Kaposiform hemangioendothelioma of paranasal sinus

Kapossiform hemangioendothelioma (KHE) of the paranasal sins (PNS) is a rare cause of recurrent epistaxis. To date, only two cases of PNS KHE have been reported in the literature, both occurring in the pediatric population. The case presented here appears to be the first case of PNS KHE occurring in an adult. A 46‐year‐old white female presented with progressively worsening unilateral recurrent epistaxis. Diagnostic histopathology confirmed it to be KHE. After a detailed workup, the tumor was completely excised en bloc (medial maxillectomy; anterior and posterior ethmoidectomy) via a lateral‐rhinotomy approach. Complete excision of the tumor with clear margins offers the best results. Laryngoscope, 124:2103–2106, 2014     

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.     

Epithelioid hemangioendothelioma of the mandible

The authors describe a locally recurrent tumor of the mandible whose microscopic structure and immunohistochemical findings corresponded to an epithelioid hemangioendothelioma. The tumorous cells had an epithelioid character, they created rudimentary vascular lumina, and they focally expressed vascular markers CD 34, CD 31, and F VIII. Locally the tumor grew aggressively. During the last recurrence, the tumor even affected the already healed and rebuilt bone graft, which was incorporated after the primary resection of the mandibular body. From the histological point of view, the tumor approximated an angiosarcoma. No metastases occurred. Electronic Publication     

Postcricoid infantile hemangioma: Management of a case in the propranolol era

We describe a rare case of a postcricoid infantile hemangioma (IH) and treatment with propranolol. A newborn female was found to have a large, postcricoid submucosal mass. The patient was brought to the operating theater for endoscopy and biopsy. The histopathology and GLUT-1 immunoreactivity confirmed the diagnosis of IH. The lesion was treated with direct injection of triamcinolone (40 mg/ml) and systemic propranolol therapy. The patient responded well, and her symptoms resolved quickly. Propanolol provides a promising therapy as a first line treatment to avoid surgery, tracheostomy, and prolonged use of steroids.     

Lobular capillary hemangioma formation: An unusual complication of submucous resection with power instrumentation of the inferior turbinate

Submucous resection with powered instrumentation (SRPI) is an effective surgical method to achieve inferior turbinate (IT) reduction with minimal morbidity. We describe a series of two cases of capillary hemangioma (pyogenic granuloma) that developed in the posterior third of the IT as a late complication after SRPI. Laryngoscope, 125:2653–2655, 2015     

Epithelioid hemangioendothelioma of the submandibular gland

Epithelioid hemangioendothelioma (EH) is a seldom tumor of vascular origin, which occurs at sites such as liver, lung or soft tissues, and has a highly unpredictable malignant potential and clinical course. We submitted a case with epithelioid hemangioendothelioma of submandibular gland. The patient was a 44-year-old woman who presented with left submandibular painly mass. Fine needle aspiration showed a mixed lymphoid population. Contrast enhanced Computerize Tomography (CT) scan revealed that to be a well-circumscribed and including microcalcification in centre 2.5 cm x 2.0 cm mass in diameter without evidence of bony invasion. Submandibular gland was excised. The pathological result was interpretated as epithelioid hemangioendothelioma of the submandibular gland. The patient was seen regularly for recurrence.     

Essentials of surgical treatment for intramasseteric hemangioma

Although rare, surgical treatment may become necessary for patients with intramasseteric hemangiomas. Possible bleeding, neural injury or postoperative dysfunction are factors limiting surgical approaches. We present the following surgical highpoints for tumors involving the masseter muscles. These include careful preoperative planning with computed tomography and magnetic resonance imaging, as well as a surgical approach that provides adequate exposure for optimal tumor resection and identification of vital anatomic structures. In particular, care must be taken to preserve branches of the facial nerve. In certain cases, preoperative embolization or ligation of vessels feeding tumor helps to minimize blood loss. Whenever possible, complete tumor should be adequately resected with a surrounding margin of normal muscle. Postoperative lymphedema can be minimized by preserving the mandibular periosteum and oral or parenteral use of medication with anti-inflammatory agents. At the University of Tokyo, continuous suction is preferred with a fenestrated drain or pressure dressing with a Penrose drain should be applied to prevent hematoma. Postoperative dysfunction, such as trismus, is prevented by supportive measures.     

Cavernous hemangioma of the tympanic membrane

Cavernous hemangioma seems to most frequently arise in the posterior portion of the external auditory canal. However, they rarely occur in the tympanic membrane. A 49-year-old male patient was referred for evaluation of right-sided pulsatile tinnitus that he'd experienced for the previous 2 years. Temporal bone computerized tomography showed an isolated soft tissue mass just lateral to the tympanic membrane. There was no evidence of bony erosion or middle ear invasion. The patient underwent excision of the mass using a postauricular approach. The mass was removed en bloc and the defect of the tympanic membrane was repaired by tympanoplasty type I. There was no recurrence after 1 year of follow-up.     

Hemangioma of the external auditory canal in an adolescent male: Report of a case and review of the literature

Hemangiomas confined exclusively to the external auditory canal (EAC) are rare, with only nine cases being described in the literature. We report a case of a hemangioma causing conductive hearing loss in a 15-year-old male. This is the first case of a hemangioma isolated to the EAC in an adolescent and the first to be described as being of the venous subtype. Imprecise terminology has historically made the diagnosis and classification of these lesions difficult. We use this opportunity to not only shed light upon this unique case, but to briefly compare current classification schemes and review the literature relating to hemangiomas involving the EAC.     

Propranolol for the treatment of airway hemangiomas: A case series and treatment algorithm

Objectives

(1) To present six patients with symptomatic airway hemangiomas treated with oral propranolol. (2) To review the diagnostic and treatment options for airway hemangiomas and propose a new management protocol.

Study design

Retrospective review.

Setting

Tertiary care children's hospital.

Subjects and methods

Pediatric patients diagnosed with obstructive airway hemangiomas treated with oral propranolol. Patients were followed for symptomatic improvement and relief of airway obstruction on imaging or laryngoscopy.

Results

Seven patients presenting with airway obstruction were treated with propranolol. One patient had a focal hemangioma confined to the subglottis. Four patients had airway hemangiomas that extended beyond the confines of the larynx and trachea. A sixth patient had a bulky supraglottic hemangioma. A seventh patient with an extensive maxillofacial lesion failed propranolol therapy and was found to have a pyogenic granuloma on final pathology after excision. Six patients had failed standard medical therapy and/or surgical interventions and were treated successfully with oral propranolol with improvements in airway symptoms and oral intake, requiring no further surgical intervention. Treatment was initiated as early as 1.5 months of age, and as late as 22 months. No adverse side effects of propranolol were noted.

Conclusions

Oral propranolol was successfully used to treat airway hemangiomas, resulting in rapid airway stabilization, obviating the need for operative intervention, and reducing the duration of systemic corticosteroid therapy while causing no obvious adverse effects. These outstanding results enable the possibility of use of a standardized diagnostic and treatment algorithm for airway hemangiomas that incorporates systemic propranolol.     

Management of complicated hemangiomas with vincristine/vinblastine: Quantitative response to therapy using MRI

Purpose

To quantify the efficacy of vincristine and vinblastine in the treatment of complicated hemangiomas.

Design

Retrospective review.

Methods

Charts were reviewed to identify patients treated with vincristine or vinblastine for complicated hemangiomas from August 2002 to October 2007. Only patients who received both a pre and post-treatment magnetic resonance imaging (MRI) were considered. A database was created which includes patient gender, age at treatment initiation, rationale for treatment, hemangioma location, number of cycles of chemotherapy received, and complications of treatment. A single pediatric radiologist calculated lesion volumes from both pre and post-treatment MRI which were compared to quantify treatment response.

Results

Seven patients (2 male, 5 female) met criteria. Mean age at treatment initiation was 20 weeks (median 14, range 5-60). Rationale for treatment included four patients (57%) with proptosis/orbital compromise and one patient each (14%) with heart failure, airway compression, and hemangiomatosis with rapid growth of multiple lesions. Patients received a mean of 2.86 cycles of chemotherapy (median 3, range 1-5).Twelve lesions were identified and analyzed for pre and post-treatment volume on MRI in the seven patients. Eleven of twelve (92%) lesions decreased in size after treatment. The mean volume ratio of hemangiomas at the conclusion of chemotherapy was 0.45 compared to pre-treatment size (median 0.18, range 0-2.19) Orbital compromise, airway compression, and cardiac failure either improved or resolved in all patients.Three complications of treatment were seen in seven patients (42%) including bacteremia with anemia, peripheral neuropathy and motor delay. All complications resolved after cessation of chemotherapy.

Conclusions

Treatment of complicated hemangiomas with vincristine or vinblastine can control growth and improve symptoms in the majority of patients. Treatment often requires multiple cycles of chemotherapy. Complications of treatment are common, but reversible.     

Facial nerve hemangioma: A rare case involving the vertical segment

This case report and literature review reports on a rare case of facial nerve hemangioma (FNH) involving the vertical facial nerve (FN) segment, and discusses the clinical presentation, imaging, pathogenesis, and management of these rare lesions. A 53‐year‐old male presented with a 10‐year history of right hemifacial twitching and progressive facial paresis (House‐Brackmann grading score V/VI). The computed tomography and magnetic resonance imaging studies confirmed an expansile lesion along the vertical FN segment. Excision and histopathologic examination demonstrated FNH. FNHs involving the vertical FN segment are extremely rare. Despite being rare lesions, we believe that familiarity with the presentation and management of FNHs are imperative. Laryngoscope, 2012     

成人咽喉部血管瘤23例临床分析

目的探讨成人咽喉部血管瘤的临床特征、诊断、治疗方法选择及治疗效果。方法对2006年4月~2018年6月期间在江南大学附属医院耳鼻咽喉头颈外科住院诊治的咽喉部血管瘤患者的临床资料进行回顾性分析。结果23例患者,男性15例,女性8例;主要临床症状为咽部异物感、声音嘶哑及咳血,部分患者无任何症状。主要发病部位为舌根、会厌谷、杓会厌皱襞,梨状窝等,可单发,也可多发。MRI一般表现为T1WI等或低信号,T2WI高信号,增强可明显强化。15例患者采用了药物注射治疗,6例采用了手术切除,2例采用了药物注射联合射频或者低温等离子手术治疗。随访2个月~1年,治愈21例,显效2例,无严重治疗并发症。结论成人咽喉部血管瘤多发生于声门上区,多数病人无特异性症状,电子喉镜和增强MRI是主要的诊断工具,根据肿瘤部位和大小采取药物注射、手术切除或者联合治疗可取得满意效果,治疗期间需注意呼吸道的管理。     

Facial nerve hemangiomas: A review

Facial nerve hemangiomas are rare benign vascular tumors with primary symptoms including progressive facial nerve palsy and hearing loss. They can involve various segments of the facial nerve, especially the geniculate ganglion (G.G.). Preoperative MRI and CT imagining is of great importance for early diagnosis of such lesions. The most accepted treatment for facial nerve hemangiomas is surgical excision1 and the type of approach depends on tumor location, preoperative hearing level and tumor size. In consideration of the difficulties in differential diagnosis, otologists should pay attention to avoid missed or incorrect diagnosis in hemangiomas of facial nerve.     

Microdebrider removal under suspension laryngoscopy: An alternative surgical technique for subglottic hemangioma

Objective

To describe microdebrider removal of subglottic hemangiomas and evaluate the efficacy of this procedure.

Methods

This retrospective study was conducted at an otorhinolaryngology department. Seven consecutive patients with subglottic hemangiomas who failed to respond to pharmacologic treatment were treated by microdebrider removal under suspension laryngoscopy. Respiratory symptoms and hemangioma status were assessed preoperatively and postoperatively.

Results

The mean age of patients at disease onset was 68 days (range, 24–120 days), and their mean age at the time of surgical treatment was 132 days (range, 55–195 days). Their initial pharmacologic treatment included steroids, propranolol, and/or antibiotics. The mean percentage of airway obstruction was initially 36% (range, 30–50%), and this value changed to 61% (50–80) before surgery. Microdebrider removal of hemangiomas was successful in all patients without any intraoperative or postoperative complication. The mean operative time was 30 min (range, 21–47 min). Three patients required orotracheal intubation for 12 or 24 h after surgery, and no reintubation or tracheotomy was required in this series. Symptoms such as stridor and inspiratory retraction were resolved approximately 72 h after surgery, but wheezing was generally resolved at a later time point. One year later, no respiratory symptom was noted in these patients, and endoscopic examination revealed a sufficient glottis opening.

Conclusions

Surgical removal with a microdebrider is advantageous as a routine surgical technique for small and moderate pediatric subglottic hemangioma.