Combined capillary hemangioma and angiodysplasia of the ileum: an unusual cause of obscure gastrointestinal bleeding with preoperative localization by double-balloon endoscopy

Vascular lesions in the small bowel with bleeding complications are usually difficult to diagnose and localize preoperatively. With the increased use of either capsule endoscopy or enteroscopy, there have been more reports of preoperative diagnosis from such lesions. The authors report a rare case of obscure gastrointestinal bleeding from a combined hemangioma and angiodysplasia identified by double-balloon enteroscopy. To the authors' knowledge, this is the first report of such combined lesions.     

结直肠海绵状血管瘤的诊断及外科治疗

目的 总结国内文献中结直肠海绵状血管瘤的临床病例特点和外科治疗方式.方法 回顾性分析北京协和医院1993年至2006年收治的4例和检索1979年至2006年国内文献报道的54例结直肠海绵状血管瘤的病例资料.结果 男女发病比例为100:1.0,43.1%的患者在婴幼儿时期发病,98.3%的患者表现为反复血便,纤维结肠镜、直肠CT和磁共振成像的诊断准确率为100%.91.4%的患者病变呈弥漫浸润性,8.6%的病变表现为局限性.82.8%的患者接受了外科干预治疗,3.5%的患者未接受治疗.结论 纤维结肠镜是诊断结直肠海绵状血管瘤的首选诊断方式.外科手术方式的选择方面应该根据患者的具体情况而定.     

Gastric hemangioma in a 5-year-old boy

We report a rare cause of gastrointestinal bleeding in a 5-year-old boy with hematemesis and melena for a month. Upper gastrointestinal endoscopy and an abdominal magnetic resonance image suggested a large hemangioma on the gastric fundus. The patient underwent an exploratory laparotomy with partial gastrectomy limited to the gastric fundus. An intraoperative endoscopy helped to ensure a complete resection of the tumor. Histopathology showed a cavernous hemangioma of the gastric fundus.     

Proximal nerve root capillary hemangioma presenting as a lung mass with bandlike chest pain: case report and review of literature

BACKGROUND: Capillary hemangiomas occasionally occur in the peripheral nervous system (PNS), presenting as mass lesions causing proximal nerve root symptoms, thus prompting neurosurgical intervention. In contrast to cavernous angiomas, which may also occasionally involve the PNS, capillary hemangiomas have little or no tendency for acute or chronic bleeding and, hence, lack the apoplectic symptomatic onset and neuroimaging features of hemosiderin deposition as seen in cavernous angiomas. CASE DESCRIPTION: A 56-year-old man presented with chronic, radiating, bandlike, chest wall pain. Chest radiograph revealed an apical lung mass suspicious for malignancy. Subsequent magnetic resonance imaging revealed an enhancing dumbbell-shaped mass extending into the spinal canal and chest cavity at T3, causing a slight degree of lung compression. Differential diagnostic considerations included schwannoma, neurofibroma, and meningioma. At operation, the spongy, reddish purple vascular tumor was easily separated from dura but had to be excised en bloc, with the T3 nerve root at the neural foramen. The large intrathoracic portion measuring 37 x 41 mm was left behind. Pathology was consistent with a capillary hemangioma. CONCLUSIONS: We present the second case in the English language literature of a dumbbell-shaped, epidural capillary hemangioma with intrathoracic extension. We review the literature on proximal nerve capillary hemangiomas and contrast these with cavernous angiomas and capillary hemangioblastomas, both of which occasionally involve proximal nerves. Unlike capillary hemangioblastomas, which may be multiple and associated with von Hippel-Lindau (VHL) syndrome, capillary hemangiomas are solitary lesions and have not been associated with an inherited disorder.     

Cavernous hemangioma of the cervix: a rare cause of vaginal bleeding

Cavernous hemangiomas of the cervix are extremely rare, benign lesions. To date, fewer than 50 cases have been reported. Here reported is a case of cavernous hemangioma of the cervix in a 53-year-old patient with the complaint of postcoital spotting. Most of these lesions are asymptomatic incidental findings, but sometimes, they may cause abnormal vaginal bleeding in the form of menometrorrhagia and postcoital spotting. They should be included in the differential diagnosis of patients with vaginal bleeding.     

Solitary jejunal cavernous hemangioma causing intermittent melena: A case report

Introduction and importanceHemangioma of the small intestine is a rare disease that causes acute and chronic intestinal bleeding and is difficult to diagnose before surgery. This report presents a case of a cavernous hemangioma confined to the jejunum of a man with intermittent melena.Case presentationA 38-year-old man presented with an episode of melena one and a half month ago. He had similar symptoms a year ago and had undergone esophagogastroduodenoscopy, colonoscopy, and abdominopelvic computed tomography, but the results were normal. Abdominopelvic computed tomography performed after hospitalization showed a small intraluminal protruding mass in the jejunum. A jejunal hemangioma was found during laparoscopy and was successfully treated with intestinal resection.Clinical discussionIt is difficult to identify the cause of gastrointestinal bleeding preoperatively in patients with normal esophagogastroduodenoscopy, colonoscopy, and abdominopelvic computed tomography results. Small bowel tumors, especially small hemangiomas, should be considered as a cause. Laparoscopy may be one option for finding and treating lesions of the small intestine in hospitals where capsule endoscopy or double-balloon intestinal enteroscopy is not available.ConclusionThis report presents a case of a cavernous hemangioma localized in the jejunum of a 38-year-old man with intermittent melena that was successfully treated with laparoscopy and intestinal resection, thereby emphasizing the usefulness of laparoscopy in hospitals where capsule endoscopy or double-balloon intestinal enteroscopy is not available.     

Cavernous Hemangioma After Stereotactic Radiotherapy

Vascular changes after radiotherapy of the central nervous system are well known. Recently, pro-liferative vascular lesions have been reported. We present the case of an 18-year-old male who developed a cavernous hemangioma 5 years after radiotherapy for a low-grade glioma at the same site in the brain. Short review of the literature and hypothesis follow the case presentation.     

An incidentally identified 15 cm cavernous hemangioma of the small intestine: Case report and literature review

IntroductionHemangiomas of the small intestine are rare, usually present with symptoms such as anemia, gastrointestinal bleeding or abdominal pain and are resected. We report resection of an incidentally identified cavernous hemangioma of the small intestine that did not present symptoms referable to the hemangioma. Although it was a large lesion, it was resected using laparoscopy and a mini-laparotomy.Presentation of caseA 29-year-old otherwise healthy man was referred for evaluation of ileal wall thickening found on a contrast-enhanced computed tomography scan obtained for the workup of chronic diarrhea. Double balloon enteroscopy (DBE) showed a cavernous hemangioma of the small intestine. The lesion was 15 cm and resected using laparoscopy and a mini-laparotomy to prevent future bleeding. The histopathological diagnosis was a cavernous hemangioma of the ileum.DiscussionThough there have been no reports of the asymptomatic patients of the disease, the recent spread of double balloon enteroscopy and capsule endoscopy will allow us to diagnose more asymptomatic hemangiomas like this patient. Also, this large lesion was able to be resected through a small incision due to its compressible nature.ConclusionFuture studies in asymptomatic patients of the disease may help to determine the optimal management for these patients. Even large hemangiomas are compressible, facilitating minimally invasive resection.     

肝脏海绵状血管瘤剥离术及组织、解剖学基础

目的探讨剥离术治疗肝海绵状血管瘤(CHL)手术方法及其相关组织学、解剖学基础。方法术中导管插至肝内Ⅱ级动脉,直接供血动脉造影;观察瘤体超微结构;肝叶切除,风干标本横切面观察及剥离新鲜标本观察瘤体㈦瘤床关系。临床治疗11例。结果瘤前均有约1mm的供血小动脉,可供方便结扎。瘤体内无肝小叶结构,证实CHL无门静脉供血及形成胆管的组织学基础。手术标本证实瘤体与瘤床间除供血小动脉外无任何粘连。结论手术剥离瘤体安全性高,无需切除肝组织,术中无大出血,术后无胆漏发生。可适用于肝脏各叶内血管瘤。安全性远高于肝叶切除治疗CHL。     

Cervical cystic lymphangioma in a patient with blue rubber bleb nevus syndrome: clinical case report and review of the literature

We present the case of a 9-year-old boy diagnosed with blue rubber bleb nevus syndrome, who showed a very large left cervical cystic lymphangioma. He was previously subjected to various treatments for lesions in the intestinal tract including blood transfusions for anemia, sclerosis, enterotomies or resections. The tumor was resected without any complications and the anatomopathologic report confirmed this diagnosis. The blue cavernous hemangioma syndrome (or blue rubber bleb nevus syndrome) is a rare disease characterized by cavernous angiomas involving the skin and gastrointestinal tract. Several cases of cystic lymphangiomas associated with this syndrome have been published recently and lymphomatous differentiation has been identified in the cells of cutaneous lesions. Given their common embryological origin, we underscore the importance of bearing in mind that it is possible for different types of vascular malformations to coexist in the same patient.     

Stereotactic radiosurgery for cavernous sinus cavernous hemangioma--case report.

A 40-year-old female presented with cavernous sinus cavernous hemangioma manifesting as left abducens and trigeminal nerve pareses. Magnetic resonance imaging revealed a left cavernous sinus tumor. The tumor was partially removed. Histological examination of the specimen confirmed cavernous hemangioma. Radiosurgery was performed using the gamma knife. The tumor markedly decreased in size after radiosurgery and morbidity was avoided. Cavernous sinus cavernous hemangiomas may be difficult to treat surgically due to intraoperative bleeding and cranial nerve injury. Stereotactic radiosurgery can be used either as an adjunct treatment to craniotomy, or as the primary treatment for small cavernous sinus cavernous hemangioma.     

非肢体血管瘤手术中阻断血供的应用

目的探讨非肢体血管瘤手术治疗时减少术中出血的方法. 方法 1998年11月～2003年11月共收治非肢体血管瘤49例,其中男21例,女28例,年龄3个月～63岁.毛细血管瘤14例,海绵状血管瘤25例,蔓状血管瘤7例,混合性血管瘤3例.根据血管瘤的性质和不同部位选择不同阻断血供的方法.头面颈部蔓状血管瘤扪清搏动的滋养血管后,于其一侧血管约1 cm处用7号丝线深缝一针,暂时缝扎致血管远端搏动消失再切除血管瘤;腹股沟及大腿上份蔓状血管瘤于瘤体组织近端腹股沟韧带上切开分离出髂总或髂外动脉、或在其远端切开分离出股动脉,通过无创血管夹阻断血流,再切除血管瘤;头面颈部、躯干部的毛细血管瘤及海绵状血管瘤瘤体周围用7号丝线间断缝扎,暂时阻断周围滋养血管,再切除血管瘤. 结果通过阻断血供,血管瘤组织不同程度缩小,术中出血明显减少.术后均获随访6个月～4年,49例中47例完全愈合,无复发,外形满意,功能恢复正常.有2例因术后第2天出血行二次手术止血,术后伤口愈合,功能基本恢复正常. 结论手术切除非肢体血管瘤之前阻断血供可明显减少术中出血,降低手术难度,为尽可能彻底切除血管瘤、减少复发创造了条件.     

Cavernous hemangioma of the anterior mediastinum: case report and 50-year review of Japanese cases.

Hemangioma is a rare tumor of the mediastinum. We report a case of cavernous hemangioma of the anterior mediastinum that was incidentally detected by chest radiography during a routine health check. A mass lesion was seen in the anterior mediastinum on computed tomography and magnetic resonance imaging of the chest. Because a thymoma with cystic degeneration was suspected preoperatively, extended thymectomy was performed. The tumor was confined to the left lobe of the thymus without invasion into the adjacent structures. It measured 42x32x17 mm, and was completely resected without any major bleeding. Pathological examination led to the diagnosis of cavernous hemangioma. A total of 61 cases of mediastinal hemangioma, including our case, reported in Japan over the past 50 years are reviewed.     

结直肠弥漫性海绵状血管瘤的诊断与治疗

目的探讨结直肠弥漫性海绵状血管瘤的诊断与治疗方法。方法1994年至2005年手术治疗6例,其中全结直肠弥漫性海绵状血管瘤1例,直肠乙状结肠弥漫性海绵状血管瘤4例,直肠弥漫性海绵状血管瘤1例。结果术后随访6月至10年,4例患者无便血,2例患者有少量的间歇性大便带血。结论肠镜和影像学检查是诊断结直肠海绵状血管瘤的重要方法,保留肛门括约肌功能手术是结直肠弥漫性海绵状血管瘤有效的治疗方法。     

A cavernous hemangioma of the rectum treated as a hemorrhoid for 1 year prior to its,diagnosis: Report of a case

We report herein the case of a 28-year-old woman with a rectal cavernous hemangioma in whom recurrent rectal bleeding and marked anemia were interpreted as being caused by her coexisting internal hemorrhoids, resulting in a delay in the correct diagnosis for 1 year. Digital examination revealed a walnut-sized, wide-based, and elastic soft mass, 3 cm proximal from the anal verge, in addition to the internal hemorrhoids. Colonofiberscopy revealed a bluish submucosal lesion with superficial capillary dilatation at the same site. A transanal local resection was performed and the histological diagnosis of the tumor was cavernous hemangioma. The patient has been well without any recurrence of rectal bleeding for 2 years and 6 months since her operation. Thus, although rectal cavernous hemangioma is rare, a lack of awareness of this condition could lead to misdiagnosis as hemorrhoids and subsequent inappropriate therapy.     

大肠海绵状血管瘤的诊治(附8例报告)

目的探讨大肠海绵状血管瘤的临床特点、诊治原则和疗效。方法回顾1997年至2004年收治的8例大肠海绵状血管瘤患者的临床资料。结果8例患者平均年龄18岁,其中直肠海绵状血管瘤4例,直肠和乙状结肠海绵状血管瘤3例,全大肠海绵状血管瘤1例。临床表现为反复发作的无痛性便血和贫血。术前行电子结肠镜、腹部X线平片、气钡双重对比造影、CT和MRI等检查明确诊断。所有患者均行手术治疗,平均随访36月(10~96月),全部治愈。结论增强对本病的认识,重视电子结肠镜在大肠海绵状血管瘤的首诊作用,影像学检查进一步确定病变部位和血管瘤的范围,手术治疗是有效的治疗手段。     

Sinusoidal hemangioma. A distinctive benign vascular neoplasm within the group of cavernous hemangiomas.

Twelve cases of sinusoidal hemangioma, a distinctive subset of the group of lesions known as cavernous hemangioma, are described. All presented as solitary subcutaneous/deep dermal lesions in adults, predominantly females. Five arose on a limb and five on the trunk; two of the latter were situated in mammary subcutaneous tissue. Histologically they were characterized by dilated, interconnecting, thin-walled vascular channels that frequently showed a pseudopapillary pattern. These vessels had a predominantly lobular architecture but peripherally showed focally ill-defined spread into subcutaneous tissue. The lining endothelium was single-layered but showed focal pleomorphism and hyperchromasia, which, combined with the pseudopapillae and apparent infiltrative pattern in areas, raised the possibility of angiosarcoma in four cases, most notably in the breast lesions. This possibility was further suggested by the presence of pseudonecrotic central infarction in two cases. Follow-up in eight cases, however, has revealed no tendency for either local recurrence or metastasis.     

肝海绵状血管瘤不同手术切除方式比较

目的 探讨切除肝血管瘤应优先选择的手术方式。方法 1995年1月至2005年12月，共手术治疗62例肝血管瘤病人，其中36例行肝血管瘤剥除术，26例行肝切除术，将两者相关因素进行比较。结果 血管瘤剥除术手术过程中失血量以及输血量均明显少于肝切除术，术后并发症也明显减少。多因素分析显示，手术方式是影响术后合并症发生的惟一独立因素。结论 血管瘤剥除术是治疗肝血管瘤的首选手术方式．     

Solitary extradural cavernous hemangiomas in the spinal canal. Report of five cases

Five cases of solitary extrathecal cavernous hemangioma in the spinal canal are reported. In one case, two coexistent massive cerebral venous medullary malformations were found. The symptomatology and prognosis of extrathecal spinal cavernous hemangiomas are discussed, as is the significance of various diagnostic measures. The patient material further permitted a rough calculation of the incidence of symptomatic extrathecal cavernous hemangiomas in the spinal canal.     

Intrasellar cavernous hemangioma. Case report

The authors present a rare entity, an intrasellar cavernous hemangioma that on neuroimages mimicked a nonfunctioning pituitary macroadenoma in a patient with a known orbital hemangioma. Such lesions can grow extraaxially within the dural sinuses, particularly the cavernous sinus, and present like tumors. A better understanding of the neuroimaging. clinical, and anatomical features of these lesions may prevent difficulties in management.