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Ko M, Kamimura K, Sakamaki A, Niwa Y, Tominaga K, Mizuno K, Terai S. Rare Mesenteric Arterial Diseases: Fibromuscular Dysplasia and Segmental Arterial Mediolysis and Literature Review. Intern Med 2019; 58:3393-3400. [PMID: 31327836 PMCID: PMC6928505 DOI: 10.2169/internalmedicine.3094-19] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/26/2019] [Accepted: 06/03/2019] [Indexed: 12/29/2022] Open
Abstract
Fibromuscular dysplasia (FMD) and segmental arterial mediolysis (SAM) are noninflammatory, nonatherosclerotic arterial diseases that cause aneurysm, occlusion, and thromboses. These diseases are rarely seen in mesenteric arterial lesions; however, as they can be lethal if appropriate management is not provided, the accumulation of clinical information from cases is essential. We herein report the cases of a 57-year-old man diagnosed with FMD and a 63-year-old man diagnosed with SAM. We conclude that an early diagnosis with imaging modalities and clinical information followed by the appropriate treatment improves the prognosis of these arterial diseases.
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Affiliation(s)
- Masayoshi Ko
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Japan
| | - Kenya Kamimura
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Japan
| | - Akira Sakamaki
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Japan
| | - Yusuke Niwa
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Japan
| | - Kentaro Tominaga
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Japan
| | - Kenichi Mizuno
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Japan
| | - Shuji Terai
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Japan
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Ko M, Kamimura K, Ogawa K, Tominaga K, Sakamaki A, Kamimura H, Abe S, Mizuno K, Terai S. Diagnosis and management of fibromuscular dysplasia and segmental arterial mediolysis in gastroenterology field: A mini-review. World J Gastroenterol 2018; 24:3637-3649. [PMID: 30166859 PMCID: PMC6113722 DOI: 10.3748/wjg.v24.i32.3637] [Citation(s) in RCA: 14] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/25/2018] [Revised: 06/17/2018] [Accepted: 06/25/2018] [Indexed: 02/06/2023] Open
Abstract
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.
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Affiliation(s)
- Masayoshi Ko
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
| | - Kenya Kamimura
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
| | - Kohei Ogawa
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
| | - Kentaro Tominaga
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
| | - Akira Sakamaki
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
| | - Hiroteru Kamimura
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
| | - Satoshi Abe
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
| | - Kenichi Mizuno
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
| | - Shuji Terai
- Division of Gastroenterology and Hepatology, Graduate School of Medical and Dental Sciences, Niigata University, Niigata 9518510, Japan
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Mitchell A, Caty V, Bendavid Y. Massive mesenteric panniculitis due to fibromuscular dysplasia of the inferior mesenteric artery: a case report. BMC Gastroenterol 2015; 15:71. [PMID: 26100669 PMCID: PMC4477478 DOI: 10.1186/s12876-015-0303-5] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/31/2015] [Accepted: 06/17/2015] [Indexed: 02/01/2023] Open
Abstract
Background Fibromuscular dysplasia (FMD) is a nonatheromatous, noninflammatory arterial disorder of unknown etiology resulting in vessel stenosis and/or aneurysm formation. The renal and cephalocervical (mainly carotid arteries) arterial beds are classically involved; involvement of visceral arteries is rare. Mesenteric panniculitis (MP) is an inflammatory process of mesenteric fat considered to be of unknown etiology. The majority of cases involve the small bowel mesentery; colorectal MP is rare. To our knowledge, no example of MP due to FMD has been described. Case presentation A 52 year old man presented with steadily worsening lower abdominal pain. Investigation revealed ischemic rectosigmoid mucosa associated with a large mesenteric mass of unknown nature. Angiography showed the disease was limited to the distribution of the inferior mesenteric artery. Subsequent symptoms of large bowel obstruction necessitated a left hemicolectomy. Pathologic examination showed bowel wall necrosis and massive panniculitis of the rectosigmoid due to FMD. Subsequent angiographic imaging of other vascular beds was negative. Conclusions Several features of this case are noteworthy: FMD limited to the inferior mesenteric artery has not been previously reported, FMD has not previously been implicated as a cause of MP, and the massive extent of panniculitis. An accompanying literature review of cases of visceral FMD, traditionally believed to almost exclusively affect females, highlights a greater than anticipated number of males (33 %), and a gender difference regarding concomitant involvement of cephalocervical and/or renal vascular beds (32 % in males versus 80 % in females). The latter observation may have implications regarding the value of radiologic screening of other vascular beds, particularly in asymptomatic males, in patients presenting with visceral artery FMD. Electronic supplementary material The online version of this article (doi:10.1186/s12876-015-0303-5) contains supplementary material, which is available to authorized users.
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Affiliation(s)
- Andrew Mitchell
- Departments of Anatomic Pathology and Cytology, Maisonneuve-Rosemont Hospital, 5415 Boulevard de L'Assomption, Montreal, QC, H1T 2M4, Canada.
| | - Véronique Caty
- Department of Radiology, Maisonneuve-Rosemont Hospital, 5415 Boulevard de L'Assomption, Montreal, QC, H1T 2M4, Canada.
| | - Yves Bendavid
- Department of Surgery, Maisonneuve-Rosemont Hospital, 5415 Boulevard de L'Assomption, Montreal, QC, H1T 2M4, Canada.
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Ellis JA, D'Amico R, Altschul D, Leung R, Connolly ES, Meyers PM. Medial lenticulostriate artery aneurysm presenting with isolated intraventricular hemorrhage. Surg Neurol Int 2011; 2:92. [PMID: 21748044 PMCID: PMC3130466 DOI: 10.4103/2152-7806.82374] [Citation(s) in RCA: 18] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/03/2011] [Accepted: 06/01/2011] [Indexed: 11/07/2022] Open
Abstract
Background: Isolated intraventricular hemorrhage (IVH) secondary to lenticulostriate artery aneurysm rupture is extremely rare. Thus, the diagnostic imaging modalities and therapeutic interventions utilized in the management of such cases are not clearly defined. Case Description: Here we describe a case of isolated or primary IVH (PIVH) in a 71-year-old woman presenting with severe headache. Emergent catheter cerebral angiography, performed after nondiagnostic computed tomography angiography (CTA), revealed the bleeding source to be a 4 × 2.6 mm distal medial lenticulostriate artery aneurysm that ruptured directly into the lateral ventricle. The poorly accessible location of the aneurysm for both endovascular and direct surgical treatment argued for conservative management. A good clinical outcome was obtained with rapid angiographic resolution of the ruptured aneurysm. Conclusion: Thus, lenticulostriate artery aneurysm rupture must be given diagnostic consideration in cases of isolated IVH. Emergent catheter cerebral angiography should be performed in cases such as this when noninvasive imaging is unrevealing. Conservative management may be a reasonable therapeutic option in patients with this kind of aneurysm, and spontaneous resolution can be observed.
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Affiliation(s)
- Jason A Ellis
- Department of Neurological Surgery, Columbia University Medical Center, NY, USA
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Kaneko K. Cerebrovascular lesion in idiopathic midaortic syndrome in children. Pediatr Nephrol 2009; 24:419. [PMID: 18752005 DOI: 10.1007/s00467-008-0987-7] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/29/2008] [Accepted: 07/30/2008] [Indexed: 11/24/2022]
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Horie T, Seino Y, Miyauchi Y, Saitoh T, Takano T, Ohashi A, Yamada N, Tamura K, Yamanaka N. Unusual petal-like fibromuscular dysplasia as a cause of acute abdomen and circulatory shock. JAPANESE HEART JOURNAL 2002; 43:301-5. [PMID: 12227706 DOI: 10.1536/jhj.43.301] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 12/16/2022]
Abstract
Fibromuscular dysplasia (FMD) is a nonatherosclerotic, noninflammatory segmental arterial occlusive disorder that involves primarily the renal and carotid arteries, and less often the coronary, iliac, and visceral arteries. We report the case of 78-year-old Japanese woman who presented with acute abdomen complicated by shock. Autopsy revealed hemorrhagic necrosis of the small intestine due to severe narrowing of the mesenteric arteries. Histologically, smooth muscles showed in-bundle hyperplasia surrounding the adventitia together with medial and perimedial fibrodysplasia of these arteries, forming the characteristic petal-like appearance of FMD. No occlusive thrombus was observed. Further, another medial fibrodysplasia type of FMD was also seen in the renal and left circumflex coronary arteries. Unusual proliferation of smooth muscles resulted in the petal-like atypical FMD at the superior mesenteric artery.
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Affiliation(s)
- Tsutomu Horie
- First Department of Medicine, Nippon Medical School, Tokyo, Japan
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Ebaugh JL, Chiou AC, Morasch MD, Pearce WH, Yao JS. Staged embolization and operative treatment of multiple visceral aneurysms in a patient with fibromuscular dysplasia--a case report. VASCULAR SURGERY 2001; 35:145-8. [PMID: 11668384 DOI: 10.1177/153857440103500211] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Abstract
The importance of following a prioritized sequential approach to patients with complex multianeurysm disease cannot be overemphasized. The following patient with multiple visceral aneurysms first had coil embolization of bilateral renal artery aneurysms and then operative excision of her remaining splenic artery aneurysms to minimize the potential morbidity of a larger operation. This case also demonstrates the potential for following levels of specific degradative enzymes associated with aneurysmal disease (matrix metalloproteinase-9 (MMP-9) in this case) preoperatively and postoperatively and in long-term follow-up to monitor for disease recurrence.
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Affiliation(s)
- J L Ebaugh
- Division of Vascular Surgery, Department of Surgery, Northwestern University Medical School, Chicago, IL 60611, USA
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Sandmann W, Schulte KM. Multivisceral fibromuscular dysplasia in childhood: case report and review of the literature. Ann Vasc Surg 2000; 14:496-502. [PMID: 10990561 DOI: 10.1007/s100169910095] [Citation(s) in RCA: 21] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/18/2022]
Abstract
We report here a 9-year-old girl with fibromuscular dysplasia of many muscular arteries including both renal and internal carotid arteries, the celiac artery, superior mesenteric artery, and one external carotid artery. She suffered from severe renovascular hypertension with beginning secondary cardiac decompensation, typical angina abdominalis, and neurological signs, including severe headaches and hemianopsia. Surgery was performed for all major vessels and the outcome is good 2.5 years after the operation. The clinical presentation, differential diagnosis, and treatment options of fibromuscular dysplasia in childhood are discussed and the literature is reviewed.
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Affiliation(s)
- W Sandmann
- Clinic for Vascular Surgery, University Clinics, Heinrich-Heine-University, Düsseldorf, Germany
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Warrasak S, Tapaneya-Olarn W, Euswas A, Sriphojanart S, Sirikulchayanonta V, Leelachaikul P. Fibromuscular dysplasia: a rare cause of cilioretinal artery occlusion in childhood. Ophthalmology 2000; 107:737-41. [PMID: 10768337 DOI: 10.1016/s0161-6420(99)00147-5] [Citation(s) in RCA: 13] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/18/2022] Open
Abstract
OBJECTIVE To report a case of cilioretinal artery occlusion with angiographic findings characteristic of the "string of beads" associated with renovascular hypertension secondary to fibromuscular dysplasia of the renal artery in a child. DESIGN Case report. INTERVENTION The patient underwent ex vivo renal artery reconstruction with saphenous vein graft and reimplantation. MAIN OUTCOME MEASURES The main clinical outcomes were control of severe hypertension, reversible hypertensive retinopathy, and improvement of vision. RESULTS Revascularization of the kidney improved renal function, and renovascular hypertension was clearly improved. Visual acuity improved to 20/200. CONCLUSIONS A child with hypertensive retinopathy and arterial occlusion in the retina should undergo investigation to rule out a surgically curable hypertension. Magnetic resonance angiography of extrarenal vessels may reveal other sites of involvement of fibromuscular dysplasia. Evaluation and early diagnosis of renovascular hypertension will prevent severe end-organ damage.
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Affiliation(s)
- S Warrasak
- The Department of Ophthalmology, Ramanthibodi Hospital Faculty, Mahidol University, Bangkok, Thailand.
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