Accepted Manuscript Massive: hemorrhagic adrenal pseudocyst Yolanda Schrank, MD Miguel Madeira, PhD PII:
S0002-9343(14)00360-X
DOI:
10.1016/j.amjmed.2014.04.021
Reference:
AJM 12500
To appear in:
The American Journal of Medicine
Received Date: 19 December 2013 Revised Date:
21 April 2014
Accepted Date: 21 April 2014
Please cite this article as: Schrank Y, Madeira M, Massive: hemorrhagic adrenal pseudocyst, The American Journal of Medicine (2014), doi: 10.1016/j.amjmed.2014.04.021. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 1 of 5 Massive: hemorrhagic adrenal pseudocyst Yolanda Schrank, MD* Miguel Madeira, PhD**
RI PT
*Division of Endocrinology and Metabolism, Department of Internal Medicine, Bonsucesso Federal Hospital, Rio de Janeiro, RJ, Brazil.
** Division of Endocrinology and Metabolism, Department of Internal Medicine, Bonsucesso Federal Hospital, Rio de Janeiro, RJ, Brazil.
Division of Endocrinology and Metabolism, Federal University of Rio de Janeiro,
SC
Rio de Janeiro, RJ, Brazil.
M AN U
Conflict of Interest: All authors have no conflict of interests.
Authorship: All authors had access to the data and a role in writing the article.
Correspondence: Miguel Madeira
Rua Professor Rodolpho Paulo Rocco 255, 9º andar, Serviço de Endocrinologia, Edifício do HUCFF. Cidade Universitária, Ilha do Fundão, Rio de Janeiro, RJ. CEP:
Phone: 55-21-22662606
TE D
21941-913
Email: [email protected]
EP
Article type: Images in Radiology
AC C
Key words: hemorraghic; adrenal; mass; pseudocyst; calcified
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 2 of 5 Images in Radiology
Massive: hemorrhagic adrenal pseudocyst
RI PT
Robert G. Stern, MD, Images in Radiology Editor Yolanda Schrank, MD,a Miguel Madeira, PhDb a
Division of Endocrinology and Metabolism, Department of Internal Medicine,
M AN U
University of Rio de Janeiro, Rio de Janeiro, RJ, Brazil.
SC
Bonsucesso Federal Hospital and bDivision of Endocrinology and Metabolism, Federal
Requests for reprints should be addressed to Miguel Madeira, PhD, Rua Professor Rodolpho Paulo Rocco 255, 9º andar, Serviço de Endocrinologia, Edifício do HUCFF. Cidade Universitária, Ilha do Fundão, Rio de Janeiro, RJ. CEP: 21941-913.
PRESENTATION
TE D
E-mail address: [email protected]
According to the usual accounts of such lesions, the mass lodged in the upper left quadrant of the patient’s abdomen should not have been palpable. A 62-year-old woman
EP
presented to the hospital with dull abdominal pain. She had hypertension but no history of trauma or malignancy. She did, however, provide a prior abdominal x-ray, which
AC C
showed a large ovoid mass with rim calcification in the upper left quadrant (Figure 1). On examination, the mass was dull to percussion and tender to palpation.
ASSESSMENT
An abdominal computed tomography (CT) scan showed a large mass, measuring 9.1 x 7.4 x 6.9 cm, with dense rim calcification in the region of the left adrenal gland (Figure 2). Its contents were not enhanced with contrast medium. The growth was in close contact with the inferior pole of the spleen and located posterior to the pancreatic tail. It caused downwards displacement of the left kidney. An adrenal lesion, such as an
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 3 of 5 adenoma, was considered, but a thorough adrenal evaluation ruled out excess hormone production—and the tumors that can cause it. Left adrenalectomy was performed via laparoscopic surgery. More than 90% of the entire surgical specimen, which weighted 190 g and measured 13 x 10 x 4 cm, was
DIAGNOSIS
RI PT
cyst (Figure 3). The patient’s postoperative course was uneventful.
On pathology examination, the cyst contained hemorrhagic fluid and was surrounded by a fibrous calcified pseudocapsule with no clear lining epithelium or endothelium. The
SC
findings confirmed a diagnosis of an adrenal pseudocyst (Figure 4).
Patients with adrenal cysts are most often diagnosed between the ages of 20 and 49 years, and women may be more vulnerable than men; a 3:1 ratio has been
M AN U
documented.1 Cysts represent perhaps 6% of all newly discovered incidentalomas. Although the first description of an adrenal cyst dates from 1670, relatively few have since been reported in the medical literature; just over 300 cases of adrenal cysts and about 125 cases of pseudocyst of the adrenal gland are on record.2 Clinically, adrenal cysts are rarely palpable, but an abdominal examination usually identifies tenderness. Four histological categories exist: endothelial cysts (45%)
TE D
are most common, followed by pseudocysts (39%), parasitic cysts (7%), and epithelial cysts (2%).3 CT, the most useful modality for detecting any type of adrenal cyst, has a diagnostic accuracy of 62.5–96%. When CT does not produce specific findings, magnetic resonance imaging (MRI) may be of value. MRI is particularly sensitive for
EP
detecting intracystic hemorrhage, which shows up as hyperintensity on both T1- and T2-weighted images.4
AC C
Pseudocysts are ordinarily benign, and they are uncommon, accounting for nearly 40% of cystic adrenal masses.2 As with our patient’s cyst, they do not have an endothelial or epithelial lining and are generally filled with fresh or coagulated blood as a result of hemorrhage into a normal or a pathologic gland.5 They tend to be found incidentally on imaging or at autopsy. When patients do have symptoms, the most common are dull regional pain and gastrointestinal complaints.6 Adrenal hypofunction or hyperfunction is seldom triggered by a pseudocyst. Patients with a large adrenal cyst can present with symptoms spurred by its bulk, mainly pain caused by its encroachment on other structures, and complications such as hemorrhage, rupture, or infection can ensue.3
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 4 of 5 While the precise cause of adrenal pseudocysts has yet to be defined, hemorrhage and infection are regarded as potential contributing factors.5 The lesions can, on occasion, be encountered in patients who also have a primary adrenal neoplasm, such as a pheochromocytoma, a malignancy, or an adenoma. These primary tumor types
RI PT
should be considered in the differential diagnosis for a cystic adrenal mass, as should lymphangioma, epithelial cysts, and parasitic cysts.5
MANAGEMENT
Treatment is generally based on the size of the pseudocyst, the patient’s symptoms and
SC
functional status, and whether there is the possibility of malignancy.4 Patients with small asymptomatic lesions might fare well with simple observation, but those with cysts exceeding 5-6 cm are candidates for a more active approach, such as percutaneous
M AN U
drainage, the treatment of choice for adrenal cysts.2 The most definitive treatment is surgical excision, which is indicated for patients with tumors that provoke adrenal hyperfunction or those who may have a malignancy. Surgery can be open or laparoscopic, though the latter approach tends to reduce hospital stay, lower analgesia requirements, and allow an earlier return to work.4 Whenever possible, the adrenal gland should be preserved.
TE D
In conclusion, the hemorrhagic adrenal pseudocyst is an infrequent clinical finding, and it is even more remarkable to find one that is giant-sized. Patients with small asymptomatic pseudocysts can be observed, but those with symptomatic
References
EP
pseudocysts or pseudocysts larger than 5-6 cm should be treated.
AC C
1. Demir A, Tanidir Y, Kaya H, Turkeri LN. A giant adrenal pseudocyst: case report and review of the literature. Int Urol Nephrol. 2006;38:167-169.
2. Khilnani GC, Kumar A, Bammigatti C, Sharma R, Gupta SD. Hemorrhagic pseudocyst of the adrenal gland causing acute abdominal pain. J Assoc Physicians India. 2008;56:379-380.
3. Ujam AB, Peters CJ, Tadrous PJ, Webster JJ, Steer K, Martinez-Isla A. Adrenal pseudocyst: diagnosis and laparoscopic management—a case report. Int J Surg Case Rep. 2011;2:306-308. 4. Salemis NS, Nisotakis K. Giant adrenal pseudocyst: laparoscopic management. ANZ J Surg. 2011;81:185-186.
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 5 of 5 5. Habra MA, Feig BW, Waguespack SG. Image in endocrinology: adrenal pseudocyst. J Clin Endocrinol Metab. 2005;90:3067-3068. 6. Momiyama M, Matsuo K, Yoshida K, et al. A giant adrenal pseudocyst presenting with right hypochondralgia and fever: a case report. J Med Case Rep.
RI PT
2011;5:135.
FIGURE LEGENDS
SC
Figure 1. Abdominal radiography demonstrated a large ovoid mass with rim calcification in the upper left quadrant.
M AN U
Figure 2. A large mass with dense rim calcification was evident in the region of the left adrenal gland on abdominal computed tomography.
Figure 3. The surgical specimen included a large adrenal cyst.
Figure 4. Pathology identified hemorrhagic fluid surrounded by a fibrous calcified
TE D
pseudocapsule with no clear lining epithelium or endothelium. These findings
AC C
EP
confirmed the diagnosis of an adrenal pseudocyst.
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
S0002-9343(14)00360-X
DOI:
10.1016/j.amjmed.2014.04.021
Reference:
AJM 12500
To appear in:
The American Journal of Medicine
Received Date: 19 December 2013 Revised Date:
21 April 2014
Accepted Date: 21 April 2014
Please cite this article as: Schrank Y, Madeira M, Massive: hemorrhagic adrenal pseudocyst, The American Journal of Medicine (2014), doi: 10.1016/j.amjmed.2014.04.021. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 1 of 5 Massive: hemorrhagic adrenal pseudocyst Yolanda Schrank, MD* Miguel Madeira, PhD**
RI PT
*Division of Endocrinology and Metabolism, Department of Internal Medicine, Bonsucesso Federal Hospital, Rio de Janeiro, RJ, Brazil.
** Division of Endocrinology and Metabolism, Department of Internal Medicine, Bonsucesso Federal Hospital, Rio de Janeiro, RJ, Brazil.
Division of Endocrinology and Metabolism, Federal University of Rio de Janeiro,
SC
Rio de Janeiro, RJ, Brazil.
M AN U
Conflict of Interest: All authors have no conflict of interests.
Authorship: All authors had access to the data and a role in writing the article.
Correspondence: Miguel Madeira
Rua Professor Rodolpho Paulo Rocco 255, 9º andar, Serviço de Endocrinologia, Edifício do HUCFF. Cidade Universitária, Ilha do Fundão, Rio de Janeiro, RJ. CEP:
Phone: 55-21-22662606
TE D
21941-913
Email: [email protected]
EP
Article type: Images in Radiology
AC C
Key words: hemorraghic; adrenal; mass; pseudocyst; calcified
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 2 of 5 Images in Radiology
Massive: hemorrhagic adrenal pseudocyst
RI PT
Robert G. Stern, MD, Images in Radiology Editor Yolanda Schrank, MD,a Miguel Madeira, PhDb a
Division of Endocrinology and Metabolism, Department of Internal Medicine,
M AN U
University of Rio de Janeiro, Rio de Janeiro, RJ, Brazil.
SC
Bonsucesso Federal Hospital and bDivision of Endocrinology and Metabolism, Federal
Requests for reprints should be addressed to Miguel Madeira, PhD, Rua Professor Rodolpho Paulo Rocco 255, 9º andar, Serviço de Endocrinologia, Edifício do HUCFF. Cidade Universitária, Ilha do Fundão, Rio de Janeiro, RJ. CEP: 21941-913.
PRESENTATION
TE D
E-mail address: [email protected]
According to the usual accounts of such lesions, the mass lodged in the upper left quadrant of the patient’s abdomen should not have been palpable. A 62-year-old woman
EP
presented to the hospital with dull abdominal pain. She had hypertension but no history of trauma or malignancy. She did, however, provide a prior abdominal x-ray, which
AC C
showed a large ovoid mass with rim calcification in the upper left quadrant (Figure 1). On examination, the mass was dull to percussion and tender to palpation.
ASSESSMENT
An abdominal computed tomography (CT) scan showed a large mass, measuring 9.1 x 7.4 x 6.9 cm, with dense rim calcification in the region of the left adrenal gland (Figure 2). Its contents were not enhanced with contrast medium. The growth was in close contact with the inferior pole of the spleen and located posterior to the pancreatic tail. It caused downwards displacement of the left kidney. An adrenal lesion, such as an
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 3 of 5 adenoma, was considered, but a thorough adrenal evaluation ruled out excess hormone production—and the tumors that can cause it. Left adrenalectomy was performed via laparoscopic surgery. More than 90% of the entire surgical specimen, which weighted 190 g and measured 13 x 10 x 4 cm, was
DIAGNOSIS
RI PT
cyst (Figure 3). The patient’s postoperative course was uneventful.
On pathology examination, the cyst contained hemorrhagic fluid and was surrounded by a fibrous calcified pseudocapsule with no clear lining epithelium or endothelium. The
SC
findings confirmed a diagnosis of an adrenal pseudocyst (Figure 4).
Patients with adrenal cysts are most often diagnosed between the ages of 20 and 49 years, and women may be more vulnerable than men; a 3:1 ratio has been
M AN U
documented.1 Cysts represent perhaps 6% of all newly discovered incidentalomas. Although the first description of an adrenal cyst dates from 1670, relatively few have since been reported in the medical literature; just over 300 cases of adrenal cysts and about 125 cases of pseudocyst of the adrenal gland are on record.2 Clinically, adrenal cysts are rarely palpable, but an abdominal examination usually identifies tenderness. Four histological categories exist: endothelial cysts (45%)
TE D
are most common, followed by pseudocysts (39%), parasitic cysts (7%), and epithelial cysts (2%).3 CT, the most useful modality for detecting any type of adrenal cyst, has a diagnostic accuracy of 62.5–96%. When CT does not produce specific findings, magnetic resonance imaging (MRI) may be of value. MRI is particularly sensitive for
EP
detecting intracystic hemorrhage, which shows up as hyperintensity on both T1- and T2-weighted images.4
AC C
Pseudocysts are ordinarily benign, and they are uncommon, accounting for nearly 40% of cystic adrenal masses.2 As with our patient’s cyst, they do not have an endothelial or epithelial lining and are generally filled with fresh or coagulated blood as a result of hemorrhage into a normal or a pathologic gland.5 They tend to be found incidentally on imaging or at autopsy. When patients do have symptoms, the most common are dull regional pain and gastrointestinal complaints.6 Adrenal hypofunction or hyperfunction is seldom triggered by a pseudocyst. Patients with a large adrenal cyst can present with symptoms spurred by its bulk, mainly pain caused by its encroachment on other structures, and complications such as hemorrhage, rupture, or infection can ensue.3
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 4 of 5 While the precise cause of adrenal pseudocysts has yet to be defined, hemorrhage and infection are regarded as potential contributing factors.5 The lesions can, on occasion, be encountered in patients who also have a primary adrenal neoplasm, such as a pheochromocytoma, a malignancy, or an adenoma. These primary tumor types
RI PT
should be considered in the differential diagnosis for a cystic adrenal mass, as should lymphangioma, epithelial cysts, and parasitic cysts.5
MANAGEMENT
Treatment is generally based on the size of the pseudocyst, the patient’s symptoms and
SC
functional status, and whether there is the possibility of malignancy.4 Patients with small asymptomatic lesions might fare well with simple observation, but those with cysts exceeding 5-6 cm are candidates for a more active approach, such as percutaneous
M AN U
drainage, the treatment of choice for adrenal cysts.2 The most definitive treatment is surgical excision, which is indicated for patients with tumors that provoke adrenal hyperfunction or those who may have a malignancy. Surgery can be open or laparoscopic, though the latter approach tends to reduce hospital stay, lower analgesia requirements, and allow an earlier return to work.4 Whenever possible, the adrenal gland should be preserved.
TE D
In conclusion, the hemorrhagic adrenal pseudocyst is an infrequent clinical finding, and it is even more remarkable to find one that is giant-sized. Patients with small asymptomatic pseudocysts can be observed, but those with symptomatic
References
EP
pseudocysts or pseudocysts larger than 5-6 cm should be treated.
AC C
1. Demir A, Tanidir Y, Kaya H, Turkeri LN. A giant adrenal pseudocyst: case report and review of the literature. Int Urol Nephrol. 2006;38:167-169.
2. Khilnani GC, Kumar A, Bammigatti C, Sharma R, Gupta SD. Hemorrhagic pseudocyst of the adrenal gland causing acute abdominal pain. J Assoc Physicians India. 2008;56:379-380.
3. Ujam AB, Peters CJ, Tadrous PJ, Webster JJ, Steer K, Martinez-Isla A. Adrenal pseudocyst: diagnosis and laparoscopic management—a case report. Int J Surg Case Rep. 2011;2:306-308. 4. Salemis NS, Nisotakis K. Giant adrenal pseudocyst: laparoscopic management. ANZ J Surg. 2011;81:185-186.
ACCEPTED MANUSCRIPT 13-1770SchrankCHS, Page 5 of 5 5. Habra MA, Feig BW, Waguespack SG. Image in endocrinology: adrenal pseudocyst. J Clin Endocrinol Metab. 2005;90:3067-3068. 6. Momiyama M, Matsuo K, Yoshida K, et al. A giant adrenal pseudocyst presenting with right hypochondralgia and fever: a case report. J Med Case Rep.
RI PT
2011;5:135.
FIGURE LEGENDS
SC
Figure 1. Abdominal radiography demonstrated a large ovoid mass with rim calcification in the upper left quadrant.
M AN U
Figure 2. A large mass with dense rim calcification was evident in the region of the left adrenal gland on abdominal computed tomography.
Figure 3. The surgical specimen included a large adrenal cyst.
Figure 4. Pathology identified hemorrhagic fluid surrounded by a fibrous calcified
TE D
pseudocapsule with no clear lining epithelium or endothelium. These findings
AC C
EP
confirmed the diagnosis of an adrenal pseudocyst.
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
AC C
EP
TE D
M AN U
SC
RI PT
ACCEPTED MANUSCRIPT
