• Archives of Plastic Surgery
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• v.46 no.1
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• pp.23-33
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• 2019

Background Venous malformations (VMs) are a common type of vascular malformation. However, their causes and management remain unclear, and few studies specific to VMs of the head and neck have been reported. This study describes our experiences with VMs of the head and neck. Methods This retrospective study included 82 patients who underwent treatment for head and neck VMs, among 222 who visited our vascular anomalies center. Medical records between 2003 and 2016 were reviewed to identify common features in the diagnosis and treatment. The diagnosis of suspected head and neck VMs was based on the results of imaging studies or biopsies, and the VMs were analyzed based on magnetic resonance imaging, computed tomography, and Doppler sonography findings. Results VMs were slightly more common in female patients (59.8%), and 45.1% of patients developed initial symptoms at the age of 10 or younger. Lesions were slightly more common on the right side (47.3%). The main sites involved were the cheek (27.7%) and lip area (25.5%). The muscle layer was commonly involved, in 98.7% of cases. Small lesions less than 5 cm in diameter accounted for 60.8% of cases, and well-defined types were slightly more prevalent at 55.4%. Improvement was observed in 77.1% of treated patients. Conclusions Early and accurate diagnosis and appropriate treatment according to individual symptoms are important for successful treatment of VMs. If treatment is delayed, the lesions can worsen, or recurrence becomes more likely. Therefore, VMs require a multidisciplinary approach for early and accurate diagnosis.

• Archives of Plastic Surgery
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• v.49 no.1
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• pp.115-120
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• 2022

Background In addition to vascular endothelial cells, vascular smooth muscle cells (VSMCs) are subject to continuous shear stress because of blood circulation. The angiogenic properties of VSMCs in extracranial arteriovenous malformations (AVMs) may exceed those of normal blood vessels if the body responds more sensitively to mechanical stimuli. This study was performed to investigate the hypothesis that rapid angiogenesis may be achieved by mechanical shear stress. Methods VSMCs were obtained from six patients who had AVMs and six normal controls. The target genes were set to angiopoietin-2 (AGP2), aquaporin-1 (AQP1), and transforming growth factor-beta receptor 1 (TGFBR1). Reverse-transcriptase polymerase chain reaction (RT-PCR) and real-time PCR were implemented to identify the expression levels for target genes. Immunofluorescence was also conducted. Results Under the shear stress condition, mean relative quantity values of AGP2, AQP1, and TGFBR1 in AVM tissues were 1.927±0.528, 1.291±0.031, and 2.284±1.461 when compared with neutral conditions. The expression levels of all three genes in AVMs were higher than those in normal tissue except for AQP1 under shear stress conditions. Immunofluorescence also revealed increased staining of shear stress-induced genes in the normal tissue and in AVM tissue. Conclusions Shear stress made the VSMCs of AVMs more sensitive. Although the pathogenesis of AVMs remains unclear, our study showed that biomechanical stimulation imposed by shear stress may aggravate angiogenesis in AVMs.

• Clinical and Experimental Pediatrics
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• v.64 no.11
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• pp.559-572
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• 2021

The International Society for the Study of Vascular Anomalies classifies vascular anomalies into vascular tumors and vascular malformations. Vascular tumors are neoplasms of endothelial cells, among which infantile hemangiomas (IHs) are the most common, occurring in 5%-10% of infants. Glucose transporter-1 protein expression in IHs differs from that of other vascular tumors or vascular malformations. IHs are not present at birth but are usually diagnosed at 1 week to 1 month of age, rapidly proliferate between 1 and 3 months of age, mostly complete proliferation by 5 months of age, and then slowly involute to the adipose or fibrous tissue. Approximately 10% of IH cases require early treatment. The 2019 American Academy of Pediatrics clinical practice guideline for the management of IHs recommends that primary care clinicians frequently monitor infants with IHs, educate the parents about the clinical course, and refer infants with high-risk IH to IH specialists ideally at 1 month of age. High-risk IHs include those with life-threatening complications, functional impairment, ulceration, associated structural anomalies, or disfigurement. In Korea, IHs are usually treated by pediatric hematology-oncologists with the cooperation of pediatric cardiologists, radiologists, dermatologists, and plastic surgeons. Oral propranolol, a nonselective beta-adrenergic antagonist, is the first-line treatment for IHs at a dosage of 2-3 mg/kg/day divided into 2 daily doses maintained for at least 6 months and often continuing until 12 months of age. Topical timolol maleate solution, a topical nonselective beta-blocker, may be used for small superficial type IHs at a dosage of 1-2 drops of 0.5% gel-forming ophthalmic solution applied twice daily. Pulse-dye laser therapy or surgery is useful for the treatment of residual skin changes after IH involution.

• Maxillofacial Plastic and Reconstructive Surgery
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• v.34 no.6
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• pp.488-493
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• 2012

Hemangioma and vascular malformation are the most common benign tumors that are caused by congenitally or traumatic events. Theses tumors represent approximately 1/3~1/4 of all hemangiomas and vascular malformations in the head and neck. There are many forms of treatment for hemangioma and vascular malformation including closed observation, surgery, radiotherapy, laser therapy, steroid therapy, compression, embolization, and sclerotherapy. Ethanolamine oleate is an unsaturated fatty acid salt that has been used as a sclerosing agent because of its excellent thrombosing properties. This paper presents 1 case of intraoral multiple venous malformations treatment with 1.25% ethanolamine oleate (3.6~9.6 mg dose) intralesionally injected for 6 to 14 weeks over 2 week intervals. After the sclerotherapy, lesions almost completely disappeared without side effects. In conclusion, sclerotherapy using ethanolamine oleate is very effective against venous malformations, and sufficiently provides alternative support for surgical and other methods.

• Clinical and Experimental Pediatrics
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• v.60 no.1
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• pp.1-9
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• 2017

Malformations of cortical development are rare congenital anomalies of the cerebral cortex, wherein patients present with intractable epilepsy and various degrees of developmental delay. Cases show a spectrum of anomalous cortical formations with diverse anatomic and morphological abnormalities, a variety of genetic causes, and different clinical presentations. Brain magnetic resonance imaging has been of great help in determining the exact morphologies of cortical malformations. The hypothetical mechanisms of malformation include interruptions during the formation of cerebral cortex in the form of viral infection, genetic causes, and vascular events. Recent remarkable developments in genetic analysis methods have improved our understanding of these pathological mechanisms. The present review will discuss normal cortical development, the current proposed malformation classifications, and the diagnostic approach for malformations of cortical development.

• Pediatric Gastroenterology, Hepatology & Nutrition
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• v.23 no.1
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• pp.115-120
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• 2020

Klippel-Trenaunay syndrome (KTS) is a rare disorder characterized by a triad of abnormal bone and soft tissue growth, the presence of a port-wine stain, and venous malformations. Gastrointestinal (GI) manifestations of KTS are relatively common and generally do not cause significant problems. However, persistence can lead to chronic GI blood loss or even massive bleeding in rare cases. The majority of the severe GI manifestations associated with KTS present as vascular malformations around the GI tract and exposed vessels can lead to serious bleeding into the GI tract. Herein, we report a case of a 16-year-old boy with severe iron deficiency anemia who was previously misdiagnosed as hemorrhoid due to small amount of chronic bleeding. The actual cause of chronic GI bleeding was from an uncommon GI manifestation of KTS as rectal polyposis.

• Journal of Korean Neurosurgical Society
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• v.38 no.3
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• pp.238-241
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• 2005

The main causes of pineal apoplexy are hemorrhage associated with pineal region tumors, vascular malformations, and pineal cysts. Cavernous malformations rarely occur in the pineal region, with only fifteen cases reported previously. Hemorrhage associated with cavernous malformation causes apoplectic event in the pineal region. We report two surgically treated cases of pineal hemorrhage associated with cavernous malformation and discuss the consideration in management of the pineal apoplexy.

• The Journal of the Korean bone and joint tumor society
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• v.17 no.2
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• pp.100-105
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• 2011

Vascular malformations may typically present with palpable mass that can be either asymptomatic or can present with symptoms including swelling and pain. On rare occasions, vascular malformation of muscle may produce joint deformities caused by contracture of the involved muscle. When vascular malformation involves the flexor muscle of the leg, ankle equinus deformity may occur. However, there are no reports of toe deformities secondary to intermuscular or intramuscular vascular malformations of flexor muscles of toe. Thus, we report a case of vascular malformation of flexor hallucis longus muscle with flexion contracture of toes in a 40-years-old woman who was treated with surgical excision.

• Journal of Chest Surgery
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• v.29 no.7
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• pp.802-806
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• 1996

Arteriovenous malformations are vascular anomalies containing a communication between artery and vein without an intervening capillary bed and also are the most dangerous of vascular malformations being hemodynamically active. Treatment must be careful usually limited and considered in the phase of activity of hemodynamics. The patient was 29-year-old female and had no specific signs and symptoms except buldging, pal- pable mass on the right posterolateral chest wall from several years ago and it was gradually growing from that time. The operation was done with ligation of the right 9th, 1 Oth intercostal arteries and dissection from other normal tissues and then excised the arteriovenous malformation mass and its feeding vessels. The pathologic result was arteriovenous malformation.

• The Journal of the Korean bone and joint tumor society
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• v.14 no.1
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• pp.56-61
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• 2008

More than half of the reported cases of congenital arteriovenous malformations involve the extremities. However, these are predominantly arteriovenous malformations of soft tissues. There are few studies on intraosseous arteriovenous malformations. The clinical features of vascular malformations are not well defined, but are likely to be extremely diverse. So, it is not easy to diagnose exactly and treat intraosseous arteriovenous malformations. The authors noted intraosseous arteriovenous malformation of tibia in a child and had a good result by the use of ethanol embolization. Therefore we include those results along with the literature review.