Aortic arch aneurysm with dissection in Cushing syndrome

Takagi, Hisato; Furukawa, Midori; Umeda, Yohtaro; Fukumoto, Yukiomi; Yoshida, Naomasa; Yamada, Takuya; Mori, Yoshio

doi:10.1016/j.jtcvs.2003.08.022

Cited by 11 publications

(10 citation statements)

References 5 publications

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“…8,9 In addition, Cushing disease is recognized to be a risk factor for the degeneration of arterial walls with rare cases of aortic dissection and splenic artery and aortic aneurysms reported in the literature. [10][11][12] To our knowledge, however, only one case of a mycotic aortic aneurysm Mycotic Pseudoaneurysm in Cushing Disease in a patient with (iatrogenic) Cushing disease has been reported, and the development of a peripheral mycotic aneurysm in a cushingoid patient has not been previously described. 13 It has been postulated that cortisol excess leads to increased risk of aneurysm formation or dissection via the effect of glucocorticoids on the arterial media.…”

Section: Discussionmentioning

confidence: 98%

“…14,15 Hypercortisolemia has also been suggested to result in the metaplastic transformation of medial smooth cells into fibroblast-like cells, thereby predisposing patients to aneurysm formation. 11 The management of pseudoaneurysms is varied; however, the presence of infection limits the management options available. Although the current consensus is that either ultrasound-guided thrombin injection or ultrasoundguided compression repair can be used to manage the vast majority of uninfected pseudoaneurysms, the presence of infection is a contraindication for thrombin use; therefore, it cannot be used to manage mycotic pseudoaneurysms.…”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Mycotic Pseudoaneurysm of the Superficial Femoral Artery in a Patient with Cushing Disease: Case Report and Literature Review

et al. 2009

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Mycotic pseudoaneurysms of the peripheral arteries are rare and can occur as an extension of localized infection or from systemic sepsis. In some cases, no obvious source of infection may be identified. Both endovascular and open surgical management options are available for this important condition. We report a mycotic pseudoaneurysm of the superficial femoral artery in a patient associated with systemic immunosuppression secondary to Cushing disease that was successfully managed with open surgical repair and autologous bypass grafting. This unusual case highlights the potential for serious adverse cardiovascular sequelae of Cushing disease and the need for awareness of such complications in this patient group.

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Section: Discussionmentioning

confidence: 98%

Section: Discussionmentioning

confidence: 99%

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Mycotic Pseudoaneurysm of the Superficial Femoral Artery in a Patient with Cushing Disease: Case Report and Literature Review

et al. 2009

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“…Although this association may be coincidental, we speculate that they may be secondary to tissue heterogeneity in glucocorticoid sensitivity, as seen in other nuclear hormone resistance syndromes. As seen in cases of Cushing's syndrome presenting with vascular aneurysms, these vascular malformation are thought to be linked to the effect of hypercortisolemia on blood vessels [27]. In mice, injection of hydrocortisone has been showed to lead to induction of ectasia, aneurysms and, in genetically susceptible animals, aortic rupture [28].…”

Section: Discussionmentioning

confidence: 99%

Glucocorticoid resistance syndrome caused by a novel <i>NR3C1</i> point mutation

et al. 2018

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Glucocorticoid resistance syndrome (GRS) is a rare genetic disorder caused by inactivating mutations of the NR3C1 gene which encodes the glucocorticoid receptor. The phenotypic spectrum is broad but typically include symptoms of adrenal insufficiency, mineralocorticoid excess and hyperandrogenism. We report a new case associated with a novel NR3C1 mutation. A 55-year-old woman with lifelong history of low body weight, hyperandrogenism and anxiety was seen at the endocrine clinic after left adrenalectomy and salpingoophorectomy for lesions suspicious of ovarian cancer and adrenal metastasis. The tumors turned out to be a 3.5 cm benign ovarian serous adenofibroma and a 3.5 cm multinodular adrenal mass. She complained of worsened fatigue and inability to recover weight lost with surgery. Pre-operative serum and urinary cortisol were elevated, but she had no stigma of Cushing's syndrome. Plasma ACTH was elevated and a 1-mcg cosyntropin stimulation test was normal. Her fatigue persisted over ensuing years and ACTH-dependent hypercortisolemia remained stable. Low dose oral dexamethasone failed to suppress endogenous cortisol. A pituitary MRI was normal but revealed incidental brain aneurysms. Bone densitometry showed profound osteoporosis. On the bases of this contradictory clinical picture, glucocorticoid resistance syndrome (GRS) was suspected. Using next generation sequencing technology, a novel heterozygous pathogenic variant in the NR3C1 gene was detected. We speculate that vascular malformations and profound osteoporosis, findings associated to cortisol excess, reflect in our patient a variable tissue sensitivity to glucocorticoids. In conclusion, in patients with clinically unexpected ACTH-dependent hypercortisolemia, primary glucocorticoid resistance (GRS) should be considered.

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“…2) Cushing's syndrome is a risk factor for dissecting aortic aneurysm. 5,6,15,16) Excess of plasma cortisol causes dissecting aortic aneurysm in some experimental models, 11,13) but the mechanism is unknown. Progress of arteriosclerosis due to hypertension, hyperlipemia, and diabetes originating in Cushing's disease may be the direct cause of the aortic dissection.…”

Section: Discussionmentioning

confidence: 99%

Vertebral Artery Dissecting Aneurysm With Cushing's Syndrome -Case Report-

Kato

Tanaka

Kuroda

et al. 2010

Neurol. Med. Chir.(Tokyo)

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A 45-year-old woman presented with subarachnoid hemorrhage of World Federation of Neurosurgical Societies grade IV. Cerebral angiography showed a dissecting aneurysm of the right vertebral artery (VA). Internal trapping of the right VA with coils was performed. The postoperative course was uneventful, but she continued to demonstrate moon facies and experience amenorrhea. Computed tomography demonstrated an adrenal tumor. Laparoscopic adrenalectomy was performed under a diagnosis of Cushing's syndrome caused by an adrenal tumor. Overproduction of cortisol caused by Cushing's syndrome may be related to the development of cerebral aneurysm.

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Aortic arch aneurysm with dissection in Cushing syndrome

Cited by 11 publications

References 5 publications

Mycotic Pseudoaneurysm of the Superficial Femoral Artery in a Patient with Cushing Disease: Case Report and Literature Review

Mycotic Pseudoaneurysm of the Superficial Femoral Artery in a Patient with Cushing Disease: Case Report and Literature Review

Glucocorticoid resistance syndrome caused by a novel <i>NR3C1</i> point mutation

Vertebral Artery Dissecting Aneurysm With Cushing's Syndrome -Case Report-

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