Rare Benign Vascular Lesion Research Articles

Diffuse Cavernous Hemangioma of the Rectosigmoid Colon Mimicking Ulcerative Colitis

Diffuse cavernous hemangioma of the rectosigmoid colon is a rare benign vascular lesion. The rectosigmoid colon is the most common site of this disease. It affects mainly young adults. The most frequent symptom is rectal bleeding, which can be confused with hemorrhoids, perianal diseases and ulcerative colitis. We present a case of diffuse cavernous hemangioma of the rectosigmoid colon mimicking ulcerative colitis.

Eccrine angiomatous hamartoma treated with multimodal vascular laser and incobotulinum.

Eccrine angiomatous hamartoma (EAH) is a rare benign vascular lesion that is distinguished histologically by vascular and eccrine overgrowth. We report the case of a 46-year-old woman with EAH who was successfully treated with multimodal incobotulinum toxin A, pulsed dye laser and long-pulsed neodymium-doped yttrium aluminium garnet laser.

Florid vascular proliferation due to focal necrotizing enterocolitis in infancy: A case report

Vascular anomalies rarely emerge in the gastrointestinal tract. The most prevalent benign vascular lesions include angiodysplasia, Dieulafoy’s lesion, vascular ectasia, vascular malformations, and hemangiomas. Florid vascular proliferation (FVP) is also a rare benign vascular lesion associated with intussusception in adults. In infants, there are only a few cases in literature. Although FVP usually presents as a mass in the intestine, it is a nonneoplastic lesion and is thought to occur due to a reactive process. In this study, we present a case of FVP due to an attack of focal necrotizing enterocolitis in a 10-month-old infant.

Multiple Cerebral Cavernous Malformation: An Unusual Cause of Vertigo.

Cerebral cavernous malformation (CM) is a rare benign vascular lesion of the brain, accounting for only 0.3-0.5% incidence in the population. Among them, only a small portion of the patients have multiple lesions (3-33%). While most lesions are asymptomatic, some patients present with seizures and hemorrhagic events. We present an atypical case of multiple cerebral CM in a patient who presented with vertigo. He did not have apparent neurological signs and symptoms until it was detected through imaging studies. His MRI showed multiple CM, with the largest lesion located in the cerebellopontine angle. A neurological consult was obtained and the patient opted for conservative treatment. He was discharged well on medications and was under active monitoring by both the neurosurgery and otorhinolaryngology teams. Intracranial cavernoma can present as vertigo without classic neurological manifestations. Any atypical presentations should alert the physician to perform further imaging to rule out sinister causes in the brain. To date, there is no reported case of intracranial cavernoma presenting as vertigo in the literature.

Breast Hemangiomas: Imaging Features With Histopathology Correlation.

Breast hemangiomas are rare benign vascular lesions. In a previously performed review of approximately 10,000 breast surgical pathology results, roughly 0.15% (15/~10 000) were hemangiomas. Hemangiomas are more frequent in women and have a documented age distribution of 1.5 to 82 years. They are most often subcutaneous or subdermal and anterior to the anterior mammary fascia but may rarely be seen in the pectoralis muscles or chest wall. On imaging, breast hemangiomas typically present as oval or round masses, often measuring less than 2.5 cm, with circumscribed or mostly circumscribed, focally microlobulated margins, equal or high density on mammography, and variable echogenicity on US. Calcifications, including phleboliths, can be seen. Color Doppler US often shows hypovascularity or avascularity. MRI appearance can vary, although hemangiomas are generally T2 hyperintense and T1 hypointense with variable enhancement. Pathologic findings vary by subtype, which include perilobular, capillary, cavernous, and venous hemangiomas. If core biopsy pathology results are benign, without atypia, and concordant with imaging and clinical findings, surgical excision is not routinely indicated. Because of histopathologic overlap with well-differentiated or low-grade angiosarcomas, surgical excision may be necessary for definitive diagnosis. Findings that are more common with angiosarcomas include size greater than 2 cm, hypervascularity on Doppler US, irregular shape, and invasive growth pattern.

Sclerosing angiomatoid nodular transformation of spleen with calcifying fibrous pseudotumor: A rare case with unusual association

Sclerosing angiomatoid nodular transformation (SANT) of the spleen is a rare benign vascular lesion of the spleen that was first described in 2004. SANT is associated with other concurrent diseases, mostly malignancies. Calcifying fibrous pseudotumor (CFPT) is a tumor-like lesion usually arising from soft tissue or peritoneal sub-serosa. Both SANT and CFPT are considered to be variants of the inflammatory myofibroblastic tumor. We report the rare case of a 24-year-old female presenting with a left abdominal mass that was clinically diagnosed as an extraintestinal gastrointestinal stromal tumor (GIST). Histopathological examination revealed SANT of spleen and CFPT. We report this case due to its rarity of occurrence and unusual association of SANT with CFPT.

Sclerosing angiomatoid nodular transformation of the spleen in a child with anemia: a case report and review of the literature

BackgroundSclerosing angiomatoid nodular transformation of the spleen is a relatively rare benign vascular lesion in both adult and pediatric age groups with unclear etiopathogenesis and variable clinical presentations. Many benign and also malignant splenic masses could mimic sclerosing angiomatoid nodular transformation, both clinically and radiologically. Herein, we report our experience with a case of sclerosing angiomatoid nodular transformation in a 3-year-old girl.Case reportA 3-year-old Iranian girl presented with abdominal pain, back pain, and constipation for 2 weeks. She was being followed up by a pediatrician due to her short stature and persistent anemia. Physical examination showed stable vital signs, short stature, pallor, and a puffy face. Laboratory evaluation showed normochromic normocytic anemia with a normal reticulocyte count, ferritin, and hemoglobin electrophoresis. Radiologic assessments revealed a hypoechoic lesion in the spleen with high vascularity, clinically suspected to be lymphoma. She was operated on, and after partial splenectomy, pathologic evaluation of the spleen showed a solitary, well-demarcated, and unencapsulated dark mass. Microscopic examination revealed micronodular appearance composed of irregular-shaped vascular spaces lined by plump endothelial cells and surrounded by concentric collagen fibers, features in keeping with sclerosing angiomatoid nodular transformation. The patient’s anemia was resolved after surgery, and no clinical or radiologic deficits were noted during the 10-month follow-up visits.ConclusionAlthough sclerosing angiomatoid nodular transformation is exceedingly rare in children, it should be considered a differential diagnosis in pediatric splenic neoplasms with concurrent hematologic manifestations, such as anemia.

Epithelioid hemangioma (ALHE) on the tongue of an infant treated with oral corticosteroids: A case report and review of the literature.

Epithelioid hemangioma or angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign vascular lesion presenting as multiple nodules on the head and neck. Surgery had been considered to be the best treatment modality for ALHE. We report the case of a 6-month-old boy with ALHE on his tongue that was treated successfully with oral prednisolone.

Sinonasal Lobular Capillary Hemangioma After Continuous Self-tests for COVID-19: A Case Report and Review of the Literature

Sinonasal lobular capillary hemangiomas (LCH) are rare benign vascular lesions commonly arising from the nasal septum. Nasopharyngeal, nasal mid-turbinate, and anterior nasal swabbing is the preferred method of screening for coronavirus disease-2019 (COVID-19). Herein, we present a case of a sinonasal LCH in a child after continuous self-tests for COVID-19, with an anterior nasal swab. The child presented with a well-defined red mass in the anterior part of the nasal septum, which was removed endoscopically. Histopathology revealed a LCH. This is the first report of a complication other than epistaxis with the use of an anterior nasal swab. Our literature review identified 32 studies reporting complications of COVID-19 screening. Cerebrospinal fluid leaks and foreign body retention are the most common ones. A proper specimen collection technique and a quick patient history with an emphasis on risk factors are the best practices to prevent complications from COVID-19 screening.

Splenic hamartoma in a patient with peripancreatic arterial arcades: A case report

AbstractSplenic hamartoma is a rare benign vascular lesion of the spleen. A splenic mass was incidentally detected in an asymptomatic 65-year-old male during an abdominal ultrasound scan. The workup of the splenic lesion included an abdominal CT angiography (CTA), contrast-enhanced ultrasound (CEUS), and microvascular flow imaging Doppler sonography. The CT scan confirmed that the splenic mass had benign characteristics, and its contrast enhancement was similar to the spleen. The CEUS was able to rule out the possibility of hemangioma. Meanwhile, chronic compression of the celiac trunk and the filling of the splenic artery from collaterals were also detected on CTA. The imaging studies suggested a splenic hamartoma, and the diagnosis was confirmed by the result of a fine needle aspiration biopsy.

S2093 Painless Voluminous Rectal Bleeding: It’s Not Always a “Tic”: A Case of a Recto-Sigmoid Hemangioma

Introduction: Hemangiomas are known as benign vascular tumors and may present as multiple or solitary lesions. Very few cases of colonic hemangiomas have been reported in literature. We present a case of rectal bleeding leading to a finding of recto-sigmoid hemangioma. Case Description/Methods: A 31-year-old female with no past medical history presented to the ED with complaints of bright red blood per rectum (BRBPR). She admitted to having 4 days of blood clots associated with bowel movements, in addition to diffuse lower abdominal pain which has occurred intermittently and progressively worsened for the past 2 years. In Cuba, she was told she had a rectal tumor which was “inoperable”. Lab work on admission was significant for iron deficiency anemia. CT of the abdomen and pelvis with oral and IV contrast revealed findings consistent with active gastrointestinal (GI) bleed in the ascending colon, as well as a 2.5 cm pelvic mass. However, an IR arteriogram revealed a large segment of angiodysplasia in the ascending colon but no evidence of active GI bleed. Transvaginal ultrasound ruled out an adnexal mass. Sigmoidoscopy revealed a large recto-sigmoid mass suspicious for an hemangioma as well as surrounding vascular ectasias. Carefully taken biopsies revealed a benign rectal hemangioma. Ultimately, the patient was stabilized and discharged, with follow-up outpatient with surgical oncology due to the extent of her disease (Figure 1). Discussion: Colonic hemangiomas are extremely rare benign vascular lesions of the GI tract. Often located in the recto-sigmoid, they may be discovered during workup for recurrent painless rectal bleed and rarely presenting as life-threatening bleeding. Large cavernous hemangiomas may be associated with Kasabach-Merritt syndrome, hemolytic anemia, thrombosis and bowel ischemia. Studies reveal that it may take on average up to 19 years before a proper diagnosis is established. There are few reported cases of local invasion to surrounding structures such as bladder and uterus. Despite being incidentally found in colonoscopy, insufflation may flatten these lesions which may be also misinterpreted as proctitis. Imaging studies such as MRI, CT and rectal EUS are helpful in determining the diagnosis. Sclerotherapy, angiographic embolization and ligation of mesenteric vessels may be attempted for small lesions. Surgical resection, if possible, is generally the treatment of choice for more extensive lesions. Due to its mimicking presentation of common GI diseases, hemangiomas risk being overlooked.Figure 1.: Extensive recto-sigmoid hemangioma with associated vascular ectasias.

Granulation tissue-type hemangioma of ureter: a highly misdiagnosed disease (a rare case report and literature review)

BackgroundUreteral granulation tissue hemangiomas are rare benign vascular lesions, and they may be clinically asymptomatic or present with massive or recurrent hematuria. Sometimes hemangiomas are difficult to distinguish from malignant ureteral tumors, and most ureteral hemangiomas are confirmed by postoperative pathological examination. This article aims to present a case of granulation tissue-type hemangioma of the ureter and briefly review the current literature on this condition.Case presentationA 30-year-old male patient presented with complaints of painless macroscopic hematuria for 2 months. Computerized tomography of the urinary system showed that the upper 1/3 of the right ureter was occupied, and then the possibility of tumor lesions was considered. The urine cytology showed occasional nuclear abnormalities and many light-stained crystals in urine. Because of suspicious radiological and cytological findings, the patient underwent the right ureteroscopy and the laparoscopic right ureteral mass resection. The postoperative pathological report showed that it was a mesenchymal tumor. The morphological and immunohistochemical staining was consistent with that of hemangioma, tending to granulation tissue hemangioma. After surgery, the patient was in a good state and recovered well at the last follow-up.ConclusionsUreteral granulation tissue hemangiomas are an easily misdiagnosed disease. Intermittent painless hematuria is an important characteristic of this disease. Therefore, we suggest that unnecessary radical surgery can be avoided when clinicians consider the possibility of benign ureteral tumors during the evaluation.

Angioma serpiginosum: Two cases in children and review of literature.

Angioma serpiginosum (AS) is a rare benign vascular lesion that typically arises in early childhood, with a prevalence in female, and then grow up over a period of months/years. It is characterized by small asymptomatic purple-red dots that cluster together and they do not disappear on diascopy. It is mainly localized on the arms but some cases on face and neck have been reported. The etiology of AS is unknown, dermoscopy may aid in the diagnosis but usually the biopsy is necessary. We report 2 cases: one male and one female with angioma serpiginosum, aged 13 and 8 years old.

Intravascular papillary endothelial hyperplasia as a rare cause of cervicothoracic spinal cord compression: A case report

BACKGROUNDIntravascular papillary endothelial hyperplasia (IPEH) is a rare benign reactive vascular lesion that grows into an expansile compressing mass. It most commonly involves the skin and subcutaneous tissue. Spinal involvement is rare, with only 11 reported cases in the literature. We report, to our knowledge, the first case of IPEH in the cervicothoracic spinal canal and present a literature review.CASE SUMMARYA 27-year-old man presented with acute-onset neck pain, numbness, and weakness in his extremities. Magnetic resonance imaging showed an epidural mass in the cervicothoracic (C6-T1) spinal canal and vertebral hemangioma (VH) involving the C7 vertebral body. C6-T1 Laminectomy and radical excision of the mass were performed. Histopathological examinations revealed papillary proliferation of vascular endothelial cells with thrombus formation, and an IPEH diagnosis was made. By his 6-mo follow-up appointment, his symptoms were relieved without recurrence. The possible pathogenesis, clinical and imaging features, differential diagnosis, and management of IPEH were reviewed.CONCLUSIONWe report, to our knowledge, the first case of IPEH in the cervicothoracic spinal canal, treated via complete resection, and showing a favorable outcome. We found a causal relationship between spinal IPEH and VH; this partly explains the mechanism of IPEH.

Angioma sepiginosus: two cases in children and review of literature

Angioma serpiginosum (AS) is a rare benign vascular lesion that typically arises in early childhood, with a prevalence in female, and then grow up over a period of months/years. It is characterized by small asymptomatic purple-red dots that cluster together and they do not disappear on diascopy. It is mainly localized on the arms but some cases on face and neck have been reported. The etiology of AS is unknown, dermoscopy may aid in the diagnosis but usually the biopsy is necessary. We report 2 cases: one male and one female with angioma serpiginosum, aged 13 and 8 years old.

Clinicopathological features of sclerosing angiomatoid nodular transformation of the spleen

PurposeTo explore the clinicopathological features of sclerosing angiomatoid nodular transformation (SANT) of the spleen. MethodsThe clinicopathological data of 26 SANT patients were analyzed. ResultThere were 15 men and 11 women, aged 23–62 years (mean: 43.9 years; median: 43 years). Twenty patients were found during health check-ups. Magnetic resonance imaging had significantly higher specificity than other imaging modes in the diagnosis of SANT. Macroscopically, the lesions were gray–red and gray–white, along with well-demarcated nodules. Microscopy showed multiple angiomatoid nodules embedded in hyperplastic fibrous tissues and dense collagen fiber; the angiomatoid structures inside the nodules had varied morphology. Patchy and nodular fresh and old hemorrhages were observed in each lesion. Proliferative fibroblasts were seen in the stroma, along with infiltration of a few mixed inflammatory cells. Serum tumor markers were negative. Fourteen patients (53.8 %) had benign or malignant lesions in other parts of the body, including the liver, kidneys, and adrenal and pituitary glands which were similar to von Hippel–Lindau (VHL) syndrome. The reasons for occurrence of SANT may be as follows: hemangioma/lymphangioma or splenic congestion with extensive hemorrhage and secondary changes. ConclusionsSANT is a rare benign vascular lesion with some clinical manifestations similar to VHL syndrome. Patients have good prognosis after tumor removal.

Bleomycin for Head and Neck Venolymphatic Malformations: A Systematic Review.

Venolymphatic malformations are rare benign vascular lesions of the head and neck. Sclerotherapy has become the first-line therapy of these lesions with bleomycin being a sclerosing agent commonly used. To perform a systematic review of the published literature to synthesize evidence on the safety and efficacy of bleomycin for the treatment of head and neck venolymphatic malformations. A systematic review of the literature (January 1995-May 2019) was performed in PubMed, Embase, and Cochrane Library databases to identify studies on sclerotherapy of venolymphatic malformations of the head and neck. A total of 32 studies with participants met the inclusion criteria among which 1121 patients were included in the systematic review. Two reviewers independently screened and extracted data and assessed the risk of bias. The primary outcome was the subjective or objective reduction of lesion size as well as minor and major complications. The bleomycin/pingyangmycin sclerotherapy achieved subjective or objective lesion size reduction in 96.3% (95% CI 94.1%-98.5%) of patients. Minor complications were observed in 16.2% and major complications in 1.1%. Bleomycin is a highly effective treatment of venolymphatic malformations of the head and neck with a low rate of major adverse events. This study represents an update on the "available" evidence, but only low-to-moderate quality studies were available. This study reviewed 32 studies performed in different parts of the world, but there was heterogeneity of the study designs and interventions.

Haemangioma of the Penis: A Review and Update of the Literature

Haemangioma of the penis (HOP) is a rare benign vascular lesion which can affect the penis of males including infants, children, and adults of up to the over 80 year-olds. HOP does manifest as: asymptomatic lump or lesion on the penis; localised pain or tenderness anywhere within the penis; a tumoral mass anywhere on the penis; pain in the penis on erection; curvature of penis on erection; bright red compressible papule or papules on penis; nodules on penis; plaques on penis; ulceration on penis; haemorrhage on penis. HOP may be associated erectile dysfunction in some cases and could affect voiding if it is associated with the urethral meatus. HOP which tends to be solitary in most cases may mimic other diseases of the penis as well as may be associated with haemangioma in the scrotum and perineal region occasionally. Diagnosis can be established by the clinical features and this can be reaffirmed by radiology imaging features with the use of Doppler ultrasound scan, CT scan and MRI scan which show low-flow within the lesion. Majority of the lesions tend to be superficial and not associated with the corpora cavernosa and spongiosum. There is no consensus opinion on the best treatment options hence various treatment options for HOP have been successfully utilized including: complete surgical excision, sclerotherapy, laser treatment, cryotherapy, and other therapies. Most cases of HOP do not recur but following sclerotherapy and laser treatment of large HOPs repeat procedures tend to be required and there may be residual / recurrent lesion that may need to be treated again. Deep and large HOPs may require complete excision with insertion of tunica graft. Very large HOPs could be more effectively treated by one stage complete surgical excision but the choice of the patient is important. The diagnosis of the lesion can be confirmed by the histopathology features of the lesion and positive immunohistochemistry staining for CD31 (strong), Factor VIII, and HHF35 (myopericytes) and minimal staining for CD34. HOP needs to be carefully differentiated from epithelioid angiosarcoma of the penis and epithelioid haemangioendothelioma. A multi-centre trial of various treatment options for HOP would be required to provide treatment guidelines for HOP taking into consideration the size of the lesion and availability of the various treatment options.

Splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case report

BackgroundSplenic cord capillary hemangioma is a rare benign vascular lesion classified as a splenic hamartoma. On the other hand, non-islet cell tumor hypoglycemia (NICTH) is one of the rare causes of spontaneous hypoglycemia and is considered to be one of the paraneoplastic syndromes. To the best of our knowledge, this is the first reported case of a splenic cord capillary hemangioma with NICTH.Case presentationA 25-year-old male was referred to our hospital with hypoglycemia. Except for his low blood sugar, there were no abnormal findings from laboratory tests, which included an endocrinological examination. Enhanced computed tomography confirmed the presence of a solid mass measuring about 6 cm in the retroperitoneum, and a tumorectomy was performed. During this operation, it became clear that the tumor turned out to be a splenic parenchyma, and as a result, a total splenectomy was performed. Microscopically, we diagnosed this as a cord capillary hemangioma, and through immunohistochemistry, we found that some tumor cells were positive for insulin-like growth factor -II. Fortunately, the hypoglycemia-related symptoms disappeared after surgical resection was performed. The patient is still alive and well without evidence of local tumor recurrence 15 years after the operation.ConclusionsSplenic cord capillary hemangioma, one of the types of splenic hamartomas, is a very rare benign vascular lesion and might be associated with hypoglycemia thought to be NICTH.

Sclerosing Angiomatoid Nodular Transformation of the Spleen: A Case Report

Objectives: Sclerosing Angiomatoid Nodular Transformation (SANT) of the spleen is a rare benign vascular lesion with unknown etiology. Our purpose is to present our experience with this condition and review the available literature regarding it. Methods: This report depicts a pathologically proven case of SANT in a 32-year-old man with a history of two previous old injuries who presented with weight loss, vomiting and with left upper quadrant pain and an enlarged spleen. Results: The patient was successfully treated by splenectomy and diagnosed with SANT based on histological and immunohistochemical findings postoperatively. Conclusion: Most patients with SANT are asymptomatic. Imaging studies are essential for detection and the management of SANT. However, the final diagnosis can only be made by histopathology. SANT does not recur after splenectomy.