Initial experience using propranolol as the sole treatment for infantile airway hemangiomas

The objective of this study is to describe the initial use of propranolol as the sole treatment for focal infantile airway hemangiomas, and to report on available literature describing the use of propranolol for airway lesions. This retrospective case series was carried out at a tertiary pediatric medical center. We obtained the following results: two children demonstrated significant response to oral propranolol therapy and avoided not only invasive surgical procedures, but also long-term administration of oral corticosteroids. This is the first report of treating infantile airway hemangiomas with only propranolol without additional surgical intervention or corticosteroid use. Review of literature reveals initial case series with similar, successful results using propranolol as an adjuvant treatment along with other medications and surgical interventions. We conclude that the initial use of propranolol as the sole treatment for infantile airway hemangioma is promising. Literature review reveals that propranolol as the sole treatment for most head and neck hemangiomas shows significant promise based on early case reports. Further studies are needed to determine the long-term effectiveness, dosing strategies, and side effect profile of propranolol treatment for hemangiomas.     

Response of infantile airway and facial hemangiomas to propranolol in a patient with PHACE syndrome

PHACE syndrome is a neurocutaneous syndrome defined by posterior fossa malformations (P), hemangiomas (H), arterial anomalies (A), cardiac defects and coarctation of the aorta (C), and eye abnormalities (E). Propranolol is currently first line therapy for treatment of head and neck hemangiomas that cause functional defects or extreme disfigurement. The safety and efficacy of this treatment have been well documented in current literature, but few authors have reported the use of propranolol in patients with PHACE syndrome. We report a case of a patient with PHACE Syndrome and associated infantile airway and facial hemangiomas who was successfully treated with propranolol and a review of the literature on this subject.     

Case report: Treatment failure using propanolol for treatment of focal subglottic hemangioma

Subglottic hemangioma is a rare, potentially life threatening tumor of infancy which poses serious treatment challenges. A number of medical and surgical therapies over the years have met with variable success, and are associated with numerous potential morbidities. A potential windfall in the management of infantile hemangiomas has arisen with the recent identification of propanolol as a highly efficacious and relatively safe new treatment modality. At least five reports in the literature have described the rapid, successful treatment of airway hemangiomas with oral propanolol. We describe the first reported treatment failure with propanolol for subglottic hemangioma in an infant who initially responded dramatically to the medication.     

Invasive sinonasal actinomycosis

Actinomycosis is a rare anaerobic bacterial infection typically caused by Actinomyces israelii. Although part of normal flora in the oral cavity, and respiratory and digestive tracts, A israelii can give rise to pathologic infections most commonly reported in the oral cavity from odontogenic causes. We present a rare case of invasive actinomycosis presenting with extensive midface destruction involving the maxilla and paranasal sinuses, with mucosal necrosis mimicking an aggressive neoplasm. The diagnosis is usually reached only after histopathologic analysis showing characteristic sulfur granules with filamentous gram‐positive, non–acid‐fast bacteria. We review the literature on its epidemiology, clinical presentation, diagnosis, treatment, and prognosis.     

Role of Propranolol in the therapeutic strategy of infantile laryngotracheal hemangioma

There are recent reports of effective treatment of cutaneous hemangiomas with Propranolol. The current study aims to assess efficacy of systemic Propranolol for subglottic hemangiomas and to discuss its place among the other available therapies. We report 2 infants with subglottic hemangiomas, which were resistant to other established medical treatments. One infant presented with PHACES association, the other with widespread cutaneous congenital hemangiomas. Both were subsequently treated with systemic Propranolol. Both patients’ subglottic hemangiomas responded dramatically to systemic Propranolol. No side effects of the therapy occurred, and a safety protocol previously designed for Propranolol prescribed for other indications was applied to our patients. Propranolol appears to be an effective treatment for subglottic hemangiomas and should now be used as a first-line treatment in subglottic hemangiomas when intervention is required.     

Acute invasive fungal rhinosinusitis: our experience with 19 patients

Acute invasive fungal rhinosinusitis (AIFR) is a potentially fatal infection that affects immunocompromised patients. Early diagnosis and treatment, including aggressive surgical debridement, antifungal medication, and correction of underlying predisposing factors are essential for recovery. The aim of this study was to review our experience with AIFR. The records of 19 patients histopathologically diagnosed with invasive fungal rhinosinusitis were retrospectively reviewed. Demographic data, presenting symptoms and signs, underlying diseases, and outcomes of the patients are presented and invasive fungal rhinosinusitis is discussed in light of the current literature.     

Treatment of hemangiomas of the face with magnesium seeds

Summary Various conservative and surgical procedures are available for the management of aggressive hemangiomas in infancy and childhood. One of them is magnesium seeding, a treatment modality developed more than 90 years ago but neglected for a long time. The technique consists of implanting 0.5- to 1-mm-thick wires of 99.8% pure magnesium into the tumor mass. During repeated courses oxidation of the metal results in fibrosis and cicatricial transformation of the tumor. Indications, technique and results are discussed.     

Treatment of hemangiomas of the face with magnesium seeds

Various conservative and surgical procedures are available for the management of aggressive hemangiomas in infancy and childhood. One of them is magnesium seeding, a treatment modality developed more than 90 years ago but neglected for a long time. The technique consists of implanting 0.5- to 1-mm-thick wires of 99.8% pure magnesium into the tumor mass. During repeated courses oxidation of the metal results in fibrosis and cicatricial transformation of the tumor. Indications, technique and results are discussed.     

Laser photocoagulation of vascular malformations of the tongue

Vascular malformations of the tongue (hemangiomas, lymphangiomas, AV fistulae, etc.) compromise a significant portion of head and neck angiodysplastic lesions. Complications requiring treatment include bleeding, pain, and difficulties related to increased tongue volume. Treatment modalities have included embolization, excision, cryotherapy, sclerosis, radiation, and chemotherapy. The more aggressive therapies often result in major functional disability to loss of lingual tissue. A series of nine patients has been treated for one or more of the complications of lingual vascular malformations with either argon of Nd:YAG laser photocoagulation. The therapeutic objective was provision of symptomatic relief with conservation of functioning lingual tissue. Results have varied from good to excellent with markedly decreased bleeding frequency as well as reduction in lesion size. No serious complications, such as bleeding or invasive infection, have been noted. Laser phototherapy is beneficial in the palliation of selected benign lingual vascular malformations.     

Management of the locally aggressive thyroid carcinoma

The management of locally advanced thyroid carcinoma can be challenging. Differing philosophies exist for the treatment of invasive disease affecting the upper aerodigestive tract. The ability to maximize local control and overall survival while minimizing the morbidity of a radical resection is the goal of care in these patients. This article will review the literature concerning the presentation, preoperative evaluation, and the subsite treatment of locally aggressive well-differentiated thyroid carcinoma.     

Head and neck manifestations of Maffucci's syndrome.

Maffucci's syndrome consists of multiple cutaneous hemangiomas, dyschondroplasia, and often enchondroma. Once considered as a rare disorder, the syndrome has been frequently recognized and reported in the last 20 years. We report a case of multiple neoplasms of the head and neck and review the literature with regard to those manifestations of Maffucci's syndrome in the head and neck area. We also discuss treatment and the potential for malignant change.     

Primary intraosseous cavernous hemangioma of the zygoma: a case report and literature review

Intraosseous hemangiomas are rare. We report the case of a 47-year-old man who presented with a gradually enlarging left zygomatic mass that had caused pain, deformity, and superficial soft-tissue swelling. Computed tomography revealed a well-circumscribed 2.0 × 2.5-cm mass with a ground-glass matrix in the left zygoma. Following surgical excision, the patient's symptoms resolved. Findings on pathologic examination of the excised tissue were consistent with an intraosseous cavernous hemangioma. We describe the features of this rare case, we discuss the pertinent radiologic features and pathophysiology of intraosseous hemangiomas, and we review the available literature.     

Propranolol for the treatment of subglottic hemangiomas

IntroductionInfantile subglottic hemangiomas are rare causes of airway obstruction. They begin to proliferate at 1–2 months of age and can cause biphasic stridor with or without respiratory distress. Diagnosis requires direct visualization by direct laryngoscopy and bronchoscopy. Various therapeutic options have been utilized for treatment, including tracheotomy, open surgical excision, laser ablation, intralesional steroid injection, systemic steroids, and now oral propranolol.MethodsWe present a retrospective chart review of infantile subglottic hemangiomas over a 5-year span (January 2005–2010) at a tertiary care pediatric hospital. IRB approval was obtained, and charts were reviewed to find patients with subglottic hemangiomas, including patient characteristics, presentation, workup, medical and surgical management, and outcomes. A case presentation demonstrates diagnostic, management, and treatment strategies and dilemmas encountered.ResultsNine patients were found to have infantile subglottic hemangiomas. Six of nine patients were treated with laser excision, with five of the six having localized subglottic hemangiomas. In 2009, three of four patients were initiated on propranolol as first-line treatment; the fourth had comorbidities which precluded this. Of the three, two showed improvement, while a third, who also had bearded hemangioma, required tracheotomy.DiscussionInfantile subglottic hemangiomas are rare but essential in the differential diagnosis of biphasic stridor. Although propranolol has been effective in treating cutaneous and airway hemangiomas, our experience suggests that this is not consistent for subglottic hemangiomas. In an area where airway compromise can be lethal, we must extend caution and monitor these patients closely as they may require adjuvant therapy.     

Carotid artery occlusion due to fulminant rhinocerebral mucormycosis

Mucormycosis is a rare disorder with a potentially lethal course caused by an opportunistic fungus of the Phycomycetes family. Rhinocerebral mucormycosis (RCM) is an acute fulminant form of invasive fungal sinusitis occurring principally in individuals who are immunologically or metabolically compromised. This form originates from one of the paranasal sinuses and may spread to the orbit or the intracranial space either by direct extension or using the unique capability of spreading along vascular walls, leaving the bony structure intact. Carotid artery occlusion is a complication of RCM. Because of the rapidity with which this disease progresses, prompt and aggressive therapy is essential. The morbidity and mortality of this disease are directly related to the length of time before diagnosis and treatment. The diagnosis is difficult because invasive mucormycosis may be present with no mucosal changes and a normal sinus CT scan. It is a histological diagnosis. Treatment of RCM includes early diagnosis, correction of underlying conditions, early and radical surgical debridement, and lipid formulation of amphotericin B. Multiple surgeries are often necessary for adequate debridement. We report a case of carotid artery occlusion due to RCM and discuss the literature on its early signs and symptoms, pathophysiology, and treatment options.     

Current concepts in the classification, diagnosis and treatment of hemangiomas and vascular malformations of the head and neck

There are many different classifications of vascular anomalies. As the correct classification of the vascular lesion has a direct influence on therapy it is difficult to decide which treatment should be considered as the treatment of choice. Based on an extensive review of the literature and personal experience of the treatment of more than 200 patients with hemangiomas or vascular malformations of the head and neck, a clinical classification is described that allows vascular lesions to be categorized in order to plan purposeful treatment. In general, hemangiomas represent the main group of vascular lesions in infancy and childhood. They are usually apparent a few weeks after birth and are characterized by an initially rapid growth of epithelial cells, followed by spontaneous involution. Hemangiomas should be differentiated from vascular malformations that are present at birth but may not be evident clinically. Spontaneous involution of vascular malformations has never been reported, whereas laser therapy can induce involution of hemangiomas at an early stage in a majority of cases. In certain situations steroids or surgical removal may seem to be the appropriate therapy of choice. In contrast, vascular malformations have to be treated according to their histopathology and location, as well as their hemodynamic features as shown radiographically with angiography. The accurate diagnosis of vascular anomalies is essential for further treatment, as shown by clinical experience at the University of Marburg.     

Supraglottic hemangioma in an infant

We present a 40 days-old infant with a hemangioma involving the epiglottis and the left aryepiglottic fold and type II laryngomalacia, which was diagnosed during evaluation for feeding difficulties. Diagnosis was based on clinical symptoms and the appearance of the lesion. It was supported by the co-existence of skin, liver and left parotid hemangiomas, and the good response to propranolol therapy. In literature review, we found that the presence of hemangiomas in the supraglottis is rarely described. This may be due to lack of symptoms and the tendency of infantile hemangiomas to involute without therapy over time.     

Infant tongue lesions: A case presentation and review of the literature

Purpose: A case presentation of a soft tissue lesion in an 8-month-old patient's left lateral tongue and review of the literature regarding infant tongue lesions. Methods: The patient's medical records were reviewed with summarization of care to date that care was received at presenting institution. A systematic literature review was then conducted using PedMed. Results: The differential diagnosis of infant tongue lesions is large, encompassing hemangiomas, lymphangiomas, thyroglossal duct cysts, teratomas, choristomas, infantile fibromatosis and rhabdomyosarcomas, among many others. Conclusion: Most infant tongue lesions are benign. However, given the aggressive propensity of infantile fibromatosis and rhabdomyosarcomas, all infant tongue lesions must be characterized.     

The blue rubber bleb nevus syndrome: a case with prominent head and neck findings

The blue rubber nevus syndrome describes a clinical entity that presents with cutaneous and visceral hemangiomas, particularly those found in the gastrointestinal tract. These lesions may appear at birth or in early childhood and can increase in size and frequency with age. Oral cavity hemangiomas occur in 59% to 64% of cases. Successful diagnosis requires a high index of suspicion, and treatment is symptomatic. This report discusses a unique presentation and a review of the literature.     

Palpebral capillary hemangioma and amblyopia. Apropos of 2 cases

Periorbital capillary hemangiomas of childhood can produce visual axis occlusion and anisometropia, resulting in amblyopia. We report our experience performing surgical resection of periorbital capillary hemangiomas. Partial resection of hemangiomas was performed in two infants under five months of age, and permitted clearing of the visual axis. This immediate effect is necessary to prevent amblyopia. Surgical resection should be considered a treatment option for managing periorbital capillary hemangiomas which threaten vision.