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Benign Cysts of the Adrenal Gland: A Case Report
THE JOURNAL OF UROLOGY
Vol. 68, No. 1, July 1952 Printed in U.S.A.
BENIGN CYSTS OF THE ADRENAL GLAND: A CASE REPORT DEAN PARKER
Benign cysts of the adrenal gland are rare. Prior to 1920, fourteen cases were reported and only five additional cases have been reported in the American literature since then. The first case was cited by Greiselius in 1670. 1 Terrier and Lecene published their case and reviewed nine others reported up to 1906. 2 They also classified adrenal cysts: 1) parasitic, 2) true glandular retention cysts, 3) cystic adenomas, 4) serous or lymphangiomatous cysts and 5) pseudocysts (hemorrhagic cysts). Only 2 cases reported, that of Levinson and this case, were diagnosed prior to surgery. The etiology of adrenal cysts is somewhat confusing and only a little clarity is gained from the review of literature. Major and Black3 reported a case of bilateral adrenal cysts which appeared to arise from dilated capillaries or sinusoids between columns of adrenal cells. The cases of Oberndorfer 4 and one by de Vecchi 5 appeared to arise from lymphangiectasia, as did the most recent case in the literature, that of Zuckner. 6 Henchen7 recorded a hemorrhagic cyst of the adrenal while Sick 8 reported a ciliated cyst which must have developed on the basis of displacement of tissue from the urogenital anlage. Stoerk, 9 and later Nowicki, 10 both expressed doubts as to the existence of true epithelial cysts. This seems logical since there is no tendency to acini formation in the cells of the adrenal gland. Reimann and Guyton11 believe that most cysts may be classified etiologically as ectasia of pre-existing vessels or lymphatics, or cystic degeneration of hematomas, or adenomas and cites cases reported by Robson and Zimmerman 12 and lglitsyn13 in support of this view. Parasitic cysts of the adrenal gland occur but rarely as shown by Barnett, 14 who in a review of 1,617 cases with echinococcus cysts found the adrenal involved in 0.5 per cent of the cases. Hemorrhagic disease of the adrenal gland is not an uncommon finding at necropsy. It is seen in the newborn associated with certain categories of anoxia. It is seen as a toxic manifestation in meningococcus infections such as the Waterhouse-Fredricksen syndrome. More rarely it is seen in cases of profound Read at annual meeting, Western Section, American Urological Association, Sun Valley, Idaho, June 27, 1951. 1 Greiselius, cited by Doran, A.H. G.: Brit. Med. J., 1: 1558, 1908. 2 Terrier and Lecene, cited by Zuckner, J.: A. M.A. Arch. Path., 50: 468-474, 1950. 3 Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. 4 Oberndorfer, cited by Zuckner, J.: A. JVI. A. Arch. Path., 50: 468-474, 1950. 5 de Vecchi, cited by Zuckner, J., Major, R.H. and Black, D.R.: A. M.A. Arch. Path., 50: 468-474, 1950. 6 Zuckner, J.: A. M. A. Arch. Path., 50: 468-474, 1950. 7 Henchen, cited by Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. 8 Sick, cited by Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. 9 Stoerk, cited by Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. Also by Zuckner, J.: A. M.A. Arch. Path., 50: 468-474, 1950. 10 Nowicki, cited by Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. Also by Zuckner, J.: A. M. A. Arch. Path., 50: 468-474, 1950. 11 Reimann, D. L. and Guyton, W. L.: Am. J. Path., 23: 479, 1947. 12 Robson, S. H. and Zimmerman, E. F.: Arch. Path., 26: 869, 1938. 13 Iglitsyn, cited by Reimann, D. L. and Guyton, W. L.: Am. J. Path., 23: 479, 1947. 14 Barnett, cited by Levison, P.: Endocrinology, 17: 372, 1933. 1
2
DEAN PARKER
shock such as following surgical shock or in cases of massive crush injuries. On a basis of frequency and the pathological findings of this case it seems more logical to place the etiology of this case under that group of hemorrhage into the adrenal with organization and subsequent cyst formation. I am opposed to classifying it as lympho-angiectasis, even though its gross appearance is very similar to Zuckner's case which he felt fell in that group. Most lymphangiomas or hemangiomas in the body are not globular, well circumscribed lesions, but are flat with many finger-like projections; and secondly, they should be lined with endothelium, although it is possible that under pressure, atrophy could have taken place. The pathological picture, that of organized hematoma, the calcification and macrophages and laminated hyaline tissue at the periphery leaves little pathological doubt that the tumor arose on the basis of an adrenal hemorrhage. DIAGNOSIS
There is no characteristic symptom. Many of the cases reported had no symptoms and were found on routine necropsy. Other symptoms have been "sense of pressure," "epigastric distress," "abdominal pain," "shock," or a palpable mass. Diagnosis is based on roentgenologic urography and a suspicion of the disease entity. TREATMENT
The treatment need not be surgical since it is a benign lesion. However, differential diagnosis is practically impossible so surgical exploration is definitely indicated. Diagnostic procedures to rule out malignancies of the adrenal gland may be helpful in the differential diagnosis. CASE REPORT
A 55 year old man had always been in good health. His past medical history was noncontributory except for an accident 1 year ago when he was struck in the face and chest with a sandblast in the railroad yards. He was taken to the hospital in questionable shock and still had some sand in the left eye and left face. At a recent routine railroad examination he was found to have albuminuria and a systolic blood pressure of 150, and hence was referred for urological consultation. On further questioning at the time of my examination, he stated that for 3 months he had noticed a full feeling in the upper right abdomen, and for 1 month pain radiated down the posterior aspect of the right leg. The only positive finding on physical examination was a questionable mass or fullness in the right upper quadrant and a palpable lower pole of the right kidney. The blood pressure was 168/110. Laboratory examination showed: Kahn, negative; blood urea nitrogen 16.3 mg. per cent; red blood cells 5,300,000; white blood cells 11,400; hemoglobin 16.0 gm. (102.8 per cent of normal). Urinary findings were: pH 5.5; specific gravity 1.019; albumin 1-2 plus; microscopic examination showed an occasional leukocyte and granular cast.
BENIGN CYSTS OF ADRENAL GLAND
3
An excretory urogram showed a round, well circumscribed mass below the diaphragm, above the right kidney which displaced the upper pole of the right kidney downward and laterally, so that the kidney lay in a horizontal rather than a vertical plane. The calyces, renal pelvis and ureter on the right side were otherwise normal. The left pyelogram was entirely normal. See figure l. On hospital admission the systolic blood pressure was 150, and the diastolic 90. A roentgenogram of the chest was normal. A renal study with retrograde pyelograms added no information to that obtained from the excretory urographic series. The 24 hour urinary excretion of the 17-ketosteroids was 17 mg. per cent. The blood pressure response to histamine and dibenamine was normal and
FIG. 1. Excretory urogram showing subdiaphragmatic mass on right, displacing upper pole of kidney.
showed no evidence of an epinephrine-producing tumor of the adrenal gland. Impression: Benign cyst of the right adrenal gland. Operation: With the patient in the right kidney position, a transthoracic approach was made through the right chest by removal of the tenth rib. The fibers of the diaphragm were incised radially from the tendinous portion. A large cyst of the right adrenal gland was easily dissected free from the upper pole of the right kidney down to its vascular pedicle which was really that of the right adrenal gland. The cyst was not attached to the upper pole of the kidney nor to its capsule, but the upper pole of the kidney was concavely indented by the downward pressure of the mass. The wound was closed in layers without drains or water seal by interrupted cotton technique. The skin edges were approximated with interrupted cotton sutures. The gross specimen consisted of a mass which was almost round. It measured
4
DEAN PARKER
15 by 13 by 12 cm. "The surface has a mottled orange-yellow color due to what appears to be flattened, irregular portions of yellow tissue either attached to the cyst wall or within the cyst wall. On one surface a small amount of definite adrenal gland can be identified and these areas of orange-yellow color suggest cortical adrenal tissue. The surface is fairly smooth with the exception of some tiny fibrous strands along with a limited amount of fatty tissue. When this specimen is opened the contents are a light colored fluid with a slightly greasy quality and contains flaky, minute glistening particles. The wall of the cyst is
Fm. 2. Gross specimen of cyst and remaining portion of right adrenal gland
Fm. 3. A, zone to left shows hyalinized capsule of cyst. B, middle zone, adrenal cortical cells. C, zone to right, capsule of adrenal gland.
a fairly thin fibrous wall in which there are islands of orange colored tissue which resembles adrenal cortex and on the inner surface of the cyst are numerous irregular small masses of similar orange colored tissue which show some degeneration. The surface of the cyst between the islands of orange colored tissue is glistening." See figure 2. Histologic examination of sections taken from the cyst show "a capsule which is essentially the same as the capsule of a normal adrenal gland with the exception of some thickening in areas; within this layer of fibrous tissue there is normal adrenal tissue with cells being mostly of the zone of fasciculata. There has been another layer of hyalinized fibrous-like tissue between normal adrenal cells and acidophilic granular debris in which them arn needle-like spaces probably
BENIGN CYSTS OF ADRENAL GLAND
5
representing spaces from which cholesterol crystals have been dissolved. Within this area there are some areas of calcific deposit. Also seen within this material here and there are a few large cells which show a vacuolated cytoplasm and which may represent degenerated adrenal cells or possibly phagocytic cells with vacuolization." Microscopic diagnosis: Adrenal cyst. See figure 3. The patient had some difficulty in coughing and bronchial aspiration was done on the first postoperative day. A chest aspiration was done on the second postoperative day. The highest elevation of temperature was lOOF and he was discharged on the eighth postoperative day. He was last seen 6 months later entirely asymptomatic. SUMMARY
A case of benign cyst of the adrenal gland has been reported and the literature has been reviewed with an attempt to classify such cysts according to etiological factors. A preoperative diagnosis was made primarily on the basis of the radiographic peculiarity of the lesion in question. This case emphasizes the advantages in the transthoracic approach and its use in upper pole renal or adrenal disease.
1330 Madison St., Seattle 4, Wash.
Vol. 68, No. 1, July 1952 Printed in U.S.A.
BENIGN CYSTS OF THE ADRENAL GLAND: A CASE REPORT DEAN PARKER
Benign cysts of the adrenal gland are rare. Prior to 1920, fourteen cases were reported and only five additional cases have been reported in the American literature since then. The first case was cited by Greiselius in 1670. 1 Terrier and Lecene published their case and reviewed nine others reported up to 1906. 2 They also classified adrenal cysts: 1) parasitic, 2) true glandular retention cysts, 3) cystic adenomas, 4) serous or lymphangiomatous cysts and 5) pseudocysts (hemorrhagic cysts). Only 2 cases reported, that of Levinson and this case, were diagnosed prior to surgery. The etiology of adrenal cysts is somewhat confusing and only a little clarity is gained from the review of literature. Major and Black3 reported a case of bilateral adrenal cysts which appeared to arise from dilated capillaries or sinusoids between columns of adrenal cells. The cases of Oberndorfer 4 and one by de Vecchi 5 appeared to arise from lymphangiectasia, as did the most recent case in the literature, that of Zuckner. 6 Henchen7 recorded a hemorrhagic cyst of the adrenal while Sick 8 reported a ciliated cyst which must have developed on the basis of displacement of tissue from the urogenital anlage. Stoerk, 9 and later Nowicki, 10 both expressed doubts as to the existence of true epithelial cysts. This seems logical since there is no tendency to acini formation in the cells of the adrenal gland. Reimann and Guyton11 believe that most cysts may be classified etiologically as ectasia of pre-existing vessels or lymphatics, or cystic degeneration of hematomas, or adenomas and cites cases reported by Robson and Zimmerman 12 and lglitsyn13 in support of this view. Parasitic cysts of the adrenal gland occur but rarely as shown by Barnett, 14 who in a review of 1,617 cases with echinococcus cysts found the adrenal involved in 0.5 per cent of the cases. Hemorrhagic disease of the adrenal gland is not an uncommon finding at necropsy. It is seen in the newborn associated with certain categories of anoxia. It is seen as a toxic manifestation in meningococcus infections such as the Waterhouse-Fredricksen syndrome. More rarely it is seen in cases of profound Read at annual meeting, Western Section, American Urological Association, Sun Valley, Idaho, June 27, 1951. 1 Greiselius, cited by Doran, A.H. G.: Brit. Med. J., 1: 1558, 1908. 2 Terrier and Lecene, cited by Zuckner, J.: A. M.A. Arch. Path., 50: 468-474, 1950. 3 Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. 4 Oberndorfer, cited by Zuckner, J.: A. JVI. A. Arch. Path., 50: 468-474, 1950. 5 de Vecchi, cited by Zuckner, J., Major, R.H. and Black, D.R.: A. M.A. Arch. Path., 50: 468-474, 1950. 6 Zuckner, J.: A. M. A. Arch. Path., 50: 468-474, 1950. 7 Henchen, cited by Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. 8 Sick, cited by Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. 9 Stoerk, cited by Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. Also by Zuckner, J.: A. M.A. Arch. Path., 50: 468-474, 1950. 10 Nowicki, cited by Major, R.H. and Black, D.R.: Am. J. Med. Sc., 156: 469, 1918. Also by Zuckner, J.: A. M. A. Arch. Path., 50: 468-474, 1950. 11 Reimann, D. L. and Guyton, W. L.: Am. J. Path., 23: 479, 1947. 12 Robson, S. H. and Zimmerman, E. F.: Arch. Path., 26: 869, 1938. 13 Iglitsyn, cited by Reimann, D. L. and Guyton, W. L.: Am. J. Path., 23: 479, 1947. 14 Barnett, cited by Levison, P.: Endocrinology, 17: 372, 1933. 1
2
DEAN PARKER
shock such as following surgical shock or in cases of massive crush injuries. On a basis of frequency and the pathological findings of this case it seems more logical to place the etiology of this case under that group of hemorrhage into the adrenal with organization and subsequent cyst formation. I am opposed to classifying it as lympho-angiectasis, even though its gross appearance is very similar to Zuckner's case which he felt fell in that group. Most lymphangiomas or hemangiomas in the body are not globular, well circumscribed lesions, but are flat with many finger-like projections; and secondly, they should be lined with endothelium, although it is possible that under pressure, atrophy could have taken place. The pathological picture, that of organized hematoma, the calcification and macrophages and laminated hyaline tissue at the periphery leaves little pathological doubt that the tumor arose on the basis of an adrenal hemorrhage. DIAGNOSIS
There is no characteristic symptom. Many of the cases reported had no symptoms and were found on routine necropsy. Other symptoms have been "sense of pressure," "epigastric distress," "abdominal pain," "shock," or a palpable mass. Diagnosis is based on roentgenologic urography and a suspicion of the disease entity. TREATMENT
The treatment need not be surgical since it is a benign lesion. However, differential diagnosis is practically impossible so surgical exploration is definitely indicated. Diagnostic procedures to rule out malignancies of the adrenal gland may be helpful in the differential diagnosis. CASE REPORT
A 55 year old man had always been in good health. His past medical history was noncontributory except for an accident 1 year ago when he was struck in the face and chest with a sandblast in the railroad yards. He was taken to the hospital in questionable shock and still had some sand in the left eye and left face. At a recent routine railroad examination he was found to have albuminuria and a systolic blood pressure of 150, and hence was referred for urological consultation. On further questioning at the time of my examination, he stated that for 3 months he had noticed a full feeling in the upper right abdomen, and for 1 month pain radiated down the posterior aspect of the right leg. The only positive finding on physical examination was a questionable mass or fullness in the right upper quadrant and a palpable lower pole of the right kidney. The blood pressure was 168/110. Laboratory examination showed: Kahn, negative; blood urea nitrogen 16.3 mg. per cent; red blood cells 5,300,000; white blood cells 11,400; hemoglobin 16.0 gm. (102.8 per cent of normal). Urinary findings were: pH 5.5; specific gravity 1.019; albumin 1-2 plus; microscopic examination showed an occasional leukocyte and granular cast.
BENIGN CYSTS OF ADRENAL GLAND
3
An excretory urogram showed a round, well circumscribed mass below the diaphragm, above the right kidney which displaced the upper pole of the right kidney downward and laterally, so that the kidney lay in a horizontal rather than a vertical plane. The calyces, renal pelvis and ureter on the right side were otherwise normal. The left pyelogram was entirely normal. See figure l. On hospital admission the systolic blood pressure was 150, and the diastolic 90. A roentgenogram of the chest was normal. A renal study with retrograde pyelograms added no information to that obtained from the excretory urographic series. The 24 hour urinary excretion of the 17-ketosteroids was 17 mg. per cent. The blood pressure response to histamine and dibenamine was normal and
FIG. 1. Excretory urogram showing subdiaphragmatic mass on right, displacing upper pole of kidney.
showed no evidence of an epinephrine-producing tumor of the adrenal gland. Impression: Benign cyst of the right adrenal gland. Operation: With the patient in the right kidney position, a transthoracic approach was made through the right chest by removal of the tenth rib. The fibers of the diaphragm were incised radially from the tendinous portion. A large cyst of the right adrenal gland was easily dissected free from the upper pole of the right kidney down to its vascular pedicle which was really that of the right adrenal gland. The cyst was not attached to the upper pole of the kidney nor to its capsule, but the upper pole of the kidney was concavely indented by the downward pressure of the mass. The wound was closed in layers without drains or water seal by interrupted cotton technique. The skin edges were approximated with interrupted cotton sutures. The gross specimen consisted of a mass which was almost round. It measured
4
DEAN PARKER
15 by 13 by 12 cm. "The surface has a mottled orange-yellow color due to what appears to be flattened, irregular portions of yellow tissue either attached to the cyst wall or within the cyst wall. On one surface a small amount of definite adrenal gland can be identified and these areas of orange-yellow color suggest cortical adrenal tissue. The surface is fairly smooth with the exception of some tiny fibrous strands along with a limited amount of fatty tissue. When this specimen is opened the contents are a light colored fluid with a slightly greasy quality and contains flaky, minute glistening particles. The wall of the cyst is
Fm. 2. Gross specimen of cyst and remaining portion of right adrenal gland
Fm. 3. A, zone to left shows hyalinized capsule of cyst. B, middle zone, adrenal cortical cells. C, zone to right, capsule of adrenal gland.
a fairly thin fibrous wall in which there are islands of orange colored tissue which resembles adrenal cortex and on the inner surface of the cyst are numerous irregular small masses of similar orange colored tissue which show some degeneration. The surface of the cyst between the islands of orange colored tissue is glistening." See figure 2. Histologic examination of sections taken from the cyst show "a capsule which is essentially the same as the capsule of a normal adrenal gland with the exception of some thickening in areas; within this layer of fibrous tissue there is normal adrenal tissue with cells being mostly of the zone of fasciculata. There has been another layer of hyalinized fibrous-like tissue between normal adrenal cells and acidophilic granular debris in which them arn needle-like spaces probably
BENIGN CYSTS OF ADRENAL GLAND
5
representing spaces from which cholesterol crystals have been dissolved. Within this area there are some areas of calcific deposit. Also seen within this material here and there are a few large cells which show a vacuolated cytoplasm and which may represent degenerated adrenal cells or possibly phagocytic cells with vacuolization." Microscopic diagnosis: Adrenal cyst. See figure 3. The patient had some difficulty in coughing and bronchial aspiration was done on the first postoperative day. A chest aspiration was done on the second postoperative day. The highest elevation of temperature was lOOF and he was discharged on the eighth postoperative day. He was last seen 6 months later entirely asymptomatic. SUMMARY
A case of benign cyst of the adrenal gland has been reported and the literature has been reviewed with an attempt to classify such cysts according to etiological factors. A preoperative diagnosis was made primarily on the basis of the radiographic peculiarity of the lesion in question. This case emphasizes the advantages in the transthoracic approach and its use in upper pole renal or adrenal disease.
1330 Madison St., Seattle 4, Wash.