Sphincter-saving procedure for treatment of diffuse cavernous hemangioma of the rectum and sigmoid colon

Two cases of diffuse cavernous hemangioma of the rectum and rectosigmoid colon are reported. Sphincter-saving procedures were applied as the surgical treatment for these two patients. The diseased rectum and distal sigmoid colon were resected 3 cm above the pectinate line. The mucosa of the rectal stump was extirpated. All visible residue from the hemangiomatous tissues on the muscular cuff, pararectal space, and pelvic wall were denuded. The normal colon was then pulled down through the rectal cuff and anastomosed with anoderm, and attached to the internal sphincter muscle. Both patients recovered uneventfully. No uncontrollable hemorrhage was encountered during rectal dissection. Both patients had good anal sensation and perfect continence. No complicated sexual or urinary disturbances were found. The sphincter-saving procedure for treatment of diffuse cavernous hemangioma of the rectum should be promoted and given first choice whenever possible. Read at the Tenth Biennial Congress of the International Society of University Colon and Rectal Surgeons, Strasburg, France, September 1984.     

Diffuse cavernous hemangioma of the rectosigmoid—CT scan,a new diagnostic modality,and surgical management using sphincter-saving procedures

Diffuse cavernous hemangioma of the rectosigmoid is a rare lesion. Preoperative recognition has been recorded but, because of lack of awareness and inconsistent diagnoses inappropriate therapy still persists. Surgical therapy is the hallmark of treatment. Abdominoperineal resection has been advocated. Three cases of diffuse cavernous hemangiomas of the rectosigmoid, recognized preoperatively and treated successfully with sphincter-saving procedures, are reported. Use of the CT scan as a consistent diagnostic tool will be presented for the first time.     

Polypoid cavernous hemangioma removed under colonoscopy

A 57‐year‐old Japanese man was admitted to Ushioda General Hospital because of a positive fecal occult blood test. Colonoscopy revealed a pedunculated polyp in the ascending colon, which was removed by snare polypectomy with electrocautery. The resected polyp was bluish and measured 25 × 20 mm. Histological examination of the polyp revealed a cavernous hemangioma. The hemangioma infiltrated into the muscular layer; however, massive hemorrhage or perforation was not revealed. Although complications of colonoscopic excision of hemangiomas appear to be rare, the number of reports is small and it is, therefore, difficult to determine the safety of colonoscopic treatment.     

Diffuse cavernous hemangioma of the rectosigmoid colon with extraintestinal involvement.

Cavernous hemangioma of the colon is a rare cause of gastrointestinal bleeding. These lesions can be encountered as solitary, multiple, or part of a more complex syndrome with cutaneous manifestations. We herein describe a 26-year-old woman with cavernous hemangioma involving the rectosigmoid area. Additional hemangiomas were identified in the pelvic structures, spine, iliac bone and spleen. This multi-visceral involvement without cutaneous manifestations represents an intermediate variety between solitary hemangioma and well-defined syndromes with cutaneous and structural anomalies. The potential presence of extraintestinal hemangiomatosis should be considered and investigated in patients with cavernous hemangioma of the colon even without cutaneous manifestations or with a limited colonic involvement.     

Hepatic cavernous hemangioma: underrecognized associated histologic features.

BACKGROUND/AIMS: Cavernous hemangiomas (CH) are typically described as solitary, well-circumscribed lesions and are reported to have a distinct fibrous interface. This study describes underrecognized histological changes of large hepatic hemangiomas that contradict this long-standing view. METHODS: Nineteen cases of hepatic resections for CH were reviewed. Stains for estrogen and progesterone receptors (ER/PR), MIB-1, alpha-smooth muscle actin, collagen IV, and elastic Van Gieson stains were applied to the lesions. RESULTS: The CHs measured 5-31 cm (mean 16.6 cm). Sixteen (84%) CHs had an irregular interface with the liver parenchyma while only three had the well-defined fibrous capsule typically described for CH. Fifteen (79%) CHs had dilated vascular spaces filled with blood 0.1-2.0 cm beyond the confines of the main CH, which we have designated hemangioma-like vessels (HLVs). The histochemical and immunohistochemical stains in both CH and HLVs were similar, with the walls of the vessels composed predominantly of collagen with some faint elastic fibers and smooth muscle, endothelium underlined by collagen IV, negative ER/PR in all components, and a proliferation rate of <5/100 endothelial cells. CONCLUSION: Irregular edges of CH and HLVs in the liver parenchyma adjacent to CH have been underrecognized. No significant differences in staining or proliferative rate were present between CHs and HLVs, suggesting the HLVs are within the spectrum of CH.     

肺海绵状血管瘤二例并文献复习

目的探讨肺海绵状血管瘤(cavernous hemangioma)的临床特点、诊断及治疗方法。方法报告我院2例肺海绵状血管瘤,并结合国内1981年1月至2010年10月近30年间文献报道的41例患者的临床资料进行分析。结果 43例患者中女32例,男11例,发病年龄13～72岁,平均(44.3±15.9)岁。临床表现为咳嗽、咳痰、痰中带血、咯血、胸闷、胸痛以及突发呼吸困难和休克等,也可无临床症状,于体检发现。临床诊断3例患者为血管瘤,其余误诊为肺癌、结核、炎性假瘤、肺囊肿、肺隔离症、何杰金氏病、错构瘤及自发性血气胸等。43例患者均行手术治疗,无手术死亡。结论肺海绵状血管瘤是一种肺部罕见良性肿瘤,临床表现缺乏特异性,术前误诊率高,影像资料有助于诊断,确诊依靠病理诊断,手术切除治疗疗效确切。     

A case of polypoid cavernous hemangioma of the sigmoid colon excised by colonoscopic polypectomy

A 28-year-old Japanese man was admitted to our institute because of sharp anal pain and a mass that prolapsed from the anus after defecation. After spinal anesthesia for emergency operation, the mass spontaneously withdrew into the anus and the pain disappeared. Surgery was postponed. Barium enema and colonoscopy revealed a pedunculated polyp in the sigmoid colon, which was removed by snare polypectomy with electrocautery. The resected polyp was granular and reddish, and measured 33 × 22 × 14 mm. Histological examination of the polyp revealed a cavernous hemangioma. Hemangioma of the colon is rare; only 52 cases have been reported in Japan. Of these patients, eight underwent endoscopie polypectomy. The present lesion is the largest thus treated that has been documented. Endoscopie polypectomy seems to be a safe and preferable procedure for the diagnosis and treatment of small, solitary polypoid hemangiomas of the colon.     

巨大肝海绵状血管瘤切除1例报告

1病例资料 患者,女,58岁,因＂上腹部膨隆伴进食后胀满不适1个月＂于2011年8月15日入院。查体：肝肋下5cm处可触及,质硬,边缘钝,表面稍有结节感,无压痛。实验室检查无明显异常,吲哚菁绿（ICG）15 min滞留率为5.8%。腹部增强CT示：     

Diffuse cavernous hemangioma of the rectum: MR imaging with endorectal surface coil and sphincter-saving surgery

We describe the clinical features, diagnostic procedures, and treatment of two patients with diffuse cavernous hemangioma of the rectum. Sphincter-saving operations were performed in both patients, with satisfactory results. Magnetic resonance imaging (MRI) with an endorectal surface coil, as well as a conventional body coil, was used to determine the extent of the hemangiomas. We recommend sphincter-saving surgery for the treatment of this benign disease that can cause life-threatening hemorrhage. MRI with an endorectal coil achieves higher-resolution images than conventional MRI.     

Retroperitoneal cavernous hemangioma resected by a pylorus preserving pancreaticoduodenectomy

A retroperitoneal hemangioma is a rare disease. We report on the diagnosis and treatment of a retroperitoneal hemangioma which had uncommonly invaded into both the pancreas and duodenum, thus requiring a pylorus preserving pancreaticoduodenectomy (PpPD). A 36-year-old man presented to our hospital with abdominal pain. An enhanced computed tomography scan without contrast enhancement revealed a 12 cm × 9 cm mass between the pancreas head and right kidney. Given the high rate of malignancy associated with retroperitoneal tumors, surgical resection was performed. Intraoperatively, the tumor was inseparable from both the duodenum and pancreas and PpPD was performed due to the invasive behavior. Although malignancy was suspected, pathological diagnosis identified the tumor as a retroperitoneal cavernous hemangioma for which surgical resection was the proper diagnostic and therapeutic procedure. Reteoperitoneal cavernous hemangioma is unique in that it is typically separated from the surrounding organs. However, clinicians need to be aware of the possibility of a case, such as this, which has invaded into the surrounding organs despite its benign etiology. From this case, we recommend that combined resection of inseparable organs should be performed if the mass has invaded into other tissues due to the hazardous nature of local recurrence. In summary, this report is the first to describe a case of retroperitoneal hemangioma that had uniquely invaded into surrounding organs and was treated with PpPD.     

Atypically enhanced cavernous hemangiomas of the liver: centrifugal enhancement does not preclude the diagnosis of hepatic hemangioma

The imaging features of an atypically enhanced hepatic hemangioma have not been well described in the literature, and the presence of such atypia may sometimes cause clinical problems in the differential diagnosis. Herein, we report a case of hepatic hemangioma demonstrating a previously unreported atypical enhancement pattern. On dynamic computed tomography during hepatic arteriography, a centrifugal enhancement pattern and subsequent peritumoral ring-shaped enhancement mimicking corona enhancement were found in cavernous hemangiomas of the liver in a 68-year-old Japanese man. Histopathological diagnosis of cavernous hemangioma of the liver was made on a biopsy specimen. Considering the importance of differentiating benign hepatic tumor from various forms of malignancy, radiologists and hepatologists should be aware of rare enhancement patterns sometimes seen in hepatic hemangioma. Establishing knowledge of the entire spectrum of atypical hepatic hemangioma may benefit the rational approach to future cases.     

肝血管瘤动脉造影诊断与栓塞治疗

为了探讨肝血管瘤(CHL)的肝动脉造影诊断及特异性鉴别诊断征象,总结栓塞治疗效果,寻求理想栓塞方法,回顾分析了25例CHL选择性肝动脉造影的表现与鉴别诊断征象,观察平阳霉素(PYM)超液态碘化油乳剂栓塞治疗的效果,随访6-48个月。CHL典型的瘤体显影形态,造影剂染色时间的特点,可与肝癌鉴别,动-静脉瘘的出现作为鉴别依据缺乏特异性;25例栓塞治疗,其中1次栓塞18例,2次栓塞7例,瘤体均缩小达50％以上,临床有效率达100％,无严重并发症发生。选择性肝动脉造影是诊断肝血管瘤较理想的方法,介入栓塞治疗应作为首选方法推广应用。     

Cavernous hemangioma in the ascending colon treated by endoscopic mucosal resection

Hemangioma of the large intestine is rare, but it is a clinically important entity because of the possibility of massive hemorrhage. The case is reported of a patient with a formed, sessile, polypoid-type cavernous hemangioma in the ascending colon that was removed successfully by endoscopic mucosal resection.     

Brachial plexus paralysis due to giant cavernous hemangioma with Kasabach-Merritt syndrome: Successful management with interferon alpha

Giant hemangiomas are rare in newborn babies and may cause some life-threatening complications such as congestive heart failure and coagulopathies including severe thrombocytopenia, microangiopathic hemolytic anemia and bleeding such as Kasabach-Merritt syndrome. We have presented here one infant case with a right lower brachial plexus paralysis due to giant hemangioma treated successfully with interferon alpha and have discussed the treatment. This is the first case of Kasabach-Merritt syndrome associated with a right lower brachial plexus paralysis treated with interferon in the literature to our knowledge. The administration of interferon-alpha might be indicated as a therapy for severe, life-threatening and giant hemangiomas such as Kasabach-Merritt syndrome, especially when there is resistance to steroid. However, more extensive data and new patients are needed to clarify this issue.     

肝海绵状血管瘤超微结构及其与临床特征的关系

目的:从肝海绵状血管瘤(CHL)超微结构探讨CHL组织起源,以及组织结构与临床特征关系.方法:对8例CHL切除标本进行电镜观察.1例按常规电镜样品制备,7例用弹力纤维特异染色的丹宁酸块染后,按常规电镜样品制备,经LKB-V超薄切片,用透射电镜H-600,JEOL-100SX和扫描电镜KYKY-2000进行观察.结果:血窦腔由单层内皮细胞覆盖,血窦腔蜿蜒迂回,呈迷宫状结构组成.正常内皮细胞为扁平状,细胞器减少,有吞饮小泡和微丝,细胞间有紧密连接和桥粒连接.内皮细胞下有弹力纤维,中膜层可见成纤维细胞和平滑肌细胞,中、外膜层有丰富胶原纤维.此与微动脉结构相似.CHL内皮细胞多肿胀,核仁边移,突向血窦腔成柱状.随程度加重,粗面内质网囊泡样改变,直至内皮细胞由内膜层脱落,坏死.间质中大量胶原纤维增生,分割弹力纤维,并可由内皮细胞缺损处填充于血窦腔内,发生纤维化.瘤体中无正常肝细胞和肝窦结构.由于瘤内无肝小叶,所以瘤体无双重供血组织结构,CHL由动脉供血.蜿蜒迂回、迷宫样血窦腔,可以解释影像学特征(肝血管造影、CT增强).内皮细胞退行性变化和间质中胶原纤维大量增生,导致CHL纤维化,并自行萎缩.结论:CHL血窦腔结构类似微动脉,起源于肝脏微动脉血管畸形病变.CHL一般不需手术治疗,也不易自发破裂出血.     

经皮微波消融术治疗的肝海绵状血管瘤患者血红蛋白尿发生及其处理*

目的 探讨经皮微波消融术治疗肝海绵状血管瘤（HCHs）患者的疗效,并探讨术后血红蛋白尿发生及处理方法。方法 2012年2月~2017年12月我院诊治的HCHs患者54例,病灶数80个。采用在超声引导下经皮微波消融术治疗。采用联苯胺试验检测血红蛋白尿。术后随访6~12 m。结果 在54例HCHs患者的80个病灶中,经皮微波消融术治疗后, 78个肿瘤完全被消除,2例被大部分消融;术后6~12个月对患者进行超声造影和CT检查随访,所有患者血管瘤均缩小,其中瘤体完全消失者41例（75.9%）,瘤体缩小超50%者8例（14.8%）,瘤体缩小大于25%但小于50%,但未见肿瘤复发者4例（7.4%）,瘤体缩小小于25%,但是未见肿瘤复发者1例（1.9%）;术中未出现严重的肝肾功能损害,术后3~4 h有8例（14.8%）患者出现血红蛋白尿,经水化和碱化尿液处理2 d后,尿色恢复正常;2例（3.7%）出现腹水,经内科综合治疗后消退;5例（9.3%）出现右上腹轻微的疼痛,未经干预自行缓解;7例（12.3%）出现恶心呕吐,经止吐处理后缓解。结论 经皮微波消融术是一种疗效确切、创伤小的治疗HCHs患者的方法,选择合适的患者进行治疗,可以达到微创治疗的目的。对少数发生的血红蛋白尿,经对症处理,预后良好,但应总结其发生的原因,予以避免。     

Sclerosed hemangioma of the liver: Report of a case and review of the literature.

A sclerosed hemangioma of the liver is an extremely rare type of benign hepatic tumor. A 77-year-old female was referred to Tokushima University Hospital with fever, abnormal liver function tests and a large liver mass. The tumor, 10 x 5 cm in size and located in segment 5-6 of the liver, was depicted as a low density tumor with enhancement by computed tomography (CT). Magnetic resonance imaging (MRI) showed it to be a tumor with a low signal on T1-weighted and a high signal on T2-weighted images. The patient was negative for hepatitis B surface antigen and hepatitis C antibody. She underwent a right hepatectomy for possible malignant liver tumors, including intrahepatic cholangiocarcinoma or fibrolamellar hepatocellular carcinoma. The following histological examination of the surgical specimen revealed the tumor to be a hepatic sclerosed hemangioma with characteristic dense collagenous tissues. We report here on the case of this unusual tumor and review the relevant literature.