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A 40-year-old woman with a 1-year history of arterial hypertension (systolic blood pressure, 170-180 mm Hg, and diastolic blood pressure, 90-100 mm Hg) requiring combination therapy with bisoprolol and lacidipine was referred to the Department of Internal Medicine at the University of Valencia. As part of the diagnostic procedures, clinical examination was followed by abdominal ultrasonography, which showed a calcified cystic mass of 10-cm diameter in the left retroperitoneal area. This finding was suggestive of hydatid cystic disease; therefore, plain chest and abdominal x-ray films, abdominal computed tomography scan, and laboratory studies for echinoccocus antibodies were performed. Results of chest radiography and immunologic testing were negative. Abdominal x-ray films and computed tomography scan demonstrated the presence of a unique, large, and densely calcified cystic mass of the left adrenal gland, which was displacing the left kidney inferiorly over its vascular pedicle (Fig 1).
Fig. 1Computed tomography scan demonstrating the presence of a unique, densely calcified cystic mass of the left adrenal gland.
Although the patient denied animal contact, the cyst was presumed to be hydatidic, and surgical exploration was indicated. Findings at midline laparotomy were a left retroperitoneal cystic lesion with a densely calcified surface and soft consistency. No other intra-abdominal masses were found, either at the liver or peritoneum. The abdominal cavity and especially the area around the cyst were carefully packed with pads soaked in 20% hypertonic saline. The cyst was not drained nor aspirated during the surgical procedure. An appropriate dissection plane between the cyst and the adrenal gland could not be found, so both the cyst and the adrenal gland were removed. It is noteworthy that the patient's condition remained hemodynamically stable without hypertensive crisis during the operation. Arterial pressure presented a progressive reduction after the operation, and on the fourth postoperative day the patient was discharged with systolic and diastolic blood pressure levels between 110 to 120 mm Hg and 70 to 80 mm Hg, respectively. Macroscopic pathologic examination showed a cystic lesion with a 1-mm thick wall, filled with hydatid sand and clear fluid. Microscopically, an acellular chitinous laminated layer and calcified areas arising from the normal adrenal tissue were observed. Once the hydatidic nature of the adrenal cyst was established, therapy with albendazole was administered during the following 3 weeks. One year later, the patient's condition remains asymptomatic without antihypertensive therapy.
Discussion
Symptomatic hydatid cystic disease of the adrenal glands is rarely observed in clinical practice. Among the 10 cases previously reported of primary hydatid cyst of the adrenal glands, the majority were asymptomatic or the symptoms resulted from space-occupying lesions.
In the case we report here, chest and abdominal imaging showed that there was only 1 cyst in the left adrenal gland; thus, this was a primary cyst. Since Spain is a highly endemic area of hydatid disease, the hydatidic etiology of the cyst was preoperatively presumed. Therefore, no attempt of preoperative puncture of the lesion was made. The link between the cyst and arterial hypertension could be only achieved after surgical removal of the cyst with the involved adrenal gland, when blood pressure returned to normal values.
Three remarkable characteristics can be pointed out in this case. The first is the location of the cyst, since only 10 cases of primary hydatid cyst of the adrenal glands have been reported. Second, the symptom leading to the diagnosis was arterial hypertension; the association between arterial hypertension and primary hydatid cyst of the adrenal glands has not been previously reported. Third, surgical removal of the cyst was curative. Therefore, in areas of endemic hydatid disease, adrenal hydatid cysts can be included among the potentially curative causes of arterial hypertension.
*Reprint requests: Luis Sabater, MD, PhD, Department of Surgery, Hospital Clínico, University of Valencia, Avda. Blasco Ibáñez, 17, 46010-Valencia, Spain.
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