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CO2 laser resurfacing of intraoral lymphatic malformations: A 10-year experience
International Journal of Pediatric Otorhinolaryngology 73 (2009) 1358–1361
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
CO2 laser resurfacing of intraoral lymphatic malformations: A 10-year experience Robert S. Glade a,*, Lisa M. Buckmiller b a b
The Children’s Hospital at OU Medical Center, Department of Otolaryngology, Head and Neck Surgery, Oklahoma City, Oklahoma, USA Vascular Anomalies Center, University of Arkansas for Medical Sciences, Arkansas Children’s Hospital, Little Rock, Arkansas, USA
A R T I C L E I N F O
A B S T R A C T
Article history: Received 12 January 2009 Received in revised form 16 June 2009 Accepted 18 June 2009 Available online 22 July 2009
Objective: To evaluate the safety and efficacy of CO2 laser resurfacing in the symptomatic treatment of intraoral lymphatic malformations (LM). Design: Retrospective review. Methods: Charts were reviewed on 26 patients (16 male, 10 female) from January 1997 to July 2007 who underwent CO2 laser resurfacing for symptomatic treatment of intraoral LM. A questionnaire was given in order to elucidate effectiveness in controlling symptoms and speed of postoperative recovery. Results: Mean age at time of first treatment was 9.2 years (median 6.8). Mean number of treatments was 3.0 (median 2.5). Average time between treatments was 9.7 months (median 5.6). Questionnaires were returned for 17 patients (65%). Common preoperative symptoms included swelling, bleeding, vesicle formation, and pain. All 17 patients reported symptomatic improvement after laser treatment. Five patients (29%) tolerated oral intake immediately, 10 (59%) the following day, and 1 (6%) was gastric tube dependent. Four patients (24%) returned to normal activity immediately after treatments, six (35%) by the following day, six (35%) within a few days, and one (6%) within a week. No postoperative complications were seen. Conclusion: CO2 laser resurfacing appears to be both safe and efficacious in treatment of symptoms related to intraoral LM. Intermittent treatments for recurrent symptoms is expected. ß 2009 Elsevier Ireland Ltd. All rights reserved.
Keywords: CO2 laser Lymphatic malformation Tongue lymphatic malformation
1. Introduction Lymphatic malformations (LM) are slow flow vascular anomalies most commonly presenting in the head and neck of children. Fifty to sixty percent are detectable at birth with 80% diagnosed before two years of age [1]. While LM are benign, they continue to expand throughout life and can compress adjacent structures. Many undergo rapid enlargement secondary to infection, hemorrhage, or trauma. Rapid growth may lead to airway compromise, dysphagia, speech difficulty, or gross cosmetic deformity. LM are classified as macrocystic, microcystic or mixed based on cyst size. While macrocystic lesions are often amenable to surgery and sclerotherapy, the infiltrative nature of microcystic lesions makes total surgical excision difficult without causing unacceptable loss of function. Subtotal resection is complicated by a high recurrence rate and sclerotherapy has also proven to be ineffective in the treatment of microcystic disease [2]. Intraoral lymphatic malformations are rare and nearly always of the mixed or microcystic classification. Tongue lesions may cause bleeding, halitosis, dysphagia, dysarthria, and airway
obstruction. Macroscopically, the hallmark of intraoral LM is mucosal vesicle formation which grossly resembles fish eggs (Fig. 1). These vesicles represent small lymph filled cysts, but may develop a purple color secondary to intracystic hemorrhage. Microscopically, endothelial-lined spaces within the epithelial, subepithelial, and muscular layers are seen. This often leads to associated muscular atrophy [3]. Acute enlargement of the tongue frequently occurs consequent to upper respiratory or even remote infections causing extraoral tongue protrusion and increased exposure to dental trauma. Repeated episodes of infection or trauma lead to inflammation and further enlargement of the tongue. Surgery has been the mainstay in the treatment LM of the tongue, but is limited secondary to the need to preserve tongue function. Without complete surgical resection, cure cannot be achieved and newer treatments target symptom management. We have successfully used the CO2 laser for tongue resurfacing at Arkansas Children’s Hospital for over 10 years in the treatment of intraoral lymphatic malformations. 2. Methods
* Corresponding author at: 800 Marshall Street, Little Rock, AR 72202-3591, USA. Tel.: +1 501 364 4790. E-mail address: [email protected] (R.S. Glade). 0165-5876/$ – see front matter ß 2009 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2009.06.013
Institution Review Board approval was first obtained for the study. Charts were reviewed on all patients with a diagnosis of LM involving the oral cavity and/or oropharynx treated at Arkansas
R.S. Glade, L.M. Buckmiller / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1358–1361
1359
Table 1 Patient data.
Fig. 1. Preoperative LM.
Children’s Hospital, Center for Vascular Anomalies from January 1997 through July 2007. Only those who underwent CO2 laser resurfacing for treatment of the mucosal component of their lesions were included. A database was created which included patient’s gender, date of birth, number of procedures, and date of procedures. Treatment in all cases used a CO2 laser (Surgipulse 60XJ with silk touch scanner, Lumenis Inc., Santa Clara, CA) set at 30 W, continuous scanning mode, with a 6 mm spot. All treatments were performed by either the senior author or a second staff physician. All cases were performed under general anesthesia with either oral or nasal endotracheal (ET) intubation. The laser was used to eliminate superficial vesicles followed by manual removal of debris and char with a moist gauze. Treatment was carried through the affected epithelial and subepithelial layer to the level of the underlying musculature of the tongue. The wound was not closed and allowed to heal by secondary intention. If oral mucosal surfaces other than the tongue were affected, that mucosa and submucosa was also removed. Strict laser precautions were employed during each procedure including use of appropriate ET tube, eye protection and appropriate placement of wet towels on the patient. All patients received a dose of intraoperative dexamethasone (0.5 mg/kg) and were extubated at the conclusion of the procedure. Patients undergoing their first treatment and those with extensive lesions were observed overnight for airway obstruction and received intravenous dexamethasone (0.5 mg/kg twice daily) and cefazolin (25 mg/kg) with continuous pulse oximeter monitoring. Discharge the following day occurred if patients demonstrated a safe airway and ability to tolerate oral feeds. All patients were discharged home on a high dose prednisone taper (2–3 mg/kg/day divided BID for one week with gradual taper over three weeks) and oral amoxicillin (25 mg/kg twice daily). Repeat treatment was performed when the patient began to experience return of symptoms. An attempt was made to locate all patients in order to obtain outcome data. Results were obtained from a questionnaire adapted from that created by Grimmer et al. [4] given by phone interview. Patients and parents were questioned regarding symptoms prior to treatment, symptoms after treatment, effectiveness in controlling symptoms, previous treatments other than CO2 laser resurfacing, and postoperative recovery including tolerating oral intake and return to normal activity. 3. Results A total of 26 patients (16 males, 10 females) met inclusion criteria undergoing total of 79 CO2 laser resurfacing procedures
Patient
M/F
Age first CO2 laser (years)
Number CO2 laser treatment
Mean time between treatments (months)
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26
M M M F M M F M F F F M M M M M F F F M M F M M M F
10.7 2.0 6.0 9.1 2.9 6.1 14.7 0.8 2.8 11.3 5.0 7.0 14.7 15.5 19.8 6.0 4.9 6.4 30.3 1.0 11.2 6.8 4.2 2.4 14.6 9.5
2 1 3 1 2 9 5 6 3 5 4 2 2 1 4 1 3 1 5 1 1 4 2 1 3 7
22.1 N/A 13.4 N/A 23.4 6.6 27.0 6.3 5.4 7.8 5.3 2.4 2.4 N/A 8.5 N/A 35.9 N/A 5.3 N/A N/A 6.3 7.1 N/A 4.4 4.5
(Table 1). The mean age at first resurfacing was 9.2 years (median 6.8 years) with a mean time between treatments of 9.7 months (median 5.6 months). Patients underwent a mean of 3.0 treatments (median 2.5), with several patients expected to require further procedures. Questionnaires were received from 17 of 26 patients (65%) (Tables 2–3). The most common pretreatment symptoms reported were swelling/large size/or growth in 16 patients (94%), 10 (59%) with bleeding, 7 (41%) with vesicle formation (although all showed vesicle formation on physical exam), and 2 (12%) with pain. All 17 patients reported improvement in symptoms after laser treatment. Five patients (29%) tolerated oral intake immediately after treatments, 10 (59%) the following day, and 1 (6%) was gastric tube dependent at the time of his surgeries. Four patients (24%) returned to normal activity immediately after treatment, six (35%) by the following day, six (35%) within a few days, and one (6%) required one week. No patients experienced postoperative airway obstruction necessitating reintubation. No complications were seen including readmission secondary to respiratory distress, inability to tolerate oral intake or infection. Postoperatively, patients typically developed a white exudate over the treated area which resolved after one to two weeks (Figs. 2–3). 4. Discussion LM are uncommon congenital lesions causing symptoms related to their anatomic location. Patients with intraoral LM often experience difficulty with breathing, speech, deglutition, as well as gross cosmetic deformity. Also, these lesions are often complicated by recurrent infections, swelling, pain, halitosis, and bleeding. Complete surgical resection, a mainstay of treatment in many LM, is rarely possible with intraoral lesions secondary to their association with vital adjacent structures and potential for high morbidity. Since cure remains difficult, treatment aims at controlling symptoms while preserving adjacent structures. Multiple conservative treatments have evolved in the treatment of LM with variable success. Systemic steroids have been
R.S. Glade, L.M. Buckmiller / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1358–1361
1360
Table 2 Response to questionnaire, symptoms. Patient
Pre-laser procedures
Pretreatment symptoms/frequency
Post-treatment symptoms/frequency
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17
Multiple excisions None None None Multiple excisions None None Excision, trach Sclerotherapy, multiple excisions None Sclerotherapy Multiple excisions None Excision Excision Excision, chemoembolization Sclerotherapy, excision
Bleeding, size/>1/week Size, bleeding, vesicles/constant Swelling, bleeding/constant Swelling, size, vesicles/once/month Size, vesicles/constant Swelling, bleeding, pain/>1/week Swelling, vesicles/>1/month Swelling, bleeding/constant Swelling, bleeding/>1/month Rapid growth, bleeding/constant Bleeding/constant Swelling, bleeding/>1/week Size, pain/>1/week Vesicles, fullness/constant Swelling, vesicles/constant Size/>1/month Swelling, bleeding, vesicles/constant
Bleeding, size/several times/year Size, drooling/once/month Bleeding/several times/year None/several times/year Vesicles, bleeding/once/month Pain/>1/month Swelling, vesicles/1/month None/several/year Swelling, bleeding/1/month Size/>1/month Size/>1/month Swelling, bleeding/1/month Size, pain/1/month Vesicles, fullness/1/month Swelling, vesicles/1/month Vesicles/several/year Vesicles, bleeding/>1 month
Table 3 Response to questionnaire, postoperative recovery.
employed, particularly with orbital lymphatic malformations, with inconsistent results [5,6]. Case reports exist of treatment with intralesional steroid injection for tongue lesions [7]. Irradiation has been used for extensive disease [8] especially within the thorax, but is not indicated for cervicofacial lesions secondary to its high morbidity. Sclerotherapy has become an excellent treatment for
macrocystic LM, but results have been unimpressive in the treatment of mixed and microcystic disease [2,9]. Newer techniques, aimed at further decreasing morbidity have been employed including radiofrequency ablation, Neodymium-YAG (Nd:YAG) laser, and CO2 laser resurfacing. Grimmer et al. reported excellent control of 11 tongue lesions with the use of radiofrequency ablation, with early postoperative oral intake and minimal postoperative pain [4]. Surgical control with this modality has been reported in subsequent studies [10,11]. April et al. showed successful treatment with the Nd:YAG laser in five patients [12]. Shapshay et al. describes safe and successful use of the Nd:YAG laser for treatment of multiple LM including one patient with a base of tongue lesion [13]. Suen and Waner reports use of Nd:YAG laser on two patients with tongue based LM with each showing post-treatment reduction of disease [14]. Successful symptomatic control of intraoral LM with CO2 laser has been shown in the past on a limited number of patients [12,3]. To the author’s knowledge, ours is the largest reported on cohort of patients with intraoral LM. We have used the CO2 laser for more than 10 years with excellent results (see Fig. 2). Received questionnaires indicate universal perceived effectiveness in treatment with CO2 laser, although only 65% of patients in this cohort could be located. Several patients, specifically those treated early in the study, had relocated or changed telephone numbers and were no longer receiving treatment at our center. This certainly could indicate a bias in our results.
Fig. 2. Two years after treatment LM.
Fig. 3. Ten days after treatment LM.
Patient
Return to oral intake
Return to normal activity
Overall improvement
1‘ 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17
Immediate Immediate Next day Next day N/A – G-tube Next day Immediate Immediate Next day Next day Next day Next day Next day Next day Immediate Few days Next day
Immediate Immediate Immediate Next day Few days Few days Few days One week Next day Next day Few days Next day Next day Few days Immediate Few days Next day
Slight Significant Slight Significant Significant Significant Significant Significant Significant Significant Significant Slight Slight Significant Significant Significant Significant
R.S. Glade, L.M. Buckmiller / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1358–1361
Treatment goals include reducing symptoms, improving function, and development of a safe airway. All patients in this study reported improvement of symptoms after treatment. Return to tolerating a normal diet and regular activity was typically immediate to within a few days. Time to return of vesicles is variable, and in and of itself does not necessitate management. Treatment is reserved when vesicles become symptomatic causing pain, bleeding, halitosis, or gross deformity. Most patients require multiple treatments secondary to symptom recurrence. Treatment appears to be safe as no postoperative complications were seen. Determination of the most effective treatment of intraoral LM remains unclear and will require future prospective studies. 5. Conclusion CO2 laser resurfacing appears to be both safe and efficacious in treatment of symptoms related to intraoral lymphatic malformations. Intermittent treatments for recurrent symptoms are expected. References [1] C.M. Bailey, Cystic hygroma, Lancet 335 (1990) 511–512 (editorial). [2] D.A. Peters, D.J. Courtemanche, M.K. Heran, J.P. Ludemann, J.S. Prendiville, Treatment of cystic lymphatic vascular malformations with OK-432 sclerotherapy, Plast. Reconstr. Surg. 118 (6) (2006) 1441–1446.
1361
[3] A. Balakrishnan, C.M. Bailey, Lymphangioma of the tongue. a review of pathogenesis, treatment and the use of surface laser photocoagulation, J. Laryngol. Otol. 105 (1991) 924–930. [4] J.F. Grimmer, J.B. Mulliken, P.E. Burrows, R. Rahbar, Radiofrequency ablation of microcystic lymphatic malformation in the oral cavity, Arch. Otolaryngol. Head Neck Surg. 132 (2006) 1251–1256. [5] B.S. Sires, C.R. Goins, R.L. Anderson, J.B. Holds, Systemic corticosteroid use in orbital lymphangioma, Ophthal. Plast. Reconstr. Surg. 17 (2) (2001) 85–90. [6] H.A. Tawfik, H. Budin, J.J. Dutton, Lack of response to systemic corticosteroids in patients with lymphangioma, Ophthal. Plast. Reconstr. Surg. 21 (4) (2005) 302– 305. [7] S. Khurana, A. Vij, A. Singal, D. Pandhi, Lymphangioma of the tongue: response to intralesional steroids, J. Dermatolog. Treat. 17 (2) (2006) 124–126. [8] A. Kandil, A.Y. Rostrom, W.A. Mourad, Y. Khafaga, A.R. Gershuny, G. el-Hosseiny, Successful control of extensive thoracic lymphangiomatosis by irradiation, Clin. Oncol. 9 (1997) 407–411. [9] A.I. Alomari, V.E. Karian, D.J. Lord, H.M. Padua, P.E. Burrows, Percutaneous sclerotherapy for lymphatic malformations: a retrospective analysis of patientevaluated improvement, J. Vasc. Interv. Radiol. 17 (2006) 1639–1648. [10] S. Roy, S. Reyes, L.P. Smith, Bipolar radiofrequency plasma ablation (coblation) of lymphatic malformations of the tongue, Int. J. Pediatr. Otorhinolaryngol. 73 (2) (2009 February) 289–293. [11] N.G. Ryu, S.K. Park, H.S. Jeong, Low power radiofrequency ablation for symptomatic microcystic lymphatic malformations of the tongue, Int. J. Pediatr. Otorhinolaryngol. 72 (11) (2008 November) 1731–1734. [12] M.M. April, E.E. Rebeiz, E.M. Friedman, G.M. Healy, S.M. Shapshay, Laser therapy for lymphatic malformations of the upper aerodigestive tract, an evolving experience, Arch. Otolaryngol. Head Neck Surg. 188 (2) (1992) 205–208. [13] S.M. Shapshay, L.M. David, S. Zeitels, Neodymium-YAG laser photocoagulation of hemangiomas of the head and neck, Laryngoscope 97 (3 Part 1) (1987) 3232– 3330. [14] J.Y. Suen, M. Waner, Treatment of oral cavity vascular malformations using the neodymium:YAG laser, Arch. Otolaryngol. Head Neck Surg. 115 (11) (1989 November) 1329–1333.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
CO2 laser resurfacing of intraoral lymphatic malformations: A 10-year experience Robert S. Glade a,*, Lisa M. Buckmiller b a b
The Children’s Hospital at OU Medical Center, Department of Otolaryngology, Head and Neck Surgery, Oklahoma City, Oklahoma, USA Vascular Anomalies Center, University of Arkansas for Medical Sciences, Arkansas Children’s Hospital, Little Rock, Arkansas, USA
A R T I C L E I N F O
A B S T R A C T
Article history: Received 12 January 2009 Received in revised form 16 June 2009 Accepted 18 June 2009 Available online 22 July 2009
Objective: To evaluate the safety and efficacy of CO2 laser resurfacing in the symptomatic treatment of intraoral lymphatic malformations (LM). Design: Retrospective review. Methods: Charts were reviewed on 26 patients (16 male, 10 female) from January 1997 to July 2007 who underwent CO2 laser resurfacing for symptomatic treatment of intraoral LM. A questionnaire was given in order to elucidate effectiveness in controlling symptoms and speed of postoperative recovery. Results: Mean age at time of first treatment was 9.2 years (median 6.8). Mean number of treatments was 3.0 (median 2.5). Average time between treatments was 9.7 months (median 5.6). Questionnaires were returned for 17 patients (65%). Common preoperative symptoms included swelling, bleeding, vesicle formation, and pain. All 17 patients reported symptomatic improvement after laser treatment. Five patients (29%) tolerated oral intake immediately, 10 (59%) the following day, and 1 (6%) was gastric tube dependent. Four patients (24%) returned to normal activity immediately after treatments, six (35%) by the following day, six (35%) within a few days, and one (6%) within a week. No postoperative complications were seen. Conclusion: CO2 laser resurfacing appears to be both safe and efficacious in treatment of symptoms related to intraoral LM. Intermittent treatments for recurrent symptoms is expected. ß 2009 Elsevier Ireland Ltd. All rights reserved.
Keywords: CO2 laser Lymphatic malformation Tongue lymphatic malformation
1. Introduction Lymphatic malformations (LM) are slow flow vascular anomalies most commonly presenting in the head and neck of children. Fifty to sixty percent are detectable at birth with 80% diagnosed before two years of age [1]. While LM are benign, they continue to expand throughout life and can compress adjacent structures. Many undergo rapid enlargement secondary to infection, hemorrhage, or trauma. Rapid growth may lead to airway compromise, dysphagia, speech difficulty, or gross cosmetic deformity. LM are classified as macrocystic, microcystic or mixed based on cyst size. While macrocystic lesions are often amenable to surgery and sclerotherapy, the infiltrative nature of microcystic lesions makes total surgical excision difficult without causing unacceptable loss of function. Subtotal resection is complicated by a high recurrence rate and sclerotherapy has also proven to be ineffective in the treatment of microcystic disease [2]. Intraoral lymphatic malformations are rare and nearly always of the mixed or microcystic classification. Tongue lesions may cause bleeding, halitosis, dysphagia, dysarthria, and airway
obstruction. Macroscopically, the hallmark of intraoral LM is mucosal vesicle formation which grossly resembles fish eggs (Fig. 1). These vesicles represent small lymph filled cysts, but may develop a purple color secondary to intracystic hemorrhage. Microscopically, endothelial-lined spaces within the epithelial, subepithelial, and muscular layers are seen. This often leads to associated muscular atrophy [3]. Acute enlargement of the tongue frequently occurs consequent to upper respiratory or even remote infections causing extraoral tongue protrusion and increased exposure to dental trauma. Repeated episodes of infection or trauma lead to inflammation and further enlargement of the tongue. Surgery has been the mainstay in the treatment LM of the tongue, but is limited secondary to the need to preserve tongue function. Without complete surgical resection, cure cannot be achieved and newer treatments target symptom management. We have successfully used the CO2 laser for tongue resurfacing at Arkansas Children’s Hospital for over 10 years in the treatment of intraoral lymphatic malformations. 2. Methods
* Corresponding author at: 800 Marshall Street, Little Rock, AR 72202-3591, USA. Tel.: +1 501 364 4790. E-mail address: [email protected] (R.S. Glade). 0165-5876/$ – see front matter ß 2009 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2009.06.013
Institution Review Board approval was first obtained for the study. Charts were reviewed on all patients with a diagnosis of LM involving the oral cavity and/or oropharynx treated at Arkansas
R.S. Glade, L.M. Buckmiller / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1358–1361
1359
Table 1 Patient data.
Fig. 1. Preoperative LM.
Children’s Hospital, Center for Vascular Anomalies from January 1997 through July 2007. Only those who underwent CO2 laser resurfacing for treatment of the mucosal component of their lesions were included. A database was created which included patient’s gender, date of birth, number of procedures, and date of procedures. Treatment in all cases used a CO2 laser (Surgipulse 60XJ with silk touch scanner, Lumenis Inc., Santa Clara, CA) set at 30 W, continuous scanning mode, with a 6 mm spot. All treatments were performed by either the senior author or a second staff physician. All cases were performed under general anesthesia with either oral or nasal endotracheal (ET) intubation. The laser was used to eliminate superficial vesicles followed by manual removal of debris and char with a moist gauze. Treatment was carried through the affected epithelial and subepithelial layer to the level of the underlying musculature of the tongue. The wound was not closed and allowed to heal by secondary intention. If oral mucosal surfaces other than the tongue were affected, that mucosa and submucosa was also removed. Strict laser precautions were employed during each procedure including use of appropriate ET tube, eye protection and appropriate placement of wet towels on the patient. All patients received a dose of intraoperative dexamethasone (0.5 mg/kg) and were extubated at the conclusion of the procedure. Patients undergoing their first treatment and those with extensive lesions were observed overnight for airway obstruction and received intravenous dexamethasone (0.5 mg/kg twice daily) and cefazolin (25 mg/kg) with continuous pulse oximeter monitoring. Discharge the following day occurred if patients demonstrated a safe airway and ability to tolerate oral feeds. All patients were discharged home on a high dose prednisone taper (2–3 mg/kg/day divided BID for one week with gradual taper over three weeks) and oral amoxicillin (25 mg/kg twice daily). Repeat treatment was performed when the patient began to experience return of symptoms. An attempt was made to locate all patients in order to obtain outcome data. Results were obtained from a questionnaire adapted from that created by Grimmer et al. [4] given by phone interview. Patients and parents were questioned regarding symptoms prior to treatment, symptoms after treatment, effectiveness in controlling symptoms, previous treatments other than CO2 laser resurfacing, and postoperative recovery including tolerating oral intake and return to normal activity. 3. Results A total of 26 patients (16 males, 10 females) met inclusion criteria undergoing total of 79 CO2 laser resurfacing procedures
Patient
M/F
Age first CO2 laser (years)
Number CO2 laser treatment
Mean time between treatments (months)
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 20 21 22 23 24 25 26
M M M F M M F M F F F M M M M M F F F M M F M M M F
10.7 2.0 6.0 9.1 2.9 6.1 14.7 0.8 2.8 11.3 5.0 7.0 14.7 15.5 19.8 6.0 4.9 6.4 30.3 1.0 11.2 6.8 4.2 2.4 14.6 9.5
2 1 3 1 2 9 5 6 3 5 4 2 2 1 4 1 3 1 5 1 1 4 2 1 3 7
22.1 N/A 13.4 N/A 23.4 6.6 27.0 6.3 5.4 7.8 5.3 2.4 2.4 N/A 8.5 N/A 35.9 N/A 5.3 N/A N/A 6.3 7.1 N/A 4.4 4.5
(Table 1). The mean age at first resurfacing was 9.2 years (median 6.8 years) with a mean time between treatments of 9.7 months (median 5.6 months). Patients underwent a mean of 3.0 treatments (median 2.5), with several patients expected to require further procedures. Questionnaires were received from 17 of 26 patients (65%) (Tables 2–3). The most common pretreatment symptoms reported were swelling/large size/or growth in 16 patients (94%), 10 (59%) with bleeding, 7 (41%) with vesicle formation (although all showed vesicle formation on physical exam), and 2 (12%) with pain. All 17 patients reported improvement in symptoms after laser treatment. Five patients (29%) tolerated oral intake immediately after treatments, 10 (59%) the following day, and 1 (6%) was gastric tube dependent at the time of his surgeries. Four patients (24%) returned to normal activity immediately after treatment, six (35%) by the following day, six (35%) within a few days, and one (6%) required one week. No patients experienced postoperative airway obstruction necessitating reintubation. No complications were seen including readmission secondary to respiratory distress, inability to tolerate oral intake or infection. Postoperatively, patients typically developed a white exudate over the treated area which resolved after one to two weeks (Figs. 2–3). 4. Discussion LM are uncommon congenital lesions causing symptoms related to their anatomic location. Patients with intraoral LM often experience difficulty with breathing, speech, deglutition, as well as gross cosmetic deformity. Also, these lesions are often complicated by recurrent infections, swelling, pain, halitosis, and bleeding. Complete surgical resection, a mainstay of treatment in many LM, is rarely possible with intraoral lesions secondary to their association with vital adjacent structures and potential for high morbidity. Since cure remains difficult, treatment aims at controlling symptoms while preserving adjacent structures. Multiple conservative treatments have evolved in the treatment of LM with variable success. Systemic steroids have been
R.S. Glade, L.M. Buckmiller / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1358–1361
1360
Table 2 Response to questionnaire, symptoms. Patient
Pre-laser procedures
Pretreatment symptoms/frequency
Post-treatment symptoms/frequency
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17
Multiple excisions None None None Multiple excisions None None Excision, trach Sclerotherapy, multiple excisions None Sclerotherapy Multiple excisions None Excision Excision Excision, chemoembolization Sclerotherapy, excision
Bleeding, size/>1/week Size, bleeding, vesicles/constant Swelling, bleeding/constant Swelling, size, vesicles/once/month Size, vesicles/constant Swelling, bleeding, pain/>1/week Swelling, vesicles/>1/month Swelling, bleeding/constant Swelling, bleeding/>1/month Rapid growth, bleeding/constant Bleeding/constant Swelling, bleeding/>1/week Size, pain/>1/week Vesicles, fullness/constant Swelling, vesicles/constant Size/>1/month Swelling, bleeding, vesicles/constant
Bleeding, size/several times/year Size, drooling/once/month Bleeding/several times/year None/several times/year Vesicles, bleeding/once/month Pain/>1/month Swelling, vesicles/1/month None/several/year Swelling, bleeding/1/month Size/>1/month Size/>1/month Swelling, bleeding/1/month Size, pain/1/month Vesicles, fullness/1/month Swelling, vesicles/1/month Vesicles/several/year Vesicles, bleeding/>1 month
Table 3 Response to questionnaire, postoperative recovery.
employed, particularly with orbital lymphatic malformations, with inconsistent results [5,6]. Case reports exist of treatment with intralesional steroid injection for tongue lesions [7]. Irradiation has been used for extensive disease [8] especially within the thorax, but is not indicated for cervicofacial lesions secondary to its high morbidity. Sclerotherapy has become an excellent treatment for
macrocystic LM, but results have been unimpressive in the treatment of mixed and microcystic disease [2,9]. Newer techniques, aimed at further decreasing morbidity have been employed including radiofrequency ablation, Neodymium-YAG (Nd:YAG) laser, and CO2 laser resurfacing. Grimmer et al. reported excellent control of 11 tongue lesions with the use of radiofrequency ablation, with early postoperative oral intake and minimal postoperative pain [4]. Surgical control with this modality has been reported in subsequent studies [10,11]. April et al. showed successful treatment with the Nd:YAG laser in five patients [12]. Shapshay et al. describes safe and successful use of the Nd:YAG laser for treatment of multiple LM including one patient with a base of tongue lesion [13]. Suen and Waner reports use of Nd:YAG laser on two patients with tongue based LM with each showing post-treatment reduction of disease [14]. Successful symptomatic control of intraoral LM with CO2 laser has been shown in the past on a limited number of patients [12,3]. To the author’s knowledge, ours is the largest reported on cohort of patients with intraoral LM. We have used the CO2 laser for more than 10 years with excellent results (see Fig. 2). Received questionnaires indicate universal perceived effectiveness in treatment with CO2 laser, although only 65% of patients in this cohort could be located. Several patients, specifically those treated early in the study, had relocated or changed telephone numbers and were no longer receiving treatment at our center. This certainly could indicate a bias in our results.
Fig. 2. Two years after treatment LM.
Fig. 3. Ten days after treatment LM.
Patient
Return to oral intake
Return to normal activity
Overall improvement
1‘ 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17
Immediate Immediate Next day Next day N/A – G-tube Next day Immediate Immediate Next day Next day Next day Next day Next day Next day Immediate Few days Next day
Immediate Immediate Immediate Next day Few days Few days Few days One week Next day Next day Few days Next day Next day Few days Immediate Few days Next day
Slight Significant Slight Significant Significant Significant Significant Significant Significant Significant Significant Slight Slight Significant Significant Significant Significant
R.S. Glade, L.M. Buckmiller / International Journal of Pediatric Otorhinolaryngology 73 (2009) 1358–1361
Treatment goals include reducing symptoms, improving function, and development of a safe airway. All patients in this study reported improvement of symptoms after treatment. Return to tolerating a normal diet and regular activity was typically immediate to within a few days. Time to return of vesicles is variable, and in and of itself does not necessitate management. Treatment is reserved when vesicles become symptomatic causing pain, bleeding, halitosis, or gross deformity. Most patients require multiple treatments secondary to symptom recurrence. Treatment appears to be safe as no postoperative complications were seen. Determination of the most effective treatment of intraoral LM remains unclear and will require future prospective studies. 5. Conclusion CO2 laser resurfacing appears to be both safe and efficacious in treatment of symptoms related to intraoral lymphatic malformations. Intermittent treatments for recurrent symptoms are expected. References [1] C.M. Bailey, Cystic hygroma, Lancet 335 (1990) 511–512 (editorial). [2] D.A. Peters, D.J. Courtemanche, M.K. Heran, J.P. Ludemann, J.S. Prendiville, Treatment of cystic lymphatic vascular malformations with OK-432 sclerotherapy, Plast. Reconstr. Surg. 118 (6) (2006) 1441–1446.
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