Introduction: Hemangioma represents an expansive lesion of the hamartoma type, classified as a benign neoplasm composed of blood vessels arranged in a disordered manner, observed in the head and neck region in approximately 65% of cases.

Objective: To present sclerotherapy with polidocanol as a safe, effective, and applicable therapeutic option in the scenario of laryngeal hemangiomas in adults.

Methods: Clinical case presentation with therapeutic review. We report a case of Laryngeal Hemangioma treated with sclerotherapy using Polidocanol in an adult patient.

Case Report: A 35-year-old female patient, a teacher, presenting with progressive dysphagia and a weight loss of 8 kg over the past 6 months, associated with dyspnea on mild to moderate exertion, and significant progressive dysphonia with psychological impact and work disability. Nasofibroscopy revealed a highly vascularized, non-pulsatile, extensive lesion with a large supraglottic extension at the level of d’Ávila areas I and II.

Discussion: Sclerotherapy with polidocanol was the therapeutic option for this case, administered in the form of foam using the Tessari technique, applied through puncture. Polidocanol is a sclerosing agent indicated for the treatment of uncomplicated varicose veins. The otolaryngologist, under microlaryngoscopy, performs the procedure with pre-defined injection sites, distributing polidocanol in the tissues, around 2.5 to 3 ml in two to three application points. The patient showed significant improvement in the hemangioma, as evidenced in the postoperative follow-up after the third application.

Conclusion: Intraoperative trans-tissue infiltration of polidocanol represented a safe therapeutic option, with a significant reduction in cavernous hemangioma and consequent relative control of the patient’s dysphonia, dyspnea, and dysphagia. Given the above, we recommend its use with appropriate application of safety criteria.

• Laryngeal hemangioma; Aerodigestive tract hemangioma; Sclerotherapy; Polidocanol

d’Avila JS, Yepez AA, D’avila DV, Perazzo PSL, De Araújo MJS, et al. (2024) Laryngeal Hemangioma - Case Report and Therapeutic Update. Ann Otolaryngol Rhinol 11(2): 1331.

Hemangioma represents an expansive lesion of the hamartoma type, classified as a benign neoplasm composed of blood vessels arranged in a disordered manner. One of its main characteristics is the uncontrolled proliferation of blood vessels. According to the International Society for the Study of Vascular Anomalies (ISSVA), hemangiomas are distinguished from other vascular malformations by their clinically slow growth behavior, associated with specific radiological and histological findings [1,2]. This type of tumor is commonly observed in the head and neck region, with an incidence of approximately 65% of cases occurring in this segment [3-7].

The most significant prevalence of hemangioma occurs in childhood, especially in females, in non-Hispanic and premature children, representing approximately 4 to 7% of benign tumors in this phase. In absolute terms, the presence of hemangioma is observed in about 2.5% of neonates [1,3,8-10]. Thus, hemangioma is considered the primary vascular tumor in childhood. Although not present at birth, there are indications of a precursor lesion, manifested by a pale region, telangiectasia, or purple lesion. Over time, these characteristics become more evident, reaching the peak of growth in the proliferative phase, which varies from 3 to 7 weeks of age to 12 to 18 months. Subsequently, hemangioma progresses to the involutive phase in the early years of life, achieving, in most cases, complete resolution around the age of 12 [1,8,9,11]. After the lesion’s involution, areas with abnormal coloration and texture associated with remaining fibroadipose reactions can still be observed [8].

It is important to note that hemangioma in adults is extremely rare, and therefore, its exact incidence is not well understood. Unlike the pediatric population, males are more affected in adults. Furthermore, unlike the pattern observed in children, the lesion does not follow the phases of proliferation and involution. The occurrence of hemangiomas in adults is directly related to vocal abuse, smoking, and vocal trauma, such as orotracheal intubation [9].

There is clear clinical evidence of a direct relationship between upper respiratory tract hemangioma and skin hemangioma, whether located in the skull, scalp, or face. Hemangioma can affect the upper airways, extending from the nose to the subglottis or trachea. The clinical presentation varies according to the location and volume of the lesion [12].

There are three classic symptoms that comprehensively represent diseases of the upper respiratory tract and their respective laryngeal levels, referred to as the “3D” symptoms. The first “D” is associated with dysphagia, indicating diseases located in the supraglottic region of the pharyngo-larynx. The second “D” is related to dysphonia, indicating lesions in the glottic region, specifically on the vocal folds. The third “D” is associated with dyspnea, with lesions affecting the subglottic region, manifesting as an obstructive process and presenting the characteristic stridor. When it comes to laryngeal hemangioma in adults, especially in the supraglottic region, the symptoms differ from the presentation in children, including dysphagia for solids and progressively for liquids, dysphonia/hoarseness, dyspnea, recurrent hemoptysis, bronchoaspiration, globus pharyngeus, and more severely, acute respiratory failure, sudden collapse, and death [1,4,9,13-15].

This is a clinical case presentation with therapeutic review. We report a case of treatment of Laryngeal Hemangioma using sclerotherapy with Polidocanol in an adult patient. The clinical case was conducted at Hospital São José - SE by the otorhinolaryngology team at Clínica Otocenter.

The patient R.S.S., 35 years old, female, and a teacher, was referred from another otolaryngology service to Hospital São José - Otocenter, in the city of Aracaju - SE. She is a native and resident of Paulo Afonso - BA.

She complained of progressive dysphagia for solids and pasty consistency for the past 6 months, accompanied by a weight loss of 8 kg. Regarding her voice, she reported progressive dysphonia, hindering her professional role as a teacher and causing relative difficulty in social communication, leading to psychological distress. She also associated the difficulty in breathing (dyspnea) with mild to moderate efforts during daily physical activities. She denied a history of alcoholism and/or smoking. General otolaryngological examination revealed no alterations.

Perceptual auditory evaluation of the voice revealed dysphonia with altered resonance characterized by muffled and unprojected voice, along with vocal fatigue during moderate phonatory efforts.

Nasopharyngolaryngeal fibroscopy revealed a multiple or single (to be clarified) tumor-like lesion with a vascular appearance and bluish coloration, non-pulsatile, and extensive. It occupied a significant volume in the supraglottic and left glottic region, at the level of Areas d’Avila I and II (Figures 1-3) [16,17].

Figure 1: Videolaryngoscopy capture image demonstrating laryngeal hemangioma

Figure 2 Videolaryngoscopy capture image demonstrating laryngeal hemangioma with supraglottic extension

Figure 3 Illustration demonstrating the anatomical areas of d’Ávila I, II and III respectively from left to right. Source: D’Avila et al., [16]

Magnetic resonance imaging (MRI) of the neck revealed a neoplastic lesion in the glottis and supraglottis, more notably on the left, with obliteration of the pyriform sinus and ipsilateral vallecula; anterior commissure and vocal folds bilaterally. No nodular lesions were found in the neck (Figures 4-6).

Figure 4: Axial MRI image showing hemangioma in the left vocal fold

Figure 5: Sagittal MRI image showing mass in laryngeal topography

Figure 6: MRI in multiple axial sections showing a hyperintense lesion in laryngeal typography.

The treatment of laryngeal hemangioma remains a challenge due to its high vascularity, recurrent nature, and the lack of widely established protocols. Therapeutic options are selected based on the patient’s age, anatomy, size, and extent of the lesion, ranging from expectant clinical approaches to surgical interventions [1,5]. It is noteworthy that due to etiological differences between hemangiomas in children and adults, there is a tendency to adopt an expectant approach for children, while adults are more frequently subjected to early surgery [1,18]. Among the available therapeutic options are beta-blockers (e.g., propranolol and timolol), systemic and intramuscular corticosteroids, ethanol injections, cryosurgery, interferon, laser surgery, ultrasonic scalpel resection, sclerotherapy, embolization, and radiotherapy [1,4,19-22].

Surgical treatment of hemangiomas is complex due to the difficulty in delineating tumor margins due to the absence of well-defined dissection planes [5]. In the larynx, surgical intervention is performed through microlaryngoscopy, with or without complementary techniques such as Andrea and Dias’ rigid endoscopies, 1995 [22]. Various surgical options are employed in the treatment of laryngeal hemangiomas, including corticosteroid injections, lesion ablation, total or subtotal surgical resection, diode, KTP, or CO2 laser therapy, cryosurgery, sclerotherapy, and robotic surgery [3,5,9,11,23].

Sclerotherapy

Sclerotherapy with polidocanol was the therapeutic option for the presented case, being used in the form of foam through puncture, with absorption in the vascular wall ranging from 90% to 95%. The literature reveals low rates of systemic complications. Polidocanol is a sclerosing agent indicated for the treatment of uncomplicated varicose veins and is also used for other indications such as sclerotherapy for esophageal, gastric, nasal, and hemorrhoidal varices. The technique involves puncture with injection into the vein or area to be treated.

Polidocanol is functional for veins with a diameter of less than 1 mm, uncomplicated reticular veins, and veins with a diameter of 1 to 3 mm, preferably located at the extremities of structures. Its action aims to block blood flow at the site, causing endothelial injury with platelet aggregation, associated with the accumulation of cellular debris and fibrin, resulting in vascular occlusion.

Laurocapram Sulphonate, a polyethylene glycol derivative combined with dodecyl alcohol, acts as a surfactant on the capillary endothelium, causing an in-situ fibrosclerotic response and local thrombotic formation. This action leads to the collapse of telangiectasias and varices [24-26]. The application occurs in the form of foam, a mixture of liquid with air, prepared immediately before the procedure. The average dose should not exceed 2 mg/ kg/day, and the application is performed intravenously, injecting 0.1 to 0.3 ml of a 1% solution. Generally, 1 to 4 sessions are required to achieve satisfactory effects in external organs.

Side effects may occur, requiring special care and administration by specialist physicians. At the peripheral level, hyperpigmentation, irritation, and even local necrosis can occur, in addition to inflammatory phenomena (phlebitis), allergic reactions, metallic taste, dizziness, and drowsiness. In esophageal sclerotherapy, complications such as stenosis, hemorrhages, ulcerations, necroses, visual disturbances, metallic taste, vascular collapse, and fever may occur. In this specific case, these complications were not evidenced, highlighting only mild respiratory difficulty during extubation in the immediate postoperative period of the second procedure, which was resolved normally after a few minutes, still in the operating room [27].

As for contraindications to sclerotherapy treatment, absolute contraindications include allergy to the sclerosing agent, acute deep vein thrombosis and/or pulmonary embolism, generalized or puncture site infection, prolonged immobilization, kidney disease, liver failure, heart failure, arteriosclerosis, and diabetes. Relative contraindications include poor general health, predisposition to allergies, pregnancy, breastfeeding, and a family history of thromboembolic events. Regarding the disadvantages of the procedure, occurrences of phlebitis and thrombosis stand out, which may result in local hardening and pain, as well as the appearance of dark spots on the skin. These spots may completely disappear or leave dark areas on the skin, especially in cases of cutaneous application [28,29].

Tessari Technique

Recognized as the Three-Way Technique, it is performed by mixing 4 ml of biological gas with 1 ml of liquid polidocanol, a surfactant detergent. To generate the foam, the solution is mixed approximately twenty times, using two disposable syringes connected by a three-way stopcock. The otolaryngologist, under microlaryngoscopy, performs the procedure with pre-defined injection sites. Under video documentation, the procedure is performed by infiltrating the medication under direct vision, distributing polidocanol into the tissues, around 2.5 to 3 ml at two to three application points. Perilesional infiltration is performed to avoid possible bleeding, and an immediate reduction in the volume of the lesion is observed, albeit in small proportion (Figures 7-9).

Figure 7: Materials needed to prepare the polidocanol solution

Figure 8: Photograph illustrating the arrangement of the interdisciplinary team in the surgical field

Figure 9: Axial MRI showing significant control after the third procedure

The control MRI after the third procedure presented the following report: “Tumor lesion with characteristics of residual and/or recurrent hemangioma. Trachea and infraglottic region without abnormalities (Figures 10,11).

Figure 10: Videolaryngoscopic images of the preoperative appearance of laryngeal hemangioma

Figure 11: Video laryngoscopic images of the post-sclerotherapy appearance

We now present the evolution of treatment from preoperative to the result after the third procedure.

The intra-tissue transoperative infiltration of polidocanol represented a safe therapeutic option, with a significant reduction in the cavernous hemangioma and consequent relative control of the patient’s dysphonia, dyspnea, and dysphagia. In light of the above, we recommend its use with the appropriate application of safety criteria. Finally, for the safe execution of the procedure, the availability of an experienced interdisciplinary team that understands all aspects of treatment is of fundamental importance. This team should include qualified otolaryngologists, vascular surgeons, and stomatologists.

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