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Adrenal Cavernous Hemangioma: A Rarely Perceived Pathology--Case Illustration and Review of Literature.

1. Introduction

Incidental adrenal masses are a growing concern, especially with the significant increase in their detection upon the many abdominal imaging modalities utilized for the workup of various patient's complaints. The prevalence of incidental adrenal masses approaches 7% in the general population [1]. Adrenal masses tend to be heterogeneous in nature and comprise of benign adenomas, secreting adenomas, lymphomas, myelolipomas, cysts, and adrenocortical carcinoma most commonly as well as other rarer pathologies such as adenomatoid tumors and sex-cord stromal tumors [2]. On the other hand, adrenal cavernous hemangiomas are unusual tumors arising from the endothelial lining of blood vessels [3]. We hereby present a case of a cavernous hemangioma diagnosed on histopathology after an adrenalectomy, as well as a review of all reported cases of this entity in the literature.

2. Case Presentation

A 83-year-old previously healthy male presented with vague abdominal and bilateral flank pain of several months duration.

The pain was dull in nature, with no recent change in weight and appetite, no reported hematuria, no gastrointestinal symptoms, and no reported episodes of headache. All basic blood work up, including complete blood count, creatinine, electrolytes, and liver function tests, were within normal range. An initial imaging with an enhanced computed tomography (CT) scan of the abdomen and pelvis was performed, revealing a right supra renal mass measuring around 8 cm in greatest dimension, possessing a significant enhancement, with a Hounsfield Unit (HU) of 15 on noncontrast phase and a 55HU on the contrast phase. No other abdominal or pelvic findings were noted.

For better characterization of such adrenal lesion, a Magnetic Resonance Imaging (MRI) of the abdomen with gadolinium was requested. Again showing was a mass in the right suprarenal space, measuring 7.3 x 6.5 x 6 cm, showing heterogeneous signal intensity on all sequences, predominately mildly increased on T2-weighted and predominately decreased signal on T1-weighted images, and no appreciable signal drop on the out-of-phase images. The lesion appeared inseparable from the lateral limb of the right adrenal gland and remained separate from the upper edge cortex of the right kidney (Figure 1).

Imaging was also accompanied by a full metabolic work-up, as usually performed for any incidentally discovered large adrenal mass, including: urine and plasma metanephrines, dexamethasone suppression test, DHEA-S, and aldosterone/renin ratio, and all yielded results were within normal range.

Due to the size of the tumor, a shared decision was made to surgically remove the adrenal gland due to a potential risk of being an adrenocortical carcinoma. The patient therefore underwent a right radical adrenalectomy through an open subcostal incision. Intraoperatively, the adrenal was noticeably friable and tended to bleed on minimal manipulation, which was expected from the preoperative imaging done (Figure 1). No significant blood loss was encountered since complete dissection of the adrenal gland was done for better hemostatic control.

On gross pathological examination, the specimen measured 8 x 7 x 3 cm and the tumor was shiny tan-yellow, in nature with distinct demarcation from the normal adrenal parenchyma. Histologically, the tumor was proven to be a hemorrhagic cavernous hemangioma (Figure 2).

Patient recovered well after his operation with no complications encountered thereafter. He was discharged home on his fourth postoperative day, to follow-up in clinic one month from discharge.

3. Discussion

Cavernous hemangiomas are unusual tumors of the endothelial linings with a propensity for skin, liver, and brain involvement [3]. Cavernous hemangiomas tend rarely to affect the genitourinary system [3].

Adrenal hemangiomas are one of the rarest snonfunctioning benign adrenal tumors that are commonly diagnosed postoperatively [4]. Although many cases of adrenal hemangiomas were presented at autopsy reports before 1869, the first surgical adrenal hemangioma was reported by Johnson and Jeppesen in 1995 [3].

Sixty-six cases of adrenal cavernous hemangiomas were published between the years 1955 and 2018 (Table 1), and identified after conducting an extensive literature review using PubMed, Medline, Embase, and Scopus databases. These cases were reviewed and summarized in Table 2. The median age of patients at diagnosis was 60 years. This neoplasm had a female preponderance with a female to male prevalence ratio of 3 to 2. No laterality preference was associated with adrenal hemangiomas. Two bilateral cases were only reported in the literature so far. Metabolic workup of adrenal neoplasms was normal in 45 of the 66 reported cases. Only 6 clinically functional adrenal hemangiomas were identified; with 3 cases of hyperaldosteronism and three other cases of subclinical Cushing's syndrome. The so far reported cases of adrenal hemangiomas, with detailed published tumor characteristics, exhibited a mean diameter of 11 cm and a mean weight of 752 grams. Of the 66 published cases, 38 were incidentalomas that were clinically silent and asymptomatic; 8 presented with vague abdominal symptoms such as bloating, epigastric pain, and heaviness, and 6 cases reported solely flank pain. Another two cases presented with spontaneous rupture of the adrenal mass with subsequent retroperitoneal hemorrhage and hematoma; a serious complication that is seldom seen.

On imaging, 32 adrenal masses were associated with speckled calcification, a historically described finding in any adrenal hemangioma; and 29 cases failed to show calcifications. Sixty-five of the reported cases were managed surgically; out of them, 47 were excised through an open approach, and the remaining 16 cases were excised laparoscopically.

Most of the cavernous hemangiomas reported in the literature are incidental findings on imaging performed for unrelated or unspecific complaints [66]. These tumors grow insidiously until they reach a large size and start producing symptoms by virtue of mass effect and mechanical pressure on adjacent organs. Vague symptoms such as fever, weight loss, and sweating are nonspecific finssdings for neoplastic lesions that are reported in adrenal hemangiomas [41, 67]. Flank pain in the setting of normal urine analysis is the most commonly reported presenting complaint in symptomatic patients. Hypertension has been identified as a presenting symptom for adrenal hemangiomas in the setting of normal adrenal functions. Six cases reported so far presented with a hyperfunctioning adrenal mass; three of them presented with signs of hyperaldosteronism such as hypokalemia, and three other cases were consistent with subclinical Cushing [20, 44].

Histopathologically, adrenal hemangiomas are stratified into two subtypes: cavernous and capillary. The cavernous subtype is composed of an enlarged mass of blood filled endothelially-lined sinusoids, displacing potentially normal tissues. Whereas in the rarer capillary subtype, it is composed of small tufts of submucosal capillaries arranged in radiating loops or lobules [20].

Historically, adrenal hemangiomas were usually identified on plain abdominal radiographies for unrelated complaints. On radiographs, these neoplasms appear as calcified masses. Calcifications, if present, are universally speckled through the entire mass as opposed to the curvilinear calcifications usually associated with adrenal pseudocysts [68]. Computed tomography can effectively define the anatomy, configuration, and volume of any adrenal mass and can partially delineate the general tissue's characteristics. On CT scanning, these masses are generally encapsulated and heterogenous with scattered calcifications [68]. Calcifications are usually correlated with benign adrenal lesions; however, some reports describe calcifications in malignant lesions as well. Therefore, calcifications become an unreliable sign to assess the malignant potential of any adrenal mass. Cavernous hemangiomas are mostly masses with smooth margins and low relative attenuation coefficient [20, 68]. However, rim-like calcifications within the suprarenal glands have been adopted radiologically as a sign of benignity of such lesions. A radiologic sign was first described by Rothberg et al., referred to as phleboliths, which are round calcifications with translucent centers. This finding is considered pathognomonic for adrenal gland hemangiomas [68, 69]. CT scan has been shown to be superior to ultrasound for suprarenal masses. The masses are usually heterogeneously echogenic on ultrasonography. Magnetic Resonance Imaging (MRI) has sometimes been used, although a CT scan is enough as an imaging modality to identify adrenal neoplasms. Cavernous hemangiomas are hypo-intense masses on T1-weighted images and hyper-intense on T2-weighted images with peripheral enhancement after contrast administration [66].

Although not required for routine diagnostic workup of any adrenal masses, angiography on adrenal hemangiomas can reveal marked neovascularity with small vascular channels, usually arranged in a rim-like manner which retain contrast in delayed films [69]. These angiomas could be of many variants which include: angiomyelolipoma (more commonly seen), angiolipomas, cavernous hemangiomas, or epithelioid hemangioendothelioma, depending on the histopathological differences. Moreover, during pathologic examination, adrenal hemangiomas could be mistaken for adrenocortical carcinoma that has undergone cystic degeneration; therefore proper assessment of the subcapsular area is paramount [25].

After identification of adrenal masses on imaging, the common practice necessitated a full hormonal and metabolic workup to rule out primary functioning adrenal neoplasms mainly pheochromocytomas. Most cases of adrenal cavernous hemangiomas are nonsecretory and hormonally silent neoplasms [44]. Due to the scarcity of this condition, no guidelines have been developed so far to guide the treatment and therapeutic management of such entity.

Tumors originating from vessels could be associated with syndromes, but these are rather neonatal tumors, and not acquired tumors such as our present case. Nevertheless, there has been a previous single report of an adrenal cavernous hemangioma associated with familial adenomatous polyposis [57].

Most adrenal hemangiomas reported in the literature were managed surgically [20]. Asymptomatic small and benign-looking masses may be treated medically and conservatively with close monitoring. However, the follow-up schedule tends to be according to physician's preference. Larger masses bear the risk of spontaneous hemorrhage and should be resected surgically [20, 44]. Early cases were operated through open adrenalectomy. However, a laparoscopic approach is favored due to better postoperative results and lesser complications [70]. Knowing that, the risk of malignancy might sway the operating surgeon against a minimally invasive approach.

4. Conclusion

Adrenal cavernous hemangioma is a rare entity that might be encountered when dealing with an adrenal pathology. Surgical resection is sometimes necessary to rule out any malignant potential and alleviate symptoms secondary to mass effect. Retroperitoneal bleeding is a concern in such pathology, especially when large lesions are detected. Observation is an alternative in cases where lesions are small, asymptomatic, and metabolically inactive, especially when confirmed by biopsy.

Ethical Approval

Written consent was obtained from the patient to publish his case including clinical and pathological images, and available to Editor-in-chief upon his request.

Conflicts of Interest

The authors declare that they have no conflicts of interest.

Authors' Contributions

N.A. and M.M. performed the literature review, J.D., N.A. and R.N. were involved in the workup and surgery, N.A., J.D., and M.M. wrote the initial draft of the manuscript. A.T. provided the histopathological slides and commentaries. All authors approved of the final manuscript prior to submission. Jad A. Degheili and Nassib F. Abou Heidar were contributed equally to this manuscript and qualify as first authors.


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Jad A. Degheili [ID], (1) Nassib F. Abou Heidar [ID], (1) Mouhammad El-Moussawi, (1) Ayman Tawil, (2) and Rami W. Nasr [ID], (1)

(1) Department of Surgery, Division of Urology, American University of Beirut-Medical Center, Beirut 11072020, Lebanon

(2) Department of Pathology and Laboratory Medicine, American University of Beirut-Medical Center, Beirut 1107 2020, Lebanon

Correspondence should be addressed to Rami W. Nasr;

Received 12 July 2019; Revised 3 October 2019; Accepted 29 November 2019; Published 26 December 2019

Academic Editor: Imtiaz A. Chaudhry

Caption: FIGURE 1: Enhanced magnetic resonance images (MRI) of the abdomen and pelvis with gadolinium. (a) Coronal view of an in-phase T1-weighted image identifying a large suprarenal mass measuring around 7.3 x 6.5 x 6 cm occupying the space of the right adrenal gland, showing a significant rim enhancement with marked vascularity. (b) Coronal view of a T2-weighted image identifying the similar right suprarenal mass with heterogeneous component inside it, possessing various signal intensities.

Caption: FIGURE 2: Histopathological images of an adrenal cavernous hemangioma cross section, showing dilated capillaries with capsule and significant fibrosis on Hematoxylin and Eosin (H&E) staining (a). Magnified image showing red blood cells inside dilated capillaries (b), divided by thick fibrous septa (c).
TABLE 1: List of all published cases of adrenal cavernous

Case number     Authors (year of publication)      Age/gender

1              Johnson and Jeppesen (1955) [3]        46/F
2                  Elliot et al. (1963) [5]           37/F
3                  Chodof et al. (1966) [6]           76/F
4                Weiss and Schulte (1966) [7]        70 /M
5                     Ruebel (1973) [8]               75/M
6                 Rothberg et al. (1978) [9]          727F
7                 Rothberg et al. (1978) [9]          74/F
8                     Vargas (1980) [10]              67/F
9                   Lee et al. (1982) [11]            59/F
10                Orringer et al. (1983) [4]          51/M
11                 Goren et al. (1986) [12]           79/F
12               Nakagawa et al. (1986) [13]          71/M
13                Guerin et al. (1988) [14]           78/F
14                Derchi et al. (1989) [15]           69/F
15                Derchi et al. (1989) [15]           60/M
16              Yoshihirio et al. (1990) [16]         78/F
17                 Honig et al. (1991) [17]           73/M
18                Takahe et al. (1991) [18]           55/M
19                 Salup et al. (1992) [19]           73/F
20                Hamrick et al. (1993) [20]          66/M
21               Sabanegh et al. (1993) [21]          60/F
22               Boraschi et al. (1995) [22]          64/M
23               Stumvoll et al. (1996) [23]          60/F
24                Marotti et al. (1997) [24]          68/F
25                Marotti et al. (1997) [24]          60/F
26                  Oh et al. (1997) [25]             56/M
27               Hayakawa et al. (1998) [26]          56/M
28                Hisham et al. (1998) [27]           61/F
29               Makiyama et al. (1998) [28]          61/F
30               Thiele and Bodie (2001) [29]         72/F
31               Yagisawa et al. (2001) [30]          52/M
32                  Xu and Liu (2002) [31]            60/M
33                Nursal et al. (2004) [32]           48/F
34                 Wang et al. (2004) [33]            63/F
35                    Forbes (2005) [34]              75/M
36                 Heis et al. (2008) [35]            50/F
37                  Ng et al. (2008) [36]             59/M
38                 Nigri et al. (2008) [37]           58/F
39             Arkadopoulos et al. (2009) [38]        75/F
40                Matsuda et al. (2009) [39]          51/M
41                Siddiqi et al. (2009) [40]          54/F
42                 Telem et al. (2009) [41]           42/F
43                Cheong and Kim (2010) [42]          66/F
44            Paluszkieweicz et al. (2010) [43]       45/M
45              Abu EL Ghar et al. (2011) [44]        44/M
46               Al Jabri et al. (2011) [45]          19/F
47                kieger et al. (2011) [46]           53/F
48                 Oishi et al. (2012) [47]           75/F
49               Quildrian et al. (2012) [48]         62/F
50                Edward et al. (2013) [49]           78/F
51                 Galea et al. (2013) [50]           84/F
52                  Noh et al. (2014) [51]            27/F
53                 Wang et al. (2014) [52]            37/M
54                Agrusa et al. (2015) [53]           49/F
55                 Wong et al. (2015) [54]            80/F
56                 Pang et al. (2015) [55]            71/F
57               Tarchouli et al. (2015) [56]         71/F
58                 Bacha et al. (2016) [57]           60/M
59               Kinebuchi et al. (2016) [58]         77/M
60                Njoumi et al. (2017) [59]           30/F
61                 Tadic et al. (2017) [60]           50/F
62                  Feo et al. (2018) [61]            70/M
63               Hashimoto et al. (2018) [62]         70/M
64                Iwamot et al. (2018) [63]           52/M
65               Lavingia et al. (2018) [64]          64/M
66                 Peng et al. (2018) [65]            31/F

Case number   Laterality      Size(cm)             Presentation

1               Right         6.5*4*3              Hypertension
2                Left            25             Incidental finding
3                Left          18*16            Abdominal mass and
4               Right          11*7*6        Acute urinary retention
                                                    (-ve met)
5               Right        8*7.3*6.5              Hematuria
6               Right         14*10*7       Long standing hypertension
7                Left           9*8             Incidental finding
8                Left            NA           Incidental finding on
                                                 barium study for
                                                  chronic anemia
9               Right         8.5*7*6          Incidental abdominal
10              Right            17            Epigastric heaviness
11              Right          9*7*5            Incidental finding
                                                   (-ve metab)
12               Left         10*18*24           Night sweat and
                                               generalised fatigue
13               Left        3*2.5*2.5             Elevated ESR
14               Left            20             Incidental finding
15              Right            18              Hepatomegaly and
                                                  abdominal pain
16               Left         6*5.5*5           Incidental finding
17               Left            NA             Incidental finding
18               Left          10*9*9           Incidental finding
19               Left            15            Syncope (incidental
20               left            9              Incidental finding
21               left          20*20            Incidental finding
22              right         10*8*6.5         Megaloblastic anemia
23              Right            8              Mineralocorticoid
                                                 excess syndrome
24               Left         14*10*10          Incidental finding
25               Left         9*7.5*5           Incidental finding
26              Right          6*5*4          Right flank discomfort
27               Left            5              Incidental finding
28              Right            25                 Flank pain
29              Right       5.5*3.5*3.5         Incidental finding
30               Left       9.5*4.2*4.5         Incidental finding
31              Right          6.5*7              Dull back pain
32              Right            17               Abdominal mass
                                                incidental finding
33               Left            13              Palpitation and
                                             unremitting hypertension
34               Left         5.5*5*4        Left upper quadrant pain
35               Left         19*18*8       Retroperitoneal hemorrhage
36              Right            10                 Flank pain
37               Left         3.1*2.9           Incidental finding
38              Right         7*4.5*3           Incidental finding
39               Left          8*6*4            Incidental finding
40               Left         4*4*3.5           Incidental finding
41              Right         2.8*2.5             Abdominal pain
42               Left            12              Left flank pain
43               Left         4.5*3.4           Incidental finding
44               Left            NA         Retroperitoneal hemorrhage
45              Right           11*6            Incidental finding
46              Right       4.3*7.3*5.4         Incidental finding
47              Right            2            Microscopic hematuria
48               Left          5*5*3            Incidental finding
                                             with positive metabolic
                                              workup for subclinical
                                                 cushing disease
49               Left       12.5*11.5*8         Incidental finding
50              Right         5.4*3.3           Incidental finding
51               Left          13*11                Flank pain
52              Right         7.8*7.8           Incidental finding
53              Right         6*5*4.5           Incidental finding
54              Right         11*7.5*7        Nonspecific abdominal
                                               symptoms (epigastric
                                            pain, nausea and vomiting)
55              Right      12.3*13.9*13.8       Incidental finding
56               Left        9.5*8*7.5          Chronic abdominal
57              Right         42*38*17        Intermittent abdominal
                                                pain and increase
                                                 abdominal girth
58               Left        17.5*17*9          Incidental finding
59               Left         5.4*4.3           Incidental finding
60              Right            7              Incidental finding
61              Right        11.5*11*11      Intermittent flank pain
                                             and abdominal discomfort
62               Left         9*6.5*7           Incidental finding
63               Left        27*17*5.5           Loss of appetite
64               Left         5*3.7*3           Incidental finding
65              Right        64*5.5*4.7         Incidental finding
66              Right            NA            Right upper quadrant
                                                  and flank pain

Case number            Surgery

1              Open right adrenalectomy
2              Open left adrenalectomy
3              Open left adrenalectomy
4              Open right adrenalectomy
5              Open right adrenalectomy
6              Open right adrenalectomy
7              Open left adrenalectomy
8              Open left adrenalectomy
9              Open right adrenalectomy
10              Exploratory laparotomy
                  with adrenalectomy
11             Open right adrenalectomy
12             Open left adrenalectomy
13             Open left adrenalectomy
14             Open left adrenalectomy
15             Open right adrenalectomy
16             Open left adrenallectomy
17              Exprolatory laparotomy
                  with adrenalectomy
18             Open left adrenalectomy
                   with splenectomy
19             Open left adrenalectomy,
                distal pancreatectomy,
                 splenectomy and left
                 radical nephrectomy
20                Open adrenalectomy
21             Open left adrenalectomy
22                Open adrenalectomy
23                Open partial right
24                Open adrenalectomy

25                Open adrenalectomy
26             Open right adrenalectomy
27             Open left adrenalectomy
28             Open right adrenalectomy
29             Open right adrenalectomy
30             Open left adrenalectomy
31                Laparoscopic right
32             Open right adrenalectomy
33               Laparotomy with left
34                Left adrenalectomy
35                    Laparotomy
36             Open right adrenalectomy
37                Laparoscopic left
38                Laparoscopic right
39             Open left adrenalectomy
40                Laparoscopic left
41                        NA
42                Laparoscopic left
43                Laparoscopic left
44                    Laparotomy
45                        NA
46                Laparoscopic right
47              No surgical management
48                Open adrenalectomy
49             Open left adrenalectomy
50                Laparoscopic right
51             Open left adrenalectomy
52                Laparoscopic right
53                Laparoscopic right
54                Laparoscopic right
55               Laparotomy and right
56                Laparoscopic left
57               Laparotomy and open
58                Open adrenalectomy
59            Laparoscopic adrenalectomy
60                Laparoscopic right
61             Open right adrenalectomy
62             Open left adrenalectomy
63                Laparoscopic left
64                Adreno-nephrectomy
65             Open right adrenalectomy
66                  Laparotomy and

TABLE 2: Summary of characteristics of previously
reported adrenal cavernous hemangioma in the

Characteristics                   Data (N = 66)

Median age (year)                     60.04
Female                              41 (62%)
Male                                25 (38%)
Right                               31 (47%)
Left                                35 (53%)
Mean size (cm)                        10.8
Mean weight (g)                       751.9
Asymptomatic                       38 (57.5%)
Vague abdominal symptoms            8 (12.1%)
Flank pain                           6 (9%)
Speckled calcifications
Present                             29 (44%)
Absent                             32 (48.5%)
Metabolic workup
Normal                              45 (68%)
Abnormal                             6 (9%)
Hyperaldo steronism                 3 (4.5%)
Subclinical Cushing's syndrome      3 (4.5%)
Surgical approach
Open                                47 (71%)
Laparoscopic                        16 (24%)
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Title Annotation:Case Report
Author:Degheili, Jad A.; Heidar, Nassib F. Abou; Moussawi, Mouhammad El-; Tawil, Ayman; Nasr, Rami W.
Publication:Case Reports in Pathology
Date:Jan 1, 2020
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