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Unilateral Adrenal Hematoma with Associated Giant Mass: A Report of Two Cases

Yoshitaka Uji*

Department of Gastrointestinal Surgery, Shin-Koga Hospital, Japan

*Corresponding Author:
Yoshitka Uji
Department of Gastrointestinal Surgery
Shin-Koga Hospital, Japan
Tel: +81-942-38-2222
E-mail: [email protected]

Received date: June 03, 2017; Accepted date: June 12, 2017; Published date: June 22, 2017

Citation: Uji Y (2017) Unilateral Adrenal Hematoma with Associated Giant Mass: A Report of Two Cases. Med Rep Case Stud 2: 140. doi: 10.4172/2572-5130.1000140

Copyright: © 2017 Uji Y. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

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Abstract

Abstract Back ground: Adrenal hematoma is a rare yet potentially life-threatening event that occurs both in traumatic conditions and in a variety of nontraumatic conditions. In the present study, we report the two cases with idiopathic unilateral adrenal hematoma, presenting as a huge adrenal mass. Case presentation: Case 1 involved a 72-year-old man who was complaining of upper abdominal pain. Abdominal computed tomography (CT) and magnetic resonance imaging (MRI) demonstrated a giant mixed density tumor in the right retroperitoneal space. Based on the radiological findings, the preoperative differential diagnoses included adrenal cancer, pheochromocytoma, an adrenal hematoma, and a primary retroperitoneal tumor. To remove the tumor we carried out an adrenalectomy on the right adrenal gland, resecting a specimen measuring 15 × 13.5 cm. The histopathological findings of the specimen revealed a hematoma with normal adrenal tissue. Case 2 involved a 53-year-old woman who complained of an uncomfortable feeling in her left upper abdomen. An Abdominal CT and MRI showed that a retroperitoneal tumor had formed a multicystic lesion. Due to the tumor’s size, the left renal vein had become compressed and therefore, was resected by left adrenalectomy. The resected specimen was 10.5 × 10 cm in size and revealed an adrenal hematoma. In the absence of any obvious etiology, the diagnoses in both cases were idiopathic adrenal hematoma. Conclusion: An accurate diagnosis of idiopathic adrenal hematoma is quite difficult to make prior to surgery. Some imaging modalities are useful in generating a differential diagnosis

Keywords

Adrenal hematoma; Adrenal hemorrhage; Magnetic resonance imaging

Introduction

Adrenal hematoma (AH) is an extremely rare condition, which is difficult to diagnose because of its nonspecific presentation [1]. It is caused by trauma, stress, sepsis, adrenal tumors, anticoagulation, hemorrhagic disorders or pregnancy. In the absence of any distinct etiology, the AH is termed ‘an idiopathic adrenal hematoma’. The lesion is usually discovered as a chronic so-called hematoma, which manifests as a giant and palpable mass. Herein, we report on the two above-mentioned cases involving patients with idiopathic unilateral adrenal hematoma and reviewing the relevant literature on this rare lesion.

Case Report

Case 1

A 72-year-old man was admitted to our hospital, suffering from acute abdominal pain. His past medical history included no anticoagulant therapy, no abdominal trauma, and no specific diseases. A physical examination on admission revealed no abnormality other than some tenderness in the right upper quadrant. His vital signs and laboratory data, including tumor markers, were within the normal range. Abdominal computed tomography (CT) showed a huge, wellcircumscribed, 15 × 15 cm, cystic lesion between the liver and right kidney in which the peripheral part of the tumor was slightly enhanced and the central part was hypovascular. The tumor was heterogeneously enhanced in the portal phase (Figure 1) with no liver metastasis or lymph node swelling being detected. Magnetic resonance imaging (MRI) showed that the signal intensity of the tumor was higher than what is typical of muscle, whereas with T1-weighted imaging, the signal intensity of the tumor was heterogeneously low (Figure 2a). With T2- weighted imaging, the tumor was seen to be diffusely high with the exception of it having a mostly hypointense rim (Figure 2b). The angiography via the right subphrenic artery revealed several encasement areas of the arteries (Figure 3). Although we were unable to make a precise diagnosis, the preoperative differential diagnoses included an adrenal adenoma, pheochromocytome, adrenal hematoma, or some other type of primary retroperitoneal tumor. Next, we performed a laparotomy through a reverse L-shaped incision finding a round, hard, cystic tumor, which was covered by a capsule. The tumor was located on the backside of the liver, and was found to be compressing the inferior and although there was some bleeding from the retroperitoneal tissue, the tumor was safely resected. In total, the operation took four hours and twenty minutes, during which 333 ml of blood was lost. The resected specimen was 15 × 13.5 cm in size and weighed 1,500 g. The cut surface of the tumor revealed a well-defined dark red and white solid mass (Figure 4a). On histopathological examination, the tumor, which consisted of an old hematoma with fibrinoid exudate and organization, was seen to involve the normal adrenal gland beneath the capsule. Despite this, no neoplastic tissue was observed. Thus, the final pathological diagnosis was simply idiopathic adrenal hematoma (Figure 4b). Due to postoperative complications, the patient experienced temporary paralysis of both lower limbs from the epidural anesthesia, which subsequently improved, allowing the patient to be discharged 25 days after surgery.

medical-reports-case-studies-circumscribed

Figure 1: Abdominal CT shows a huge, well-circumscribed, 15 × 15 cm cystic lesion between the liver and right kidney. The peripheral part of the tumor was slightly enhanced and the central part was hypovascular.

medical-reports-case-studies-heterogeneously

Figure 2a: MRI shows that the signal intensity of the tumor was heterogeneously low on T1-weighted imaging.

medical-reports-case-studies-hypointense

Figure 2b: T2-weighted imaging shows that the tumor was diffusely high except there was a mostly hypointense rim.

medical-reports-case-studies-encasement

Figure 3: The angiography via the right subphrenic artery indicates several encasement areas of the arteries (arrow).

medical-reports-case-studies-macroscopic

Figure 4a: Macroscopic finding of the excised tumor. The resected specimen was 15 × 13.5 cm in size and weighed 1,500 g.

medical-reports-case-studies-histopathological

Figure 4b: On histopathological examination, the tumor involved the normal adrenal gland beneath the fibrous capsule. It consisted of an old hematoma with fibrinoid exudate and organization (hematoxylin-eosin staining, ×40).

Case 2

A 53-year-old woman was admitted to our hospital suffering from discomfort due to a giant mass in the upper left quadrant of her abdomen. Her past medical history included no anticoagulant therapy, no abdominal trauma, and no specific diseases. A physical examination on admission revealed no abnormalities. Her vital signs and laboratory data, including tumor markers, were within the normal range. An Abdominal CT showed a 10.5 × 8.7 cm cystic lesion between the aorta and left kidney, in which the periphery was slightly enhanced and the central part was hypovascular, thus indicating central necrosis in the arterial phase (Figure 5a). As, the tumor was found to be compressing the left renal vein in the portal phase (Figure 5b) we performed an MRI which showed that the signal intensity of the tumor was slightly high on T1-weighted imaging (Figure 6a). In addition, on T2-weighted imaging, the tumor showed a multicystic lesion with a thick capsule containing several septations (Figure 6b). We were able to make preoperative differential diagnoses, which included an adrenal cyst, a hematoma, or some other type of primary retroperitoneal tumor. A laparotomy was performed through a median incision where the tumor was found to have invaded the left renal vein and the mesenterium of the colon. Consequently, the invaded part of the left vein and the inferior mesenteric vein were resected simultaneously with the tumor. The tumor was safely excised after resecting the left ovarian vein in an operation that took four hours and thirty-two minutes and saw 1,250 ml of blood loss. The resected specimen was 10.5 × 10 cm in size and weighed 980 g. The cut surface of the tumor revealed both a welldefined dark red color and a number of blood clots. Histopathological examination showed that the inside of the tumor was consisted of fresh hematoma and filled with red blood cells. Thus, the final pathological diagnosis was simply an idiopathic adrenal hematoma (Figures 7a and 7b). The patient was discharged without any complications 21 days after the operation.

medical-reports-case-studies-hypovascular

Figure 5a: Abdominal CT shows a cystic lesion between the aorta and left kidney. The peripheral part was slightly enhanced and the central part was hypovascular.

medical-reports-case-studies-compressing

Figure 5b: The tumor was located compressing the left renal vein (arrow) in the portal phase.

medical-reports-case-studies-weighted

Figure 6a: MRI showed that the signal intensity of the tumor was of slightly high intensity on T1-weighted imaging.

medical-reports-case-studies-multicystic

Figure 6b: T2-weighted imaging shows that the tumor had a multicystic lesion with a thick capsule containing several septations.

medical-reports-case-studies-macroscopic

Figure 7a: Macroscopic finding of the excised tumor. The resected specimen was 10.5 × 10 cm in size and weighed 980 g.

medical-reports-case-studies-histopathological

Figure 7b: Histopathological examination showed that the inside of the tumor was consisted of fresh hematoma and filled with red blood cells (hematoxylineosin staining, ×40).

Discussion

The incidence of AH identified at autopsy has been reported to be 0.14-1.1% [2]. In one of the biggest published series, Vella et al. summarized 141 cases of AH in a 25-year period at the Mayo Clinic [3].

They classified AH into seven categories; namely, incidentaloma (28 cases), spontaneous AH (16 cases), AH associated with antiphospholipid and heparin-associated thrombocytopenia (20 cases), postoperative AH (14 cases), AH associated with anticoagulant therapy (3 cases), AH associated with trauma (4 cases), and AH associated with severe stress or sepsis (56 cases). According to their criteria, spontaneous AH presents as acute and spontaneous hemorrhage of the adrenal gland, with acute abdominal pain in the absence of prior trauma or anticoagulant therapy. “Incidentaloma” presents as an apparent nonfunctioning adrenal mass discovered by imaging studies performed for other reasons. Clinical manifestations of AH can vary widely depending on the degree and rate of hemorrhage, as well as the amount of adrenal cortex compromised by the hemorrhage. Clinical symptoms are often accompanied with physical findings of fever, hypotension or abdominal mass, as discussed elsewhere [4,5]. The diagnosis of AH is often complicated by its nonspecific presentation and tendency to occur in the setting of acute illness and other complicating medical conditions. In regard to the radiological findings of AH, CT proved to diagnose AH in a relatively acute phase with abdominal or flank pain [6]. Hoeffel et al. [7] insisted that MRI was more useful than CT for the diagnosis of spontaneous unilateral AH, with the most important finding being the high intensity on T1-weighted images, especially if the high signal is located mainly peripherally. Furthermore, heterogeneously low signals were characteristic on T2-weighted images. They also suggested that gadolinium-enhanced MRI might be helpful to distinguish between a collection of fluid such as blood, and a neoplasm such as pheochromocytoma or carcinoma. At the same time, the appearance of hematomas on MRI depends on their stage of evolution. They evolve over time into the different components of acute, subacute, and chronic hemorrhage (deoxyhemoglobin, methemoglobin, and hemosiderin) [8]. In acute bleeding (less than seven days) deoxyhemoglobin appears isointense or hypointense on T1-weighted images and has a low signal intensity on T2-weighted images. Subacute blood (seven days to seven weeks) in the form of methemoglobin is hyperintense on T1-weighted images. Initially, methemoglobin is intracellular and has low signal intensity on T2-weighted images. Subsequently, as the red cells lyse and the methemoglobin becomes extracellular, it has high signal intensity on T2-weighted images. Chronic hematoma (more than seven weeks) has low signal intensity on both T1- and T2-weighted images because of the presence of hemosiderin. With this classification, our MRI in case 1 showed a chronic stage hematoma, and our MRI in case 2 showed a subacute stage hematoma. Therefore, each stage of the hematoma in the above cases was able to be proven through actual histopathological examination. Although MRI may be useful for showing the contents of the tumor, idiopathic adrenal hematomas are difficult to diagnose prior to surgery. Although imaging modalities such as CT and MRI are helpful in detecting adrenal hematomas, it is difficult to determine whether the hemorrhage is associated with tumors or not [9]. In Japan, Koizumi et al. [10] described 14 cases of idiopathic AH from 1983 to 2010. Of these 14 cases, 13 had preoperatively been suspected to be adrenal tumors that included malignant lesions, but were then found to be ‘idiopathic adrenal hematomas’ on pathological examination after surgery. Only one patient was diagnosed with an idiopathic AH without surgery. Therefore, surgery was strongly recommended for a definite diagnosis as well as for treatment.

Conclusion

It is difficult to make a precise preoperative diagnosis of idiopathic adrenal hematomas or hemorrhages. Although MRI can be useful, we should be considered when differentiating this rare lesion from an unknown adrenal tumor.

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