Abstract
Primary adrenal epithelioid angiosarcoma (PAEA) is an exceedingly rare and high-grade malignant neoplasm with an epithelioid appearance morphologically. To the best of our knowledge, only 35 cases of PAEA have previously been reported in English literature. Here, we present our 7-year experience (2010-2017) with adrenal lesions at our Clinical Pathological Diagnosis Center. Of more than 1,800 adrenal gland specimens, a total of 2 adrenal epithelioid angiosarcoma cases were identified. We reviewed the literature for 13 similar cases. All of the patients appeared to have morphological features of epithelioid cells including vascular endothelium immunophenotype, hemorrhage/necrosis, and anastomosing vascular channels. They underwent adrenalectomy without chemotherapy or radiotherapy. One was still well at 3-year follow up but the other had died at 1-month follow up. One of our cases was first described with accompanying reactive adrenal cortex hyperplasia in a local area. Moreover, the mechanisms of epithelioid angiosarcoma are still uncertain and require further study. Clinical prognosis is poor, therefore early accurate diagnosis and complete resection of this tumor may be very helpful for patients.
Keywords: Primary adrenal epithelioid angiosarcoma, adrenal cortex hyperplasia, morphology, immunohistochemistry, poor prognosis
Introduction
PAEA is an extremely rare disorder characterized by sheets of irregular endothelial cells with predominantly or exclusively epithlioid appearance. It most often occurs in the sixth decade of life and has a male predilection [1-11]. Most primary adrenal angiosarcoma (PAA) are the epithelioid type. Of the 37 reported cases, 35 cases presented epithlioid appearance [1-4]. PAEA shows an aggressive clinical course and poor prognosis, with 5-year overall survival rates of 24% to 31% [1]. Prognostic factors and appropriate treatment strategies require further validation by more cases.
Most adrenal angiosarcoma represent metastases from another primary tumor site, therefore, exclusion of other original sites is vital for pathologists to diagnose PAEA. In addition, PAEA can be easily confused with adrenal cortical carcinoma, malignant melanoma, and epithelioid sarcoma-like hemangioendothelioma (ESLH), given its rarity and morphologic overlap. Immunohistochemistry is an available ancillary tool for us currently.
In this study, we presented 2 unusual cases of PAEA with detailed clinical, histological, and immunophenotypic features. We reviewed the literature for 5 recent years (2012-2017) and found 13 similar cases. Our two cases present similar pathological features with irregular epithlioid cells, characterized by abundant eosinophilic cytoplasm, enlarged nuclei, and prominent nucleoli. Immunophenotype demonstrated that all epithelioid cells were positive for CD31, ERG and Fli-1. To date, no previous cases have been reported that accompanied with reactive adrenal cortex hyperplasia.
Materials and methods
Two cases of PAEA were obtained from the Department of Pathology of Ningbo Clinical Pathological Diagnosis Center, China. Glass slides and paraffin blocks were available from the specimens of left adrenalectomies in June 2014 and August 2017. Follow up times were 1 month and 3 years. Two expert pathologists (S.Y.W and Y.S.Y) confirmed a consensus diagnosis of epithlioid angiosarcoma, according to the 2013 (4th edition) World Health Organization (WHO) classification criteria of soft tissue tumors. Use of these human tissue samples was reviewed and approved by our Institutional Ethics Board.
Immunhistochemical (IHC) studies were performed on formalin-fixed paraffin-embedded sections (4-um-thick) by avidin-biotin-complex immunoperoxidase technique. Paraffin sections stained with hematoxylin-and-eosin (H&E) and IHC were viewed with an upright microscope and photographed by OLYMPUS BX53. Antibodies are detailed in Table 1. Appropriate positive and negative controls were used for each antibody. Tumor reactivity for immunohistochemical antibodies was scored as follows: -, all tumors cells were negative; tumors cells +, 5-25% of tumors cells were positive; ++, 26-50% of tumors cells were positive; and +++, >50% of tumors cells were positive. Only tumors cells with distinct nuclear staining for ERG, Fli-1, Ki67, S-100, PAX-8, calretinin, and P53; distinct cell membrane staining for CK (pan), CK5/6, and D2-40; distinct cell cytoplasmic staining for F8, CK7, Melan-A, inhibin α, syn, CgA , HMB45, vimentin, and SMA; and distinct cell membrane and/or cytoplasmic staining for CD31 and CD34 were recorded as positive.
Table 1.
Antibodies used for immunohistochemical staining
| Antibody | Clone number | Source | Dilution |
|---|---|---|---|
| CD31 | JC/70A | MAB, Fuzhou, China | 1:300 |
| CD34 | QBEnd/10 | MAB, Fuzhou, China | 1:500 |
| ERG | EP111 | ZM, Beijing, China | 1:600 |
| Fli-1 | MRQ-1 | ZM, Beijing, China | 1:400 |
| F8 | None* | MAB, Fuzhou, China | 1:500 |
| CK (pan) | AE1/AE3 | MAB, Fuzhou, China | 1:400 |
| Ki67 | MIB-1 | Dako, Glostrup, Denmark | 1:200 |
| CK5/6 | D5/16B4 | MAB, Fuzhou, China | 1:400 |
| CK7 | OV-TL12/30 | ZM, Beijing, China | 1:500 |
| Vimentin | V9 | MAB, Fuzhou, China | 1:600 |
| Melan-A (A103) | A103 | MAB, Fuzhou, China | 1:500 |
| S-100 | 4c4.9 | MAB, Fuzhou, China | 1:400 |
| Syn | SP11 | MAB, Fuzhou, China | 1:500 |
| CgA | EP38 | ZM, Beijing, China | 1:300 |
| Melanoma (HMB45) | HMB45 | MAB, Fuzhou, China | 1:600 |
| PAX-8 | MRQ-50 | ZM, Beijing, China | 1:400 |
| D2-40 | D2-40 | Dako, Glostrup, Denmark | 1:600 |
| Calretinin | None* | ZM, Beijing, China | 1:600 |
| P53 | DO-7 | Leica, Germany | 1:800 |
| SMA | 1A4 | MAB, Fuzhou, China | 1:400 |
Neither F8 nor calretinin has a clone number due to rabbit polyclonal antibody.
Results
Clinical features
Clinical features of all cases are summarized in Table 2. In case 1, a 60-year-old man had been diagnosed with high blood pressure ten years prior and achieved good control by regularly taking medicine. Upon physical examination in June 2014, computed tomography (CT) scan of the chest and abdomen demonstrated a palingenetic mass of the left adrenal gland (Figure 1A) and was interpreted as cortical adenoma, not excepting for malignancy. No other primary tumor sites were revealed. A complete blood count, aldosterone, and electrolytes were unremarkable. Cortisol was within normal limits and measured 399.8 nmol/L. A laparoscopic left adrenalectomy was then performed. Chemotherapy and radiation therapy were never given. Postopratively, the patient made an uneventful recovery and follow up at 3 years showed no tumor recurrence.
Table 2.
Reported cases and two cases of epithelioid angiosarcoma in renal gland
| Cases | Age/sex (y) | Size (cm) | Cite | Concomitant adrenal lesion | Relation to the adjacent organs | Chemotherapy/radiotherapy | Clinical outcome |
|---|---|---|---|---|---|---|---|
| 1 [9] | 61/M | NA | R | None | NA | NA | NA |
| 2 [10] | 35/M | 6 | R | Adrenocortical adenoma (F), IM | Without evidence of local tissue invasion or metastatic spreading | Adriamycin/ifosfamide x 4 cycles | Alive, NED at 2 years |
| 3 [11] | 69/F | 14 | R | None | NA | NA | NA |
| 4 [7] | 62/F | 13.7 | CL | NA | NA/NA | NA | |
| 5 [8] | 42/M | 14 | R | CL | Severe adhesions of the adrenal mass to the right kidney and liver | None/Yes | Metastases to the right chest wall, liver and right mid-abdominal wall |
| 6 [5] | 68/M | 8 | L | None | In contact with the gastric wall, left renal vein and splenic vessels | (Anthracyclines)/Yes | Died of neoplastic cachexia at 7 months, metastases to vertebral and costal |
| 7 [4] | 63/M | 8.5 | R | Adrenocortical adenoma (NF), IM | None | NA/NA | NA |
| 8 [3] | 60/F | 5.5 | L | Adrenocortical adenoma (NF), CF | None | NA/NA | Alive, NED at 9 months after the primary resection |
| 9 [2] | 53/M | 15 | L | None | NA | Yes/Yes | Died of disease at 3 months, metastases to lung, brain, liver |
| 10 [2] | 74/F | 8.5 | L | None | NA | None/none | Died of disease at 5 months |
| 11 [2] | 75/F | 11 | L | None | NA | None/none | Died of disease at 16 months, metastases to lung, bone |
| 12 [2] | 75/F | NA | L | None | NA | None/none | Alive with disease at 19 months, metastases to brain at 18 months |
| 13 [1] | 66/M | 3.4 | R | Adrenocortical adenoma (NF), CF | Without evidence of local tissue invasion or metastatic spreading | Paclitaxel/none | Metastases to pleura at 3 months, died with another disease at 1.5 years |
| Our case 1 | 60/M | 6.5 | L | None | None | None/none | Alive, NED at 3 years |
| Our case 2 | 60/M | 8.0 | L | Adrenal cortex hyperplasia and CL | Adhesions to the capsule of kidney and spleen | None/none | Died of disease at 1 month |
NA, Not available; F, Functioning; NF, Non-functioning; IM, adrenal epithelioid angiosarcoma and adrenocortical adenoma were intermingled; CF, angiosarcomatous lesion appeared confined within the adrenocortical adenoma; NED, No evidence of recurrence or metastatic disease; CL, Cystic lesions.
Figure 1.
CT scan of the mass indicated by an arrow; regions as follows: A. The left adrenal gland of case 1; B. The left adrenal gland of case 2 and no clear demarcation between the lesion and adjacent tissues.
In case 2, a 60-year-old man was admitted to the ICU of our hospital because of severe pain in his left lumbar region in August 2017. CT revealed a huge solid mass with cystic areas, peripheral leakage around the lesion in his left adrenal gland, and no clear demarcation between the adrenal mass and adjacent organs including the left kidney, spleen, and bottom of the stomach serosal surface (Figure 1B). No suspicious lesions were identified elsewhere in the body, as with case 1. A left adrenalectomy, left nephrectomy, and splenectomy were then performed. Hemoglobin declined frequently and blood transfusion was performed many times postoperatively. The patient had a rapidly progressive and fatal course, dying one month after diagnosis.
Histopathology
The tumors measured 6.0 cm and 8.5 cm in maximum dimension. Macroscopically, the cut surface of the two cases was typically dark red to yellow intermingling with regions of hemorrhage and necrosis. Cystic lesions were seen in case 2. Yellow orange adrenal parenchyma was grossly identified adjacent to the mass in both cases. Histopathologically, the appearance was of sheets of irregular epithelioid cells invading the adrenal gland or the vascular lumen lined by atypical epithelioid cells (Figure 2A-D), only a thinned cortex remained. Marked necrosis, hemorrhage, and anastomosing vascular channels with red cells lined by irregular epithelioid cells could be easily seen. The irregular epithelioid cells were mostly characterized by abundant eosinophilic cytoplasm, enlarged nuclei, prominent nucleoli, and increased mitotic activity (5-40/10 HPF) (Figure 2E, 2F). Moreover, a reactive proliferation of adrenocortical cells without mitoses was seen in a local area in case 2 and showed negative for inhibin α (Figure 2G, 2H). The tumor was confined to the adrenal gland in case 1 (stage II) and invaded adjacent organs (such as the capsule of kidney and spleen) in case 2 (stage III). Tumor stage was according to French Fédération Nationale des Centres de Lutte Contre le Cancer (FNCLCC) classification.
Figure 2.
Microscopic features of adrenal epithelioid angiosarcoma of case 1 (A, B) and case 2 (C-H). Irregular epithelioid cells invaded the adrenal gland and lined up along the vascular lumen (A, B) or appeared a sheet-like pattern (C, D). Cells were characterized by abundant eosinophilic cytoplasm, enlarged nuclei, prominent nucleoli, and increased mitotic activity (E, F). Tumor with reactive adrenal cortex hyperplasia (G) exhibited that irregular epithelioid cells were negative for inhibin α but cells of reactive adrenal cortex hyperplasia were positive for inhibin α (H).
Immunohistochemical studies
The results of immunostaining are shown in Table 3 and Figure 3. Atypical epithelioid cells in all specimens expressed CD31 and/or CD34 and CK (Figure 3A, 3B, 3F) but Melan-A and calretinin were negative (exclude data not available). In addition, both cases showed nuclear co-expression of ERG and Fli-1 (Figure 3C, 3D) and strongly and diffusely expressed CD31 with a distinctive membranous pattern and staining intensity similar to that seen in normal vessels. The Ki67 proliferative index was 20-30% (Figure 3E). Irregular epithelioid cells were negative for Melan-A and HMB45 and positive for vimentin (Figure 3G-I).
Table 3.
Reported cases and our cases of immunohistochemistry
| CK | CD31 | CD34 | F8 | ERG | Fli-1 | Ki67 | Melan-A | S-100 | HMB45 | Calretinin | |
|---|---|---|---|---|---|---|---|---|---|---|---|
| 1 | NA | NA | NA | NA | NA | NA | NA | NA | NA | NA | NA |
| 2 | + | + | + | + | NA | NA | NA | NA | NA | NA | NA |
| 3 | + | + | + | NA | NA | NA | NA | - | - | - | NA |
| 4 | NA | + | + | NA | NA | NA | 90% | NA | NA | NA | NA |
| 5 | + | + | - | - | NA | NA | 60% | NA | NA | NA | NA |
| 6 | Focal + | + | - | + | NA | NA | NA | NA | - | NA | NA |
| 7 | + | + | - | NA | NA | NA | 90% | - | - | - | - |
| 8 | Focal + | + | Focal + | + | NA | NA | NA | - | NA | NA | NA |
| 9 | + | + | + | NA | NA | NA | NA | NA | NA | NA | NA |
| 10 | + | - | + | NA | NA | NA | NA | NA | NA | NA | NA |
| 11 | + | - | + | NA | NA | NA | NA | NA | NA | NA | NA |
| 12 | + | - | + | NA | NA | NA | NA | NA | NA | NA | NA |
| 13 | + | + | + | + | + | + | NA | NA | NA | NA | - |
| Our case 1 | Focal + | ++ | + | + | + | + | 20% | - | Focal + | - | - |
| Our case 2 | Focal + | ++ | - | + | +++ | ++ | 20% | - | - | - | - |
NA, Not available.
Figure 3.
Immunohistochemical staining appearance of tumors in our two cases. The irregular epithlioid cells strongly and diffusely expressed vascular differentiation markers CD31, CD34, ERG, and Fli-1 (A-D). The Ki67 proliferative index is about 20% (E). A few scattered cells were positive for CK (pan) (F). The cells showed negative for Melan-A and HMB45 (G, H) and showed positive for vimentin (I).
Discussion
PAEA is a rare disease initially described by Kareti and his colleagues in 1988 [12]. Thirteen other cases have been reported in a recent 5 year span. Clinical and pathological characteristics are demonstrated in Tables 2 and 3. Among all of the cases, 4 reported cases had a collision tumor in which PAEA was combined with adrenocortical adenoma and our case 2 accompanied with reactive adrenal cortex hyperplasia. Nine patients were male and six were female. Twelve patients were over 60 years old. All cases appeared to have morphological features of epithelioid cells including vascular endothelium immunophenotype, hemorrhage, and necrosis, and anastomosing vascular channels with red cells.
Diagnosis of adrenal epithelioid angiosarcoma (AEA) can be challenging, given its rarity and morphology, with histological mimics including adrenal cortical carcinoma, metastatic carcinoma, malignant melanoma, epithelioid sarcoma-like hemangioendothelioma, and so forth. In our cases, the first differential diagnosis was adrenal cortical carcinoma. Both have atypical large cells with abundant eosinophilic cytoplasm, enlarged nuclei, and prominent nucleoli. Immunohistologically, adrenal cortical carcinoma has been ruled out by negativity for adrenal cortical differentiation markers (Melan-A and calretinin) and positivity for vascular differentiation markers (CD31, CD34, Fli-1, and ERG) which can serve as ancillary tools in confirmation of AEA [13-15]. AEA may also be mistaken as metastatic carcinoma [4,5,8,16-18]. Therefore, an appropriate immunohistochemical panel of stains is crucial in making the correct diagnosis. Considering the variability of staining, more than one endothelial marker may be necessary to support a diagnosis. Metastatic carcinoma is negative for all of these endothelial markers. Most importantly, there were no primary lesions in our cases. Misleadingly, AEA may also be considered as malignant melanocytoma. However, anastomosing vascular channels are always seen in the former but the latter is absent. The former is positive for CD31, CD34, ERG, or Fli-1 and negative for S-100 and HMB45. The latter is opposite, as presented in Table 2. Additionally, ESLH is a distinctive indolent vascular neoplasm characterized by predilection for young adults, multifocal involvement of soft tissue in extremities, epithelioid cytomorphology, and co-expression of keratins/endothelial markers [19]. Mitoses of ESLH are always less than 5/50HPF, while AEA has an aggressive clinical course, prominent atypical cells, and mitoses. Diffuse FOSB expression may be helpful in distinguishing ESLH from epithelioid angiosarcoma [19].
However, the pathogenetic mechanism of PAEA has not been fully clarified. We only found three studies related to pathogenetic mechanism. Exposure to arsenicals has been descried to be associated with PAEA by Livaditou A et al. in 1991 [20]. In 2014, Criscuolo M et al. [5] reported one case of a vinyl chloride-exposed worker with PAEA, indicating that vinyl chloride may be a predisposing factor of PAEA. In our two cases, there was no history of arsenicals or vinyl chloride exposure potentially associated with development of angiosarcoma, neither in the other 12 cases presented in Table 2. Cornejo KM et al. [2] reported three cases that were positive for MYC protein expression by IHC in PAEA in 2015, of which only one cases was found to contain polysomy of chromosome 8 [2] and found to be associated with a chromosome 8/MYC gene abnormality. It implied that the pathogenetic mechanism of PAEA may be related to chromosome 8/MYC gene amplification or rearrangement.
PAEA is an aggressive neoplasm with poor prognosis. 5-year overall survival ranges from only 24% to 31% [1]. Three of the 15 patients were revealed to have adhesions of the adrenal mass to adjacent organs [5,8], in operating or through CT scan. Six of 13 reported patients were confirmed with metastases to distant organs including lung, bone, vertebral and costal, brain, and liver [1,2,5,8] and only one patient was alive at 19-month follow up [2]. However, there is no current uniform treatment but the main method is surgical resection. Optimal adjuvant chemotherapy and radiotherapies have not yet been clearly identified. Four of 13 reported patients were treated with chemotherapy such as adriamycin/ifosfamide, anthracyclines, or paclitaxel. One patient was administered four cycles of adriamycin/ifosfamide and remained alive with no sign of recurrence at 2-year follow up [10]. The remaining three patients died of neoplastic cachexia at 7 months [5], 3 months [2], and 1.5 years [1], respectively. Four patients without chemotherapy achieved complete tumor resection, of which two died of the disease at 5- and 16-months and one was alive with the disease at 19-month follow up [2]. Of note, two patients that received chemotherapy and radiotherapy died of neoplastic cachexia at 7 months and 3 months [2,5]. Furthermore, our two patients achieved complete tumor resection without chemotherapy orradiotherapy. One was healthy at 3 years and one died at 1 month follow up. Based on analysis, there seems to be no significant difference in clinical outcome between patients with or without chemotherapy/radiotherapy after surgery.
In conclusion, we present two unusual cases of PEA with epithelioid morphology, with case 2 accompanied with reactive adrenal cortex hyperplasia and showing aggressive clinical course. PAEA may be easily misdiagnosed given its rarity, morphology, and immunohistochemical expression of CK. More case research is necessary to better understand biological characteristics, clinical outcomes, and the pathogenetic mechanism. Close follow up is equally important. Of note, early detection, accurate diagnosis, and complete tumor resection is crucial for patient prognosis.
Disclosure of conflict of interest
None.
References
- 1.Takizawa K, Kohashi K, Negishi T, Taguchi K, Yamada Y, Nakamura M, Oda Y. A exceptional collision tumor of primary adrenal angiosarcoma and non-functioning adrenocortical adenoma. Pathol Res Pract. 2017;213:702–705. doi: 10.1016/j.prp.2017.04.017. [DOI] [PubMed] [Google Scholar]
- 2.Cornejo KM, Hutchinson L, Cyr MS, Nose V, McLaughlin PJ, Iafrate AJ, Sadow PM. MYC analysis by fluorescent in situ hybridization and immunohistochemistry in primary adrenal angiosarcoma (PAA): a series of four cases. Endocr Pathol. 2015;26:334–341. doi: 10.1007/s12022-015-9385-4. [DOI] [PubMed] [Google Scholar]
- 3.Hayashi T, Gucer H, Mete O. A mimic of sarcomatoid adrenal cortical carcinoma: epithelioid angiosarcoma occurring in adrenal cortical adenoma. Endocr Pathol. 2014;25:404–409. doi: 10.1007/s12022-014-9330-y. [DOI] [PubMed] [Google Scholar]
- 4.Hendry S, Forrest C. Epithelioid angiosarcoma arising in an adrenal cortical adenoma: a case report and review of the literature. Int J Surg Pathol. 2014;22:744–748. doi: 10.1177/1066896914532541. [DOI] [PubMed] [Google Scholar]
- 5.Criscuolo M, Valerio J, Gianicolo ME, Gianicolo EA, Portaluri M. A vinyl chloride-exposed worker with an adrenal gland angiosarcoma: a case report. Ind Health. 2014;52:66–70. doi: 10.2486/indhealth.2013-0044. [DOI] [PMC free article] [PubMed] [Google Scholar]
- 6.Kedzierski L, Hawrot-Kawecka A, Holecki M, Dulawa J. Angiosarcoma of the adrenal gland. Pol Arch Med Wewn. 2013;123:502–503. [PubMed] [Google Scholar]
- 7.Sebastiano C, Zhao X, Deng FM, Das K. Cystic lesions of the adrenal gland: our experience over the last 20 years. Hum Pathol. 2013;44:1797–1803. doi: 10.1016/j.humpath.2013.02.002. [DOI] [PubMed] [Google Scholar]
- 8.Sung JY, Ahn S, Kim SJ, Park YS, Choi YL. Angiosarcoma arising within a long-standing cystic lesion of the adrenal gland: a case report. J. Clin. Oncol. 2013;31:e132–136. doi: 10.1200/JCO.2012.44.0800. [DOI] [PubMed] [Google Scholar]
- 9.Derlin T, Clauditz TS, Habermann CR. Adrenal epithelioid angiosarcoma metastatic to the epicardium: diagnosis by 18F-FDG PET/CT. Clin Nucl Med. 2012;37:914–915. doi: 10.1097/RLU.0b013e318262af6b. [DOI] [PubMed] [Google Scholar]
- 10.Lepoutre-Lussey C, Rousseau A, Al Ghuzlan A, Amar L, Hignette C, Cioffi A, Zinzindohoue F, Leboulleux S, Plouin PF. Primary adrenal angiosarcoma and functioning adrenocortical adenoma: an exceptional combined tumor. Eur J Endocrinol. 2012;166:131–135. doi: 10.1530/EJE-11-0791. [DOI] [PubMed] [Google Scholar]
- 11.Schreiner AM, Hoda RS. Primary adrenal epithelioid angiosarcoma showing rhabdoid morphology on air-dried smears. Diagn Cytopathol. 2012;40(Suppl 2):E162–164. doi: 10.1002/dc.21690. [DOI] [PubMed] [Google Scholar]
- 12.Kareti LR, Katlein S, Siew S, Blauvelt A. Angiosarcoma of the adrenal gland. Arch Pathol Lab Med. 1988;112:1163–1165. [PubMed] [Google Scholar]
- 13.Zambo I, Vesely K. WHO classification of tumours of soft tissue and bone 2013: the main changes compared to the 3rd edition. Cesk Patol. 2014;50:64–70. [PubMed] [Google Scholar]
- 14.Jo VY, Fletcher CD. WHO classification of soft tissue tumours: an update based on the 2013 (4th) edition. Pathology. 2014;46:95–104. doi: 10.1097/PAT.0000000000000050. [DOI] [PubMed] [Google Scholar]
- 15.Carstens HB, Ghadially FN, Henderson DW, Stirling JW. Case for the panel. Weibel-Palade body-like lamellar structure in angiosarcoma. Ultrastruct Pathol. 1995;19:137–143. [PubMed] [Google Scholar]
- 16.Ben-Izhak O, Auslander L, Rabinson S, Lichtig C, Sternberg A. Epithelioid angiosarcoma of the adrenal gland with cytokeratin expression. Report of a case with accompanying mesenteric fibromatosis. Cancer. 1992;69:1808–1812. doi: 10.1002/1097-0142(19920401)69:7<1808::aid-cncr2820690724>3.0.co;2-e. [DOI] [PubMed] [Google Scholar]
- 17.Croitoru AG, Klausner AP, McWilliams G, Unger PD. Primary epithelioid angiosarcoma of the adrenal gland. Ann Diagn Pathol. 2001;5:300–303. doi: 10.1053/adpa.2001.27917. [DOI] [PubMed] [Google Scholar]
- 18.Wenig BM, Abbondanzo SL, Heffess CS. Epithelioid angiosarcoma of the adrenal glands. A clinicopathologic study of nine cases with a discussion of the implications of finding “epithelial-specific” markers. Am J Surg Pathol. 1994;18:62–73. [PubMed] [Google Scholar]
- 19.Hung YP, Fletcher CD, Hornick JL. FOSB is a useful diagnostic marker for pseudomyogenic hemangioendothelioma. Am J Surg Pathol. 2017;41:596–606. doi: 10.1097/PAS.0000000000000795. [DOI] [PubMed] [Google Scholar]
- 20.Livaditou A, Alexiou G, Floros D, Filippidis T, Dosios T, Bays D. Epithelioid angiosarcoma of the adrenal gland associated with chronic arsenical intoxication? Pathol Res Pract. 1991;187:284–289. doi: 10.1016/S0344-0338(11)80785-5. [DOI] [PubMed] [Google Scholar]