Benign Vascular Proliferation Research Articles

68Ga-PSMA-11 PET/CT in Cherry Hemangiomas.

A 63-year-old man was remitted for a 68Ga-PSMA-11 PET/CT scan due to biochemical recurrence with a PSA of 0.32 ng/mL 1 year after radical prostatectomy of locally advanced Gleason 6 (3 + 3) ISUP 2 pT3a pN0 prostate cancer. 68Ga-PSMA-11 PET/CT showed multiple cutaneous and subcutaneous uptake foci in the upper body. Physical examination revealed numerous dome-shaped, ruby-red papules. These were consistent with a previous diagnosis of cutaneous hemangiomas. Cherry hemangiomas (also known as Campbell de Morgan spots) are the most common type of benign vascular proliferation of the skin. Due to the nonspecific 68Ga-PSMA-11 uptake of vascular lesions, careful interpretation should be considered in order to avoid a potential pitfall in nonmalignant conditions.

Whole exome sequencing of placental chorangioma

IntroductionPlacental chorangioma is a benign non-trophoblastic vascular proliferation of the placental chorion favored to represent hamartoma-like or hyperplastic capillary lesions. As the exact pathophysiology has not been established, we investigated the molecular characteristics of placental chorangiomas using exploratory whole exome sequencing. MethodsThree cases were retrospectively selected and whole exome sequencing was performed on macrodissected lesions. DNA extraction, DNA quantification, library preparation and sequencing were performed with IDT xGen™ Exome Hybridization Panel v2 for library capture. Sequencing data was analyzed with an in-house bioinformatics pipeline for single-nucleotide variants and insertions/deletions. ResultsAll neonates were delivered at term and had birth weights ranging from 11th-35th percentile for gestational age. All mothers presented with hypertensive disorder during pregnancy. Chorangiomas ranged from 0.7 cm to 5.1 cm and were well-circumscribed near the fetal surface. Case 1 showed a background of chorangiosis and acute subchorionitis, while case 2 had foci of chronic lymphocytic villitis. Whole exome sequencing did not reveal any significant pathologic variants. DiscussionsThe absence of molecular alteration in placental chorangioma is likely indicative of the reactive/non-neoplastic nature of this lesion. The presence of compromised blood flow in the form of hypertensive disorders in our cases may be one of its underlying pathophysiologic mechanisms.

Efficacy of available treatments for periungual and subungual pyogenic granulomas: A systematic review

Efficacy of available treatments for periungual and subungual pyogenic granulomas: A systematic review

Cutaneous Epithelioid Angiomatous Nodule with Unusual Localization

Abstract: Brenn and Fletcher described cutaneous epithelioid angiomatous nodule (CEAN) as a benign vascular proliferation that was in 2004. It is very rare condition. It is characterized by endothelial cells that have histiocytoid appearance. It is generally found in the dermis or sub cutis and rarely, in visceral organs. Despite the etiology of CEAN is not clear, it is considered as a reactive entity and no association with either infection, trauma or immunosuppression has been described yet. A 76-year-old woman visited hospital for routine examination. The physician noticed an erythematous nodule on the upper part of her right labium minus which appears like a cystic lesion. Histopathological examination revealed a relatively circumscribed, unencapsulated, unilobular vascular lesion which is in the superficial dermis. There were also moderately lymphoplasmacytic infiltration on the background. There were also scattered extravasated erytrocytes around. Positive stained for CD34, CD31 and negative stained for HHV-8 (performed to rule out a possibility of Kaposi sarcoma), pancytokeratin and s-100 were prominent in epithelioid cells. The overall histopathological appearance was consistent with CEAN and there was not any previous report CEAN occurring at this area.

IADVL SIG Pediatric Dermatology (Academy) Recommendations on Hemangioma of Infancy (HOI).

Hemangioma of infancy (HOI) is a benign vascular proliferation. Though resolution is the norm, potential complications make an accurate diagnosis and early management of importance. The Indian association of dermatologists, venereologists and leprologists (IADVL) special interest group (SIG) paediatric dermatology in association with IADVL academy did an extensive analysis of the literature on the clinical features, diagnosis, and management of HOI published between 2010 and 2021. Online meetings were conducted from February 2021 to March 2022 to reach a consensus on these recommendations which are made from an Indian perspective.

Large right temporal mass in young adolescent

A 16-year-old White woman presented with a facial lesion on the right side (Fig 1) that rapidly enlarged over 3 months. Patient denies previous trauma and personal or family history of melanoma. Physical examination revealed a well-circumscribed right temporal mass with a base measuring approximately 2 cm. No palpable lymphadenopathy of cervical, submandibular, or submental clusters were noted. The excisional biopsy sample stained with hematoxylin and eosin (Fig 2, A) revealed ulcerated skin with underlying inflammation and benign vascular proliferation with a variety of patterns; there were clusters of spindle cells and small capillary vessels to large dilated vessels.

The capillary lobule variant of radiation-associated angiosarcoma in the setting of breast cancer: A diagnostic pitfall.

Post-radiation angiosarcoma is an iatrogenic event seen in the setting of breast cancer treatment. Histopathologically, there are morphologic variants of angiosarcoma that mimic benign entities, including the capillary lobule variant of post-radiation angiosarcoma. We present the largest case series to date of this histopathologic variant of post-radiation angiosarcoma. Cases of the capillary lobule variant of post-radiation angiosarcoma from institutional/consultation archives from 2008 to June 2022 were reviewed. For inclusion, tumors had to occur in irradiated skin and exhibit a multi-lobular proliferation of tightly packed capillary-like vessels, as previously described in this variant. Prior ancillary studies were also reviewed. Eight cases met the criteria. All occurred in women treated with radiation for breast cancer (median age 75 years). All cases had similar findings, including a multi-lobular proliferation of tightly packed vessels, infiltrative cords, and atypical single endothelial cells. A conventional angiosarcoma pattern was also seen in five cases. All cases tested were positive for vascular markers (CD31, CD34, and/or ERG) and MYC. MYC amplification was shown by FISH in all cases tested. Smooth muscle actin (SMA) was positive in pericytes in the capillary lobules in all five cases tested and areas of conventional angiosarcoma in two of three cases. The capillary lobule variant of angiosarcoma is a rare and therefore potentially under-recognized variant of post-radiation angiosarcoma. The lobular architecture and SMA positivity may mimic benign vascular proliferations. Careful attention to histopathologic features and ancillary tests may facilitate accurate diagnosis.

Topical treatment of pyogenic granulomas in a pediatric population: A single-institution retrospective review

To the Editor: Pyogenic granulomas (PG) are common benign vascular proliferations of the skin and mucous membranes. Because of their propensity to grow and bleed, surgical management is favored, but it can be costly, traumatic, result in scarring, or require an in-office or operative suite setting. Noninvasive treatment modalities include topical beta-blockers,1 imiquimod,2 and corticosteroids.3 By promoting vasoconstriction and downregulating proangiogenic factors such as vascular endothelial growth factor A, matrix metalloproteinase 1, and interleukin 6, topical corticosteroids potentially target several key pathways implicated in the PG pathogenesis.

Splenic Hamartoma in a Teenage: A Rare Case Report

Splenic hamartoma is a rare, benign vascular proliferation that is often found incidentally while working up for other complaints or at autopsy. Women more commonly present with symptoms related to mass effect than man. Although rendering a diagnosis can be difficult, endothelial cells that are positive for CD8 are a key feature that differentiate hamartoma from other vascular lesions of the spleen. Clinical, radiologic, and histologic correlation is essential to ensure this benign lesion is not mistaken for malignancy. In our case, a teenage was diagnosed with splenic hamartoma.

Cutaneous reactive angiomatosis associated with intravascular cryoprotein deposition as the presenting finding in a patient with underlying lymphoplasmacytic lymphoma: A case report and review of the literature

Cutaneous reactive angiomatosis, a group of disorders defined by benign vascular proliferation, is associated with a number of systemic processes, including intravascular occlusion by cryoproteins. We report a case of a 64-year-old female patient who presented with a 1-year history of nontender petechiae of the bilateral arms and lower legs. Dermoscopic evaluation showed increased vascularity with a globular pattern. Over a period of months, her findings progressed to erythematous to violaceous plaques with admixed hypopigmented stellate scarring of the bilateral lower extremities, forearms, and lateral neck. Biopsy showed increased thin-walled, small dermal blood vessels with focal inter-anastamosis. Some vessels were occluded by eosinophilic globules suspicious for cryoprotein. Subsequent laboratory studies confirmed a diagnosis of type 1 cryoglobulinemia, prompting a bone marrow biopsy that revealed lymphoplasmacytic lymphoma. Herein, we report the fourth case of angiomatosis secondary to intravascular cryoproteins as the initial presentation of an underlying hematologic malignancy. We also present a review of the literature and emphasize the need for thorough initial workup and close and prolonged clinical monitoring for underlying systemic disease in these patients.

Percutaneous ethanol sclerotherapy is a promising treatment for recalcitrant angiolymphoid hyperplasia with eosinophilia

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign vascular proliferation, which manifests as characteristic red nodules and papules, mostly located on the scalp and periauricular regions. Patients seek treatment for both aesthetic and functional reasons, as lesions may ulcerate, bleed and itch. Many therapeutic approaches have been reported, with variable success, and relapse remains a troublesome issue. The aim of this study was to report our experience treating ALHE using percutaneous ethanol sclerotherapy (PES). We present a retrospective case series of three patients treated with PES (1-2 treatment sessions each). All patients had tried and failed other treatments prior to this intervention, but following PES treatment, all patients demonstrated significant improvement, which was sustained at follow-up (range 8-17 months after first treatment). Adverse effects were tolerable and transient. This case series demonstrates PES as a promising treatment for recalcitrant ALHE.

Topical corticosteroids for noninvasive treatment of pyogenic granulomas

Pyogenic granulomas are benign vascular proliferations of the skin and mucous membranes that tend to bleed easily. They typically require procedural treatments that can be difficult for patients with intellectual disabilities or behavioral concerns to tolerate. In our practice, we have found the use of topical clobetasol to be effective to induce regression of cutaneous pyogenic granulomas. We present here a case of an adolescent patient with autism and two bleeding pyogenic granulomas who poorly tolerated a biopsy of the first lesion and could not tolerate subsequent procedures. Topical therapy with clobetasol effectively managed the second pyogenic granuloma, an approach representative of a noninvasive practice utilized in our clinic.

Rare TERT Promoter Mutations Present in Benign and Malignant Cutaneous Vascular Tumors

Mutations in the promoter of the telomerase reverse transcriptase (TERT) gene have been described as the most common hot-spot mutations in different solid tumors. High frequencies of TERT promoter mutations have been reported to occur in tumors arising in tissues with low rates of self-renewal. For cutaneous vascular tumors, the prevalence of TERT promoter mutations has not yet been investigated in larger mixed cohorts. With targeted next-generation sequencing (NGS), we screened for different known recurrent TERT promoter mutations in various cutaneous vascular proliferations. In our cohort of 104 representative cutaneous vascular proliferations, we identified 7 TERT promoter mutations. We could show that 4 of 64 (6.3%) hemangiomas and vascular malformations harbored TERT promoter mutations (1 Chr.5:1295228 C > T mutations, 1 Chr.5:1295228_9 CC > TT mutation, and 2 Chr.5:1295250 C > T mutations), 1 of 19 (5.3%) angiosarcomas harbored a Chr.5:1295250 C > T TERT promoter mutation, and 2 of 21 (9.5%) Kaposi’s sarcomas harbored TERT promoter mutations (2 Chr.5:1295250 C > T mutations). To our knowledge, this is the first general description of the distribution of TERT promoter mutations in a mixed cohort of cutaneous vascular tumors, revealing that TERT promoter mutations seem to occur with low prevalence in both benign and malignant cutaneous vascular proliferations.

GNA14, GNA11, and GNAQ Mutations Are Frequent in Benign but Not Malignant Cutaneous Vascular Tumors.

Cutaneous vascular tumors consist of a heterogeneous group of benign proliferations, including a range of hemangiomas and vascular malformations, as well as heterogeneous groups of both borderline and malignant neoplasms such as Kaposi’s sarcoma and angiosarcomas. The genetics of these tumors have been assessed independently in smaller individual cohorts making comparisons difficult. In our study, we analyzed a representative cohort of benign vascular proliferations observed in a clinical routine setting as well as a selection of malignant vascular proliferations. Our cohort of 104 vascular proliferations including hemangiomas, malformations, angiosarcomas and Kaposi’s sarcoma were screened by targeted next-generation sequencing for activating genetic mutations known or assumed to be potentially relevant in vascular proliferations. An association analysis was performed for mutation status and clinico-pathological parameters. Frequent activating hotspot mutations in GNA genes, including GNA14 Q205, GNA11 and GNAQ Q209 were identified in 16 of 64 benign vascular tumors (25%). GNA gene mutations were particularly frequent (52%) in cherry (senile) hemangiomas (13 of 25). In angiosarcomas, activating RAS mutations (HRAS and NRAS) were identified in three samples (16%). No activating GNA or RAS gene mutations were identified in Kaposi’s sarcomas. Our study identifies GNA14 Q205, GNA11 and GNAQ Q209 mutations as being the most common and mutually exclusive mutations in benign hemangiomas. These mutations were not identified in malignant vascular tumors, which could be of potential diagnostic value in distinguishing these entities.

Giant pyogenic granuloma complicating a folliculitis decalvans

<p class="abstract">Pyogenic granuloma or lobular capillary hemangioma is a common and benign vascular proliferation that affects the skin and mucosa. It that can take many forms and be misleading in some cases. Its giant variant is very rare. The pathogenesis is unknown but multiple factors can play a role on its onset. It is considered to be a pseudotumoral reaction to various stimuli, such as trauma, hormonal changes or the use of some medications. We report the case of a 35 years old male patient who presented with a large, ulcerated tumefaction of the scalp evolving since 4 months. The physical examination revealed a crescent shaped large ulcerated tumor with signs folliculitis decalvans in the rest of the scalp. A skin biopsy was made and confirmed the diagnosis of pyogenic granuloma. The treatment consisted of a shave excision of the entire lesion. No recurrences were noted after two years of follow-up. This case illustrates the possible association of pyogenic granuloma with underlying dermatoses such as folliculitis decalvans.</p>

Skin tumors in xeroderma pigmentosum: Evaluation of a large series and a literature review

Xeroderma pigmentosum (XP) is a rare genodermatosis with a lifelong propensity to develop malignant skin tumors. In this retrospective study, 24 XP patients were evaluated with regard to frequency and clinicopathological features of benign and malignant skin tumors. Seventeen patients had at least one malignant skin tumor diagnosed: basal cell carcinoma (BCC) in 13 patients (n=72), basosquamous carcinoma in three patients (n=4), squamous cell carcinoma in six patients (n=13), keratoacanthoma in three patients (n=15), and melanoma in six patients (n=18). Most melanomas (n=15) were in situ lesions. Several benign skin tumors were noted such as tricholemmoma (n=1), trichoepithelioma (n=1), trichoblastoma (n=1), follicular infundibulum tumor (n=1), keratoacanthoma-like follicular lesion (n=1), adnexal tumors with folliculosebaceous (n=1) and tricholemmal differentiation (n=1), and neurofibroma (n=1). Benign vascular proliferations including pyogenic granulomas (n=8), widespread telangiectasias, and senile angioma-like lesions were also observed in 3, 5, and 5 patients, respectively. Similar to many reports, BCC was found to be the most common malignant skin tumor. The high prevalence of benign adnexal tumors of follicular differentiation, some of them showing mixed histopathological features and various vascular proliferations in our series raises the question of whether they indicate a formerly undescribed association with XP.

A Review of Proliferative Vascular Disorders of the Central Nervous System of Animals.

In disease, blood vessel proliferation has many salient roles including in inflammation, when granulation tissue fills superficial defects, or in the recanalization of an occluded blood vessel. Sometimes angiogenesis goes awry-granulation can be exuberant, and plexiform proliferation of vascular components can contribute to pulmonary hypertension. This review focuses on the diverse manifestations of pathologic vascular overgrowth that occur in the brain, spinal cord, and meninges of animals from birth until old age. Entities discussed include systemic reactive angioendotheliomatosis in which glomeruloid vascular proliferations are encountered in various organs including the central nervous system (CNS). The triad of CNS vascular malformations, hamartomas, and benign vascular proliferations are an especially fraught category in which terminology overlap and the microscopic similarity of various disorders makes diagnostic classification incredibly challenging. Pathologists commonly take refuge in "CNS vascular hamartoma" despite the lack of any unique histopathologic features and we recommend that this diagnostic category be abandoned. Malformative lesions that are often confusing and have similar features; the conditions include arteriovenous malformation, cavernous angioma, venous angioma, and capillary telangiectases. Meningioangiomatosis, a benign meningovascular proliferation with dual components, is a unique entity seen most commonly in young dogs. Last, accepted neoplastic conditions range from lower-grade locally acquired growths like hemangioblastoma (a tumor of mysterious interstitial stromal cells encountered in the setting of abundant capillary vasculature proliferation), the rare hemangioendothelioma, and the highly malignant and invariably multifocal metastatic hemangiosarcoma. Additionally, this review draws on the comparative medical literature for further insights into this problematic topic in pathology.

Treatment Modalities for Cherry Angiomas: A Systematic Review.

Cherry angiomas are benign vascular proliferations of endothelial cells associated with aging. Currently, no mainstay of treatment for these vascular anomalies exists. To review existing evidence-based therapies for the treatment of cherry angiomas. A literature search in May 2019 was performed with PubMed Database and Cochrane Library using the following terms: "cherry angioma," "senile hemangioma," "senile angioma," "cherry hemangioma," and "Campbell de Morgan spots." Ten studies included in this systematic review reported laser therapy and nonlaser therapy as efficacious treatments for cherry angiomas. Among the laser therapies, pulsed dye laser (PDL) was preferred over potassium-titanyl-phosphate (KTP) and electrodessication (ED), based on decreased procedure-related pain. The neodymium-doped yttrium aluminum garnet (Nd:YAG) laser 1064 nm produced less pigmentary complications, whereas KTP and PDL risked pigmentary changes in darker-skinned individuals. Nonlaser therapies included cryotherapy, sclerotherapy, electrosurgery (i.e., ED, electrocoagulation), and radiofrequency ablation. No therapy proved to be superior. A variety of therapeutic modalities exist for the treatment of cherry angiomas. However, a limited number of high-quality studies explored the efficacy of treatments and compared treatment modalities. Light-based methods such as argon, KTP, Nd:YAG, intense pulsed light, and PDL, along with non-light-based interventions such as cryotherapy, electrosurgery, and sclerotherapy effectively treated cherry angiomas.

Primary Nodal Lymphangioma: A Rare Case Report and Literature Review

Abstract Introduction/Objective Lymphangiomas are benign nodal vascular tumors commonly seen in the skin, mucosa, and soft tissue; however, its occurrence in lymph nodes is extremely rare, with only a few cases reported in English literature. Methods We report a case of a 20-year-old female who presented with a 6 cm right upper thigh mass below the inguinal region that has been gradually increasing in size over the past three months. Ultrasound showed a cystic appearance. An excisional biopsy was performed to yield a thin-walled cystic mass filled with clear fluid. The mass appeared to be connected to a regional lymph node. Histologic examination revealed a multiloculated thin-wall cyst involving the hilum of the lymph node. The cyst is lined by flat lymphatic endothelial cells with focally disorganized smooth muscle at the wall of large channels and peripheral lymphoid aggregates. The rest of the lymph node shows reactive change with vascular proliferation and congestion. Results Lymphangiomas are soft, relatively well-circumscribed hamartomatous malformations due to the failure of the lymphatic system to communicate with the venous system. This leads to abnormal rests of lymphatic tissue that proliferate and accumulate fluid, causing their cystic morphology. Lymphangiomas are classified as capillary, cavernous, and cystic forms. Imaging techniques such as ultrasonography, mammography, and magnetic resonance imaging may aid in clinical diagnosis and follow-up. Histologically, cavernous lymphangiomas show numerous narrow cavernous spaces lined by a single layer of attenuated endothelial cells, and containing amorphous eosinophilic fluid with few lymphocytes. These spaces may contain blood resulting in misdiagnosis of this lesion as cavernous hemangioma; however, increased lymphoid cells in the stroma, more irregular cavernous spaces, support the diagnosis of lymphangioma. Conclusion Lymphangiomas are slowly progressive painless lesions that frequently recur in cases of incomplete excision. It is important to identify this primary nodal lymphangioma to avoid misdiagnosis of benign vascular proliferation, benign vascular tumor, or malignant vascular tumor.

Characteristic appearance of spindle cell hemangiomatosis, often misdiagnosed as venous malformation: A retrospective study of 11 cases.

Spindle cell hemangiomatosis is a benign multifocal vascular proliferation that mostly occurs in the distal extremities. It is a relatively rare disease and causes difficulties in clinical diagnosis and differential diagnosis of venous malformation. We retrospectively assessed the medical history, and clinical features, imaging features, pathological features and follow up of 11 patients diagnosed pathologically with spindle cell hemangiomatosis after surgery. There are two types of clinical appearances in spindle cell hemangiomatosis in the distal extremities: bleb-like nodules and varix-like nodules. Bleb-like nodules are mostly superficially located in the palm or interphalangeal joints with obvious hemorrhage in lesions and mainly composed of cavernous spaces rather than spindle cells. Varix-like nodules are located in the back of the hands or arms with normal skin color and mainly composed of solid areas of accumulated spindle cells. Surgery is the standard therapy for spindle cell hemangiomatosis, while sclerotherapy is invalid. Despite the tendency to develop new lesions, there are no residual lesions at the surgical site during follow up. Bleb-like nodules in the palm and interphalangeal joints are a typical clinical appearance in spindle cell hemangiomatosis which can help make clinical and a differential diagnosis of venous malformation. Surgery is the standard therapy for spindle cell hemangiomatosis with no residual lesions remaining at the surgical sites.