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Utility of radiofrequency ablation for haemorrhagic lingual lymphangioma
International Journal of Pediatric Otorhinolaryngology (2008) 72, 953—958
www.elsevier.com/locate/ijporl
CASE REPORT
Utility of radiofrequency ablation for haemorrhagic lingual lymphangioma Nicolas Leboulanger a,b,c,*, Gilles Roger a,b,c, Alexandre Caze a,b,c, ´a-Noe ¨l Garabedian a,b,c Odile Enjolras d, Franc¸oise Denoyelle a,b,c, Ere a
AP-HP, Armand-Trousseau Children Hospital, Pediatric ENT Department, 75012 Paris, France UPMC University Paris 06, F-75005 Paris, France c INSERM U-587, Paris, France d Department of Maxillofacial and Reconstructive Surgery, Armand-Trousseau Children’s Hospital, 75012 Paris, France b
Received 29 January 2008; received in revised form 29 February 2008; accepted 4 March 2008 Available online 2 May 2008
KEYWORDS Radiofrequency; Lymphangioma; Haemorrhage; Tongue; Mouth; Child
Summary Lymphangiomas of the mouth and tongue pose considerable therapeutic problems. Their complete exeresis is not feasible, and they can be a major functional impediment and cause face and jaw deformities. A risk of secondary growth is classically described after surgical reduction. Invasion of lingual mucosa, often papillomatous, results in accidental biting, recurrent bleeding and pain. Here, we report on 7 cases of children with haemorrhagic mucosal lingual lymphangiomas, one with noteworthy drop in the haemoglobin, treated by surface radiofrequency reduction. With a short follow-up (3—13 months), a functional improvement was observed in every case, as a complete disappearance of bleeding in 5/7 cases, and a moderate local relapse in 2/7 cases. This technique did not induce progressive regrowth. It provides a new therapeutic tool for the treatment of lingual microcystic lymphangioma. # 2008 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Lymphangiomas are localised malformations of the lymphatic system frequently located in the head and neck. Many of these lesions are congenital. They can * Corresponding author at: Service d’ORL et de chirurgie cervico-faciale, Ho ˆpital Armand-Trousseau, 26 avenue du Docteur Arnold-Netter, 75012 Paris, France. Tel.: +33 144736114; fax: +33 144736108. E-mail address: [email protected] (N. Leboulanger).
come to light after surgery or trauma. The great majority are diagnosed before age two years [1]. Two theories have been advanced to describe the formation of the lymphatic system [2]. The first is the centrifugal theory, whereby two endothelial buds grow from the jugular sacs and develop into lymph vessels. The second is the centripetal theory whereby an anastomotically developing lymphatic system eventually joins up to the central venous system. Whichever the case, a lymphangioma results from the impossibility for part of the lymphatic network
0165-5876/$ — see front matter # 2008 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2008.03.013
954
N. Leboulanger et al.
to connect into the central network, with a resulting build-up of lymph and excessive expansion of the vessels. Lymphangiomas are classified according to their clinical presentation into macrocystic (cavities larger than about 2 cm3), microcystic (cavities smaller than about 2 cm3), and mixed, combining these two types. Lingual localisation presents specific therapeutic problems because of the almost exclusively microcystic character of the lesions and the marked functional problems they cause (obstruction, bleeding, pain, feeding problems, oedema, lingual extorsion, dental problems, breathing problems, jaw deformities, etc.) [3]. Various treatments have been attempted and proposed. Complete surgical exeresis is not possible because of the unacceptable degree of mutilation it would entail. Partial glossectomy, performed regularly, is a serious operation with a high rate of relapse or secondary growth and often results in a morphological modification of the tongue. Post-operative healing is painful and relatively long. The injection of sclerosing agents can be useful for macrocystic lesions but is not appropriate for microcystic lesions [4—6]. Low radiofrequency treatment, developed more recently, is a useful approach to the treatment of lingual lymphangiomas. Its principle is the delivery, by the local route via an electrode and a conducting medium, of low radiofrequency energy that induces local tissue destruction and creates a secondary fibrosis. This technique, called ‘coblation’ (for ‘controlled ablation’) is already used for tonsillectomy. It has been described by several groups [3,7]. Here, we report on 7 cases of microcystic lingual lymphangioma with haemorrhage subjected to surface radiofrequency treatment at the Armand-Trousseau Hospital between 2005 and 2007.
We used the ENTEC1 Coblator# Plasma Surgery System probe with an Evac 70# probe (E 5070-01) (AthroCare Corp.). The power setting was 7 for vaporisation and 4 for coagulation. The conduction medium was a solution of isotonic saline serum with controlled continuous irrigation. The areas treated were exclusively those infiltrated by the lymphangioma, including the tip of the tongue, its dorsal, lateral and ventral surfaces and the floor of the mouth. The Wharton canal openings were preserved. The coblation remained superficial so as to create a non-budded tongue surface without attempting deep vaporisation. Even when the underlying muscle was exposed we left the treated area to heal spontaneously without surgical preparation. Hemostasis was also carried out with the coblator. All the patients were extubated on the operating table and spent their first post-operative night in their rooms. We had no problems with airways or heavy post-operative bleeding (minimal blood seepage with spontaneous healing needing no more than simple monitoring without further surgery). The post-operative treatment comprised a short course of antibiotics (amoxicillin + clavulanic acid), corticotherapy for 48 h (prednisolone 1 mg 2/day), and class III analgesics (morphine in the recovery room, then nalbuphine for 2—3 days as needed), promptly replaced by class II analgesics. Pain was evaluated on a visual analogue scale (VAS) adapted to the age of the child. Feeding was resumed on the evening after surgery with a cold, soft regimen of the type used after tonsillectomy. Mouthwashes with dilute chlorhexidine (e.g. 0.05 ml/10 ml chlorhexidine digluconate, Hextril#) were given starting on Day 1, three per day. All the children were discharged on Day 2 postoperatively.
2. Materials and methods
3. Clinical case studies
All the patients were treated in the ENT department of the Armand-Trousseau Hospital. The 7 children were treated by the same protocol under general anaesthesia with orotracheal intubation. We planned for potential intubation difficulties owing to the morphological modifications present in these children, and so all the necessary equipment was at hand for fibroscopic intubation. This did not prove necessary, and the children underwent intubation in the usual way under mask anaesthesia. None of the children needed peri-operative tracheotomy.
3.1. Case no. 1 A one-year-old girl with an extended congenital lymphangioma of the mouth affecting the base of the tongue, the valleculi and the epiglottis. However, the lesions were sufficiently stable not to notably impede respiratory function. At the end of 2006, the tongue lesions were such that bites and resulting bleeding occurred daily (Figs. 1 and 2). This repeated microhaemorrhage was accompanied by a gradual drop in the haemoglobin (loss of 3 points in 8 weeks).
Utility of radiofrequency ablation for haemorrhagic lingual lymphangioma
Fig. 1 Pre-operative appearance: microcystic lingual lymphangiomatous infiltration with ulceration, iterative bleeding and biting. The patient cannot close her mouth.
At age 16 months the lingual surface was coblated, after which biting and bleeding ceased (Fig. 3). The patient subsequently suffered a regrowth with an increase in volume, but without haemorrhagic vesicles or bleeding.
955
Fig. 3 Post-operative appearance after two weeks. The volume of the tongue has clearly diminished. The mouth can be closed. The number of surface vesicles is much lower and the functional impairment less marked. Cessation of haemorrhage.
3.2. Case no. 2 A boy in whom microcystic lesions were discovered at birth at the base of the tongue, the floor of the mouth, the free edge of the tongue and the valleculi, with a macrocystic lesion of the left sub-maxillary region. The lesions were stable up to age 4 months, when a first growth occurred that was treated medically (antibiotics, corticoids, proton pump inhibitors). Several further growths occurred with a progressively weaker lingual regression. At 11 months, the lingual lymphangiomatous infiltration was such that the tongue could no longer fit in the mouth, bled daily on contact and impeded swallowing. A single coblation session was applied at age 16 months. The result was favourable at two years postoperatively. No more lingual extrusion, normal feeding and markedly reduced frequency of microtrauma. Since then the sub-maxillary lesion has increased in volume and now presents an indication for sclerotherapy.
3.3. Case no. 3
Fig. 2 Pre-operative: same patient, microcystic lingual lymphangiomatous infiltration.
A girl aged 10 years presenting an exclusively lingually localised congenital microcystic lymphangioma. A first surgical treatment, a partial glossectomy, had been performed at age 6 months.
956 Following several lymphangiomatous growths a second partial glossectomy was performed at age two years. The patient was then lost to follow-up for several years. At age 10 years, she was brought in by her parents with a voluminous, massively infiltrated tongue with daily biting and bleeding, a lower jaw deformity and a speaking impediment. A coblation session was conducted some time afterwards, with a favourable outcome on the tongue volume and no more bleeding. Improvement of the functional impairment and phonation was less marked, however. Seven months after surgery, a few lingual vesicles reappeared, but in a much smaller area than that initially affected. Another coblation session will very probably prove necessary.
3.4. Case no. 4 A girl aged 8 years presenting a lingual and pelvilingual microcystic lymphangioma and a sub-maxillary macrocystic lymphangioma. The lesion was present at birth. She had been given a first sclerosing injection in the cervical region at age 15 months, with a favourable outcome. A partial glossectomy was performed at age 3 years, of limited efficacy, followed by a new sclerosing injection at age 7 years. The following year she presented a marked lingual infiltration with repeated bleeding, frequent trauma and biting, and eating difficulties. A lingual coblation session was conducted a short time afterwards. There were no post-operative complications, and functional improvement was immediate. There was no more bleeding, and eating was easier. Biting did not disappear but became much less frequent.
N. Leboulanger et al.
Fig. 4
Per-operative view: dorsal bud.
were several surgical treatments during childhood: exeresis by cervicotomy and reductive glossoplasty. The patient presented a marked mandibular deformity for which orthodondic treatment and orthognatic surgery were planned. He presented numerous lymphomangiomatous lesions of the dorsal and ventral surfaces of the tongue, not very painful but bleeding frequently. There was no noteworthy fall in haemoglobin count. A coblation session was applied to remove all projecting lymphangiomatous surface lesions. Five months post-operatively the overall volume of the tongue was unchanged, but the projecting lesions had disappeared and the symptoms were relieved.
3.7. Case no. 7 A girl aged 7 years presenting an essentially lingual lymphangioma, located mainly on the dorsal surface of the tongue and on its right free edge. There was also a small, barely visible right laterocervical
3.5. Case no. 5 A girl aged 6 years presenting a stable lingual lymphoma with unimpaired dental articulation, and of moderate size. She had never had surgical treatment. In a few months a voluminous dorsal bud appeared (Fig. 4), causing the patient considerable discomfort. The lesion was painful and bled on contact. Lingual coblation removed the bud (Fig. 5) and cause the symptoms to disappear completely. Follow-up at 5 months showed no relapse. The other lingual lesions, which were not troublesome, remained stable.
3.6. Case no. 6 A 14-year-old male adolescent presenting a cervicofacial lymphangioma discovered at birth. There
Fig. 5 Per-operative view, after coblation: dorsal bud removed.
Utility of radiofrequency ablation for haemorrhagic lingual lymphangioma
Fig. 6
Pre-operative front view.
localisation that had never grown or needed treatment. The lingual localisation underwent monthly growths with increase in volume and in the number of surface vesicles that bled and were painful. No tongue surgery had ever been performed. Despite the volume of the tongue, the girl was able to close her mouth and feed normally (Fig. 6). We applied a single lingual coblation session that destroyed the exophytic vesicular lesions on the right lateral and dorsal surfaces. There were no post-operative complications. Four months after surgery the overall volume of the tongue had decreased (Fig. 7). Four lymphangiomatous vesicles reappeared on the right lateral edge of the tongue, but with only one incident of traumatic bleeding.
4. Discussion The use of radiofrequency in lymphangioma is some 10 years old. Two main techniques have been proposed.
Fig. 7
Post-operative front view.
957
Initially, high frequency radiofrequency has been used in the aim to reduce the size of the tongue; the electrode is introduced directly into the lingual tissue and several passes are carried out in depth. This method induces a deep secondary fibrosis that preserves the surface mucosa and is comparable to that used for turbinate reduction or radiofrequency velar coblation for ronchopathy. It has the disadvantage of low accuracy and it cannot be used for mucosal treatment. However, it allows an overall reduction in the size of the tongue. The second technique, described recently and reported here [7], makes use of a coblation probe comparable to that used for tonsillectomy or tonsil reduction (low frequency radiofrequency). The energy is applied to the surface via a conduction medium (here saline serum), and over a smaller volume, which allows greater local efficacy and better accuracy. It permits a specific reduction of mucosal lymphangiomatous lesions without affecting underlying muscle, and maintains satisfactory haemostasis. The quantity of energy delivered can be more closely controlled than in the first method. Both methods are complementary and can be combined. The utility of surface radiofrequency coblation of lymphangiomas of the mouth, and in particular of the tongue, lies in its ease of implementation and straightforward post-operative phase. The patients described here all had specific massive lingual infiltration with functional impact and bleeding. This low-volume but daily repeated bleeding was a significant functional impairment also affecting those close to the patient, and having a general impact on well-being. The series described by Grimmer et al. [7] concerned 11 cases of lymphangioma of the mouth, with wider-ranging localisation. Their functional results were favourable, with a very straightforward postoperative phase. Likewise, pain and bleeding were low here, and eating was resumed by Day 1. The use of powerful antalgics (III) was limited to the immediate postoperative period. The longest time elapsed since surgery is currently two years, with in two cases a return of local symptoms at a milder level. However, this technique has no effect on overall lingual hypertrophy, or on iterative increases in volume, or on any secondary mandibular deformities. Patients and their families must be well informed that the treatment is in no way curative, and that regrowth, aggravation of the lesion and the recurrence of haemorrhagic lingual vesicles are possible.
958
N. Leboulanger et al.
Further work will be necessary to fully validate these findings and the treatment indication in the long term.
5. Conclusion Surface radiofrequency coblation is a useful technique for improving functional impairment (bites, hindrance due to large-volume vesicles and bleeding), and in particular reducing bleeding, due to certain lingual lymphangiomas. Mucosal lingual lesions respond well to the treatment, with an uncomplicated post-operative phase. Although the functional improvement is marked, its stability over time remains to be assessed by a long-term patient follow-up. It must be explained to the patients and their families that no long-term review of the treatment is yet available.
6. IRB disclosure The authors of this article certify to have no financial interest or involvement of any kind in the subject matter or materials discussed in this manuscript.
References [1] C. Gigue `re, N. Bauman, R. Smith, New treament options for lymphangioma in infants and children, Ann. Otol. Rhinol. Laryngol. 111 (December) (2002) 1066—1075. [2] Th. Kennedy, M. Whitaker, P. Pellitteri, W.E. Wood, Cystic hygroma/lymphangioma: a rational approach to management, Laryngoscope 111 (November (11 Pt 1)) (2001) 1929—1937. [3] B. Cable, E.A. Mair, Radiofrequency ablation of lymphagiomatous macroglossia, Laryngoscope 111 (October (10)) (2001) 1859—1861. [4] D. Herbreteau, M.C. Riche, O. Enjolras, M. Khayata, F. Lemarchand-Venencie, M. Borsik, Percutaneous embolization with Ethibloc of lymphatic cystic malformations with a review of the experience in 70 patients, Int. Angiol. 12 (March (1)) (1993) 34—39. [5] N.N. Mathur, I. Rana, R. Bothra, R. Dhawan, G. Kathuria, T. Pradhan, Bleomycin sclerotherapy in congenital lymphatic and vascular malformations of head and neck, Int. J. Pediatr. Otorhinolaryngol. 69 (January (1)) (2005) 75— 80. [6] T. Okazaki, S. Iwatani, T. Yanai, H. Kobayashi, Y. Kato, T. Marusasa, Treatment of lymphangioma in children: our experience of 128 cases, J. Pediatr. Surg. 42 (February (2)) (2007) 386—389. [7] J.F. Grimmer, J.B. Mulliken, P. Burrows, R. Rahbar, Radiofrequency ablation of microscopic lymphatic malformation in the oral cavity, Arch. Otolaryngol. Head Neck Surg. 132 (November (11)) (2006) 1251—1256.
Available online at www.sciencedirect.com
www.elsevier.com/locate/ijporl
CASE REPORT
Utility of radiofrequency ablation for haemorrhagic lingual lymphangioma Nicolas Leboulanger a,b,c,*, Gilles Roger a,b,c, Alexandre Caze a,b,c, ´a-Noe ¨l Garabedian a,b,c Odile Enjolras d, Franc¸oise Denoyelle a,b,c, Ere a
AP-HP, Armand-Trousseau Children Hospital, Pediatric ENT Department, 75012 Paris, France UPMC University Paris 06, F-75005 Paris, France c INSERM U-587, Paris, France d Department of Maxillofacial and Reconstructive Surgery, Armand-Trousseau Children’s Hospital, 75012 Paris, France b
Received 29 January 2008; received in revised form 29 February 2008; accepted 4 March 2008 Available online 2 May 2008
KEYWORDS Radiofrequency; Lymphangioma; Haemorrhage; Tongue; Mouth; Child
Summary Lymphangiomas of the mouth and tongue pose considerable therapeutic problems. Their complete exeresis is not feasible, and they can be a major functional impediment and cause face and jaw deformities. A risk of secondary growth is classically described after surgical reduction. Invasion of lingual mucosa, often papillomatous, results in accidental biting, recurrent bleeding and pain. Here, we report on 7 cases of children with haemorrhagic mucosal lingual lymphangiomas, one with noteworthy drop in the haemoglobin, treated by surface radiofrequency reduction. With a short follow-up (3—13 months), a functional improvement was observed in every case, as a complete disappearance of bleeding in 5/7 cases, and a moderate local relapse in 2/7 cases. This technique did not induce progressive regrowth. It provides a new therapeutic tool for the treatment of lingual microcystic lymphangioma. # 2008 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Lymphangiomas are localised malformations of the lymphatic system frequently located in the head and neck. Many of these lesions are congenital. They can * Corresponding author at: Service d’ORL et de chirurgie cervico-faciale, Ho ˆpital Armand-Trousseau, 26 avenue du Docteur Arnold-Netter, 75012 Paris, France. Tel.: +33 144736114; fax: +33 144736108. E-mail address: [email protected] (N. Leboulanger).
come to light after surgery or trauma. The great majority are diagnosed before age two years [1]. Two theories have been advanced to describe the formation of the lymphatic system [2]. The first is the centrifugal theory, whereby two endothelial buds grow from the jugular sacs and develop into lymph vessels. The second is the centripetal theory whereby an anastomotically developing lymphatic system eventually joins up to the central venous system. Whichever the case, a lymphangioma results from the impossibility for part of the lymphatic network
0165-5876/$ — see front matter # 2008 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2008.03.013
954
N. Leboulanger et al.
to connect into the central network, with a resulting build-up of lymph and excessive expansion of the vessels. Lymphangiomas are classified according to their clinical presentation into macrocystic (cavities larger than about 2 cm3), microcystic (cavities smaller than about 2 cm3), and mixed, combining these two types. Lingual localisation presents specific therapeutic problems because of the almost exclusively microcystic character of the lesions and the marked functional problems they cause (obstruction, bleeding, pain, feeding problems, oedema, lingual extorsion, dental problems, breathing problems, jaw deformities, etc.) [3]. Various treatments have been attempted and proposed. Complete surgical exeresis is not possible because of the unacceptable degree of mutilation it would entail. Partial glossectomy, performed regularly, is a serious operation with a high rate of relapse or secondary growth and often results in a morphological modification of the tongue. Post-operative healing is painful and relatively long. The injection of sclerosing agents can be useful for macrocystic lesions but is not appropriate for microcystic lesions [4—6]. Low radiofrequency treatment, developed more recently, is a useful approach to the treatment of lingual lymphangiomas. Its principle is the delivery, by the local route via an electrode and a conducting medium, of low radiofrequency energy that induces local tissue destruction and creates a secondary fibrosis. This technique, called ‘coblation’ (for ‘controlled ablation’) is already used for tonsillectomy. It has been described by several groups [3,7]. Here, we report on 7 cases of microcystic lingual lymphangioma with haemorrhage subjected to surface radiofrequency treatment at the Armand-Trousseau Hospital between 2005 and 2007.
We used the ENTEC1 Coblator# Plasma Surgery System probe with an Evac 70# probe (E 5070-01) (AthroCare Corp.). The power setting was 7 for vaporisation and 4 for coagulation. The conduction medium was a solution of isotonic saline serum with controlled continuous irrigation. The areas treated were exclusively those infiltrated by the lymphangioma, including the tip of the tongue, its dorsal, lateral and ventral surfaces and the floor of the mouth. The Wharton canal openings were preserved. The coblation remained superficial so as to create a non-budded tongue surface without attempting deep vaporisation. Even when the underlying muscle was exposed we left the treated area to heal spontaneously without surgical preparation. Hemostasis was also carried out with the coblator. All the patients were extubated on the operating table and spent their first post-operative night in their rooms. We had no problems with airways or heavy post-operative bleeding (minimal blood seepage with spontaneous healing needing no more than simple monitoring without further surgery). The post-operative treatment comprised a short course of antibiotics (amoxicillin + clavulanic acid), corticotherapy for 48 h (prednisolone 1 mg 2/day), and class III analgesics (morphine in the recovery room, then nalbuphine for 2—3 days as needed), promptly replaced by class II analgesics. Pain was evaluated on a visual analogue scale (VAS) adapted to the age of the child. Feeding was resumed on the evening after surgery with a cold, soft regimen of the type used after tonsillectomy. Mouthwashes with dilute chlorhexidine (e.g. 0.05 ml/10 ml chlorhexidine digluconate, Hextril#) were given starting on Day 1, three per day. All the children were discharged on Day 2 postoperatively.
2. Materials and methods
3. Clinical case studies
All the patients were treated in the ENT department of the Armand-Trousseau Hospital. The 7 children were treated by the same protocol under general anaesthesia with orotracheal intubation. We planned for potential intubation difficulties owing to the morphological modifications present in these children, and so all the necessary equipment was at hand for fibroscopic intubation. This did not prove necessary, and the children underwent intubation in the usual way under mask anaesthesia. None of the children needed peri-operative tracheotomy.
3.1. Case no. 1 A one-year-old girl with an extended congenital lymphangioma of the mouth affecting the base of the tongue, the valleculi and the epiglottis. However, the lesions were sufficiently stable not to notably impede respiratory function. At the end of 2006, the tongue lesions were such that bites and resulting bleeding occurred daily (Figs. 1 and 2). This repeated microhaemorrhage was accompanied by a gradual drop in the haemoglobin (loss of 3 points in 8 weeks).
Utility of radiofrequency ablation for haemorrhagic lingual lymphangioma
Fig. 1 Pre-operative appearance: microcystic lingual lymphangiomatous infiltration with ulceration, iterative bleeding and biting. The patient cannot close her mouth.
At age 16 months the lingual surface was coblated, after which biting and bleeding ceased (Fig. 3). The patient subsequently suffered a regrowth with an increase in volume, but without haemorrhagic vesicles or bleeding.
955
Fig. 3 Post-operative appearance after two weeks. The volume of the tongue has clearly diminished. The mouth can be closed. The number of surface vesicles is much lower and the functional impairment less marked. Cessation of haemorrhage.
3.2. Case no. 2 A boy in whom microcystic lesions were discovered at birth at the base of the tongue, the floor of the mouth, the free edge of the tongue and the valleculi, with a macrocystic lesion of the left sub-maxillary region. The lesions were stable up to age 4 months, when a first growth occurred that was treated medically (antibiotics, corticoids, proton pump inhibitors). Several further growths occurred with a progressively weaker lingual regression. At 11 months, the lingual lymphangiomatous infiltration was such that the tongue could no longer fit in the mouth, bled daily on contact and impeded swallowing. A single coblation session was applied at age 16 months. The result was favourable at two years postoperatively. No more lingual extrusion, normal feeding and markedly reduced frequency of microtrauma. Since then the sub-maxillary lesion has increased in volume and now presents an indication for sclerotherapy.
3.3. Case no. 3
Fig. 2 Pre-operative: same patient, microcystic lingual lymphangiomatous infiltration.
A girl aged 10 years presenting an exclusively lingually localised congenital microcystic lymphangioma. A first surgical treatment, a partial glossectomy, had been performed at age 6 months.
956 Following several lymphangiomatous growths a second partial glossectomy was performed at age two years. The patient was then lost to follow-up for several years. At age 10 years, she was brought in by her parents with a voluminous, massively infiltrated tongue with daily biting and bleeding, a lower jaw deformity and a speaking impediment. A coblation session was conducted some time afterwards, with a favourable outcome on the tongue volume and no more bleeding. Improvement of the functional impairment and phonation was less marked, however. Seven months after surgery, a few lingual vesicles reappeared, but in a much smaller area than that initially affected. Another coblation session will very probably prove necessary.
3.4. Case no. 4 A girl aged 8 years presenting a lingual and pelvilingual microcystic lymphangioma and a sub-maxillary macrocystic lymphangioma. The lesion was present at birth. She had been given a first sclerosing injection in the cervical region at age 15 months, with a favourable outcome. A partial glossectomy was performed at age 3 years, of limited efficacy, followed by a new sclerosing injection at age 7 years. The following year she presented a marked lingual infiltration with repeated bleeding, frequent trauma and biting, and eating difficulties. A lingual coblation session was conducted a short time afterwards. There were no post-operative complications, and functional improvement was immediate. There was no more bleeding, and eating was easier. Biting did not disappear but became much less frequent.
N. Leboulanger et al.
Fig. 4
Per-operative view: dorsal bud.
were several surgical treatments during childhood: exeresis by cervicotomy and reductive glossoplasty. The patient presented a marked mandibular deformity for which orthodondic treatment and orthognatic surgery were planned. He presented numerous lymphomangiomatous lesions of the dorsal and ventral surfaces of the tongue, not very painful but bleeding frequently. There was no noteworthy fall in haemoglobin count. A coblation session was applied to remove all projecting lymphangiomatous surface lesions. Five months post-operatively the overall volume of the tongue was unchanged, but the projecting lesions had disappeared and the symptoms were relieved.
3.7. Case no. 7 A girl aged 7 years presenting an essentially lingual lymphangioma, located mainly on the dorsal surface of the tongue and on its right free edge. There was also a small, barely visible right laterocervical
3.5. Case no. 5 A girl aged 6 years presenting a stable lingual lymphoma with unimpaired dental articulation, and of moderate size. She had never had surgical treatment. In a few months a voluminous dorsal bud appeared (Fig. 4), causing the patient considerable discomfort. The lesion was painful and bled on contact. Lingual coblation removed the bud (Fig. 5) and cause the symptoms to disappear completely. Follow-up at 5 months showed no relapse. The other lingual lesions, which were not troublesome, remained stable.
3.6. Case no. 6 A 14-year-old male adolescent presenting a cervicofacial lymphangioma discovered at birth. There
Fig. 5 Per-operative view, after coblation: dorsal bud removed.
Utility of radiofrequency ablation for haemorrhagic lingual lymphangioma
Fig. 6
Pre-operative front view.
localisation that had never grown or needed treatment. The lingual localisation underwent monthly growths with increase in volume and in the number of surface vesicles that bled and were painful. No tongue surgery had ever been performed. Despite the volume of the tongue, the girl was able to close her mouth and feed normally (Fig. 6). We applied a single lingual coblation session that destroyed the exophytic vesicular lesions on the right lateral and dorsal surfaces. There were no post-operative complications. Four months after surgery the overall volume of the tongue had decreased (Fig. 7). Four lymphangiomatous vesicles reappeared on the right lateral edge of the tongue, but with only one incident of traumatic bleeding.
4. Discussion The use of radiofrequency in lymphangioma is some 10 years old. Two main techniques have been proposed.
Fig. 7
Post-operative front view.
957
Initially, high frequency radiofrequency has been used in the aim to reduce the size of the tongue; the electrode is introduced directly into the lingual tissue and several passes are carried out in depth. This method induces a deep secondary fibrosis that preserves the surface mucosa and is comparable to that used for turbinate reduction or radiofrequency velar coblation for ronchopathy. It has the disadvantage of low accuracy and it cannot be used for mucosal treatment. However, it allows an overall reduction in the size of the tongue. The second technique, described recently and reported here [7], makes use of a coblation probe comparable to that used for tonsillectomy or tonsil reduction (low frequency radiofrequency). The energy is applied to the surface via a conduction medium (here saline serum), and over a smaller volume, which allows greater local efficacy and better accuracy. It permits a specific reduction of mucosal lymphangiomatous lesions without affecting underlying muscle, and maintains satisfactory haemostasis. The quantity of energy delivered can be more closely controlled than in the first method. Both methods are complementary and can be combined. The utility of surface radiofrequency coblation of lymphangiomas of the mouth, and in particular of the tongue, lies in its ease of implementation and straightforward post-operative phase. The patients described here all had specific massive lingual infiltration with functional impact and bleeding. This low-volume but daily repeated bleeding was a significant functional impairment also affecting those close to the patient, and having a general impact on well-being. The series described by Grimmer et al. [7] concerned 11 cases of lymphangioma of the mouth, with wider-ranging localisation. Their functional results were favourable, with a very straightforward postoperative phase. Likewise, pain and bleeding were low here, and eating was resumed by Day 1. The use of powerful antalgics (III) was limited to the immediate postoperative period. The longest time elapsed since surgery is currently two years, with in two cases a return of local symptoms at a milder level. However, this technique has no effect on overall lingual hypertrophy, or on iterative increases in volume, or on any secondary mandibular deformities. Patients and their families must be well informed that the treatment is in no way curative, and that regrowth, aggravation of the lesion and the recurrence of haemorrhagic lingual vesicles are possible.
958
N. Leboulanger et al.
Further work will be necessary to fully validate these findings and the treatment indication in the long term.
5. Conclusion Surface radiofrequency coblation is a useful technique for improving functional impairment (bites, hindrance due to large-volume vesicles and bleeding), and in particular reducing bleeding, due to certain lingual lymphangiomas. Mucosal lingual lesions respond well to the treatment, with an uncomplicated post-operative phase. Although the functional improvement is marked, its stability over time remains to be assessed by a long-term patient follow-up. It must be explained to the patients and their families that no long-term review of the treatment is yet available.
6. IRB disclosure The authors of this article certify to have no financial interest or involvement of any kind in the subject matter or materials discussed in this manuscript.
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