Adrenal Cavernous Hemangioma: A Rarely Perceived Pathology--Case Illustration and Review of Literature.
1. IntroductionIncidental 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.
https://doi.org/10.1155/2019/8463890
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; rn05@aub.edu.lb
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
hemangiomas.
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
discomfort
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
calcification
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
finding)
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
(primary
hyperaldosteronism)
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
distention
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
adrenalectomy
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
adrenalectomy
32 Open right adrenalectomy
33 Laparotomy with left
adrenalectomy
34 Left adrenalectomy
35 Laparotomy
36 Open right adrenalectomy
37 Laparoscopic left
adrenalectomy
38 Laparoscopic right
adrenalectomy
39 Open left adrenalectomy
40 Laparoscopic left
adrenalectomy
41 NA
42 Laparoscopic left
adrenalectomy
43 Laparoscopic left
adrenalectomy
44 Laparotomy
45 NA
46 Laparoscopic right
adrenalectomy
47 No surgical management
48 Open adrenalectomy
49 Open left adrenalectomy
50 Laparoscopic right
adrenalectomy
51 Open left adrenalectomy
52 Laparoscopic right
adrenalectomy
53 Laparoscopic right
adrenalectomy
54 Laparoscopic right
adrenalectomy
55 Laparotomy and right
adrenalectomy
56 Laparoscopic left
adrenalectomy
57 Laparotomy and open
adrenalectomy
58 Open adrenalectomy
59 Laparoscopic adrenalectomy
60 Laparoscopic right
adrenalectomy
61 Open right adrenalectomy
62 Open left adrenalectomy
63 Laparoscopic left
adrenalectomy
64 Adreno-nephrectomy
65 Open right adrenalectomy
66 Laparotomy and
adrenalectomy
TABLE 2: Summary of characteristics of previously
reported adrenal cavernous hemangioma in the
literature.
Characteristics Data (N = 66)
Median age (year) 60.04
Sex
Female 41 (62%)
Male 25 (38%)
Laterality
Right 31 (47%)
Left 35 (53%)
Mean size (cm) 10.8
Mean weight (g) 751.9
Symptoms
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 |
| Words: | 5535 |
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