Successful Management of a Large Orbital Hemangioma in an Infant: A Case Report.

Orbital hemangiomas are benign vascular tumors commonly affecting infants and young children, often manifesting with proptosis, eyelid swelling, or discoloration. Propranolol has emerged as the primary therapy due to its efficacy in promoting regression and minimizing complications. Here, we present a case of a previously healthy six-month-old male infant with progressive right eyelid swelling and discoloration. Magnetic resonance imaging confirmed a large orbital hemangioma. The patient was referred to an ophthalmology center where treatment with propranolol resulted in substantial improvement. Early recognition and initiation of propranolol therapy are crucial in managing orbital hemangiomas in pediatric patients. This case underscores the successful outcome achievable with pharmacologic intervention and emphasizes the importance of long-term follow-up for monitoring and optimizing patient outcomes.

Predictors of poor response to oral propranolol in infantile hemangiomas.

BACKGROUND: Propranolol is the first-line treatment for infantile hemangiomas (IH). Cases of propranolol-resistant infantile hemangiomas are rarely reported. The purpose of our study was to investigate the predictive factors for poor response to propranolol. METHODS: A prospective analytical study was conducted between January 2014 and January 2022 including all patients with IH who received oral propranolol therapy at a dose of 2-3 mg/kg/day maintained for at least 6 months. RESULTS: A total of 135 patients with IH were treated with oral propranolol. Poor response was reported in 18 (13.4%) of the patients: 72% were girls and 28% were boys. Overall, 84% of the IH were mixed, and hemangiomas were multiple in three cases (16%), nasal tip hemangiomas accounted for four cases (22%), and 15 patients (83%) had segmental hemangiomas. There was no significant association between the age or sex of the children and type of response to treatment (p > 0.05). No significant association was found between the type of hemangioma and the therapeutic outcome as well as the recurrence after treatment discontinuation (p > 0.05). Multivariate logistic regression analysis revealed that nasal tip hemangiomas, multiple hemangiomas, and segmental hemangiomas were at greater risk of poor response to beta-blockers (p < 0.05). CONCLUSION: Poor response to propranolol therapy has rarely been reported in the literature. In our series, it was approximately 13.4%. To our knowledge, no previous publications have focused on the predictive factors of poor response to beta-blockers. However, the reported risk factors for recurrence are discontinuation of treatment before 12 months of age, mixed or deep type IH, and female gender. In our study, the predictive factors for poor response were multiple type IH, segmental type IH, and location on the nasal tip.

A multi-step approach to the treatment of giant scalp congenital hemangiomas: a report of two cases.

This is a review of two cases of neonatal giant scalp congenital hemangioma. Both patients were treated with propranolol using a similar multi-step approach that included transarterial embolization of the supplying arteries followed by surgical resection of the lesion. In this report, we discuss the treatments, complications, and clinical outcomes of interventions and surgical procedures.

"Face(s)" of a PHACE(S) Syndrome Patient before and after Therapy: Particular Case Report and Review of Literature.

A rare, uncommon disorder called PHACE(S) (P-posterior fossa anomalies, H-hemangioma, A-arterial anomalies, C-cardiac anomalies, E-eye anomalies, and S-sternal cleft) of unknown etiology was rarely reported. Children are susceptible to developing PHACE(S) syndrome from the moment they are born. It may be challenging for a physician to appropriately diagnose and treat children with PHACE due to the multifaceted nature of the disease and the extensive range of consequences that may be associated with it. A one-month-old newborn girl was admitted to hospital with extensive, multiple facial infantile hemangiomas, ulceration of the lower lip hemangioma-like lesion, cardiovascular, sternal, and neurological concomitant malformations. Five days following the initial application of the medication, systemic treatment with propranolol and topical treatment with silver sulfadiazine produced their first noticeable benefits. The lip ulceration was mostly healed and facial hemangioma started to regress. The regression continued under therapy and this effect persists for 6 months since Propranolol therapy ended. No cardiovascular or neurological clinical events have been registered during follow-up. The present case has three peculiarities: (1) high number of facial hemangiomas; (2) presence of subependymal cyst not yet reported in the literature associated with PHACE syndrome; and (3) lack of cardiovascular events during therapy knowing that these events frequently appear in PHACE syndrome patients.

Real-time optical imaging of the hypoxic status in hemangioma endothelial cells during propranolol therapy.

Infantile hemangioma (IH) is the most common microvascular tumor of infancy involving the area of head and neck. One of the most important independent risk factors of IH is the hypoxia microenvironment. Fluorescent chemosensor provides a noninvasive intervention, high spatiotemporal resolution, ultrasensitive response, and real-time feedback approach to reveal the hypoxic status of cells. Our research group developed an ultrasensitive fluorescent chemosensor, HNT-NTR, and investigated the potential ability of imaging the hypoxic status of hemangioma-derived endothelial cells (HemECs). In this study, we successfully visualized the propranolol (PRN) treatment in HemECs using NHT-NTR with "Turn-off" sensing method. This chemosensor exhibited high sensitivity and selectivity for optical imaging of hypoxic status with fast responsiveness, real-time feedback and durable photostability of the fluorescent signal. It was also confirmed that HNT-NTR could monitor nitroreductase in vivo. Paramountly, we expected this chemosensor to offer an available optical method for imaging of the hypoxic status and visualizing the therapeutic status of PRN therapy in IH with the hypoxia-imaging capability.

A Clinicopathological Study to Assess the Role of Intralesional Sclerotherapy Following Propranolol Treatment in Infantile Hemangioma.

Context: As propranolol has emerged as first-line therapy for problematic infantile hemangioma, the number of non-responders and partial responders to propranolol therapy is also increasing. Aims: The study was conducted to evaluate the response of intralesional bleomycin, triamcinolone, and a combination of both as second line of treatment for the residual hemangioma following propranolol therapy. Settings and Design: A prospective comparative study was conducted in patients who were either non-responders or partial responders to previous propranolol treatment. Materials and Methods: The patients randomly received injection bleomycin, injection triamcinolone, and combination of both bleomycin and triamcinolone. The response to treatment was recorded clinically by using photographs. The pathological response was assessed by calculating pre-treatment and post-treatment microvessel density in biopsy of lesion from the non-cosmetic sites using immunohistochemistry. Statistical Analysis Used: χ2 test was used to test the association between the variables. The utility of microvessel diameter (MVD) in terms of clinical response to the therapy was predicted by using receiver operating characteristic (ROC) curve. Results: Out of the 134 patients, 42 received bleomycin and 44 received triamcinolone and were treated with a combination of both. The overall clinical response was better in the combination group compared with the bleomycin group (P = 0.018) and triamcinolone group (P = 0.0005), respectively, after 6 months of follow-up. There was no difference in clinical response between the triamcinolone and bleomycin groups. Change in MVD correlated with the clinical response. Conclusion: The combination of bleomycin and triamcinolone is effective and safe for the treatment of residual hemangioma.

Evidence-Based Medicine in the Treatment of Infantile Hemangiomas.

Over the past decade, the treatment of infantile hemangiomas has undergone dramatic breakthroughs. This review critically evaluates the latest literature that supports the myriad treatment options for infantile hemangiomas. It chronicles the fading role of steroid therapy and evolution of propranolol use as the major treatment modality. Although propranolol is helping this disease become more of a medical disease and less of a surgical dilemma, the report also reveals a continued search to find nonsystemic treatment options. In summary, this is an evidence-based medicine review for the treatment of infantile hemangiomas.

Propranolol therapy in a case of capillary hemangioma.

Capillary hemangioma is one the most common tumors of eyelid and orbit reported in pediatric age group. Oral propranolol therapy is latest addition to the armamentarium of treatment options available to ophthalmologists in treating capillary hemangiomas. We report the successful response to propranolol therapy to a 5-year-old child with capillary hemangioma involving lids, orbit, and the paranasal sinuses. A long-term follow-up is necessary for the prognostic efficacy of the therapy.