are usually well demarcated and completely separated from adjacent structures [6] , when total removal can be attempted. But when there is gross adhesion to contiguous anatomical structures, close proximity to major vessels or unexpected operative difficulties due to the expansive volume, [7] total removal may be associated with significant morbidity and mortality. [8] In these situations, initial decompression with careful removal of the wall in parts may have to be chosen, as we have done in this particular case.
| References | | |
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| 2. | Hirano S, Kawaguchi S, Mikawa I et al. Primary retroperitoneal teratoma in an adult: ultrasonographic, computer tomographic and magnetic resonance imaging demonstrations. Hinyokika Kiyo 1988:34: 2031-2034. |
| 3. | Terada Y. Kato A, Kishi H et al. Nuclear magnetic resonance imaging of a benign cystic teratoma in the retroperitoneum. J Urol 1987; 137: 106-108. |
| 4. | Kanizsai B, Turi Z, Orley J et al. Sonographic diagnosis of a retroperitoneal dermoid cyst in a young girl. Ultrasound Obstet Gynecol 1998; 12: 367-368. |
| 5. | Dewar G, Arnold M, Li AK. Retroperitoneal dermoid presenting as an infected pancreatic cyst. Aust NZ J Surg 1990; 60: 488-489. |
| 6. | Yokoyama O, Nakajima K, Nakashima T et al. Benign retroperitoneal cystic teratoma with postoperative ascites - a case report. Hinyokika Kiyo 1988; 34: 1639-1643. |
| 7. | Danieli D, Serio G, Tenchini P et al. Cystic neoformations of the retroperitoneal space. Chir Ital 1983; 35: 157-179. |
| 8. | Sharpe LA, Van Oppen DJ. Laparoscopic removal of a benign pelvic retroperitoneal dermoid cyst. J Am Assoc Gynecol Laparosc 1995; 2: 223-226. |
| 9. | Bellin MF, Duron JJ, Curet P et al. Primary retroperitoneal teratoma in the adult : Correlation of features with CT and Pathology. |
| 10. | Crantson PE, McPherson SH. Para-adrenal teratoma : CT presentation. South Med J 1989; 82: 518-519. |
[Figure - 1], [Figure - 2], [Figure - 3], [Figure - 4]
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| Durand, P. and Malone, A. and Courtney, D. | | Clinical Anatomy. 2013; 26(5): 638-640 | | [Pubmed] | |
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