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World Journal of Gastroenterology 2018年第32期24卷 3637-3649页

作者： Masayoshi Ko Kenya Kamimura Kohei Ogawa Kentaro Tominaga Akira Sakamaki Hiroteru Kamimura Satoshi Abe Kenichi Mizuno Shuji Terai Division of Gastroenterology and Hepatology Graduate School of Medical and Dental Sciences Niigata University

The vascular diseases including aneurysm, occlusion, and thromboses in the mesenteric lesions could cause severe symptoms and appropriate diagnosis and treatment are essential for managing patients. With the development and improvement of imaging modalities, diagnostic frequency of these vascular diseases in abdominal lesions is increasing even with the small changes in the vasculatures. Among various vascular diseases, fibromuscular dysplasia(FMD) and segmental arterial mediolysis(SAM) are noninflammatory, nonatherosclerotic arterial diseases which need to be diagnosed urgently because these diseases could affect various organs and be lethal if the appropriate management is not provided. However, because FMD and SAM are rare, the cause, prevalence, clinical characteristics including the symptoms, findings in the imaging studies, pathological findings, management, and prognoses have not been systematically summarized. Therefore, there have been neither standard diagnostic criteria nor therapeutic methodologies established, to date. To systematically summarize the information and to compare these disease entities, we have summarized the characteristics of FMD and SAM in the gastroenterological regions by reviewing the cases reported thus far. The information summarized will be helpful for physicians treating these patients in an emergency care unit and for the differential diagnosis of other diseases showing severe abdominal pain.

关键词： fibromuscular dysplasia Segmental arterial mediolysis Mesenteric lesion diagnosis Humans

Optical coherence tomography-guided percutaneous transluminal angioplasty in renal artery fibromuscular dysplasia

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Journal of Geriatric Cardiology 2020年第3期17卷 173-174页

作者： Wai Kin Chi Tan Guang Ming Bryan P Yan Division of Cardiology Department of Medicine and TherapeuticsPrince of Wales Hospitalthe Chinese University of Hong KongHong KongChina

A 40-year-old woman was referred for resistant hypertension despite three anti-hypertensive medications including a diuretic at maximum tolerated *** has no family history of fibromuscular dysplasia(FMD).Contrast-enha... 详细信息

关键词： fibromuscular dysplasia Optical coherence tomography Renal artery stenosis

Liac arterial fibromuscular dysplasia:a systematic review

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中国循环杂志 2018年第S1期33卷 162-163页

作者： Luyun Fan Huimin Zhang Jun Cai National Center for Cardiovascular Diseases Fuwai HospitalChinese Academy of Medical Sciences and Peking Union Medical CollegeBeijingChina

Background liac arterial fibromuscular dysplasia(FMD)were rarely reported and their demographic,clinical and imaging features have not been precisely described resulting in uncertain therapeutic *** A literature review was performed using Pubmed,Web of Science and Embase *** articles in English published since 1990 with full-text and detailed demographic,clinical,imaging and therapeutic information regarding iliac arterial FMD were included.

关键词： fibromuscular dysplasia uncertain therapeutic methods precisely described

Segmental arterial mediolysis: A clinical-pathologic review, its role in fibromuscular dysplasia and description and differential diagnosis of the masquerader-muscular artery cystic necrosis

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World Journal of Cardiovascular Diseases 2013年第1期3卷 64-81页

作者： Richard E. Slavin Cascade Pathology Group Department of Pathology Emanuel Hospital and Health Center Portland USA

Segmental arterial mediolysis (SAM) is an uncommon non-inflammatory iatrogenic arteriopathy caused by alpha-1 adrenergic agonists or Beta-2 agonists able to release norepinephrine from the peripheral nervous system. Causative agents include adrenergic agonists used to control blood pressure, B-2 tocolytic agonists, and ractopamine used as a repartitioning agent in animal husbandry. The liberated norepinephrine both injures and stimulates a robust reparative response in the muscular arteries in the abdomen, brain base, and coronary arteries. This response may be augmented by endothelin-1 formed in the arterial adventitia. Three types of arterial lesions develop in the injurious stage: 1) apoptotic induced mediolysis, 2) separation of the outer media from the adventitia and 3) the formation of arterial gaps. The latter enlarge, particularly in elderly patients, to form gap-aneurysms complicated by dissections and dissecting an- eurysms that when ruptured cause the calamitous hemorrhages that clinically announce SAM. The other types of injury remain clinically silent but with repair develop sequelae and can metamorphose into fibromuscular dysplasia. The sequelae are mainly asymptomatic but may cause arterial stenosis and ischemic lesions. The definitive diagnosis of SAM re- quires histological conformation but misinterpreta- tion of smooth muscle vacuolar change has caused di- agnostic errors. Muscular artery cystic necrosis a newly named non-inflammatory muscular artery ar- teriopathy may be confused with SAM both clinically and pathologically. This arteriopathy represents the muscular artery equivalent of cystic media necrosis of the elastic arteries since it exhibits similar morphol- ogic features and can occur concomitantly with this entity. Adrenergic agents to counter hemorrhagic shock in SAM are contraindicated since they may intensify injury and create new lesions. The use of norepinehrine antagonists introduces a new, but as yet untested, treatment option for

关键词： Segmental Arterial Mediolysis Norepinephrine Adrenergic Agonists Ractopamine Arterial Apoptosis Aneurysms Dissecting Hematomas Arterial fibromuscular Dysplasia Muscular Artery Cystic Necrosis

Spontaneous Bilateral Carotid Artery Dissection Revealing Multifocal fibromuscular Dysplasia

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Case Reports in Clinical Medicine 2019年第9期8卷 250-257页

作者： W. Bnouhanna W. Fakir M. Rahmani M. Benabdeljlil S. Aï di Department of Neurology A and Neuropsychology Mohamed V University H&ocirc pital des Spécialités Ibn Sina University Hospital Rabat Morocco

Background: fibromuscular dysplasia (FMD) is a rare idiopathic segmental non-arteriosclerotic and non-inflammatory arterial disease of small to medium-sized vessels. Although it is considered a benign entity, FMD can lead to potentially severe cerebrovascular complications. We present an atypical clinical case of ischemic stroke (IS) caused by bilateral carotid dissection revealing multifocal FMD. Case Report: A 43-year-old right-handed female with no medical history suddenly developed somnolence with left-sided weakness. Clinical examination revealed left hemiplegia and left central facial paralysis with an NIHSS score: 22. Cerebral tomography (CT) with arterial angiogram revealed abilateralis of the middle cerebral arteries (MCA), a double dissection of the internal carotids (IC), aneurysmal ectasia of the left IC, and aneurysm of the M2 segment of the MCA. Brain magnetic resonance imaging (MRI) with arterial angiography confirmed the bilateral dissection. A cerebral angiography (CA) revealed a radiological pattern of multifocal FMD. The angiography of the aorta and its branches showed adiffuse dysplasia of the renal arteries. The patient was put under antiaggregants. The modified Rankin score (mRS) after three months was three. Conclusion: Cerebrovascular FMD has typical angiographic features, the “string of beads” appearance being the most common. Although the risk of a stroke in patients with cervical FMD is low, it can lead to a severe stroke and the long-term risks are unknown. Patient registries should be encouraged to better understand the mechanisms and evolutionary profile of FMD and alert clinicians to this potentially disabling condition.

关键词： fibromuscular Dysplasia Carotid Disease Stroke

Segmental arterial mediolysis, reparative phase: An analysis and case report showing conversion to fibromuscular dysplasia with renal infarction

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World Journal of Cardiovascular Diseases 2014年第2期4卷 50-60页

作者： Richard E. Slavin Julian del Cerro Gonzalez Jose Manuel Machin Angel Robles Rita Maria Regojo Marie Luisa Diez Department of Internal Medicine La Paz University Hospital Madrid Spain Department of Internal Medicine University Hospital Guadalajara Spain Department of Pathology Legacy Emanuel Hospital Portland Oregon USA Department of Pathology University Hospital Guadalajara Spain Family Practice Physician Guadalajara Health Center Cervantes Branch Guadalajara Spain Interventional Radiologist University Hospital Guadalajara Spain

Segmental arterial mediolysis (SAM), an uncommon arteriopathy putatively caused by norepinephrine released by alpha-1 adrenergic agonists or some Beta-2 agonists capable of releasing norepinephrine from the peripheral sympathetic nervous system potentially can present ischemic and organ injury symptoms caused by sequelae created in its reparative phase in lieu of catastrophic hemorrhages announced in its injurious phase. The case documents this presentation—the patient presenting renal infarcts and ischemic lesions causing abdominal angina, hypertension and a nephrectomy event developing 10 years after prolonged ritodrine treatment for premature labor. This agent may have directly caused SAM or sensitized the patient to agonists causing SAM encountered at a latter date. A variety of lesions derived from injurious phase arterial injuries characterize reparative phase SAM. All were encountered in a hilar branch of the resected renal artery. These included side-by-side sequela aneurysms grossly forming a large fusiform aneurysm, granulation tissue filling adventitial medial tear spaces in which a dissecting hematomas developed, medial muscle loss centered to the outer media repaired with fibrous tissue, arterial stenosis created by reparative intimal plaques, and arterial thrombo-embolism. These lesions were mirrored in accompanying radiologic studies. The accompanying renal vein exhibited changes consistent with repair of the spastic venous angiopathy that often accompanies abdominal SAM. This angiopathy, putatively induced by Endothelin-1, suggested that this agent played a role in the genesis of the arterial lesions. Angiographic resolution of non-treated sequelae occurred in 5 months either spontaneously or due to treatment with bosentem. Conclusions: The histologic and angiographic changes demonstrate that the clinical onset of reparative SAM may be significantly delayed to produce ischemic lesions, renal infarction and in this case report, medial fibromuscular dy

关键词： Segmental Arterial Mediolysis fibromuscular Dysplasia Ritodrine Renal Infarcts Dissecting Hematoma Intestinal Angina Endothelin-1 Bosentem

An Unusual Case of Stroke: A Case Report and Review of Current Literature

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Open Journal of Internal Medicine 2015年第2期5卷 5-10页

作者： Alistair Mackett Saugata Das Muhibbur Chowdhury Peter Phillips Ipswich Hospital Ipswich UK

fibromuscular dysplasia (FMD) is a non-atherosclerotic, non-inflammatory vascular disease. FMD once considered rare, is increasingly a condition recognised as an incidental finding. It is an important cause of stroke especially in younger, female patients and therefore a high index of suspicion is required. FMD has distinct pathological findings along with some pathognomonic angiographic findings (“string of beads” appearance). The mainstay of cerebrovascular FMD treatment is antiplatelet therapy but intervention with percutaneous transluminal angioplasty is indicated under some circumstances. We present an interesting case of a woman in her 60 s who underwent extensive investigation to identify the underlying aetiology of her stroke.

关键词： fibromuscular Displasia Stroke

Two Unusual Cases of Sudden Death in Children

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Open Journal of Pathology 2013年第2期3卷 69-72页

作者： Maria-Chiara Osterheld Marie-Hélène Perez Institute of Pathology University of Lausanne Lausanne Switzerland

We report two unusual cases of sudden unexpected death in children. Histopathologic examination showed intimal fibroplasias, a variant of fibromuscular dysplasia of the right coronary artery associated in both cases with fatty infiltration of the right ventricular myocardium. The significance of this particular combination of two lesions known to induce a sudden death in young people is discussed.

关键词： Arrhythmogenic Right Ventricular Dysplasia-Like Cardiomyopathy fibromuscular Coronary Artery Dysplasia Sudden Death