Periocular capillary hemangioma treated with low dose oral propranolol - presentation and outcome of 30 patients

Purpose: To evaluate the presentation and outcome of periocular capillary hemangioma treated with low-dose oral propranolol. Method: Thirty cases of periocular capillary hemangioma prospectively studied from 1st June 2015 to 31st May 2017 who received oral propranolol on an outpatient basis. Hemangioma causing any threat to vision or disfigurement was included and age below 3 months and multiple lesions were excluded. Starting dose of propranolol was 1 mg/kg and increased to 2 mg/kg after 2 weeks as a maintenance dose. The tapering dose was 1 mg/kg of body weight before discontinuing the medication. Treatment was continued till the child is 1 year of age or no further change in color or size of the lesion in two successive follow-ups. Results: Presenting age was 6.36 ± 3.36 months (ranged 3–24 months) with female predominance (70%). In 86.6% of cases, the vision was Central Steady and Maintained and cycloplegic refraction showed marked astigmatism in 3 children which resolved after treatment. Forty-six percent of children showed color change as an initial response to treatment. Most children (33.3%) responded completely within 5 months after starting the treatment. One third patients (33.3%) showed 100% resolution, 50% showed 90% to 70% resolution. Pretreatment and post-treatment lesion size was1.60 ± 0.86 cm2 and 0.30 ± 0.40 cm2 respectively (p - value < 0.0005). None showed any significant adverse effect of oral propranolol. Conclusion: Low-dose oral propranolol is an effective and cost-effective treatment modality for periocular capillary hemangioma and is safe as an outpatient basis.

An unusual deep seated haemangioma successfully treated with propranolol

Infantile hemangiomas (IH) are most common vascular tumors. It usually resolves on its own, treatment becomes necessary if there is disease progression. Oral propranolol is a medical therapeutic option for complicated IH with impressive efficacy and generally good tolerance. We report a case of deep seated IH of the cheek in a 4 month old successfully treated with propranolol.

Management of Upper Airway Infantile Hemangiomas: Experience of One Italian Multidisciplinary Center

Airway infantile hemangiomas (IHs) can represent a life-threatening condition since the first months of life. They may be isolated or associated to cutaneous IHs, and/or part of PHACES syndrome. Diagnosis, staging, and indication to treatment are not standardized yet despite the presence in the literature of previous case series and reviews. The diagnosis might be misleading, especially in the absence of cutaneous lesions. Airway endoscopy is the gold standard both for diagnosis and follow-up since it allows evaluation of precise localization and entity of obstruction and/or stricture. Proliferation of IH in the infant airways manifests frequently with stridor and treatment is required as soon as possible to prevent further complications. The first line of therapy is oral propranolol, but duration of treatment is not yet well-defined. All considered, we report the experience of our multidisciplinary center from 2009 to date, on 36 patients affected by airway IHs, and successfully treated with oral propranolol. Thus, the authors propose their experience for the management of airway IHs, specifically early diagnosis, when to perform endoscopy, how to interpret its findings, and when to stop the treatment.

Color Doppler Evaluation of Arterial Resistive Index in Infantile Hemangioma: A Useful Parameter to Monitor the Response to Oral Propranolol?

Infantile hemangioma (IH) is the most common benign vascular tumor in childhood. In more than 85% of all cases, IHs undergo spontaneous involution, but nearly 10–12% of IHs develop complications and require immediate therapy. Oral propranolol is currently the first-line treatment for IHs. Color Doppler ultrasound is the gold standard in the diagnosis of deep IH, and it is used to evaluate the morphological change and the modification of vascularization that occur during its evolution and treatment. To date, only few data in the literature described the changes of intralesional arterial resistive index (RI) during treatment with propranolol; particularly, some authors have shown an increase of intralesional arterial RI in IHs with clinical regression during treatment with propranolol. The objective of this paper is to evaluate the changes of RI of the intralesional arteries of the IHs during the treatment with oral propranolol. We retrospectively analyzed a total of 64 IHs in 60 patients treated with oral propranolol with a good clinical response. Gray-scale ultrasonography and color Doppler imaging were performed before and during the therapy. The intralesional RIs were measured before and during the treatment. For each lesion, we recorded the RI values, and then we calculated the mean RI value for any single lesion. We compared the mean RI value observed at the baseline with the mean RI value of the last detectable sampling at color Doppler. We also compared between them the mean RI values observed during intermediate ultrasound. The RI values were compared in 44 lesions, with at least two significant samplings of RI. In the 44 lesions compared, we did not find statistically significant variations in the mean RI values between the baseline control and the values recorded at the last post-treatment control. The time trend of mean RI values of the intermediate color Doppler analysis performed between the first pre-treatment control and the last measurable control did not show any statistically significant variation in the trend of mean RI values. Contrarily to what has been described by some authors, in our experience, we have not observed an increase of RI in IHs treated with oral propranolol.

The Effectiveness of Propranolol in Managing Hemorrhagic Choroidal and Exudative Retinal Detachment following Ahmed Glaucoma Valve Implantation in Sturge-Weber Syndrome: Case Report and Literature Review

Sturge-Weber syndrome (SWS) is a congenital neurological disorder that is characterized by hamartomas involving the skin, brain, and eyes and marked by the presence of the facial port-wine stain, which consequently leads to various ocular complications. Among all ocular comorbidities, glaucoma is the most frequently witnessed in SWS patients with a prevalence of 30%–70%. If glaucoma is refractory to conventional medical management, surgical intervention can be considered. Common complications of glaucoma procedures in SWS are choroidal detachment and suprachoroidal hemorrhage. Moreover, we report a 6-year-old girl, known case of unilateral congenital glaucoma secondary to SWS. Despite being on maximal antiglaucoma drops and undergoing multiple surgical interventions, the patient had uncontrolled intraocular pressure of her right eye. A decision to proceed with Ahmed glaucoma valve implantation (AGVI) to the right eye was made. In the immediate postoperative period, the patient developed hemorrhagic choroidal detachment and exudative retinal detachment. A trial of oral propranolol (1.5–2 mg/kg/day) was then initiated for 4 months. After 30 days from oral propranolol course initiation, we started noticing a significant improvement of the hemorrhagic choroidal and exudative retinal detachment. Spontaneously, a marked reduction in subretinal fluid and suprachoroidal hemorrhage was also seen. Thus, the improvement was correlated with the propranolol therapy. Here, we report a significant improvement of the postoperative complications of AGVI in a patient with SWS, following 4 months of oral propranolol course (1.5–2 mg/kg/day). Further studies are needed to determine the dosage, duration, and optimal mechanism by which propranolol works in this situation.

Topical Timolol for Infantile Haemangioma of the Orbit

Infantile haemangiomas (IHs) are the most common benign tumours of the eyelid and orbits in infancy. Beta-blockers, in the form of oral propranolol, have become first-line treatment in severe cases with functionally significant or disfiguring IH. However, adverse drug reactions of oral propranolol in infants are reported in 1 in 11 and serious or potentially life-threatening systemic side effects in 1 in 38, including dyspnoea, hypotension, hyperkalaemia, hypoglycaemia, and cyanosis, therefore requiring careful and close monitoring during the course of systemic treatment. More recently, two large meta-analyses have shown topical beta-blockers, such as timolol maleate 0.5%, to be as effective as oral propranolol in superficial IH, but with no or significantly fewer adverse effects, and have advocated that topical beta-blockers replace oral propranolol as the first-line treatment of superficial IH. We have previously reported the therapeutic response of deep periocular IH to primary topical timolol maleate 0.5% monotherapy. Here we also describe the first successful treatments of large orbital IHs with primary topical timolol maleate 0.5% monotherapy in four infants, resulting in immediate cessation of progression and rapid clinical improvement or resolution in all cases. No adverse effects and no recurrence during long-term follow-up of up to 2.5 years after cessation were seen in any of the patients treated with topical timolol maleate 0.5%.

Infantile parotid haemangioma: a report of three cases

Aim of this case: To highlight the presentation of rapidly enlarging parotid haemangioma in three children who were all successfully managed with oral propranolol. Case report: Parotid haemangioma in infancy, albeit rare, has been vastly reported due to it being the most common tumour of the salivary gland in infants. Despite that, the presence of a rapidly enlarging tumour in “the little ones” may be daunting, leading to countless unwarranted procedures by attending physicians. Herein, we present three cases of infantile rapidly tumefying parotid lesions which were diagnosed clinically with the support of imaging, and successfully managed by promptly initiating oral propranolol. Conclusion: Although several recent reports have been reviewed, the challenges in diagnosis and management of the condition need to be individualised.

Mediastinal and subglottic hemangioma in an infant: a case report and literature review

We performed a retrospective analysis of the clinical manifestations, laboratory and imaging examinations, treatment, and prognosis of a male infant who was diagnosed with mediastinal and subglottic hemangioma in our hospital. The clinical features of this patient were coughing, wheezing, and dyspnea. Enhanced computed tomography of the neck and chest showed a diffuse abnormality in the right-upper mediastinum. He was diagnosed with a hemangioma after a physical examination combined with bronchoscopy. The clinical symptoms were relieved by oral propranolol. We also investigated the clinical characteristics, treatment, and prognosis of mediastinal and subglottic hemangioma in infants in the previous literature, and searched for case reports of this disease in China and in other countries. We only identified three previous cases of mediastinal and subglottic hemangioma in infants, indicating that this condition is rare. In the proliferative stage, surrounding organs and tissues are compressed, which can be life-threatening. Most of these children develop wheezing, shortness of breath, dyspnea, cyanosis, and other symptoms within 2 months. Enhanced computed tomography and magnetic resonance imaging combined with soft bronchoscopy can confirm the diagnosis of this disease, and oral propranolol achieves a favorable effect.