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Article is available online at http://www.webio.hu/por/2005/11/4/0242 CASE REPORT
Cystic Lymphangioma of the Right Adrenal Gland
Lora Esberk ATES,1 Yersu KAPRAN,1 Yesim ERBIL,2 Umut BARBAROS,2 Ferhunde DIZDAROGLU1 1Department of Pathology, 2Department of General Surgery, Istanbul Medical Faculty, Istanbul University Lymphangiomas are benign malformations of the
woman admitted to the hospital, complaining of
vessels. They are commonly located in the neck, axil-
weakness, putting on weight, and lumbago. Her lab-
lary region and mediastinum. Lymphangioma of the
oratory findings were within normal limits. Radio-
adrenal gland is very rare. These lesions were first
logical examination revealed a 7 cm cystic lesion
discovered as incidental autopsy findings. As the
located in the right adrenal gland. Right adrenalecto-
imaging techniques have improved, they now appear
my was performed. Histopathological examination
as incidental findings at abdominal ultrasonography
and immunohistochemical analysis of the cystic
and computed tomography scan examinations. They
lesion was consistent with a lymphangioma. (Patho-
are usually asymptomatic. We present a 26-year-old
logy Oncology Research Vol 11, No 4, 242–244) Key words: Adrenal gland, adrenal cyst, lymphangioma Case report
In December 2003, right adrenalectomy was performed.
The patient was discharged from the hospital a week later A 26-year-old woman reported to the hospital, com- with normal physical and laboratory findings. On patho- plaining of weakness, putting on weight, and lumbago.
logic examination the adrenalectomy specimen weighed Physical examination was normal. In laboratory findings 40 gram and measured 9x4x3.5 cm. On the cut surface, cortisol level was 1.3 µg/dl (normal range 5-25 µg/dl) normal adrenal gland measured 4x0.7x0.3 cm, and a and after 8 mg dexamethasone suppression test cortisol multi-septae cystic lesion of 8 cm in diameter was detect- level was 2 µg/dl (normal range 3 µg/dl<), testosterone ed adjacent to the gland (Figure 2). Microscopic examina- was 2.4 pg/ml (normal range 1-10 pg/ml), dehy- tion revealed a multi-cystic lesion laid with flat endothe- droepiandrosteron sulfate was 80 mg/dl (normal range35-450 mg/dl), 24-h urinary excretion of vanillyl man-delic acid was 1.6 mg/24 h (normal range 1.4-8.8 mg/24h), 24-h urinary excretion of metanephrine was 116µg/24 h (normal range 20-345 mg/24 h),normetanephrine was 174 µg/24 h (normal range 30-440mg/24 h), hematocrit was 36.5%, hemoglobin was 11.1g/dl, platelets were 229,000/µl. Abdominal ultrasonog-raphy (US) revealed a 7 cm, lobulated cystic mass withseptae and sharp contours, located in the right adrenalgland. Computed tomography (CT) scan and magneticresonance (MR) findings verified the 7 cm cystic lobu-lated mass associated to vena cava inferior (Figure 1). Received: Dec 8, 2004; accepted: July 21, 2005Correspondence: Lora Esberk ATES, Istanbul Universitesi, Istan-bul Tip Fakultesi, Temel Bilimler Binasi, Patoloji Anabilim Dali, Figure 1. Contrast-enhanced axial computed tomography image
Capa, Istanbul, 34390, Turkiye, Tel.: +90 212 414 24 00, fax: +90 shows hypodense, lobulated solid mass of 6.5x5 cm in diameter 212 631 13 67, e-mail: loraates@hotmail.com Cystic Lymphangioma of the Right Adrenal Gland lial cells adjacent to the normal-appearing adrenal cortex.
The cystic spaces were filled with proteinous fluid (Figure3). Immunohistochemically these cells stained positivelyfor CD31 (Neomarker, clone JC/70A, 1/50 dilution) andCD34 (Neomarker, clone QBEnd/10, 1/50 dilution) (Fig-ure 4). The cells were positive for smooth muscle actin(Neomarker, clone 1A4; same as asm1, 1/50 dilution),which circumscribed the cyst. Histopathological diagnosiswas cystic lymphangioma in the right adrenal gland. Discussion
Lymphangiomas are benign malformations of vessels, most frequently discovered in childhood. They are mostcommonly located in the neck, axillary region and medi-astinum (95%). The remaining 5% are found in the abdom- Figure 2. Macroscopic appearance of multi-septae cystic lesion
inal cavity.9 Four histological subtypes of lymphangiomas have been described: cystic, capillary, cavernous and vas-culolymphatic malformation.1 These types are consideredas a spectrum of the same disease. Combinations of thesetypes may be seen in the same lesion. The presence ofendothelial lined lymphatic channels separated by connec-tive tissue is the dominating histological feature. Cysts of the adrenal glands are very rare. They were first described by Plaut, whose case studies included autopsyfindings.7,8 The incidence in autopsy studies ranges from0.064% to 0.18%.10,12 As the imaging techniques improved,adrenal cysts are discovered as incidental findings onabdominal US and CT examinations.4,6,12 Adrenal cysts have been classified into four main groups:6 endothelial cysts (45%), pseudocysts (39%),epithelial (9%) and parasitic cysts (7%). Pseudocysts occurwith hemorrhage in a normal adrenal gland or an adrenaltumor. Hemorrhage can occur secondary to trauma, bleed-ing disorder, burns, shock or toxemia. Epithelial cysts are Figure 3. Histologically cystic lesion covered by flat endothelial
comprised of cystic adenomas, glandular or retention cells adjacent to the normal-appearing adrenal gland cysts, and cystic transformation of embryonic remnants.
Parasitic cysts are most commonly due to echinococcalinfection with associated disseminated systemic hydatido-sis. Endothelial cysts have a recognizable endothelial lin-ing, and most of them are lymphangiomatous, however,they are not frequently encountered.5 They are character-ized by multiloculated cystic and endothelial lined cavi-ties.11 The endothelial lining reacts with Factor VIII-relat-ed antigen, CD31 and CD34.2,13 Adrenal cysts are usually asymptomatic. When they are symptomatic, symptoms are related to size and position ofthe cyst, and can include pain, gastrointestinal disturbanceor a palpable mass. Laboratory findings are nonspecific.
Rarely small adrenal cysts may be associated with Cushingsyndrome, virilization or pheochromocytoma.6 The imag-ing methods are useful for the clinical management of anadrenal cyst. 4,10 Thin-walled (= 3 mm) cysts under 6 cmin diameter, with homogenous near-water density can be Figure 4. Flat endothelial cells are positive for CD31.
safely managed conservatively, irrespective of the pres- 5. Lack EE: Other neoplasm and tumor-like lesions of the adren- al glands. In: Atlas of Tumor Pathology, Tumors of the Adren- Some authors recommend aspiration of the contents of al Gland and Extraadrenal Paraganglia. (Ed Rosai J), ArmedForces Institute of Pathology, Washington DC, 1997 adrenal cysts for their diagnosis and management instead 6. Longo JM, Jafri SZ, Bis KB: Adrenal lymphangioma: a case of surgical excision if the suspicion of malignancy is low, or the lesion is nonfunctional and asymptomatic.3,11,14 7. Plaut A: Hemangiomas and related lesions of the adrenal gland.
Potential aggressive behavior of lymphangiomas has Virchows Arch Path Anat 335: 345-355, 1962 been described, but it is very uncommon.1,8 8. Plaut A: Locally invasive lymphangioma of adrenal gland.
9. Poncelet V: Retroperitoneal cystic lymphangioma. J Belge References
10. Rozenblit A, Morehouse HT, Amis ES: Cystic adrenal lesions.
1. Fauquenot-Nollen AM, Plaisier ML, Tjon A, Tham RT: Com- bined thoracic and abdominal lymphangioma in an adult. JBR- 11. Satou T, Uesugi T, Nakai Y, et al: Case of adrenal lymphangioma with atypical lymphocytes in aspirate cytology. Diagn 2. Gaffey MJ, Mills SE, Fechner RE, et al: Vascular adrenal cysts.
A clinicopathologic and immunohistochemical study of endothe- 12. Tagge DU, Baron PL: Giant adrenal cyst: Management and lial and hemorrhagic (pseudocystic) variants. Am J Surg Pathol review of the literature. Am Surg 63: 744-746, 1997 13. Torres C, Ro JY, Batt MA, et al: Vascular adrenal cysts: a clini- 3. Katz RL, Mackay B, Zornoza J: Fine needle aspiration cytology copathologic and immunohistochemical study of six cases and of the adrenal gland. Acta Cytol 28: 269-282, 1984 review of the literature. Mod Pathol 10: 530-536, 1997 4. Khoda J, Hertzanu Y, Sebbag G, et al: Adrenal cysts: diagnosis 14. Tung GA, Pfister RC, Papanicolaou N, et al: Adrenal cysts: imag- and therapeutic approach. Int Surg 78: 239-242, 1993 ing and percutaneous aspiration. Radiology 173: 107-110, 1989

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