Synovial hemangioma presenting with chronic painful infrapatellar mass: a case report.

BACKGROUND: Synovial hemangiomas are rare benign vascular anomalies surrounded by a synovial lining and were first described by Bouchut in 1856. These neoplasms can develop in the intra-articular region, resulting in effusions and knee pain. However, their cause remains unknown. Prompt diagnosis and intervention are critical to prevent chondral damage. Histopathological examination is used to achieve the diagnosis, which is often delayed because of a lack of specific clinical signs. This report describes a unique case in which a painful infrapatellar mass was diagnosed as a synovial hemangioma. The absence of typical magnetic resonance imaging (MRI) findings highlights the importance of arthroscopic excision for diagnosis and symptom relief. CASE PRESENTATION: A 20-year-old woman presented with persistent anterior left knee pain that became exacerbated when she climbed stairs. Despite previous pain management and physical therapy, she developed a painful lump beneath her patella that worsened over time. She had also undergone arthrocentesis, but this did not relieve her pain. Physical examination revealed a palpable, immobile 5-cm mass along the patellar tendon with limited knee flexion and extension and normal ligament stability. T1-weighted fat-saturated MRI of the left knee with gadolinium-based contrast revealed a lobulated intra-articular mass in Hoffa's fat pad that resembled a soft tissue chondroma. A biopsy of the mass was performed to provide histopathological evidence, confirming the benign nature of the mass. The subsequent excisional arthroscopy, combined with incision enlargement for mass removal, confirmed the histopathologic diagnosis of synovial hemangioma based on the presence of numerous dilated blood vessels and venous proliferation within sections of the synovium. Recovery was complete, and no residual tumor was detected on follow-up MRI after 1 year. CONCLUSION: This case study emphasizes the importance of arthroscopic excision over open surgery for patients with synovial hemangioma. The minimally invasive nature of arthroscopy combined with the well-encapsulated nature and location of the mass facilitates complete resection.

Placental ACE2 Expression: A Possible Pathogenetic Mechanism for Infantile Hemangiomas.

ACE2 is a mono-carboxypeptidase with remarkable vasculo-protective properties, and its expression in the human placenta plays a central role in blood pressure homeostasis and fetal perfusion. Therefore, an alteration in the placental expression of ACE2 could be responsible for reduced placental perfusion and infantile hemangioma (IH) development. Study placentae were collected from patients affected by IHs who were referred to our Dermatology Clinic from 2016 to 2022, while control placentae were randomly collected while matching cases for gestational age. Immunohistochemical investigations were performed with a recombinant anti-ACE2 rabbit monoclonal antibody. A total of 47 placentae were examined, including 20 study placentae and 27 control ones. The mean placental weight was significantly lower in the study group (380.6 g vs. 502.3 g; p = 0.005), while subclinical chorioamnionitis occurred more frequently in the study group (20% vs. 0%, p = 0.03). The mean ACE2 expression was dramatically lower in the study group (χ2 = 42.1 p < 0.001), and the mean placental weight was significantly lower when ACE2 was not expressed compared to the 25-75% and >75% classes of expression (p < 0.05). This study demonstrated that ACE2, as a marker for tissue hypoxia, is dramatically hypo-expressed in placentae belonging to mothers who delivered one or more babies with IH compared to the controls.

Cardiac Evaluation before and after Oral Propranolol Treatment for Infantile Hemangiomas.

Background: Most recent clinical practice guidelines addressing the management of infantile hemangiomas (IHs) recommend oral propranolol, a non-selective beta-adrenergic antagonist, as first-line treatment. However, few reports have provided continuous follow-up data regarding cardiac evaluations. Methods: Sixty-four patients diagnosed with IHs and treated with oral propranolol before 2 years of age at the Department of Pediatrics, Kangbuk Samsung Hospital (Seoul, Republic of Korea), with regular examinations between 2017 and 2021, were included. Cardiac evaluations, including electrocardiography, Holter monitoring, chest X-ray, and echocardiography, were performed. Results: Sixty-four patients with IHs successfully underwent continuous follow-up cardiac evaluations. The median age at diagnosis was 2 weeks (1 day to 34.3 weeks). The median age at treatment initiation was 13.6 weeks (2.4-87.9 weeks), the mean longitudinal diameter of hemangioma at diagnosis was 2.8 ± 2.1 cm (0.3-12.0 cm), and the mean percentage of size decrease after 1 year of oral propranolol treatment was 71.8%. None of the 64 patients experienced severe adverse side effects during propranolol treatment. There was no statistically significant differences in echocardiographic function and electrocardiographic data after treatment. Conclusions: Propranolol treatment ≥6 months was effective and safe without significant cardiac toxicity in the treatment of patients with infantile hemangiomas.

Anesthetic Management and Considerations in a Rare Case of Parietal Bone Hemangioma.

Parietal bone hemangiomas represent a minority of diagnosed brain tumors. These lesions require careful management under anesthesia due to their vascularity and cranial location. We discuss a 31-year-old female with chronic headaches who underwent surgery for the removal of a large parietal bone hemangioma, necessitating considerations for stable hemodynamics, intracranial pressure (ICP), and bleeding risks. There is no standard anesthetic for these cases, so a mixed anesthetic approach was used, combining intravenous anesthesia with sevoflurane, aimed at optimizing control during the procedure.

Recent Advances in Targeted Therapies for Infantile Hemangiomas.

Targeted therapy for infantile hemangiomas (IHs) has been extensively studied as they can concentrate drugs, increase therapeutic efficacy and reduce drug dosage. Meanwhile, they can extend drug release times, enhance drug stability, decrease dosing frequency, and improve patient compliance. Moreover, carriers made from biocompatible materials reduced drug immunogenicity, minimizing adverse reactions. However, current targeted formulations still face numerous challenges such as the non-absolute safety of carrier materials; the need to further increase drug loading capacity; the limitation of animal hemangioma models in fully replicating the biological properties of human infantile hemangiomas; the establishment of models for deep-seated hemangiomas with high incidence rates; and the development of more specific targets or markers. In this review, we provided a brief overview of the characteristics of IHs and summarized the past decade's advances, advantages, and targeting strategies of targeted drug delivery systems for IHs and discussed their applications in the treatment of IHs. Furthermore, the goal is to provide a reference for further research and application in this field.

GNA14 and GNAQ somatic mutations cause spinal and intracranial extra-axial cavernous hemangiomas.

Extra-axial cavernous hemangiomas (ECHs) are complex vascular lesions mainly found in the spine and cavernous sinus. Their removal poses significant risk due to their vascularity and diffuse nature, and their genetic underpinnings remain incompletely understood. Our approach involved genetic analyses on 31 tissue samples of ECHs employing whole-exome sequencing and targeted deep sequencing. We explored downstream signaling pathways, gene expression changes, and resultant phenotypic shifts induced by these mutations, both in vitro and in vivo. In our cohort, 77.4% of samples had somatic missense variants in GNA14, GNAQ, or GJA4. Transcriptomic analysis highlighted significant pathway upregulation, with the GNAQ c.626A>G (p.Gln209Arg) mutation elevating PI3K-AKT-mTOR and angiogenesis-related pathways, while GNA14 c.614A>T (p.Gln205Leu) mutation led to MAPK and angiogenesis-related pathway upregulation. Using a mouse xenograft model, we observed enlarged vessels from these mutations. Additionally, we initiated rapamycin treatment in a 14-year-old individual harboring the GNAQ c.626A>G (p.Gln209Arg) variant, resulting in gradual regression of cutaneous cavernous hemangiomas and improved motor strength, with minimal side effects. Understanding these mutations and their pathways provides a foundation for developing therapies for ECHs resistant to current therapies. Indeed, the administration of rapamycin in an individual within this study highlights the promise of targeted treatments in treating these complex lesions.

Recent Advances in Photodynamic Therapy for Vascular Abnormalities.

Abstract Background: Photodynamic therapy (PDT) is a minimally invasive therapy that was gradually established as a first-line treatment for vascular abnormalities. Its action depends on the appropriate wavelength of light and photosensitizer to produce toxic oxygen species and cause cell death. Objective: Several new clinical improvements and trends in PDT have been described in recent years. The aim of this review is to provide an overview of the current data from clinical trials. Methods: In this review, we introduce and generalize the wavelength, duration, dose, strength, and photosensitizer of PDT for the treatment of vascular abnormalities, such as circumscribed choroidal hemangiomas (CCH), choroidal neovascularization (CNV) and capillary malformation (CM). Results: The systematic review findings indicate that the application of PDT is a safe effective method to treat CCH, CNV and CM. However, PDT also has early onset side effects and late onset side effects. Conclusions: Based on the discussion of the effectiveness of PDT, we conclude that PDT has great potential for clinical use, although PDT has possible side effects.

Coexistence of mastoid, frontal and vertebral hemangiomas in a patient with diabetic neuropathy: Possible correlation between diabetic angiopathy and intraosseous neoangiogenesis.

Bony hemangiomas are benign vascular lesions with an expansive growth; usually they tend to obliterate the entire bony cavity. They are typical lesion of the spinal bones, but they can rarely arise within other bones of the neurocranium. Diabetic microangiopathy is a condition characterized by the development of aberrant vessel tangles anastomosed to each other due to dysregulated neoangiogenesis. We report the case of a 56-year-old woman, suffering from type 2 diabetes mellitus, admitted to the neurology department due to a reported worsening of paresthesias and dysesthesias of the upper and lower limbs. She performed a contrast-enhanced brain CT scan that showed the presence, at the level of the right mastoid process, of an hypervascular angioma. A subsequent MRI study of the brain and spine showed the presence of multiple bone angiomas, at the level of the right frontal theca and C7, Th3, and Th7 vertebral bodies. Due to the absence of further symptoms and clinical and radiological signs of intracranial compression, the patient did not perform surgery. A radiological follow-up was advised. Although possible pathophysiological correlations between diabetes and vertebral hemangiomas are mentioned in literature, vascular lesions of this type involving vertebrae and skull base simultaneously can be discovered in a patient with chronic diabetic disease. As long as these lesions remain asymptomatic, surgical treatment is not indicated, and the patient is followed over time with radiological follow-up.

Venous Malformation in the Anterior Mediastinum.

Venous malformations (VMs) located in the anterior mediastinum are rare. Thus, diagnosis using imaging is often challenging, and they are typically diagnosed only after total tumor resection. Herein, we report a case of VM located in the anterior mediastinum diagnosed using computed tomography (CT) and magnetic resonance imaging (MRI). A 56-year-old woman presented for further evaluation of an anterior mediastinal mass observed during a chest CT. On CT, the mass was observed to have scattered calcifications and early and persistent enhancement with contrast material pooling dorsally in the delayed phase. On MRI, the mass was isointense on T1-weighted imaging and hyperintense on T2-weighted imaging without flow voids. From these images, we suspected the mass to be a VM, but the possibility of an arterial malformation/fistula could not be ruled out. Initially, a contrast material was injected via the arm, but to improve differentiation, it was also injected via the leg. The 4D-CT of the leg indicated no early enhancement of the mass; however, gradual enhancement was observed. This led to a definite diagnosis of VM. As she had no symptoms, we opted for a CT follow-up, and the mass remained stable for one year post-diagnosis. This case report underscores the usefulness of injecting contrast material through the leg in distinguishing VM from AVM/Fs in the anterior mediastinum.

Multimodal imaging of a case of retinal cavernous hemangioma.

PURPOSE: Herein, we reported a case of retinal cavernous hemangioma (RCH) and its multimodal imaging. CASE PRESENTATION: Many grape-like lesions were incidentally found along the distal portion of the vascular arcade in a 23-year-old man's superonasal quadrant of the retina. The best-corrected visual acuity was 20/20 in both eyes. The fluorescein angiography showed saccular lesions that slowly filled with hypofluorescence in the early stage and clusters of aneurysms with hyperfluorescence and a special "fluorescein cap" appearance in the late stage. The optical coherence tomography angiography (OCTA) B-scan revealed multilocular cavernous spaces with different sizes in the inner retinal layers, varying signals, and an epiretinal membrane. The enface OCTA of retina showed grape-like changes and multiple highly reflective signal cavities with clear and complete boundaries. CONCLUSION: Different multimodal images are better for understanding the characteristics of RCH and more convenient for its diagnosis and follow-up.

Neonatal nasal necrosis: Case series and brief review.

There is limited information available on pressure-related neonatal nasal injuries. We present three neonates born with erythema and purpura of the nasal tip that subsequently ulcerated, then evolved into a thick eschar. Each healed well with conservative management but left behind significant scarring. The sharp demarcation and location of the lesions were suggestive of hypoxic tissue damage akin to halo scalp ring alopecia. Further investigation is necessary to elucidate the etiology and optimal management of this condition.

Hepatic Hemangioma: Review of Imaging and Therapeutic Strategies.

Hepatic hemangiomas are the most common benign liver tumors. Typically, small- to medium-sized hemangiomas are asymptomatic and discovered incidentally through the widespread use of imaging techniques. Giant hemangiomas (>5 cm) have a higher risk of complications. A variety of imaging methods are used for diagnosis. Cavernous hemangioma is the most frequent type, but radiologists must be aware of other varieties. Conservative management is often adequate, but some cases necessitate targeted interventions. Although surgery was traditionally the main treatment, the evolution of minimally invasive procedures now often recommends transarterial chemoembolization as the treatment of choice.

Surgical strategies in the management of aggressive spinal haemangiomas: Retrospective case series with literature review and a practical treatment algorithm.

Purpose: We studied the clinico-radiological features and treatment outcomes of patients with aggressive spinal haemangiomas. Methods: We undertook a retrospective review of 24 patients with aggressive spinal haemangiomas managed at our centre from 2004 to 2016. The cohort was divided into two groups. Group1 included patients managed from 2004 to 2009 while Group 2 was those treated between 2010 and 2016. Clinicoradiological features and treatment outcomes were studied. Results: Back pain (24/24) and myelopathy (18/24) were the most common presenting complaints. Over 80% (20/24) of patients, had involvement of the thoracic spine and more than 50% (13/24) had severe spasticity, being Nurick grade 4&5 at presentation. The various treatment modalities used were laminectomy with or without instrumented posterior fusion (10/24), corpectomy with instrumented fusion (10/24) and alcohol injection alone (4/24). Patients who were treated with surgery had significant clinical improvement at follow-up in both groups. Patients who underwent alcohol injection did not have any improvement in symptoms at follow-up. There was a change in our strategy in the later part of the series from a two staged anterior and posterior approach to a single staged posterior-only approach to address vertebral body disease with preoperative angioembolization. Conclusion: Haemangiomas are benign lesions with locally aggressive behavior in some cases. Results of conservative approaches such as alcohol injection in management of these lesions are discouraging. Aggressive surgical decompression combined with preoperative adjuncts such as angioembolization with or without stabilization reduces intra operative blood loss and results in good neurological recovery even in patients with severe myelopathy.

A Novel Zn(II) Coordination Polymer: Fluorescence Performances, Molecular Docking and Loaded with Bleomycin-Hydrogels on Hemangiomas.

In this study, we successfully synthesized an innovative 1D-Zn(II) coordination polymer, denoted as [Zn(L)(H2O)2]n, employing the Schiff base {4-[(2-hydroxy-3-methoxy-benzylidene)-amino]-benzoic acid} (H2L). The Schiff base was obtained through the reaction of 4-aminobenzoic acid and o-vanillin under slow volatilization conditions. The resulting compound exhibits remarkable green fluorescence emission properties, indicating its potential as a novel fluorescent and sensing material. Hydrogels based on hyaluronic acid (HA) and carboxymethyl chitosan (CMCS), denoted as HA/CMCS hydrogels, were synthesized using a chemical method. Additionally, we utilized bleomycin as a model drug to synthesize a novel bleomycin metal gel and assessed its anti-hemangioma activity. Molecular docking simulations revealed that the Zn complex can form stable bonds with the key target, involving the methoxy and carboxyl groups on the Zn complex.

Endoscopic endonasal transsphenoidal surgery for unusual sellar lesions: eight cases and review of the literature.

Background: Preoperative imaging for some unusual lesions in the sellar region can pose challenges in establishing a definitive diagnosis, impacting treatment strategies. Methods: This study is a retrospective analysis of eight cases involving unusual sellar region lesions, all treated with endoscopic endonasal transsphenoidal surgery (EETS). We present the clinical, endocrine, and radiological characteristics, along with the outcomes of these cases. Results: Among the eight cases, the lesions were identified as follows: Solitary fibrous tumor (SFT) in one case, Lymphocytic hypophysitis (LYH) in one case, Cavernous sinus hemangiomas (CSH) in one case, Ossifying fibroma (OF) in two cases; Sphenoid sinus mucocele (SSM) in one case, Pituitary abscess (PA) in two cases. All patients underwent successful EETS, and their diagnoses were confirmed through pathological examination. Postoperatively, all patients had uneventful recoveries without occurrences of diabetes insipidus or visual impairment. Conclusion: Our study retrospectively analyzed eight unusual lesions of the sellar region. Some lesions exhibit specific imaging characteristics and clinical details that can aid in preoperative diagnosis and inform treatment strategies for these unusual sellar diseases.

A rare pediatric scrotal hemangioma: A case report and comprehensive analysis of n Bloc Excision as definitive treatment.

INTRODUCTION AND IMPORTANCE: Hemangiomas, abnormal growths of vascular endothelial cells, are common vascular tumors in children. In contrast, their prevalence is 1-3 % in full-term newborns. Their unique location and associated complications can cause significant distress for patients and families. Prompt treatment is crucial to prevent complications such as fertility issues, hemorrhage, or other related problems. This case study explores the diagnosis and management of scrotal hemangiomas, emphasizing the importance of timely intervention. CASE PRESENTATION: A 10-year-old boy with a scrotal hemangioma since birth underwent surgical excision due to increased discomfort caused by a new lesion. 2.5 × 2.3 cm hemangioma was successfully removed through En Bloc Excision. The patient experienced a swift recovery, and histopathologic analysis confirmed the diagnosis of cavernous hemangioma. This case underscores the importance of careful monitoring and decisive intervention in managing scrotal hemangiomas. CLINICAL DISCUSSION: Scrotal hemangiomas often present challenges in diagnosis, mimicking malignant tumors. Clinical examination, ultrasonography, and histopathology are crucial for accurate diagnosis. Various surgical techniques, including En Bloc Excision, have been employed, with postoperative care tailored to individual cases. Precise preoperative diagnosis is essential to distinguish scrotal hemangiomas from other conditions and guide appropriate management strategies. CONCLUSION: This case study highlights the management of a scrotal hemangioma through En Bloc Excision, emphasizing the importance of considering definitive treatment options. The comprehensive scrotal examination is crucial for accurate diagnosis and effective management of this rare vascular tumor.

The efficacy of extracted Tanshinone compounds for infantile hemangiomas of the skin: results from a pilot study.

Background: Infantile hemangiomas (IHs) on skin are conventionally treated with beta blockers, pulsed dye laser (PDL), or surgery, either invasive or limited to clinical conditions. Our preclinical studies suggested that Tanshinone, extracted from Salvia miltiorrhiza (Tanshin), had a beneficial effect on IHs. Thus, we conducted a pilot clinical study to evaluate the safety and efficacy of topical Tanshinone compounds on superficial IHs. Methods: The single-armed pilot study included a total of 29 infants diagnosed with IHs. Thrice daily (at an interval of 6-8 hours) topical applications of Tanshinone were used for each patient. The primary response was the skin erythema index assessed by investigators using SkinColorCatch colorimeter instrument (Delfin). The Achauer score and the satisfaction of parents were also evaluated. Results: A total of 29 infants, 22 females (76%) and 7 males (24%), with a median age of 60 days (interquartile range, 45 to 99 days) were included. The position of IHs was distributed in the trunk (44.8%), head (34.5%), and limbs (20.7%). After 6 months of IHs treatment, the decrease in skin erythema index (baseline: 566.79±854.67 vs. after treatment: 467.97±1,118.39, P<0.001) was indicated. A total of 79.31% [23/29] of parents of the participants reported satisfaction on the responses after treatment. No serious side effects were documented. Conclusions: The topical use of Tanshinone compounds might be a potentially effective and noninvasive therapy in treating IHs.

Familial multiple lipomatosis associated with multiple cherry hemangiomas and moles: a rare case report.

Lipomas are common benign mesenchymal tumours, whereas lipomatoses are uncommon. Familial multiple lipomatosis (FML) is a rare syndrome characterized by multiple usually painless lipomas which may be associated with other conditions. FML is considered to be genetic, with various patterns of inheritance suggested. In this case report, we described a case of multiple familial lipomatosis that was misdiagnosed as dercum's disease.

Efficacy of 0.5% timolol maleate gel in the management of infantile hemangiomas.

Objective: This study aimed to evaluate the efficacy and safety of 0.5% timolol maleate gel, reported as an efficacious option for management of infantile hemangiomas (IH) in children. Methods: A retrospective study was conducted among patients diagnosed with IH from January 2019 to December 2021. All patients were treated 0.5% timolol gel. Data parameters, including photographs, at baseline and the final or most recent follow-up visit, were reviewed. Outcomes based on photographic assessment were categorized as excellent, good, fair or poor. Results: Sixty-four children with 76 lesions were enrolled. Median age was eight months (two months to 36 months) with most lesions (75.0%) presenting during the first year of life. Female preponderance (84.4%) was seen and the cervicofacial region was most commonly involved (52.6%). The majority of lesions (54, 84.4%) were solitary and most were treatment naïve (n=61, 80.3%). Excellent, good, fair, and poor responses were seen in 24 (31.5%), 39 (51.3%), 6 (7.9%), and 7 (9.2%) lesions. No complications were seen and no statistically significant difference was observed with respect to gender, age group, region involved and treatment naïve versus previously treated patients. Conclusion: Timolol maleate 0.5% gel is an effective and safe treatment option for IH irrespective of location of lesion, age and history of prior treatment.

From Severe Anemia to Intestinal Hemangiomatosis, a Bumpy Road-A Case Report and Literature Review.

Gastrointestinal hemangiomas (GIH) are unusual vascular tumors found anywhere alongside the GI tract, the small bowel being the most common site. Diagnosis requires good clinical insight and modern imaging. This is a comprehensive review of the literature, starting from a new pediatric case diagnosed through exploratory laparotomy after complex imaging techniques failed. This research was conducted on published articles from the past 25 years. We identified seventeen original papers (two series of cases with three and two patients, respectively, and fifteen case reports). The female/male ratio was 1.5. The youngest patient was a 3-week-old boy, and the was oldest a 17-year-old girl. The most common localization was the jejunum (eight cases), followed by the ileum (four), colon (three), stomach (two), and rectum (one). Seven children had cavernous and four had capillary hemangiomas. Eight patients presented gastrointestinal bleeding, seven had refractory anemia, such as our index patient, three had recurrent abdominal pain, and two had bowel obstruction. Surgical assessment was successful in fifteen cases; three cases experienced great outcomes with oral propranolol, one child was treated successfully with sirolimus, and for one patient, endoscopic treatment was the best choice. The authors present the case of a female patient admitted to the Pediatrics Department of "Grigore Alexandrescu" Emergency Children's Hospital from 25 February to 28 March 2019 for severe anemia, refractory to oral iron treatment, and recurrent blood infusions. No clear bleeding cause had been found. Although very uncommon, intestinal hemangiomas can express puzzling, life-threatening symptoms. We should keep in mind this disorder in cases of unresponsive chronic anemia.