ORIGINAL ARTICLE
Long-term Outcome of Radiofrequency Ablation
for Intraoral Microcystic Lymphatic Malformation
Sang W. Kim, MD; Katie Kauvanough, MD; Darren B. Orbach, MD, PhD;
Ahmad I. Alomari, MD, MSc; John B. Mulliken, MD; Reza Rahbar, DMD, MD
Objective: To determine long-term outcome of radio-
frequency (RF) ablation of microcystic lymphatic malformation (LM) of the oral cavity for control of recurrent infection and bleeding.
Design: Institutional review board–approved retrospec-
tive study,
Setting: Tertiary pediatric medical center,
Patients: Twenty-six patients with intraoral microcys-
tic LM were treated with RF ablation from August 2002
through August 2010.
dure. Ten patients (38%) began eating on POD 1, and
virtually every patient was on full oral intake at discharge. Fourteen patients (55%) required only acetaminophen for pain control, 11 (41%) required acetaminophen with codeine, and 1 (4%) required
oxycodone. The mean follow-up time was 47 months after treatment. At the most recent clinic evaluation, 13 patients (50%) were symptom free, 8 (31%) were stable and
improved without need for future treatment, and 5 (19%)
required further treatment. One-half of patients in the
study group underwent more than 1 RF procedure for
recurrence. The number of RF ablations in this series were
1 procedure (n=13), 2 procedures (n=7), 3 procedures
(n=2), 4 procedures (n=2), and 6 or 7 procedures (n=2).
Intervention: Radiofrequency ablation of intraoral LM.
Main Outcome Measures: Postoperative stay, diet,
pain; control of bleeding and/or infection; recurrence; and
indication for retreatment.
Results: The most common complaints necessitating initial RF ablation were recurrent infection (n=10 [37%])
and bleeding (n=9 [33%]). The most common problems requiring further ablation were bleeding (n=11
[41%]) and cosmetic deformity not affecting function (n=8
[31%]). Fourteen patients (55%) were discharged home
on postoperative day (POD) 3; the remaining 11 (45%)
were discharged home on POD 4. Thirteen patients (52%)
resumed oral diet immediately on the day of the proce-
Author Affiliations:
Department of Otolaryngology
and Communication
Enhancement (Drs Kim,
Kauvanough, and Rahbar),
Division of Vascular and
Interventional Radiology
(Drs Orbach and Alomari), and
Departments of Plastic Surgery
and Surgery (Dr Mulliken),
Children’s Hospital Boston, and
Department of Otolaryngology,
Massachusetts Eye and Ear
Infirmary (Dr Kim), Harvard
Medical School, Boston,
Massachusetts.
L
Conclusions: Radiofrequency ablation is an effective treatment for localized, superficial microcystic LM in the oral
cavity. Pediatric patients tolerate the treatment with rapid
postoperative recovery and minimal complications. The majority of patients required a short hospital stay for observation of the airway. Virtually every patient resumed oral
diet by the time of discharge. Radiofrequency ablation is
the treatment of choice at Children’s Hospital Boston (CHB)
for patients who present with symptomatic, superficial, and
localized intraoral microcystic LM. For lesions involving
deeper structures, multimodal treatments including surgical and sclerotherapy may be necessary.
Arch Otolaryngol Head Neck Surg. 2011;137(12):1247-1250
YMPHATIC MALFORMATION
(LM) most commonly occurs in the head and neck region and is categorized based
on the radiographic size of
the cysts into microcystic “lymphangioma” (⬍2 cm3), macrocystic (ⱖ2 cm3),
or combined lesions. The microcystic and
macrocystic lesions are indistinguishable
when examined for histologic features and
with immunohistochemical staining for
markers of lymphatic endothelium.1 Clinically, microcystic lesions tend to have more
mucosal involvement and tend to recur.
Macrocystic lesions are amenable to sclerotherapy or resection, whereas excision of
microcystic LM is always incomplete.
ARCH OTOLARYNGOL HEAD NECK SURG/ VOL 137 (NO. 12), DEC 2011
1247
Approximately 50% of LMs are seen at
birth as an asymptomatic mass, and by the
age of 2 years, 90% of the lesions present
with clinical symptoms. Spontaneous regression is seldom observed, with rare exception in some cases of macrocystic LM.
The typical clinical course for microcystic
LM is intermittent swelling secondary to
trauma or inflammation with pain and
bleeding. In addition to deformity, functional consequences of intraoral microcystic LM include difficulty in speaking, poor
oral hygiene, malocclusion, dysphagia, and
sometimes respiratory compromise.2
Radiofrequency (RF) ablation, or “coblation,” is a relatively new technology that
is being used in various areas of otolarynWWW.ARCHOTO.COM
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Table 1. Pediatric Patients With Lymphatic Malformation
of Oral Cavity
All patients were extubated in the operating room and encouraged to take liquids as tolerated. Patients were discharged home
with the same pain regimen used during hospitalization and
given 7 to 10 days of oral antibiotics.
No. of Patients
(N=26)
Characteristic
Total coblation procedure
Sex
Male
Female
Location of disease treated with coblation
Oral tongue
Floor of mouth
Lip
Base of tongue
Buccal
Age at coblation treatment, mean (range)
RESULTS
54
13
13
19
6
4
3
2
10.4 y (29 mo to 18 y)
gology. Owing to its mechanism of low-temperature tissue destruction, the thermal injuries to surrounding tissues are limited, and therefore patients may have a better
course of recovery. Two modes of RF ablation have been
described. The high-frequency mode is used for destruction of deep tissue, resulting in reduction in size from
secondary fibrosis without affecting the mucosal surface. In low-frequency mode, energy is transmitted via
conduction medium in small volumes for accuracy and
for the removal of a thin superficial layer, with minimal
thermal injury to nearby tissue.
Several articles have shown promising outcomes in treating LM in the oral cavity using the RF ablation technique,
including one from our group. We report herein our longterm outcome and experience using RF ablation for microcystic LM in the oral cavity. For selective patients with lesions that are localized and superficial, we have observed
excellent results with RF ablation in terms of postoperative
recovery, complication rate, and clinical outcome.
METHODS
The study group comprised of 26 children with intraoral LM who
underwent RF ablation treatment at the Children’s Hospital Boston, Boston, Massachusetts, from August 2002 through August
2010. Medical records were reviewed for the location of lesions,
previous treatments, and presenting signs and symptoms. Electronic medical records and inpatient medication records were
accessed to document duration of hospitalization, resumption
of oral intake, and pain regimen on the day of discharge; 21 patients had complete electronic medical records available. For outcome data, 4 of 26 patients were excluded owing to a follow-up
duration of less than 4 months. Of the remaining 22 patients, a
total of 50 RF ablation treatments were completed. Disease status was categorized based on examination by the senior otolaryngologist (R.R.) on follow-up clinic visits. On the basis of this
evaluation, patients were selected for RF ablation procedure according to the extent of the lesion, specifically those that are limited to superficial and localized lesions.
Radiofrequency ablation treatment was performed by the
senior otolaryngologist with the patient under general anesthesia with oral or nasal intubation. Intraoperative antibiotics
and corticosteroid (dexamethasone, 0.5 mg/kg) were administered. Microcystic LM was removed in layers from superficial to deep in broad stroke with continuous saline irrigation
using the coblator (Evac 70 Plasma Wand; ArthroCare Corp).
A total of 26 patients with localized, superficial microcystic lymphatic malformation in the oral cavity underwent RF ablation treatment at our institution (Table 1).
There was an equal sex distribution in the study group.
Some of these patients required more than 1 RF ablation treatment.
The most common location of the LM was the tongue
(n=19 [53%]). Thirteen patients had involvement of multiple anatomic sites; in Table 1 we report only the anatomic sites that met the selective criteria for RF ablation.
These were localized, superficial microcystic LM involving mucosal surface of oral cavity based on clinical assessment. For LM lesions that were more extensive than those
selected for RF ablation, patients in our series have undergone 31 surgical resections, 11 sclerotherapy procedures,
and 6 laser treatments. The youngest patient in our series
at the time of RF ablation was 29 months old and the oldest was 18 years old; the mean age was 10.4 years. Among
the 26 patients, a total of 54 RF ablation procedures were
performed during the 8-year period.
Presenting signs and symptoms that prompted RF ablation are listed in the Figure. Indications were divided into
presenting problems and residual problems requiring furthertreatment.Themostcommonpresentingproblemswere
recurrent bleeding and infection unresponsive to several
courses of antibiotics, affecting daily life or school activities.
Thirteen patients underwent a single RF ablation treatment. Seven patients underwent 2 RF ablative treatments: 3 reported satisfactory symptom control at the follow-up visit, 3 reported intermittent bleeding, and 1 had
persistent episodes of recurrent infection with swelling
and bleeding. Two patients underwent 3 RF ablation treatments: 1 reported satisfactory control at the follow-up
visit and the other had intermittent problems. Two patients had 4 RF ablation treatments and both reported
satisfactory control. One patient underwent 6 RF ablation treatments, and another patient underwent 7 RF ablation treatments. They reported resolution of symptoms or some intermittent problems, respectively.
One patient was transferred to the intensive care unit
from floor care status because of marked tongue edema on
examination on postoperative day (POD) 1. The patient
was treated with conservative medical management (intravenous antibiotic and corticosteroid), with resolution
of swelling, and was able to be transferred to floor care within
48 hours. On POD 5, the patient again developed tongue
edema, prompting a transfer to the intensive care unit for
airway monitoring. Swelling resolved without affecting the
airway, and the patient was discharged home on POD 10.
In retrospect, this patient had a lymphatic-venous malformation that presented with intralesional bleeding postoperatively. The majority of patients were tolerating oral diet
by POD 1, as given in Table 2. Postoperative oral diet regimen consisted of advancing diet from clear sips to soft con-
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A
B
C
Figure. Microcystic lymphatic malformation in base of the tongue. A. Preoperative; B, immediate postoperative; and C, at 2-year follow-up visit.
Table 2. Postoperative Course Following
Radiofrequency Ablation a
Table 3. Clinical Outcome of Radiofrequency Ablation
for Intraoral Microcystic Lymphatic Malformation a
Patients, No. (%)
(n= 22)
Variable
Days of hospitalization
2
3
⬎3
Start of oral diet
POD 0
POD 1
POD 2
⬎POD 2
Pain regimen on the day of discharge
Tylenol
Tylenol with codeine
Oxycodone
Complications
Transfer to ICU (% of total procedures)
Airway intervention required
11 (50)
10 (45)
1 (5)
12 (55)
8 (38)
1 (5)
1 (5)
Variable
Value
Duration of follow-up, mean, y
Duration between repeated procedures, mo
Disease status at the most recent clinic follow-up visit,
No. (%)
No symptom complaints
Some minor symptoms
Significant symptoms/revision procedure planned
4.3
24.1
aPatient
data available for 22 patients (50 treatments).
12 (55)
9 (41)
1 (5)
2 (4)
0
Abbreviations: ICU, intensive care unit; POD, postoperative day.
a Patient data available for 22 patients.
sistency. All patients demonstrated at least 400 mL of total
oral intake prior to discharge. By the time of discharge, pain
was not a significant source of complaint.
Symptom-free state is defined as not having any episode of bleeding and/or infection. This is based on parents’ recount during the subsequent follow-up clinic visit
with the senior otolaryngologist. All patients are followed up within 2 weeks postoperatively, then every 3
months or sooner if necessary. The clinical outcome data
on Table 3 are based on assessment at the most recent
clinic follow-up visit, at least 6 months after the procedure. Status of symptom control was categorized into 3
tiers: no complaints, minor symptoms, and major symptoms and/or revision procedure planned. The majority
of patients were in a satisfactory symptom-free state at
their most recent clinic visit (Figure). Eight patients (31%)
continued to have intermittent symptoms such as bleeding and/or infection, which were effectively managed with
oral antibiotic and had no significant impact on the quality of life. For the 5 of patients (19%) categorized as having major symptoms, recurrent episodes of infection,
swelling, and/or bleeding were affecting quality of life,
and revision RF ablation was planned.
11 (50)
7 (31)
4 (19)
COMMENT
Our preliminary report on RF ablation treatment for intraoral microcystic LM was promising.3 In this longterm follow-up series, most patients recovered from the
procedure without major pain and promptly resumed oral
diet, with few postoperative problems.
Several treatment modalities have been described for LM
including resection, sclerotherapy, cryotherapy, electrocautery, RF ablation, corticosteroid administration, and embolization. Recurrence rates following attempted resection
range from 50% to 100%.4-7 Numerous sclerosing agents
have been used in the treatment of macrocystic LM including bleomycin sulfate, alcoholic solution of zein (Ethibloc;
Ethicon), tetracycline, dextrose, interferon alfa-2a, fibrin
sealant, and OK-432 (Picibanil; Chugai Pharmaceutical Co
Ltd).8,9 OK-432 is thought to leave minimal perilesional fibrosis, making subsequent resection less hazardous.9 OK432 is more effective for macrocystic than microcystic lesions, with 86% vs 66% reported efficacy rates, respectively.8
Alternatives for the treatment of microcystic lesions include: carbon dioxide laser, potassium titanyl phosphate
laser, and RF ablation therapy in high- and low-power
settings.3,8-12 Laser treatment has the advantages of less postoperative edema, less surrounding tissue trauma, and less
blood loss compared with resection. Nevertheless, recurrence rate following laser treatment has been reported to
range from 50% to 66%.8 A few reports have described RF
ablation as an option for management of microcystic LM
in the oral cavity.3,11,12 Roy et al11 described 3 children who
underwentRFablationoftongueLMwithoutcomplications;
all 3 showed no evidence of recurrence at the 1-year follow-
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up visit. Grimmer et al3 described 11 children who had RF
ablation; 62% reported in a survey that RF ablation was more
effective at controlling symptoms than previous procedures
(surgical excision, laser, or sclerotherapy).
Selection of the patient is critical in considering RF ablation treatment for LM. While there is no standardized
medical regimen in our practice, patients in the series are
typically treated with systemic antibiotics for acute infection and optimization of oral hygiene with mouth rinse and
frequent dental care prior to and after the procedure. Recurrent symptoms, including acute bleeding, infection, enlargement affecting function, or cosmesis requiring frequent antibiotic therapy, were the indication for RF ablation
treatment for superficial mucosal intraoral LM lesions.
In those patients who required repeated RF ablation
treatment, it was notable that the indication was different
than that for their initial treatment. Specifically, cosmetic
deformity became the main indication for secondary treatment. Given how well patients tolerated the RF ablation
during their initial treatment, patients and family developed higher expectations for overall symptom control and
opted for subsequent revision RF ablations.
While the aim of this study was to describe our experience in treating superficial microcystic LM lesions involving all intraoral subunits, it is notable that the majority (53%) of the intraoral lesions in our series involved the
oral tongue. Management of the microcystic LM involving the oral tongue is particularly challenging because of
its propensity for recurrent infection, hemorrhage, airway
compromise, feeding difficulties, and its effect on speech.
Microcystic LM of the oral tongue has been classified based
on the extent of lesion to plan for treatment and predict
outcome.13,14 Recently Wiegand et al13 developed a classification system for LM of the oral tongue into 4 stages:
(1) superficial microcystic LM; (2) lesion involving tongue
muscle; (3) lesion involving tongue and the floor of mouth;
and (4) extensive lesion involving tongue, floor of mouth,
and other cervical structures. In their series, carbon dioxide laser ablation has demonstrated an excellent curative
treatment for stage 1 and some stage 2 lesions. Detailed postoperative course (ie, timing of resuming oral diet, pain regimen) following carbon dioxide laser ablation was not available. For more advanced lesions, combined and/or staged
approaches were necessary.13 Patients in our series who have
been selected for RF ablation for their oral tongue lesions
were mainly stage 1 lesions limited to the superficial mucosa based on clinical evaluation by the senior author. For
patients with more extensive microcystic LM of the oral
tongue, RF ablation is not the ideal primary treatment modality of choice. The characteristic feature of the RF ablation—low-temperature tissue destruction with limited injury to surrounding tissue—also serves as a limitation for
lesions that are deep and involving underlying muscle.
The rate of complications was very low; however, 1 patient required transfer to the intensive care unit for respiratory observation. We recommend that every patient who
undergoes RF ablation treatment of the oral cavity be observed in the hospital during the immediate postoperative
period, with a low threshold for intensive monitoring if the
patient develops any respiratory symptoms. Possible complications include postoperative hemorrhage leading to acute
swelling and airway obstruction.
Our experience with RF ablation treatment for localized,
superficial microcystic LM in pediatric patients has been
very promising. Postoperatively, patients did not have significant pain issues, tolerated feeding almost immediately,
and had few airway complications secondary to tissue swelling. Radiofrequency ablation is an excellent tool for management of intraoral LM for selected patients. These are ones
with symptomatic, superficial microcystic lesions. For lesions involving deeper structures, multimodal treatments
including surgical and sclerotherapy may be necessary.
Submitted for Publication: April 25, 2011; final revision
received August 8, 2011; accepted September 18, 2011.
Correspondence: Reza Rahbar, DMD, MD, Department
of Otolaryngology and Communication Enhancement,
Children’s Hospital Boston, Harvard Medical School, 300
Longwood Ave, Ste LO367, Boston, MA 02115 (reza
[email protected]).
Author Contributions: Dr Rahbar had full access to all the
data in the study and takes responsibility for the integrity
of the data and the accuracy of the data analysis. Study concept and design: Kim. Analysis and interpretation of data: Kauvanough, Orbach, Alomari, Mulliken, and Rahbar. Drafting of the manuscript: Kim. Critical revision of the manuscript
for important intellectual content: Kim, Kauvanough, Orbach,
Alomari, Mulliken, and Rahbar. Study supervision: Kim,
Kauvanough, Orbach, Alomari, Mulliken, and Rahbar.
Financial Disclosure: None reported.
Previous Presentation: This study was presented in the
poster session at the American Society of Pediatric Otolaryngology meeting; April 29–May 1, 2011; Chicago,
Illinois.
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