Men, Clara J.; Ediriwickrema, Lilangi S.; Paik, Ji Sun; Murdock, Jennifer; Yen, Michael T.; Ng, John D.; Liu, Catherine Y.; Korn, Bobby S.; Kikkawa, Don O.
To examine the role of adjuvant surgical resection of infantile hemangiomas after systemic β-blocker therapy.
This is a multicentered retrospective study. Standard protocol for oral propranolol was employed by the referring physicians. Ocular indications for surgery included ptosis obstructing the visual axis, high degrees of astigmatism causing amblyopia, or disfigurement from residual tumor. Patients underwent complete excision or debulking.
Eleven girls and 4 boys were surgically treated with mean operative age of 34.4 months. Patients were followed for a mean of 19.6 months after surgery. Four patients required surgical treatment due to an inability to tolerate medical therapy secondary to drug-related side effects (including bradycardia). The other 11 patients proceeded to surgery due to residual eyelid and orbital lesions despite medical treatment. All 15 patients underwent orbitotomy for residual hemangioma excision. Four patients also underwent simultaneous levator advancement at the time of excision. In all cases, there was resolution of ptosis with clearing of the visual axis. No complications were incurred during the surgical treatment and there were no hemangioma recurrences.
This is the first study to report surgical management of periocular infantile hemangiomas recalcitrant to standard therapy in the β-blocker era. In patients with infantile hemangioma who have failed medical therapy, adjuvant surgical treatment still plays an important role. For patients with persistent tumor causing ocular sequelae, surgical intervention aimed at soft tissue debulking and ptosis repair can be successful in achieving excellent functional and aesthetic outcomes with minimal side effects.
For patients with periocular infantile hemangiomas with residual soft tissue deformity following propranolol therapy, surgical treatment plays an important role in improving functional and cosmetic outcomes with minimal side effects.
Bradfield, Yasmin; Burkat, Cat N.; Albert, Daniel M.; Potter, Heather A.D.
A 7-year-old healthy girl presented for an evaluation of a left vascular scleral mass. The lesion appeared spontaneously with no history of trauma, coagulopathy, or topical medication use. It was nontender, enlarging, and did not extend intraocularly. Her OS vision was 20/20, and the remainder of her eye examination was normal. Evaluation of the ocular mass included B-scan ultrasound, ultrasound biomicroscopy, anterior segment optical coherence tomography (OCT), and orbital MRI. The anterior segment OCT demonstrated vessels within the mass with no defined capsule. The orbital MRI confirmed a lesion isolated to the scleral layers of the globe, with low blood flow. The patient had a partial response to oral propranolol. Because the lesion vessels began to extend into her corneal endothelium, there was a concern for malignancy. A biopsy confirmed a benign intrascleral capillary hemangioma. Discontinuation of the propranolol demonstrated stability of the lesion 6 months later.
Jamshidian-Tehrani, Mansooreh; Nabavi, Amin; Taghavi, Mostafa; Sharif-Kashani, Shervin; Kasaee, Abolfazl; Sadeghi-Tari, Ali; Mirhosseini, Mohammad; Nowroozzadeh, M. Hossein
Purpose: To evaluate the clinical and ultrasonographic response of periocular infantile capillary hemangioma during treatment with oral propranolol.
Methods: Patients with infantile periocular hemangioma and visual or cosmetic concerns were enrolled in this prospective interventional case series. Propranolol was given at a dose of 2 mg/kg per day for at least 6 months. Evaluation of treatment response was performed at month 3 (time point 1) and month 6 (time point 2). Gray scale ultrasonography and color Doppler imaging were performed at baseline and month 3.
Results: Thirty-one patients with mean age of 4.1 ± 2.3 months were eligible for analysis. Complete or near complete clinical resolution was observed in 4 patients (12.9%) at time point 1 and 21 patients (67.7%) at time point 2. Longitudinal diameter, transverse diameter, thickness, arterial peak systolic velocity, and end diastolic velocity reduced significantly from baseline to 3-month follow up. Complete clinical response at time point 2 was significantly higher in patients with peak systolic velocity reduction >50% from baseline to month 3 than patients with peak systolic velocity reduction of 10% to 50% and <10%.
Conclusions: Propranolol is safe and effective for infantile periocular hemangioma. Ultrasonography and color Doppler imaging are useful modalities to monitor and predict the treatment response.
Al-Haddad, Christiane; El Salloukh, Nasrine Anais; El Moussawi, Zeinab
Purpose of review To evaluate the medical literature on the use of β-blockers, through different routes, for the treatment of periorbital infantile hemangiomas and to summarize the recommendations available on dosage and monitoring.
Recent findings β-blockers for the treatment of infantile hemangioma are now considered to be first-line treatment. Growing literature on the role of oral propranolol confirmed its efficacy but also presented its multiple side-effects including hypotension, bradycardia, hypoglycemia, and bronchospasm. No universal guidelines exist concerning pretreatment evaluation, dosage, monitoring, and duration of treatment but different protocols have been created.
In the aim of minimizing side-effects, other routes of administration and more selective β-blockers have emerged. Many studies showed promising results for topical timolol especially in the treatment of superficial hemangiomas. Few studies evaluated intralesional propranolol. Limited data exist on the use of more selective β-blockers promising similar results to propranolol with fewer side-effects.
Summary Oral β-blockers are now the mainstay of treatment for periorbital hemangiomas but still with no consensus on their administration and monitoring. The topical form or more selective β-blockers may be the solution to minimize side-effects.
Giorgio Albanese, Padma Mohandas, Louise Wells, Jane Ravenscroft, Jothsana Srinivasan, Shery Thomas, Timothy Taylor & Katya Tambe
Aim: The aim of this study is to report a retrospective case series on orbital infantile haemangiomas (OIH). Radiological features and treatment with oral propranolol (OP) are illustrated along with an updated literature review. Methods: A retrospective chart review of six children, diagnosed with OIH from November 2015 to October 2016, was carried out. Only children with deep documented orbital involvement were included. All patients underwent magnetic resonance imaging (MRI) under general anaesthesia. OP was administered to the infants according to the Nottingham Children’s Hospital guideline. As per the guideline, a preliminary paediatric assessment was performed and a 1 mg/kg test dose was administered, followed by definitive treatment at a dosage of 2 mg/kg in three divided doses.Results: Average age at presentation was within the first 3 weeks of life. T1 hypointensity, T2 hyperintensity, avid enhancement with contrast, and the presence of flow-voids appear a fixed pattern of OIH on MRI. Response to treatment was noticed within 4 weeks in all children, and two of them (33.3%) responded within the first 7 days. In two children (33.3%), the haemangioma became clinically undetectable by the seventh month of treatment, while the other four (66.6%) experienced an almost complete regression of the OIH by the last follow-up. No complications were found.Conclusions: Our series strengthens the understanding that MRI is the preferred imaging modality in the investigation of OIH, showing vascular features, detailed orbital extension, and possible associated malformations. OP is the treatment of choice for OIH, and our study confirms its safety and effectiveness.
Sang Min Lee, Sang In Khwarg, Jung Hyo Ahn
Epithelioid hemangioma (also known as angiolymphoid hyperplasia with eosinophilia) is an unusual, benign, vascular lesion of endothelial cell proliferation, which usually arises in the head and neck region.1–4 Several treatments exist for epithelioid hemangioma of the orbit, including systemic/intralesional steroid therapy, radiotherapy, and chemical therapy.1–4 However, surgical excision remains the most common treatment option. Recently, systemic propranolol has been proposed as an alternative therapy in cases of incomplete tumour removal or cases of recurrence after surgery.
McClintic, Elysa A. M.S. M.D.; Ting, Andrew M.D.; Yeatts, R. Patrick M.D.
An 8-year-old female patient presented with left upper eyelid swelling and erythema. Magnetic resonance imaging revealed an orbital mass involving the left lacrimal gland with subsequent incisional biopsy leading to the diagnosis of angiolymphoid hyperplasia with eosinophilia. Initially prescribed an oral corticosteroid, alternative management was sought after 4 months due to unwanted side effects of steroid therapy. Oral propranolol (2 mg/kg/day) was initiated with concurrent steroid taper. Interval decrease in lesion size was observed on subsequent magnetic resonance imaging with complete resolution of subjective symptoms (Fig. 1). She remains stable 14 months after starting beta-blocker therapy. To our knowledge, our case is the second case report suggesting oral beta-blocker may be an alternative therapy for orbital angiolymphoid hyperplasia with eosinophilia.