- Email: [email protected]
Cavernous Hemangioma of the Adrenal Gland
0022 ~ 534 7 /26 / l 3fi ~-D341 $02 .orJ / 0 THE ,JOtJP:.NAI. G:' 1,JROLOGV
C0py:cight
1986 by The Viillia~1s
'i,?Vilkins
HE1V1AN GI O:NIA ELIAHU GOREN, DAVID
THE ADRENAL GLAND
RENATA M. REIF
AND
AVRAHAM EIDELMAN*
From the Department of Urology and Pathology, Assaf Haro/eh Medical Center, Zerifin and Sachler School of Medicine, Tel-Auiu University, Tel-Auiu, Israel
ABSTRACT
Adrenal hemangiomas are rare and seldom found ante mortem. We report a case of adrenal hemangioma that was discovered incidentally and removed surgically. The pertinent literature 1s reviewed. Hemangiomas of the adrenal gland are rare. Before 1955 only 7 cases had been reported in the literature, all of which had been diagnosed at autopsy. 1 In 1955 Johnson and Jeppesen reported the first case of an adrenal mass excised surgically, which proved to be an adrenal hemangioma on pathological examination. 2 Since then 8 other cases have been reported. 1- 8 We describe another case of adrenal hemangioma that was removed surgically. CASE REPORT
A 79-year-old woman was hospitalized for evaluation of a mass above the right discovered incidentally during excretory urography for recurrent urinary tract infecexcept for dysuria. History was tions. She was ~ ·'=~•,,= unremarkable except os1cecmclros1s and multiple fractures following repeated mild trauma. Physical examination revealed no palpable abdominal masses or other pathological findings. Blood pressure was 130/60. Laboratory data included hemoglobin 12. 7 gm./100 ml., serum blood urea nitrogen 10.3 mg./100 creatinine 0.8 mg./100 ml., and normal serum calcium, phosphorus and alkaline phosphatase levels. Hormonal studies based on a 24hour urine collection revealed 17-ketosteroids 1.6 mg. (normal 4 to 11 mg./24 hours), cortisol 78 µg. (normal 55 to 205 µg./ 24 hours) and vanillylmandelic acid spot test negative. An abdominal plain film showed no calcifications. An IVP revealed a suprarenal lesion displacing the right kidney inferiorly. Ultrasound demonstrated the right kidney to measure 10 x 4 x 5. 7 cm., and above the upper pole of the a solid nonhomogeneous mass about 6 CffL in diameter was seen (fig. Computerized tomography confirmed the finding of a demarcated mass with an internal "vrn,.,dvcu,e,_y rounded, of 24 Hounsfield units for an area of higher density Injection contrast material caused marked enhancement of the dense area. The mass was consistent with an adrenal tumor without of spread to the surrounding tissues. a thoracoabdominal inc1s1on The adrenal mass v;as vena cava and renal upper from which it was freed without difficulty. Exploration revealed no enlarged regional lymph nodes or other abdominal masses. Convalescence was uneventful and the patient was discharged from the hospital 2 weeks postoperatively. The gross specimen consisted of an oval, encapsulated, moderately firm mass measuring 9 x 7 x 5 cm. and weighing 120 gm. The cut surface showed that most of the mass was homogeneous and pinkish with no recognizable structure but with 0
Accepted for publication August 23, 1985. . *Requests for reprints: Urology Department, Assaf Harofeh Medical Center, Zerifin 703,50, Israel.
341
FIG. 1. Longitudinal ultrasonography shows suprarenal mass (left) above normal right kidney.
FIG. 2. CT shows right adrenal mass
areas of recent hemorrhage. At the periphery a thin rim of yellowish tissue was seen, compatible with adrenal cortical tissue, widening on the medial aspect into a crescentic compressed adrenal gland containing recent hemorrhages (fig. 3). The histological examination showed that the amorphous areas were compatible with old hemorrhages, including iron pigment deposition in several sites. The peripheral yellowish rim represented adrenal cortical tissue with some pressure atrophy and recent hemorrhages. In several areas accumulations of dilated blood vessels were found, which were compatible with a cavernous hemangioma. There was no evidence of malignancy (fig. 4).
342
GOREN AND ASSOCIATES DISCUSSION
Among the various adrenal cysts endothelial cysts are further subdivided into hemangiomas and the more common lymphangiomas. A review of the literature revealed that patient age ranged from 25 to 79 years, the size of the tumor from 2 to 22 cm. in diameter and the weight from a few grams to 5,000 gm. All of the cases described have involved the adrenal cortex and all patients except 2 were women. Bilateral involvement has been reported only twice. 9 • 10 Hemangiomas of the urinary tract occur infrequently but they have been reported in the bladder, 11 ureter 12 and prostate. 13 Other sites include the perineum, liver, ovary, uterus and skull. 3 Most of the hemangiomas, as in our case, were incidential findings or they were recognized because of the mechanical effects of a mass on the surrounding structures.
FIG. 3. Gross section of mass (9 X 7 cm.) shows crescent of adrenal cortex (upper rim of specimen). Bulky remainder is large organized hematoma. Arrow indicates hemangioma.
In 8 cases radiological findings of adrenal calcifications were noted. Rothberg and associates believed that these calcifications were owing to the formation of phleboliths. 6 The history of our patient included osteoporosis and multiple fractures apparently caused by mild repeated trauma. It is likely that trauma resulting in rib fractures was a possible contributing factor in the etiology of the adrenal hemorrhages in addition to the pre-existing hemangioma.
REFERENCES
1. Vargas, A. D.: Adrenal hemangioma. Urology, 16: 389, 1980. 2. Johnson, C. C. and Jeppesen, F. B.: Hemangioma of the adrenal. J. Urol., 74: 573, 1955. 3. Weiss, J. M. and Schulte, J. W.: Adrenal hemangioma: a case report. J. Urol., 95: 604, 1966. 4. Nagatani, M., Harada, T. and Takeda, T.: Case of successfully removed giant hemangioma of the adrenal gland. Naika, 24: 383, 1969. 5. Ruebel, A. A.: Adrenal hemangioma. Urology, 2: 289, 1973. 6. Rothberg, M., Bastidas, J., Mattey, W. E. and Bernas, E.: Adrenal hemangiomas: angiographic appearance of a rare tumor. Radiology, 126: 341, 1978. 7. Zeman, V. and Hruby, J.: Cavernous hemangioma of the adrenal gland. Rozhl. Chir., 58: 195, 1979. 8. Orringer, R. D., Lynch, J. A. and McDermott, W. V.: Cavernous hemangioma of the adrenal gland. J. Surg. Oncol., 22: 106, 1983. 9. Payne, J. F.: Vascular tumours of the liver, supra-renal capsules, and other organs. Trans. Path. Soc. London, 20: 203, 1869. 10. Muller-Stuhler, M.: Bilateral cavernous hemangioma of suprarenals. Virch. Arch. f. Path. Anat., 290: 177, 1933 (Cited by Vargas 1 ). 11. Esguerra, A., Carvajal, A. and Mouton, H.: Pelvic arteriography in the diagnosis of hemangioma of the bladder. J. Urol., 109: 609, 1973. 12. Uhlir, K: Hemangioma of the ureter. J. Urol., llO: 647, 1973. 13. Sundarasivarao, D., Banerjea, S., Nageswararao, A. and Rao, N. V.: Hemangioma of the prostate: a case report. J. Urol., 110: 708, 1973.
FIG. 4. Microscopic sections. A, cavernous hemangioma (left) adjacent to adrenal cortical tissue (right). B, another area of hemangioma (left). C, high power view of endothelial lining of vascular lumina. H & E stain.
C0py:cight
1986 by The Viillia~1s
'i,?Vilkins
HE1V1AN GI O:NIA ELIAHU GOREN, DAVID
THE ADRENAL GLAND
RENATA M. REIF
AND
AVRAHAM EIDELMAN*
From the Department of Urology and Pathology, Assaf Haro/eh Medical Center, Zerifin and Sachler School of Medicine, Tel-Auiu University, Tel-Auiu, Israel
ABSTRACT
Adrenal hemangiomas are rare and seldom found ante mortem. We report a case of adrenal hemangioma that was discovered incidentally and removed surgically. The pertinent literature 1s reviewed. Hemangiomas of the adrenal gland are rare. Before 1955 only 7 cases had been reported in the literature, all of which had been diagnosed at autopsy. 1 In 1955 Johnson and Jeppesen reported the first case of an adrenal mass excised surgically, which proved to be an adrenal hemangioma on pathological examination. 2 Since then 8 other cases have been reported. 1- 8 We describe another case of adrenal hemangioma that was removed surgically. CASE REPORT
A 79-year-old woman was hospitalized for evaluation of a mass above the right discovered incidentally during excretory urography for recurrent urinary tract infecexcept for dysuria. History was tions. She was ~ ·'=~•,,= unremarkable except os1cecmclros1s and multiple fractures following repeated mild trauma. Physical examination revealed no palpable abdominal masses or other pathological findings. Blood pressure was 130/60. Laboratory data included hemoglobin 12. 7 gm./100 ml., serum blood urea nitrogen 10.3 mg./100 creatinine 0.8 mg./100 ml., and normal serum calcium, phosphorus and alkaline phosphatase levels. Hormonal studies based on a 24hour urine collection revealed 17-ketosteroids 1.6 mg. (normal 4 to 11 mg./24 hours), cortisol 78 µg. (normal 55 to 205 µg./ 24 hours) and vanillylmandelic acid spot test negative. An abdominal plain film showed no calcifications. An IVP revealed a suprarenal lesion displacing the right kidney inferiorly. Ultrasound demonstrated the right kidney to measure 10 x 4 x 5. 7 cm., and above the upper pole of the a solid nonhomogeneous mass about 6 CffL in diameter was seen (fig. Computerized tomography confirmed the finding of a demarcated mass with an internal "vrn,.,dvcu,e,_y rounded, of 24 Hounsfield units for an area of higher density Injection contrast material caused marked enhancement of the dense area. The mass was consistent with an adrenal tumor without of spread to the surrounding tissues. a thoracoabdominal inc1s1on The adrenal mass v;as vena cava and renal upper from which it was freed without difficulty. Exploration revealed no enlarged regional lymph nodes or other abdominal masses. Convalescence was uneventful and the patient was discharged from the hospital 2 weeks postoperatively. The gross specimen consisted of an oval, encapsulated, moderately firm mass measuring 9 x 7 x 5 cm. and weighing 120 gm. The cut surface showed that most of the mass was homogeneous and pinkish with no recognizable structure but with 0
Accepted for publication August 23, 1985. . *Requests for reprints: Urology Department, Assaf Harofeh Medical Center, Zerifin 703,50, Israel.
341
FIG. 1. Longitudinal ultrasonography shows suprarenal mass (left) above normal right kidney.
FIG. 2. CT shows right adrenal mass
areas of recent hemorrhage. At the periphery a thin rim of yellowish tissue was seen, compatible with adrenal cortical tissue, widening on the medial aspect into a crescentic compressed adrenal gland containing recent hemorrhages (fig. 3). The histological examination showed that the amorphous areas were compatible with old hemorrhages, including iron pigment deposition in several sites. The peripheral yellowish rim represented adrenal cortical tissue with some pressure atrophy and recent hemorrhages. In several areas accumulations of dilated blood vessels were found, which were compatible with a cavernous hemangioma. There was no evidence of malignancy (fig. 4).
342
GOREN AND ASSOCIATES DISCUSSION
Among the various adrenal cysts endothelial cysts are further subdivided into hemangiomas and the more common lymphangiomas. A review of the literature revealed that patient age ranged from 25 to 79 years, the size of the tumor from 2 to 22 cm. in diameter and the weight from a few grams to 5,000 gm. All of the cases described have involved the adrenal cortex and all patients except 2 were women. Bilateral involvement has been reported only twice. 9 • 10 Hemangiomas of the urinary tract occur infrequently but they have been reported in the bladder, 11 ureter 12 and prostate. 13 Other sites include the perineum, liver, ovary, uterus and skull. 3 Most of the hemangiomas, as in our case, were incidential findings or they were recognized because of the mechanical effects of a mass on the surrounding structures.
FIG. 3. Gross section of mass (9 X 7 cm.) shows crescent of adrenal cortex (upper rim of specimen). Bulky remainder is large organized hematoma. Arrow indicates hemangioma.
In 8 cases radiological findings of adrenal calcifications were noted. Rothberg and associates believed that these calcifications were owing to the formation of phleboliths. 6 The history of our patient included osteoporosis and multiple fractures apparently caused by mild repeated trauma. It is likely that trauma resulting in rib fractures was a possible contributing factor in the etiology of the adrenal hemorrhages in addition to the pre-existing hemangioma.
REFERENCES
1. Vargas, A. D.: Adrenal hemangioma. Urology, 16: 389, 1980. 2. Johnson, C. C. and Jeppesen, F. B.: Hemangioma of the adrenal. J. Urol., 74: 573, 1955. 3. Weiss, J. M. and Schulte, J. W.: Adrenal hemangioma: a case report. J. Urol., 95: 604, 1966. 4. Nagatani, M., Harada, T. and Takeda, T.: Case of successfully removed giant hemangioma of the adrenal gland. Naika, 24: 383, 1969. 5. Ruebel, A. A.: Adrenal hemangioma. Urology, 2: 289, 1973. 6. Rothberg, M., Bastidas, J., Mattey, W. E. and Bernas, E.: Adrenal hemangiomas: angiographic appearance of a rare tumor. Radiology, 126: 341, 1978. 7. Zeman, V. and Hruby, J.: Cavernous hemangioma of the adrenal gland. Rozhl. Chir., 58: 195, 1979. 8. Orringer, R. D., Lynch, J. A. and McDermott, W. V.: Cavernous hemangioma of the adrenal gland. J. Surg. Oncol., 22: 106, 1983. 9. Payne, J. F.: Vascular tumours of the liver, supra-renal capsules, and other organs. Trans. Path. Soc. London, 20: 203, 1869. 10. Muller-Stuhler, M.: Bilateral cavernous hemangioma of suprarenals. Virch. Arch. f. Path. Anat., 290: 177, 1933 (Cited by Vargas 1 ). 11. Esguerra, A., Carvajal, A. and Mouton, H.: Pelvic arteriography in the diagnosis of hemangioma of the bladder. J. Urol., 109: 609, 1973. 12. Uhlir, K: Hemangioma of the ureter. J. Urol., llO: 647, 1973. 13. Sundarasivarao, D., Banerjea, S., Nageswararao, A. and Rao, N. V.: Hemangioma of the prostate: a case report. J. Urol., 110: 708, 1973.
FIG. 4. Microscopic sections. A, cavernous hemangioma (left) adjacent to adrenal cortical tissue (right). B, another area of hemangioma (left). C, high power view of endothelial lining of vascular lumina. H & E stain.