Superficial vascular anomalies of the face: apropos of six cases

The classification of superficial vascular anomalies of the face recognizes two major categories: hemangiomas and vascular malformations. A multidisciplinary approach to these malformations is necessary. MRI of venous vascular malformations, pulsed Doppler and arteriography of arteriovenous malformations are key examinations in the exploration strategy for superficial vascular malformations. Hemangiomas always regress. Surgery is generally needed only for late sequelae although in some cases early surgery is beneficial. Percutaneous sclerotherapy and surgery are indicated for venous malformations in order to minimize or improve skin, muscle and bone distorsions. Arteriovenous malformations are the most dangerous vascular anomalies of the face. Superselective arterial embolization prepares the surgical excision of the nidus, complete resection is essential. We review 6 cases of superficial vascular malformations of the face treated surgically and discuss the diagnosis features and the treatment modalities of these vascular anomalies.     

Angiography in the diagnosis and management of extracranial vascular lesions of the head and neck.

The angiographic features of various lesions of the head and neck are presented. Angiographically, cavernous hemangiomas display large venous lakes with calcified phleboliths. Arteriovenous malformations reveal massive tumor stain with well delineated feeding vessels from multiple systems. Chemodectomas and juvenile nasopharyngeal angiofibromas are clearly vascular with homogenous tumor staining in the capillary phase. Angiography of cavernous hemangioma, AVM, chemodectoma, and angiofibroma is diagnostic and may preclude the need for tissue biopsy. Angiographically neurilemmomas are less vascular with non-homogenous tumor stain. Carcinomas are typically avascular. The use and benefits of arterial embolization in the management of these lesions is presented.     

Childhood haemangiomas of the head and neck

Haemangiomas of the head and neck in children may be of several histological types, the clinical course depending on the group to which the haemangioma belongs. Treatment may be required if the haemangioma interferes with the airway, as in the subglottic group, or if the lesion becomes ulcerated with subsequent haemorrhage, as in capillary cavernous haemangiomas. Most haemangiomas require no immediate treatment as they involute spontaneously, though parental reassurance will be of paramount importance. Those lesions which persist may be amenable to treatment at a later date, the laser probably offering the best long-term results in terms of cosmesis as in the case of port-wine stains. Superselective embolization is becoming the treatment of choice for arterial haemangiomas. This paper is designed to clarify the histological and clinical features of these tumours and their management in view of considerable confusion in the literature encountered in our study.     

Management of postcricoid and upper esophageal hemangioma

To discuss management of postcricoid and upper esophageal hemangiomas in infants. Four children presenting with progressive feeding and/or respiratory disturbance underwent endoscopy including systematic esophagoscopy revealing the hemangioma of the postcricoid area and extending to the lumen of the upper esophagus. In the three children with severe disturbance, systemic steroids were insufficient and open surgical excision brought the hemangioma under control. No significant stenosis occurred despite prolonged progressive oral feeding up to 1 month after surgery. The fourth child whose disturbance was moderate, was managed by intralesional steroids. Various solutions have been proposed for the treatment, i.e. conservative approach or partial or complete destruction or excision, management of postcricoid hemangioma by intralesional steroids or by open surgical excision. Indications will require greater series.     

Propranolol for airway hemangiomas: Case report of novel treatment

Infantile hemangiomas arising in the trachea are rare. These lesions pose a management dilemma as several treatment options can provide safe management. Propranolol, a nonselective beta‐blocker, has recently been introduced as a novel modality for the treatment of proliferating hemangiomas. This report illustrates the successful management of tracheal hemangiomas using oral propranolol in a young patient with otherwise treatment‐resistant airway lesions. Despite various endoscopic therapeutic attempts, the patient remained stridulous with airway disease that persisted into the involution phase of the average hemangioma cycle. Within 6 weeks of beginning oral propranolol (2 mg/kg/day), her airway compromise was eliminated and she had complete resolution of endoscopically visible disease. No side effects from propranolol occurred. We propose that oral propranolol should be considered for use in airway hemangiomas. Laryngoscope, 2009     

Hard palate necrosis after bilateral internal maxillary artery embolization for epistaxis

Superselective embolization is an effective method of treating epistaxis that is refractory to conservative treatment. Soft tissue necrosis is a rare complication owing to the extensive collateral blood supply of the head and neck. We describe the case of a patient who developed unilateral necrosis of the mucosa overlying the hard palate after undergoing bilateral internal maxillary artery embolization. The presence of a nasopharyngeal balloon and bilateral nasal packs for 2 days after embolization may have compressed collateral vessels in the soft palate and nose and contributed to this complication. Packing should be removed as soon as possible after embolization.     

Pediatric postcricoid vascular malformation: a diagnostic and treatment challenge

Hemangioma of the upper aerodigestive tract is a rarely reported occurrence in the pediatric literature. To date, there have been three published case reports of postcricoid hemangiomas contributing to unexplained dysphagia and respiratory distress. We present three children with a history of swallowing difficulty and stridor who were found to have an occult postcricoid mass. Valsalva maneuvers confirmed the suspicion of a vascular malformation in both patients. Transoral laser therapy (KTP and CO2) was used to ablate the lesions. The patients are symptom-free at 5 months, 5 years, and 2 weeks, respectively. The diagnostic challenge in evaluating these children and the therapeutic choices are described.     

Clinical aspects of osseous hemangiomas of the skull base

Osseous hemangiomas are distinctly unusual tumors of the skull base. They clinically mimic other, more common lesions in this region, and their diagnosis is rarely made preoperatively with currently available techniques. Three cases of osseous hemangiomata, 2 involving the geniculate region and 1 arising from the jugular bulb, are described in relation to the perioperative assessment and management of these lesions. A review of 21 previously reported skull base hemangiomas is included.     

Postcricoid hemangioma of childhood: report of four cases

Hemangiomas are the most common tumor of infancy, and the vast majority occur in the head and neck region. In children, laryngeal hemangiomas typically occur below the level of the true vocal folds, in the region of the subglottis, and other sites are exceedingly rare. We present four cases of hemangiomas located in the postcricoid region of the hypopharynx. Because of the location of these lesions, children may present with obstructive symptoms such as dysphagia, intermittent aspiration, hypersalivation, or recurrent respiratory infections. Clinical diagnosis is relatively easily made with flexible laryngoscopy, as the lesions have a propensity to enlarge with crying or straining. When these patients are examined under general anesthesia in a relaxed state, however, the lesions are typically much smaller, and can even go unnoticed. Unlike other reported cases, the postcricoid hemangiomas in our patients were not causing any symptoms and were simply incidental findings. Thus, we believe that the true incidence of postcricoid hemangiomas is likely higher than reports suggest. To our knowledge, we report the longest follow-up (6 years) of a patient with a postcricoid hemangioma and are the first to describe the natural course of such a lesion.     

Interdisciplinary concept for classification and treatment of vascular anomalies in the head and neck]

PURPOSE: In patients with extended vascular anomalies in the head and neck, therapeutic decisions may pose a challenge to maxillofacial surgeons, dermatologists, and interventional radiologists. We analyzed the value of an interdisciplinary classification and treatment concept. PATIENTS AND METHODS: The classification distinguishes hemangiomas and vascular malformations. Whereas hemangiomas are endothelial proliferations, vascular malformations are considered to be developmental anomalies, which are further classified into high-flow or low-flow lesions and according to the vascular channels into capillary, venous, or lymphatic malformations. Since 2000 we have provided interdisciplinary consultation for patients with vascular anomalies. In patients with hemangiomas and venous malformations, the clinical diagnosis is confirmed by color-coded duplex sonography and magnetic resonance imaging; angiography is performed as part of the treatment planning for patients with arteriovenous malformations. Patients with hemangiomas are treated surgically by cryosurgery or laser surgery or conservatively according to lesion size and behavior. In patients with venous malformations, percutaneous sclerotherapy is combined with surgical reduction; patients with arteriovenous malformations undergo transarterial embolization prior to surgical excision of the nidus. RESULTS:A total of 73 patients attended the interdisciplinary consultation. This group included 53 patients with facial hemangiomas, 7 with venous malformations, 2 with capillary malformations, 5 with lymphatic malformations, and 6 with high-flow arteriovenous malformations. CONCLUSIONS: The interdisciplinary protocol increases diagnostic accuracy and helps to establish individual treatment plans for patients with extended vascular anomalies.     

Submandibular gland hemangioma: clinicopathologic features and a review of the literature

We conducted a retrospective study to determine the incidence and characteristics of submandibular gland hemangioma at our institution. We reviewed the records of all patients who had undergone submandibular gland excision from January 1998 through December 2006. We found a total of 230 such cases. Of these, submandibular gland hemangioma was found in 4 patients (1.7%)-3 women and 1 man, aged 20 to 47 years (mean 34.8). Their duration of symptoms had ranged from 26 to 78 months (mean: 49.3). These symptoms had included submandibular swelling, pain or discomfort, and features of sialadenitis. Computed tomographic angiography had revealed that the hemangiomas were supplied by the facial and lingual arteries. Two of the 4 patients had undergone preoperative vascular embolization, but it had failed to significantly reduce the amount of intraoperative blood loss. Final histopathologic examination had revealed that all 4 lesions were cavernous hemangiomas and that they had replaced the normal glandular structure. No recurrence was seen during a follow-up that ranged from 47 to 72 months (mean: 56.3).     

Therapy of juvenile angiofibroma

We describe 12 juvenile angiofibromas treated predominantly by surgical excision. The course of the disease and therapy are analysed. Four interventions on 3 patients were done after pre-operative embolization. Superselective digital subtraction arteriography allowed detailed analysis of all feeding vessels, and a search for anastomoses between extra- and intracranial arteries and endarterial occlusion of tumour vessels. Intra-operative blood loss was reduced and visibility in the field of operation was improved.     

Systemic steroids for the management of obstructive subglottic hemangioma.

Subglottic hemangioma is a recognized cause of paediatric upper airway obstruction. We present 14 patients with subglottic hemangioma treated between 1984 and 1997,4 of whom had associated extralaryngeal hemangiomatous lesions (28%). The degree of upper airway obstruction ranged between 20% and 90%. Patients with subglottic hemangioma who had obstruction of the laryngeal lumen more than 25% and those with obstructive symptoms were treated with systemic steroids. The patients were followed clinically, radiographically, and with repetitive bronchoscopies. Nine of 10 patients (90%) have responded clinically to systemic steroids. There were no major complications from the systemic steroid treatment. One patient developed a cushingoid face that was reversed after the cessation of steroid therapy. The purpose of this study is to show that systemic steroids, with or without short-term intubation after diagnostic bronchoscopy, can be used as a safe and effective alternative in the management of obstructive paediatric subglottic hemangiomas.     

Treatment of oral cavity vascular malformations using the neodymium:YAG laser

Although rare, hemangiomas and lymphangiomas of the oral cavity are difficult management problems. Surgical resection is still commonly thought of by many when considering treatment. Several articles in 1986 reported good results using the neodymium:yttrium-aluminum-garnet laser for these lesions. Using this laser, we have treated six patients with hemangiomas or lymphangiomas of the oral cavity over the past 4 years. Although most patients have required several treatments, the response has been excellent overall. This report confirms the results of previous studies.     

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.     

Evaluation and management of benign, non-congenital tongue masses in children.

Lingual tumors are rare, primarily benign, lesions in the pediatric population. Congenital lesions, such as hemangiomas, lymphatic malformations, dermoids, hamartomas and thyroglossal ducts cysts, are seen more commonly. Primary, non-congenital lingual neoplasms are less common in children. We present three patients with benign lingual neoplasms. Evaluation, management, pathology and follow-up are discussed.     

Infantile supraglottic hemangioma: a case report

Hemangiomas of the airway are benign vascular lesions that can involve any site from the nares to the tracheobronchial tree. Most of these lesions are seen in the subglottic area in infants. Supraglottic infantile hemangiomas are very rare. We report a case of supraglottic hemangioma in a 2-month-old boy who had been admitted to our hospital with inspiratory stridor and dyspnea. The hemangioma involved the left arytenoid and aryepiglottic fold. A tracheostomy was performed, and the patient was followed up endoscopically every 6 months thereafter The hemangioma disappeared when the child was 30 months old. Subsequently, a Montgomery T-tube was placed for 6 months to assist in maintaining normal breathing. The patient remains disease-free during ongoing follow-up. We also discuss the management strategies for infantile laryngeal hemangiomas.     

Epipharyngeal teratoma in infancy.

Epipharyngeal teratomas are rare congenital lesions that often cause respiratory distress, stridor at birth. The management of such anomalies should include establishment of secure upper airways, radiographic exclusion of midline CNS malformations and early surgical excision to prevent malignant transformation, asphyxia or permanent facial distortion. The diagnosis of congenital stridor, dyspnea and rhinitis should be performed very carefully.     

Kavernöses Hämangiom der Glandula parotis im Erwachsenenalter

Parotid hemangiomas are rare in adults. Whereas both capillary and cavernous hemangiomas are seen in children, only cavernous hemangiomas have thus far been reported in adults. Clinically, cavernous hemangiomas usually present as slowly growing, soft or firm, movable, painless parotid masses. Severe pain and swelling can occur, however, depending on the size of the hemangioma or in particular in the presence of acute hemorrhage or thrombosis. CT and MRI are the diagnostic tools of choice. Prior to surgery, magnetic resonance angiography or intra-arterial digital subtraction angiography should be performed to investigate the vascular supply of the tumor. Surgical excision is the treatment of choice for small lesions. Large cavernous hemangiomas usually require superficial or total parotidectomy. Especially in the case of extended lesions, the facial nerve may be difficult to identify and should be monitored intraoperatively.