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Hemorrhagic shock due to ruptured lower limb vascular malformation in a neurofibromatosis type 1 patient:A case report

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World Journal of Clinical Cases 2022年第31期10卷 11597-11606页

作者： Li-Ping Shen Gang Jin Rang-Teng Zhu Han-Tao Jiang Department of Clinical Laboratory Taizhou Hospital of Zhejiang Province affiliated to Wenzhou Medical UniversityTaizhou 318000Zhejiang ProvinceChina Department of Clinical Laboratory Enze HospitalTaizhou Enze Medical Center(Group)Taizhou 318000Zhejiang ProvinceChina Department of Orthopedics Taizhou Hospital of Zhejiang Province Affiliated to Wenzhou Medical UniversityTaizhou 318000Zhejiang ProvinceChina Department of Orthopedics Enze HospitalTaizhou Enze Medical Center(Group)Taizhou 318000Zhejiang ProvinceChina

BACKGROUND Neurofibromatosis type 1(NF-1)is a common autosomal dominant genetic *** is characterized by café-au-lait spots and cutaneous *** NF-1 typically involves the skin,nerves,bones,and eyes,vascular manifestation in the form of devastating hemorrhage can occur *** SUMMARY We present the case of a 47-year-old female with NF-1 who had a ruptured right lower limb arterial *** presented with sudden right lower limb swelling for two hours and symptoms of hemorrhagic shock on *** physical examination revealed a right lower limb presenting as elephantiasis and visible dark-brown pigmentation over a large *** tomography angiography showed right lower limb arteriovenous ***,the patient underwent emergency right lower limb digital subtraction angiography(DSA)and vascular embolization after blood ***,after DSA,vascular embolization,and repeated blood transfusions,the anemia and right lower limb swelling and tenderness did not *** a result,the patient underwent right lower extremity above-knee *** amputation,the patient's hemoglobin level improved significantly without blood transfusion,and she was discharged from the hospital after the incision *** pathological examination suggested neurogenic *** other complications had occurred 1-year *** vascular malformation and rupture are fatal complications of *** may not provide complete relief,the patient might need to undergo neurofibroma resection or amputation.

关键词： Neurofibromatosis type 1 vascular malformation Hemorrhagic shock vascular embolization Above-knee amputation Case report

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Small intestinal vascular malformation bleeding:A case report with imaging findings

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World Journal of Gastroenterology 2014年第38期20卷 14076-14078页

作者： Jun Cui Liu-Ye Huang Shu-Juan Lin Long-Zhi Yi Cheng-Rong Wu Bo Zhang Department of Gastroenterology Yan tai Yu Huang Ding Hospital

The small intestine is approximately 5-6 m long and occupies a large area in the abdominal cavity. These factors preclude the use of ordinary endoscopy and X-ray to thoroughly examine the small intestine for bleeding of vascular malformations. Thus,the diagnosis of intestinal bleeding is very difficult. A 47-year-old man presented at the hospital 5 mo ago with dark stool. Several angiomas were detected by oral approach enteroscopy,but no active bleeding was observed. Additionally,no lesions were detected by anal approach enteroscopy;however,gastrointestinal tract bleeding still occurred for an unknown reason. We performed an abdominal vascular enhanced computed tomography examination and detected ileal vascular malformations. Ileum angioma and vascular malformation were detected by a laparoscopic approach,and segmental resection was performed for both lesions,which were confirmed by pathological diagnosis. This report systemically emphasizes the imaging findings of small intestinal vascular malformation bleeding.

关键词： Small intestine vascular malformation Bleeding Imaging Diagnosis

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Venous vascular malformation of the parotid gland

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世界耳鼻咽喉头颈外科杂志英文版 2024年第4期10卷 350-352页

作者： Hakimi Amir A. Wu Eric L. Giurintano Jonathan Department of Otolaryngology-Head and Neck Surgery Medstar Georgetown University Hospital Washington District of Columbia USA

Venous vascular malformations of the parotid gland are very rare vascular tumors, especially among adults. This case report discusses the presentation and challenges of diagnosing a venous vascular malformation of the parotid gland in a patient with previous oncologic history. Our patient is a 45-year-old female with a history of breast cancer, meningioma, and secreting pituitary adenoma who presented with a malformation on magnetic resonance imaging, incidentally demonstrating a T2 intense mass in the superficial lobe of the right parotid gland. She was otherwise asymptomatic. Ultrasound-guided fine needle aspiration was nondiagnostic. She underwent a right inferior superficial parotidectomy. Histologic section analysis demonstrated a well-delineated vascular tumor made of large vascular spaces with thin walls surrounded by a parotid tissue consistent with a venous vascular malformation of the parotid gland. Venous vascular malformations are exceedingly rare, especially among adults. Ultrasound-guided fine needle aspiration can be of limited value. Parotidectomy may be electively considered for diagnostic and therapeutic purposes.

关键词： parotidectomy salivary gland tumor vascular malformation

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Mandibular Embryonal Rhabdomyosarcoma Confused with a vascular malformation: About a Case

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Journal of Biosciences and Medicines 2023年第6期11卷 103-107页

作者： Massilia Bouhmidi Hajar Boudarbala Ayad Ghannam Aziza Elouali Abdeladim Babakhouya Maria Rkain Noufissa Benajiba Mother-Child Hospital CHU Mohamed VI Oujda Morroco

Malignant mesenchymal tumors (MTM) in children represent 5% to 10% of malignant tumors in children. They constitute a heterogeneous group of tumors of various differentiations depending on their supposed tissue of origin. They mainly include tumors of muscular origin, those derived from connective, vascular, nervous, or adipose tissue. Rhabdomyosarcoma (RMS) is the most common mesenchymal tumor in children and adolescents (60% to 70% of them). And it accounts for 5.8% of all malignant solid tumors in children. Almost half of rhabdomyosarcomas occur in the head and neck. The prognosis for this type of tumor is particularly poor. A case of rhabdomyosarcoma in the mandible with extension to the abdominal wall and unilateral testis in a 6-month-old infant is reported with evolution since birth. It is a purplish lesion at the level under the right chin which was initially taken for vascular malformation, evolving very quickly towards a mandibular mass deforming the painful face with inflammatory signs, followed by the appearance of a hard swelling under the skin on the left flank taking on the same aspect of the mandibular mass. This observation illustrates the need to know how to systematically think about tumor causes in the face of atypical aspects and to carry out an anatomopathological examination.

关键词： Rhabdomyosarcoma Mandible Mesenchymal Tissue vascular malformation Abdominal Wall Testis

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Thalidomide for refractory gastrointestinal bleeding from vascular malformations in patients with significant comorbidities

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World Journal of Clinical Cases 2020年第15期8卷 3218-3229页

作者： Alexis Mae Bayudan Chien-Huan Chen Department of Medicine Washington University School of MedicineSt.LouisMO 63108United States Division of Gastroenterology Department of Internal MedicineWashington University School of MedicineSt.LouisMO 63110United States

BACKGROUND Refractory gastrointestinal bleeding(GIB)secondary to gastrointestinal vascular malformations(GIVM)such as gastrointestinal angiodysplasia(GIAD)and gastric antral vascular ectasia(GAVE)remains challenging to treat when endoscopic therapy *** thalidomide has been suggested as a treatment option for refractory *** To determine the outcome of patients treated with thalidomide for refractory GIB due to *** IRB approved,single center,retrospective review of electronic medical records from January 2012 to November *** age>18 years old,who had>3 episodes of GIB refractory to medical or endoscopic therapy,and who had been treated with thalidomide for at least 3 mo were *** primary endpoint was recurrence of GIB 6 mo after initiation of *** Fifteen patients were included in the study,all with significant cardiac,hepatic,or renal *** cause of GIB was GIAD in 10 patients and GAVE in 5 *** patients were lost to follow *** the 13 patients followed,38.5%(n=5)had no recurrent GIB or transfusion requirement after treatment with ***,84.6%(n=11)of patients had a reduction in transfusion requirements and hospitalizations for *** was discontinued in 2 patients due to cost(n=1)and medication interaction(n=1).Reported adverse reactions included fatigue(n=3),neuropathy(n=2),dizziness(n=1),and constipation(n=1).Six patients died during follow up due to unknown cause(n=4)and sepsis(n=2).CONCLUSION Thalidomide appears to be an effective treatment for refractory GIB due to GIAD or GAVE in a Western population with significant comorbidities.

关键词： vascular malformation Thalidomide Refractory gastrointestinal bleeding Gastric antral vascular ectasia Angiodysplasia

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Concentration is more important than dose when using intralesional Pingyangmycin injection for treatment of vascular malformations

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上海口腔医学 2010年第2期19卷 220-221页

作者：周琴郑家伟杨秀娟王延安 Department of Oral and Maxillofacial Surgery Ninth People’s HospitalCollege of StomatologyShanghai Jiao Tong University School of Medicine

vascular anomalies represent a wide spectrum of pathology with different clinico-pathological characteristics. Approximately 60% of them occur in the head and neck region. A variety of therapeutic modalities have been... 详细信息

关键词： vascular malformation Pingyangmycin Sclerotherapy

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Sirolimus treatment for neonate with blue rubber bleb nevus syndrome:A case report

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World Journal of Clinical Cases 2021年第23期9卷 6929-6934页

作者： Si-Si Yang Ming Yang Xiao-Jie Yue Jin-Fa Tou Department of Neonatal Surgery The Children’s HospitalZhejiang University School of MedicineHangzhou 310000Zhejiang ProvinceChina Department of Burn and Plastic Surgery The Children’s HospitalZhejiang University School of MedicineHangzhou 310000Zhejiang ProvinceChina

BACKGROUND Blue rubber bleb naevus syndrome(BRBNS)is a rare disease that usually presents with multiple venous malformations in the skin and gastrointestinal *** located in the gastrointestinal tract always result in chronic gastrointestinal bleeding and severe *** successful management of BRBNS with sirolimus had been reported in many institutions,due to its impact on signaling pathways of ***,the experience in treatment of neonates with BRBNS was *** SUMMARY A 38-day-old premature female infant born with multiple skin lesions,presented to our center complaining of severe anemia and *** examination demonstrated that hemoglobin was 5.3 g/dL and contrast-enhanced abdominal computed tomography showed multiple low-density space-occupying lesions in the right lobe of the *** was diagnosed as having BRBNS based on typical clinical and examination *** patient was treated by transfusions twice and hemostatic drugs but symptoms of anemia were difficult to alleviate.A review of BRBNS case reports found that patients had been successfully treated with *** the patient was treated with sirolimus at an average dose of 0.95 mg/m2/d with a target drug level of 10-15 ng/*** 28 mo of treatment,the lesion was reduced,hemoglobin returned to normal,and there were no adverse drug *** This case highlights the dosing regimen and plasma concentration in neonates,for the current common empiric dose is high.

关键词： Cutaneous hemangioma vascular malformation Sirolimus Rapamycin Blue rubber bleb nevus syndrome Case report

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Hemangioma in the lower labial vestibule of an eleven-year-old girl:A case report

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World Journal of Clinical Cases 2022年第5期10卷 1617-1622页

作者： Ashwag Yagoub Aloyouny Afrah Jaber Alfaifi Shahad Mohammed Aladhyani Ahad Ali Alshalan Hadeel Mohammed Alfayadh Hend Mahmoud Salem Department of Basic Dental Science Princess Nourah Bint Abdulrahman UniversityRiyadh 11671Saudi Arabia Oral Medicine Special Care Dentistry Prince Sultan Military Medical CityRiyadh 12211Saudi Arabia Department of Dentistry Ministry of HealthRiyadh 11176Saudi Arabia

BACKGROUND Hemangioma is a vascular benign tumour of endothelial *** appears commonly in the first decade of life with increases incidence in *** is not common to happen in the oral cavity and it is extremely rare to appear in the labial *** SUMMARY We present a case of an 11-year-old girl who complained of a painful,slowly growing mass which was consistent with the capillary hemangioma in the left mandibular *** tumor such as hemangioma in the mandibular vestibule is extremely rare;hence,the clinical definitive diagnosis is very ***,radiographic imaging and histopathologic analysis are crucial to reach to the final diagnosis for proper *** Comprehensive clinical evaluation,proper diagnostic imaging and microscopic analysis of the mass establish a precise diagnosis of the hemangioma for better management.

关键词： Capillary hemangioma vascular malformation Labial vestibule Childhood Case report

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Easily misdiagnosed complex Klippel-Trenaunay syndrome: A case report

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World Journal of Clinical Cases 2023年第4期11卷 922-930页

作者： Ling-Li Li Rui Xie Fu-Qing Li Cheng Huang Bi-Guang Tuo Hui-Chao Wu Department of Gastroenterology Digestive Disease HospitalAffiliated Hospital of Zunyi Medical UniversityZunyi 563003Guizhou ProvinceChina

BACKGROUND Klippel-Trenaunay syndrome(KTS) is a congenital vascular malformation with a complicated etiology. It is sporadic and clinically rare in occurrence. The typical characteristics are capillary malformation(also known as port-wine stain),varicose veins and malformations, and bony and/or soft tissue hypertrophy with or without lymphatic malformation, which are known as the “classic clinical triad”. Herein, a rare case of KTS characterized by crossed-bilateral limb hypertrophy accompanied by intermittent hematochezia and hematuria is *** SUMMARY We described a 37-year-old female with KTS. She was admitted to our hospital owing to the gradual enlargement of the left lower extremity along with intermittent hematochezia and hematuria. The patient was diagnosed to have hemorrhoid bleeding by other hospitals and treated with conventional hemostatic drugs, but continued to have intermittent gastrointestinal bleeding and hematuria. Therefore, she visited our hospital to seek further treatment. During hospitalization, relevant imaging and laboratory examinations and colonoscopy were performed. In combination with the patient’s history and relevant examinations, we considered that the patient had a complex form of KTS. We recommended a combined diagnosis and treatment from the vascular, interventional,anorectal, and other departments, although she declined any further treatment for financial *** The clinical manifestations of KTS are extensive and diverse and chiefly include the typical triad. However, vascular malformations of KTS can also involve several parts and systems such as digestive and urogenital systems. Therefore, the atypical manifestations and rare complications necessitate the clinician’s attention and are not to be ignored.

关键词： Gross hematuria Hematochezia Klippel-Trenaunay syndrome Limb hypertrophy vascular malformation Case report

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Effective treatment of gastrointestinal bleeding with thalidomide- Chances and limitations

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World Journal of Gastroenterology 2016年第11期22卷 3158-3164页

作者： Juergen Bauditz Department of Internal Medicine Helios Klinik Anhalt/Zerbst Department of Gastroenterology Hepatology and InfectiologyCharité Campus MitteCharité University Hospital

For more than 50 years bleeding from gastrointestinal angiodysplasias has been treated by hormonal therapy with estrogens and progesterons. After a randomized study finally demonstrated that hormones have no effect on bleeding events and transfusion requirements, therapy has switched to endoscopic coagulation. However, angiodysplasias tend to recur over months to years and endoscopy often has to be repeated for long time periods. Thalidomide, which caused severe deformities in newborn children in the 1960 s, is now increasingly used after it was shown to suppress tumor necrosis factor alpha, inhibi t angiogenesis and to be also effective for treatment of multiple myeloma. In 2011 thalidomide was proven to be highly effective for treatment of bleeding from gastrointestinal angiodysplasias in a randomized study. Further evidence by uncontrolled studies exists that thalidomide is also useful for treatment of bleeding in hereditary hemorrhagic telangiectasia. In spite of this data, endoscopic therapy remains the treatment of choice in many hospitals, as thalidomide is still notorious for its teratogenicity. However, patients with gastrointestinal bleeding related to angiodysplasias are generally at an age in which women have no child-bearing potential. Teratogenicity is therefore no issue for these elderly patients. Other side-effects of thalidomide like neurotoxicity may limit treatment options but can be monitored safely.

关键词： Thalidomide Angiodysplasia vascular malformation Gastrointestine Bleeding Therapy Angiogenesis vascular endothelial growth factor

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