Bipolar radiofrequency-induced interstitial thermoablation for oral cavity vascular malformations: preliminary results in a series of 5 children.
We report our assessment of the effectiveness of bipolar radiofrequency-induced interstitial thermoablation (BRIT) for the treatment of certain oral cavity vascular malformations in 5 children. Two of these patients had lymphangiomatous macroglossia (LM), 1 had lymphangioma circumscriptum (LC), and 2 had a venous malformation (VM). Each patient underwent BRIT at least twice; treatment was delivered at 4- to 8-week intervals according to each patient's circumstances. The 2 patients with LM required three treatment sessions; although their tongue volume decreased after each session, both still required a partial glossectomy to achieve a satisfactory reduction in volume. The patient with LC underwent two BRIT treatments, which reduced the size of the lesion by half; the remainder was excised. The 2 patients with a VM (1 buccal and 1 lingual) responded well to BRIT, and their malformations almost completely disappeared. Our early results with BRIT suggest that it is an effective treatment for oral cavity vascular malformations--more so for patients with venous rather than lymphangiomatous lesions.
Vascular malformations represent errors of embryonic development. They can be capillary, lymphatic, venous, arterial, and combinations thereof. (1) Vascular malformations in the tongue, which are always congenital, can cause severe swallowing and articulation difficulties. (2)
Lymphatic malformations, also known as lymphangiomas, are composed of dysplastic vesicles or pouches filled with lymphatic fluid. Macrocystic, microcystic, and combination forms have been described. (1) Some 75% of all cases occur in the head and neck region. (3) Lingual lymphatic malformations are the most common lymphatic anomalies of the tongue. (3) They are frequently localized to the anterior two-thirds of the tongue. (4) They characteristically enlarge after an episode of upper respiratory tract infection. (3)
Lymphangiomas within the oral cavity are the most common cause of pediatric macroglossia. (2) This condition may go unnoticed until after dentition develops or even after puberty. Significant airway obstruction may lead to the need for a tracheostomy.
Lymphangiomatous macroglossia (LM) has been treated with aspiration of the lymphatic fluid and either instillation or direct injection of sclerosing agents such as Ethibloc, pure ethanol, sodium tetradecyl sulfate, hypertonic saline, OK-432 (a killed strain of group A Streptococcus), bleomycin, or boiling water. However, each of these agents has been found to be ineffective, especially for microcystic lesions. Moreover, the use of sclerosing agents is associated with numerous side effects, including blistering, full-thickness mucosal necrosis, neural damage, and systemic complications such as cardiac arrest and renal toxicity. Therefore, surgery has traditionally been the preferred treatment for LM, despite the relatively recent successes of radiofrequency ablation. (5,6)
Another vascular malformation is lymphangioma circumscriptum (LC). It has been postulated that LC consists essentially of a collection of large lymphatic cisterns. (7) These cisterns lie deep in the subcutaneous plane, and they communicate with the superficial vesicles via dilated dermal lymphatics. (7) Traditional treatment consists of surgical excision, laser therapy, sclerotherapy, and radiotherapy.
Venous malformations (VMs) are also present at birth, but they are not always evident. VMs grow slowly in proportion to the growth of the child, and they often enlarge during puberty. Oral VMs typically involve the tongue, palate, and oropharynx, but they rarely impair speech. Open-bite deformity and dental malalignment of varying degrees are characteristic. (4) VMs tend to recanalize and recur. For treatment, injections of sclerosing agents similar to those used for LMs can be used, but they are not particularly effective in this case, either. When sclerotherapy is used, surgical resection of the remaining lesion is still typically required afterward. (5)
Temperature-controlled radiofrequency volumetric tissue reduction was introduced to otolaryngology practice in 1998. (8) Since then, this simple, safe, minimally invasive, and easily performed procedure has prompted continuing research for uses in a variety of conditions. In this article, we describe our early experience with the use of bipolar radiofrequency-induced interstitial thermoablation (BRIT) for the treatment of lymphangiomatous and venous lesions of the oral cavity.
We used BRIT to treat 5 children--3 girls and 2 boys, aged 4 to 16 years--who had a vascular malformation of the oral cavity (table). A 5-year-old girl and a 4-year-old boy were treated for LM; a 9-year-old girl was treated for buccal LC; and a 14-year-old girl and a 16-year-old boy were treated for a buccal and lingual VM, respectively. The 3 patients with a lingual lesion were treated under general anesthesia, and the 2 patients with a buccal lesion received local infiltration anesthesia.
BRIT was delivered via a power-control unit (CelonLab ENT; Celon AG Medical Instruments; Teltow, Germany) with a disposable bipolar radiofrequency applicator (Celon ProSleep Plus). The energy was delivered submucosally to the tongue (power level 7) or buccal mucosa (power level 15) as indicated. The delivery of energy was terminated automatically by the radiofrequency generator according to tissue resistance.
Repeat applications were performed in a similar fashion at 4- to 8-week intervals in accordance with each patient's needs. The 3 patients with tongue lesions were followed in the hospital for at least 1 week. Throughout their postoperative hospitalization, they were given corticosteroids, paracetamol, and cefazolin.
None of the 5 patients required a tracheostomy, and no serious complications were noted.
Patient 1 (LM). A 5-year-old girl was brought to our clinic with macroglossia (figure 1). She was experiencing difficulty eating and speaking. Other complications included drooling and intermittent bleeding from the anterior part of the tongue. Physical examination revealed that the lingual enlargement was localized to the anterior two-thirds of the tongue. The tongue exhibited diffuse microcystic disease, and it was firm on palpation. Tongue movements were limited.
[FIGURE 1 OMITTED]
The patient underwent three applications of BRIT. During the first session, a total of 553 J of radiofrequency energy was applied to 6 points on the tongue. Thereafter, 535 J was applied to 8 points during the second session, and 749 J was delivered to 10 points during the third. Reactive enlargement of the tongue was observed after each session, but it resolved in approximately 2 weeks. Although a significant decrease in tongue volume was noted at the end of BRIT, it was not enough, and therefore a two-step partial glossectomy was performed. The pathologic examination of the excised specimen was consistent with a capillary lymphangioma.
Patient 2 (LM). A 4-year- old boy presented with macroglossia (figure 2). He also was experiencing difficulty eating and speaking, as well as drooling and intermittent bleeding from the anterior part of the tongue. He had previously undergone surgery that involved a partial excision of the tongue. As was the case with patient 1, physical examination revealed that the lingual enlargement was localized to the anterior two-thirds of the tongue and that the tongue was firm on palpation, but this patient's tongue movements were normal.
[FIGURE 2 OMITTED]
Patient 2 also underwent three sessions of BRIT. During each session, a total of 440 J of radiofrequency energy was applied to 6 areas. Only a moderate response to BRIT was seen, and another partial glossectomy was performed. During the postoperative period, a hematoma developed and drainage was performed. The pathologic examination of the specimen was consistent with a capillary lymphangioma.
Patient 3 (buceal LC). A 9-year-old girl presented with facial cellulitis and a right-sided buccal mucosal lesion (figure 3). The cellulitis was treated with an antibiotic, and it resolved. Findings on biopsy of the mucosal lesion were consistent with LC.
The LC was treated with a total of 450 J of BRIT applied to 6 points. During a second session, 480 J was applied to 6 points. After BRIT, the lesion had shrunk to half its original size, which represented a moderate improvement. The remainder was excised.
Patient 4 (buccal VM). A 14-year-old girl presented with a 2.0 x 2.5-cm buccal VM. The lesion was solitary, low-flow, and easily compressible (figure 4, A).
After the patient received 315 J of BRIT, the lesion showed considerable regression (figure 4, B). A second session was planned to completely eradicate the lesion, and 275 J was delivered. Upon completion of BRIT, the mucosal surface of the lesion was smooth, and the bluish hue returned to a normal pink.
Patient 5 (lingual VM). A 16-year old boy presented with two separate VMs of the tongue. One lesion was located on the middle one-third of the dorsum, and the other was on the lateral aspect on the same side and at the same level as the first.
During three sessions of BRIT, a total of 420, 400, and 450 J, respectively, were applied. At the end of the therapy, the lesion had almost completely disappeared, and the color of the mucosa returned to normal, leaving the patient satisfied with the treatment.
The role of radiofrequency thermotherapy for the treatment of oral vascular malformations is not well described in the literature, since it is a relatively new radiofrequency technique for this indication. (9,10) As a result, clinicians might not be completely familiar with all the terminology. Here is a brief description of some terms:
* The term thermotherapy applies to a procedure in which tissue is heated to a temperature of 60[degrees]C or higher. This process results in coagulation.
* The term interstitial means that BRIT exerts its effects inside the tissue. An electrode is inserted through the surface of the tissue, and energy is delivered to a deeper layer, thus avoiding damage to the surface.
* Radio frequency-induced in this case indicates that the heating process is generated by high levels of radiofrequency in alternating currents (approximately 500 kHz).
[FIGURE 3 OMITTED]
Radiofrequency surgery has become a popular option for treating adult obstructive sleep apnea syndrome, where inducing scar formation in the tongue base, pharyngeal tonsils, inferior turbinate, and soft palate helps alleviate symptoms. It has already been used successfully in the treatment of hepatic malignancies, (11-13) hepatic cavernous hemangiomas, (14) and some head and neck tumors. (15,16)
[FIGURE 4 OMITTED]
The rationale for using BRIT to treat vascular malformations is based on it volume-reduction effect on tissue. (17,18) Cable and Mair used a monopolar radiofrequency generator (Somnus Medical Technologies; Sunnyvale, Calif.) in 2 patients and introduced radiofrequency tissue ablation as an effective method for microcystic LM. (6)
We used bipolar treatment in 2 patients with LM. At the end of three sessions, both patients exhibited a reduction in tongue size, an increase in motion, and improvement in swallowing function. However, since both of these patients initially presented with a significant degree of lingual enlargement, BRIT alone was not sufficient, and both required a partial glossectomy.
In our patient who had buccal LC, the response to BRIT was only moderate, and surgical excision was needed. In the 2 patients with a VM, the response to BRIT was satisfactory and no additional therapy was needed. Our experience has shown that even though BRIT was effective for both lymphangiomatous lesions and VMs, it was more effective for the latter.
While BRIT alone was not sufficient for eradicating the LMs and the LC, which were fairly large, it might be able to adequately treat patients with small lymphangiomatous lesions, as well as those in whom airway obstruction is not observed and in those for whom surgical excision would be difficult. Even when surgery is required, tissue reduction with BRIT might be beneficial because it would reduce the amount of bleeding related to scar tissue formation and it would allow for easier postoperative reconstruction.
Leboulanger et al reviewed 7 cases of pediatric lingual lymphangioma treated with surface radiofrequency reduction; all patients' symptoms were improved, but 2 of the 7 children experienced a local relapse. (19)
In a more recent study, Kim et al reviewed 26 cases in order to report the long-term results of radiofrequency ablation for the treatment of lymphatic malformations of the oral cavity in children and adolescents. (20) All the malformations were localized superficial symptomatic lesions. At a mean follow-up of 47 months, one-half of the patients were symptom-free. Among the remaining 13 patients, 8 were improved and required no further treatment, while 5 did require further treatment. All patients tolerated the procedure well, and almost all had a short recovery time.
The number of patients in our series is obviously too small to allow for definitive conclusions to be reached about the use of BRIT for oral cavity VMs. Nevertheless, the fact that it was effective in both patients holds promise.
In conclusion, the results of our early experience with BRIT suggest that it is an effective method of reducing oral cavity vascular malformations, particularly VMs. The number of thermoablation sessions that any given patient will require will depend on the size of the lesion and its response to therapy. Further studies with more patients are needed to establish radiofrequency thermoablation as an effective therapeutic modality for patients with vascular malformations.
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(11.) Rossi S, Di Stasi M, Buscarini E, et al. Percutaneous radiofrequency interstitial thermal ablation in the treatment of small hepatocellular carcinoma. Cancer J Sci Am 1995;1(1):73-81.
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Senol Civelek, MD; Ibrahim Sayin, MD; Ibrahim Ercan, MD; Burak Omur Cakir, MD; Suat Turgut, MD
From the 1st ENT Clinic, Sisli Etfal Teaching and Research Hospital, Istanbul (Dr. Civelek, Dr. Ercan, Dr. Cakir, and Dr. Turgut); and the ENT Clinic, Bakirkoy Teaching and Research Hospital, Istanbul (Dr. Sayin). The cases described in this article occurred at the Sisli Etfal Teaching and Research Hospital.
Corresponding author: Ibrahim Sayin, MD, ENT Clinic, Bakirkoy Teaching and Research Hospital, Tevfik Saglam Cad. No: 11 Zuhuratbaba, Postal Code 34417, Istanbul, Turkey. Email: dribrahimsayhin@ yahoo.com
Table. Summary of cases Age (yr) No. of BRIT Pt. sex Condition sessions 1 5/F Lymphangiomatous 3 macroglossia 2 4/M Lymphangiomatous 3 macroglossia 3 9/F Buccal lymphangioma 2 circumscriptum 4 14/F Buccal venous 2 malformation 5 16/M Lingual venous 3 malformation Pt. Response Subsequent surgery 1 Significant reduction Partial glossectomy 2 Moderate reduction Partial glossectomy 3 Moderate; lesion shrunk Remainder excised by half 4 Complete None 5 Almost complete None
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|Title Annotation:||ORIGINAL ARTICLE|
|Author:||Civelek, Senol; Sayin, Ibrahim; Ercan, Ibrahim; Cakir, Burak Omur; Turgut, Suat|
|Publication:||Ear, Nose and Throat Journal|
|Article Type:||Case study|
|Date:||Nov 1, 2012|
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