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Surgical management of large venous malformations of the lower face
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British Journal of Oral and Maxillofacial Surgery 51 (2013) 752–756
Surgical management of large venous malformations of the lower face Bernardo Hontanilla ∗ , Shan-Shan Qiu, Diego Marre Department of Plastic and Reconstructive Surgery, Clínica Universidad de Navarra, Navarra, Spain Accepted 15 June 2013 Available online 4 July 2013
Abstract We describe the benefits of an early surgical approach to large (more than 3 cm) venous malformations in the lower face, and discuss the advantages over conservative treatment. Fifty-eight patients with venous malformations of the lower face were treated in this hospital between 2005 and 2010 with sclerotherapy (lipidocanol), or láser, or resection, or all three. Only patients with recurrent malformations and a history of previously ineffective conservative treatment were included in the study (n = 17). Follow-up ranged from 23–65 months (mean 40). Functional and cosmetic outcomes and recurrence were recorded on a single questionnaire. Seventeen patients with a history of recurrent malformations, which had previously been treated ineffectively with conservative treatment and were more than 3 cm in diameter, benefited from early and wide resection. No recurrences were recorded during follow-up. Patients were satisfied with the postoperative cosmetic and functional results. Large malformations are both deforming and functionally disabling. These patients, who initially do not respond to conservative treatment, benefit from early definitive resection. Level of evidence: 4 (case series with comparison). © 2013 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Venous malformation; Surgery; Lower face
Introduction Vascular malformations are developmental errors that are composed of enlarged dysplastic blood vessels.1,2 Venous malformations are the most common slow-flow vascular malformations that are referred to specialised centres. Most of them are sporadic, and unifocal (93%), although 1% are multifocal. There is no sex preponderance. They are lightto-dark-blue lesions that can be emptied by compression or when they are in the upright position. Palpation is not painful unless they thrombose. They can affect any tissue or organ, including cutaneous and subcutaneous tissue, muscle, joint, or intestine. They can also threaten life because of bleeding, ∗ Corresponding author at: Department of Plastic and Reconstructive Surgery, Clinica Universidad de Navarra, Av. Pio XII 36, 31008 Pamplona, Spain. Tel.: +34 948255400; fax: +34 948296500. E-mail address: [email protected] (B. Hontanilla).
expansion, or obstruction of vital structures. If they are in the pharynx or larynx they can compromise the airway and cause snoring or even sleep apnoea. The common non-surgical treatments are sclerotherapy,3 embolisation,4,5 and laser.6 Excision is indicated in localised lesions that allow complete resection without appreciable haemorrhage and mutilating postoperative defects.7 Currently, despite the different therapeutic options for venous malformations, there is a lack of consensus about which is the gold standard for each case. As far as venous malformations of the lip are concerned, many factors should be considered when deciding on treatment, such as cosmesis, functional impairment, and the resultant morbidity after excision (if this option is indicated). The lips are a fundamental functional and aesthetic component of the face and it is therefore of paramount importance that their anatomical subunits should be restored to function, and labial function restored.7 Some authors have shown in the past that these two objectives
0266-4356/$ – see front matter © 2013 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.bjoms.2013.06.008
B. Hontanilla et al. / British Journal of Oral and Maxillofacial Surgery 51 (2013) 752–756
753
Table 1 Details of patients. Year of presentation
Sex
Age (years)
Site
Previous treatments
Follow-up (months)
Size (cm)
Type of reconstruction
4
F
27
Lower lip
65
4 × 4.2
3
M
4
23
3 × 3.1
3
M
24
Lower lip and chin Lower lip
Sclerotherapy (2 sessions) Laser Nd-Yag (2 sessions) Sclerotherapy (9 sessions) Sclerotherapy (2 sessions)
30
3.3 × 2.6
6
M
20
Sclerotherapy (2 sessions)
45
4×5
4
M
34
Upper and lower lip Lower lip
52
3.7 × 2
3
M
17
Lower lip
Sclerotherapy (3 sessions) Laser Nd-Yag (1 session) Sclerotherapy (4 sessions)
36
3 × 3.3
2
F
6
Sclerotherapy (5 sessions)
43
3.5 × 4
2
F
9
Upper lip and chin Lower lip
25
2.6 × 4
4
M
12
Lower lip
46
3.2 × 3.5
3
M
14
Lower lip
Sclerotherapy (3 sessions) Laser Nd-Yag (2 sessions) Sclerotherapy (5 sessions) Laser Nd-Yag (1 session) Sclerotherapy (3 sessions)
51
3.4 × 3.4
4
M
8
Lower lip
Sclerotherapy (5 sessions)
45
3×3
2
F
5
Upper lip and chin
Sclerotherapy (4 sessions) Laser Nd-Yag (2 sessions)
35
4×3
4
M
15
Sclerotherapy (6 sessions)
32
3.5 × 4
2
F
6
Upper and lower lip Lower lip
Sclerotherapy (2 sessions)
51
3.2 × 4.3
3
M
18
Lower lip
Sclerotherapy (3 sessions)
48
3×3
4
M
21
Lower lip
27
4×2
2
F
5
Lower lip
Sclerotherapy (4 sessions) Laser Nd-Yag (2 sessions) Sclerotherapy and laser
28
4 × 2.3
Triangular excision and direct closure Excision and direct closure Triangular excision and direct closure Excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure. Free gracilis transplant Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure
VM = venous malformation; no recurrences were detected.
can be obtained with less invasive approaches. However, the indication for each case is still not clear and the debate about whether resection is necessary is still open. Here we present our experience of the surgical management of large venous malformations of the lower face (upper and lower lips, and chin), and report cosmetic outcome, recurrence, and the benefits of early resection.
Patients and methods We reviewed the medical records of 58 patients who presented to our hospital between 2005 and 2010 with venous malformations of the lower face that had been treated with sclerotherapy (polidocanol), or laser, or resection, or a combination. Recurrence, and functional and cosmetic outcomes, were recorded on a single questionnaire. Age, sex, size of the lesion in relation to the volume of the lip, functional impairment, and history of previous treatments were recorded for all patients.
We have included only the 17 patients of our total of 58 who gave a history of recurrent venous malformation that had previously been treated with sclerotherapy or laser to reduce the size of the malformation and had no history of surgical treatment. These 17 patients had large venous malformations (more than 3 cm) located in the lower face, and their mean age at operation was 19 years (range 4–34). The rest of our 41 patients had venous malformations less than 3 cm in size that were resolved by sclerotherapy or laser, or both, and followed by minor operations. The 17 patients had had sclerotherapy to reduce the size of large venous malformations with no particular improvement. We have not included malformations in other sites (mouth, pharynx, neck, or upper face). The patients presented with recurrence 2–4 months after the last session of either sclerotherapy or laser, and were offered resection as these treatments had failed. Preoperatively patients had been given several sessions of sclerotherapy (2–9) and 5 had been treated with laser (Table 1). All patients were operated on under general anaesthesia by the first author. Complete resection was possible in
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B. Hontanilla et al. / British Journal of Oral and Maxillofacial Surgery 51 (2013) 752–756
all patients, and special care was taken to avoid unnecessary blood loss, particularly in children. After excision the resulting defect was closed primarily as the lesions affected the lower face. One patient, whose venous malformation involved the facial nerve, required reanimation of the smile with a free gracilis muscle transfer. Patients were usually discharged on the second or fourth postoperative day if there were no signs of haematoma. No complications that required revision were recorded during follow-up. All the specimens were sent for histological analysis to confirm the clinical diagnosis. Follow-up ranged from 23 to 65 months (mean 40). Cosmesis and recurrence were recorded on postoperative visits. Standard photographs were taken preoperatively and 6 months postoperatively to evaluate the outcome. Serial magnetic resonance images (MRI) were taken at 6 monthly intervals to detect recurrences. It is our protocol to obtain MRI for all patients whose malformations were more than 5 cm deep in the oral submucosa, pterigoid space, or deep structures, to find out whether there was a subclinical recurrence. A single questionnaire8,9 was sent to all patients. Complete clinical success was defined as disappearance of all complaints, according to the patients or their parents, or both. Clinical failure was defined as no change or worsening of one of the symptoms assessed after treatment (pain, swelling, function, and cosmesis). The clinical effect of treatment for the different symptoms was graded on a 4-point scale: 1 = worsening of symptoms; 2 = no change; 3 = partial improvement; and 4 = complete relief. Finally, patients were asked whether they were satisfied with their treatment. Data were entered in Excel® without the patient’s name. Follow-up time was calculated as time from completion of treatment to March 2010.
Table 2 Signs and symptoms before and after treatment by radical resection (n = 17).
Present before treatment Complete relief Partial relief No relief Worse
Pain
Swelling
Functional impairment
Cosmesis
6 4 1 1 0
17 17 0 0 0
17 14 2 0 1
17 17 0 0 0
Fig. 1. (A) A 4-year-old boy with a venous malformation of the entire lower lip. (B) The same boy 23 months after operation (published with the permission of the parents).
Results
mild hypoaesthesia of the lower lip, which resolved spontaneously. Another patient had a commissuroplasty several months after resection to improve cosmesis and oral control. The oral sphincter was competent and mouth opening adequate (an interincisal distance of >35 mm) in all cases. In addition, the patient who required facial reanimation developed good commissural excursion with symmetry at rest and when smiling (Fig. 2). None of the patients have shown any signs of recurrence on serial MRI 6, 12, and 36 months postoperatively.
Histological study confirmed the clinical diagnosis in all cases. Malformations were characterised by enlarged venous channels lined by a single flattened layer of endothelial cells surrounded by sparse, irregularly distributed, smooth muscle cells. Apparently no malformation affected the bones or lymphatics. Completeness of the excision was checked by serial MRI and then the size was compared with that of the excised piece. All 17 patients returned the questionnaire. All patients reported being satisfied with the treatment and would opt to undergo treatment again. Good clinical success and satisfaction of patients were closely related: all 17 patients who were clinical successes reported that they were satisfied with their treatment. Signs and symptoms before and after excision of the malformation are listed in Table 2. Swelling, pain, function, and cosmesis were satisfactory after excision. Bulk was reduced and the contour and function of the lip were improved in all patients (Fig. 1). One patient had
Fig. 2. (A) A 5-year-old girl with a venous malformation of the upper and lower lips and the chin. (B) The smiling patient 2 years postoperatively (published with the permission of the parents).
B. Hontanilla et al. / British Journal of Oral and Maxillofacial Surgery 51 (2013) 752–756
Discussion Vascular malformations can be broadly classified into high and low flow, and the former includes capillary, venous, and lymphatic malformations. Unlike vascular tumours, they display no proliferative cellular activity and, given that they do not usually involute, they require treatment at some point. For extensive cervicofacial lesions the management is not easy because of the functional and aesthetic importance of the different parts of the face, and the presence of several delicate structures (such as the facial nerve) that are sometimes captured inside the mass. Currently, combined treatment is usually suggested for such cases, and several reports have described different treatments, although there is still a controversy about which is the most suitable.10–13 Options include sclerosing agents, resection, or laser, alone or in combination. Recently, some authors have advocated conservative treatment for such lesions, particularly when resection carries high morbidity. However, the results after conservative treatment in terms of recurrence have not been completely satisfactory for any kind of vascular malformation. For this reason, we add some clarifying indications to offer simple and effective management for large vascular malformations located in the lower face. However, some authors have reported that for very large tumours complete resolution cannot be obtained with any of the techniques currently available.14 Nowadays non-surgical management is preferred by several authors who have reported good to excellent results, with reduction in the size of the lesion and a low incidence of scarring or other complications. Derby and Low in 1997 proposed the use of laser treatment,15 and classified their patients according to the size of the tumour (<10 mm and 10 mm or more). They obtained good cosmetic results although they mentioned recurrences at 2–6 months. They did not achieve complete resolution in any of their cases. We think that their conclusions can be applicable only to small lesions based on the classification by size. Similarly, Sarig et al. reported greater success with laser treatment than with resection and sclerotherapy,6 also a lower incidence of complications. Although laser can shrink vascular malformations, it is not curative. As the malformations do not regress spontaneously, they must be completely removed for a definitive cure. Ueda et al.16 functionally reconstructed the upper and lower lips using 2 free flaps, a forearm flap and a functional free gracilis muscle transfer, after resection of an arteriovenous malformation. They obtained good functional eating and speaking. Sclerotherapy can be an alternative for an extensive venous malformation, either alone or combined with resection. Several studies have reported its effectiveness for reducing size and improving symptoms, but without achieving complete resolution.3,4 Ethanol was one of the main sclerosants used to reduce size. However, to reduce side effects different foams have been included, including sodium tetradecyl sulphate and polidocanol by ultrasound-guided sclerotherapy.17 Injection sclerotherapy using sodium tetradecyl sulphate foam
755
offers an effective treatment option for the management of children who present with oral and pharyngeal venous malformations.18 However, malformations that were morphologically characterised by microcystic, septated vessels did not respond to foams, and these vascular malformations are best treated by resection.19 However, sclerotherapy combined with resection can be a useful way to prevent massive blood loss and provide a less aggressive resection. Reconstruction of labial defects remains a therapeutic challenge, as it has 2 fundamental requirements: achievement of good cosmesis and conservation of labial function.14 A malformation that affects the lip can severely affect a patient’s quality of life by causing cosmetic and functional impairment. In such cases, patients must be offered the most suitable and definitive approach. Conservative treatment of these cases in particular can be time-consuming and of doubtful efficacy. For large tumours, resection should be the first choice even though sclerotherapy first can be helpful to overcome complications such as haemorrhage and the mutilating effect of the procedure. We think that to attempt to treat these lesions solely non-surgically is not entirely correct, as neither sclerotherapy nor laser can eliminate the malformation completely, and although they can shrink the tumour and give symptomatic improvement, recurrence is often evident only a few months after treatment. In our study, all patients had had previous sclerotherapy, or laser, or both, in the hope of obtaining a good cosmetic result. However, recurrence was the rule, and so resection was offered to provide a definitive solution. After complete excision none of the patients included have so far had a recurrence. These results agree with those reported by Dubois et al.12 and Papadopoulos et al.20 in which the authors reported a 26% cure with sclerotherapy alone and a 50% cure when combined with resection. In our series the function of the oral sphincter and the overall appearance of the lip and face were satisfactory in the patients’ opinions. Because of the difficulty in managing venous malformations, the large interindividual variations in size and shape, and the variation in the effects of sclerotherapy and laser, it is difficult to inform patients about the realistic expectations of the different treatments. Given our results, we are able to tell patients that initial partial or complete relief of malformations over 3 cm in size in the lower face is expected after resection in all cases. However, in other studies there is a considerable chance of complications occurring during percutaneous treatment in 40% of patients, most of which are mild and temporary, whereas major complications occurred in 18% of patients.8
Conflict of interest We have no financial and personal relationships with other people or organisations that could inappropriately influence (bias) our work including employment, consultancies, stock ownership, honoraria,
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B. Hontanilla et al. / British Journal of Oral and Maxillofacial Surgery 51 (2013) 752–756
paid expert testimony, patent applications/registrations, and grants or other funding.
Ethics statement The work has been approved by the appropriate ethical committees related to the institution in which it was performed and that subjects gave informed consent to the work.
References 1. Mulliken JB, Glowacki J. Hemangiomas and vascular malformations in infants and children: a classification based on endothelial characteristics. Plast Reconstr Surg 1982;69:412–20. 2. Kaban LB, Mulliken JB. Vascular anomalies of the maxillofacial region. J Oral Maxillofac Surg 1986;44:203–13. 3. Mendonca DA, McCafferty I, Nishikawa H, Lester R. Venous malformations of the limbs: the Birmingham experience, comparisons and classification in children. J Plast Reconstr Aesthet Surg 2012;63:383–9. 4. Juszkat R, Zabicki B, Checinski P, Gabriel M, Matar N. Endovascular treatment of arteriovenous malformation. Aesth Plast Surg 2009;33:639–42. 5. Gelbert F, Enjolras O, Deffrenne D, Aymard A, Mounayer C, Merland JJ. Percutaneous sclerotherapy for venous malformations of the lips: a retrospective study of 23 patients. Neuroradiology 2000;42:692–6. 6. Sarig O, Kimel S, Orenstein A. Laser treatment of venous malformations. Ann Plast Surg 2006;57:20–4. Erratum in: Ann Plast Surg 2006; 57:665. 7. Aubá C, Hontanilla B. Prevention of a foot amputation: a large arteriovenous malformation reconstructed with a composite free flap. Plast Reconstr Surg 2007;119:96e–100e. 8. van der Linden E, Pattynama PM, Heeres BC, de Jong SC, Hop WC, Kroft W. Long-term patient satisfaction after percutaneous treatment of peripheral vascular malformations. Radiology 2009;251:926–32.
9. van der Linden E, Otoide-Vree M, Pattynama MT. Percutaneous treatment of peripheral vascular malformations in children: long-term clinical outcome. Cardiovasc Intervent Radiol 2012;35:316–24. 10. Van Doorne L, De Maeseneer M, Stricker C, et al. Diagnosis and treatment of vascular lesions of the lip. Br J Oral Maxillofac Surg 2002;40:497–503. 11. Riche MC, Hadjean E, Tran-Bahuy P, Merland JJ. The treatment of capillary venous malformations using a new fibrosing agent. Plast Reconstr Surg 1983;71:607–12. 12. Dubois JM, Sebag GH, De Prost Y, Teillac D, Chretien B, Brunelle FO. Soft tissue venous malformations in children: percutaneous sclerotherapy with Ethibloc. Radiology 1991;180:195–8. 13. Del Pozo J, Pazos JM, Fonseca E. Lower lip hypertrophy secondary to port-wine stain: combined surgical and carbon dioxide laser treatment. Dermatol Surg 2004;30:211–4. 14. Malard O, Corre P, Jégoux F, et al. Surgical repair of labial defect. Eur Ann Otorhinolaryngol Head Neck Dis 2010;127:49–62. 15. Derby LD, Low DW. Laser treatment of facial venous vascular malformations. Ann Plast Surg 1997;38:371–8. 16. Ueda K, Oba S, Nakai K, Okada M, Kurokawa N, Nuri T. Functional reconstruction of the upper and lower lips and commisure with a forearm flap combined with a free gracilis muscle transfer. J Plast Reconstr Aesthet Surg 2009;62:e337–40. 17. Blaise S, Charavin-Cocuzza M, Riom H, et al. Treatment of low-flow vascular malformations by ultrasound-guided sclerotherapy with polidocanol foam: 24 cases and literature review. Eur J Vasc Endovasc Surg 2011;41:412–7. 18. Stimpson P, Hewitt R, Barnacle A, Roebuck DJ, Hartley B. Sodium tetradecyl sulphate sclerotherapy for treating venous malformations of the oral and pharyngeal regions in children. Int J Pediatr Otorhinolaryngol 2012;76:569–73. 19. Markovic JN, Kim CY, Lidsky ME, Shortell CK. A 6-year experience treating vascular malformations with foam sclerotherapy. Perspect Vasc Surg Endovasc Ther 2012;24:70–9. 20. Papadopoulos O, Konofaos P, Tsantoulas Z, Chrisostomidis C, Frangoulis M, Karakitsos P. Lip defects due to tumor excision: apropos of 899 cases. Oral Oncol 2007;43:204–12.
British Journal of Oral and Maxillofacial Surgery 51 (2013) 752–756
Surgical management of large venous malformations of the lower face Bernardo Hontanilla ∗ , Shan-Shan Qiu, Diego Marre Department of Plastic and Reconstructive Surgery, Clínica Universidad de Navarra, Navarra, Spain Accepted 15 June 2013 Available online 4 July 2013
Abstract We describe the benefits of an early surgical approach to large (more than 3 cm) venous malformations in the lower face, and discuss the advantages over conservative treatment. Fifty-eight patients with venous malformations of the lower face were treated in this hospital between 2005 and 2010 with sclerotherapy (lipidocanol), or láser, or resection, or all three. Only patients with recurrent malformations and a history of previously ineffective conservative treatment were included in the study (n = 17). Follow-up ranged from 23–65 months (mean 40). Functional and cosmetic outcomes and recurrence were recorded on a single questionnaire. Seventeen patients with a history of recurrent malformations, which had previously been treated ineffectively with conservative treatment and were more than 3 cm in diameter, benefited from early and wide resection. No recurrences were recorded during follow-up. Patients were satisfied with the postoperative cosmetic and functional results. Large malformations are both deforming and functionally disabling. These patients, who initially do not respond to conservative treatment, benefit from early definitive resection. Level of evidence: 4 (case series with comparison). © 2013 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Venous malformation; Surgery; Lower face
Introduction Vascular malformations are developmental errors that are composed of enlarged dysplastic blood vessels.1,2 Venous malformations are the most common slow-flow vascular malformations that are referred to specialised centres. Most of them are sporadic, and unifocal (93%), although 1% are multifocal. There is no sex preponderance. They are lightto-dark-blue lesions that can be emptied by compression or when they are in the upright position. Palpation is not painful unless they thrombose. They can affect any tissue or organ, including cutaneous and subcutaneous tissue, muscle, joint, or intestine. They can also threaten life because of bleeding, ∗ Corresponding author at: Department of Plastic and Reconstructive Surgery, Clinica Universidad de Navarra, Av. Pio XII 36, 31008 Pamplona, Spain. Tel.: +34 948255400; fax: +34 948296500. E-mail address: [email protected] (B. Hontanilla).
expansion, or obstruction of vital structures. If they are in the pharynx or larynx they can compromise the airway and cause snoring or even sleep apnoea. The common non-surgical treatments are sclerotherapy,3 embolisation,4,5 and laser.6 Excision is indicated in localised lesions that allow complete resection without appreciable haemorrhage and mutilating postoperative defects.7 Currently, despite the different therapeutic options for venous malformations, there is a lack of consensus about which is the gold standard for each case. As far as venous malformations of the lip are concerned, many factors should be considered when deciding on treatment, such as cosmesis, functional impairment, and the resultant morbidity after excision (if this option is indicated). The lips are a fundamental functional and aesthetic component of the face and it is therefore of paramount importance that their anatomical subunits should be restored to function, and labial function restored.7 Some authors have shown in the past that these two objectives
0266-4356/$ – see front matter © 2013 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
http://dx.doi.org/10.1016/j.bjoms.2013.06.008
B. Hontanilla et al. / British Journal of Oral and Maxillofacial Surgery 51 (2013) 752–756
753
Table 1 Details of patients. Year of presentation
Sex
Age (years)
Site
Previous treatments
Follow-up (months)
Size (cm)
Type of reconstruction
4
F
27
Lower lip
65
4 × 4.2
3
M
4
23
3 × 3.1
3
M
24
Lower lip and chin Lower lip
Sclerotherapy (2 sessions) Laser Nd-Yag (2 sessions) Sclerotherapy (9 sessions) Sclerotherapy (2 sessions)
30
3.3 × 2.6
6
M
20
Sclerotherapy (2 sessions)
45
4×5
4
M
34
Upper and lower lip Lower lip
52
3.7 × 2
3
M
17
Lower lip
Sclerotherapy (3 sessions) Laser Nd-Yag (1 session) Sclerotherapy (4 sessions)
36
3 × 3.3
2
F
6
Sclerotherapy (5 sessions)
43
3.5 × 4
2
F
9
Upper lip and chin Lower lip
25
2.6 × 4
4
M
12
Lower lip
46
3.2 × 3.5
3
M
14
Lower lip
Sclerotherapy (3 sessions) Laser Nd-Yag (2 sessions) Sclerotherapy (5 sessions) Laser Nd-Yag (1 session) Sclerotherapy (3 sessions)
51
3.4 × 3.4
4
M
8
Lower lip
Sclerotherapy (5 sessions)
45
3×3
2
F
5
Upper lip and chin
Sclerotherapy (4 sessions) Laser Nd-Yag (2 sessions)
35
4×3
4
M
15
Sclerotherapy (6 sessions)
32
3.5 × 4
2
F
6
Upper and lower lip Lower lip
Sclerotherapy (2 sessions)
51
3.2 × 4.3
3
M
18
Lower lip
Sclerotherapy (3 sessions)
48
3×3
4
M
21
Lower lip
27
4×2
2
F
5
Lower lip
Sclerotherapy (4 sessions) Laser Nd-Yag (2 sessions) Sclerotherapy and laser
28
4 × 2.3
Triangular excision and direct closure Excision and direct closure Triangular excision and direct closure Excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure. Free gracilis transplant Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure Triangular excision and direct closure
VM = venous malformation; no recurrences were detected.
can be obtained with less invasive approaches. However, the indication for each case is still not clear and the debate about whether resection is necessary is still open. Here we present our experience of the surgical management of large venous malformations of the lower face (upper and lower lips, and chin), and report cosmetic outcome, recurrence, and the benefits of early resection.
Patients and methods We reviewed the medical records of 58 patients who presented to our hospital between 2005 and 2010 with venous malformations of the lower face that had been treated with sclerotherapy (polidocanol), or laser, or resection, or a combination. Recurrence, and functional and cosmetic outcomes, were recorded on a single questionnaire. Age, sex, size of the lesion in relation to the volume of the lip, functional impairment, and history of previous treatments were recorded for all patients.
We have included only the 17 patients of our total of 58 who gave a history of recurrent venous malformation that had previously been treated with sclerotherapy or laser to reduce the size of the malformation and had no history of surgical treatment. These 17 patients had large venous malformations (more than 3 cm) located in the lower face, and their mean age at operation was 19 years (range 4–34). The rest of our 41 patients had venous malformations less than 3 cm in size that were resolved by sclerotherapy or laser, or both, and followed by minor operations. The 17 patients had had sclerotherapy to reduce the size of large venous malformations with no particular improvement. We have not included malformations in other sites (mouth, pharynx, neck, or upper face). The patients presented with recurrence 2–4 months after the last session of either sclerotherapy or laser, and were offered resection as these treatments had failed. Preoperatively patients had been given several sessions of sclerotherapy (2–9) and 5 had been treated with laser (Table 1). All patients were operated on under general anaesthesia by the first author. Complete resection was possible in
754
B. Hontanilla et al. / British Journal of Oral and Maxillofacial Surgery 51 (2013) 752–756
all patients, and special care was taken to avoid unnecessary blood loss, particularly in children. After excision the resulting defect was closed primarily as the lesions affected the lower face. One patient, whose venous malformation involved the facial nerve, required reanimation of the smile with a free gracilis muscle transfer. Patients were usually discharged on the second or fourth postoperative day if there were no signs of haematoma. No complications that required revision were recorded during follow-up. All the specimens were sent for histological analysis to confirm the clinical diagnosis. Follow-up ranged from 23 to 65 months (mean 40). Cosmesis and recurrence were recorded on postoperative visits. Standard photographs were taken preoperatively and 6 months postoperatively to evaluate the outcome. Serial magnetic resonance images (MRI) were taken at 6 monthly intervals to detect recurrences. It is our protocol to obtain MRI for all patients whose malformations were more than 5 cm deep in the oral submucosa, pterigoid space, or deep structures, to find out whether there was a subclinical recurrence. A single questionnaire8,9 was sent to all patients. Complete clinical success was defined as disappearance of all complaints, according to the patients or their parents, or both. Clinical failure was defined as no change or worsening of one of the symptoms assessed after treatment (pain, swelling, function, and cosmesis). The clinical effect of treatment for the different symptoms was graded on a 4-point scale: 1 = worsening of symptoms; 2 = no change; 3 = partial improvement; and 4 = complete relief. Finally, patients were asked whether they were satisfied with their treatment. Data were entered in Excel® without the patient’s name. Follow-up time was calculated as time from completion of treatment to March 2010.
Table 2 Signs and symptoms before and after treatment by radical resection (n = 17).
Present before treatment Complete relief Partial relief No relief Worse
Pain
Swelling
Functional impairment
Cosmesis
6 4 1 1 0
17 17 0 0 0
17 14 2 0 1
17 17 0 0 0
Fig. 1. (A) A 4-year-old boy with a venous malformation of the entire lower lip. (B) The same boy 23 months after operation (published with the permission of the parents).
Results
mild hypoaesthesia of the lower lip, which resolved spontaneously. Another patient had a commissuroplasty several months after resection to improve cosmesis and oral control. The oral sphincter was competent and mouth opening adequate (an interincisal distance of >35 mm) in all cases. In addition, the patient who required facial reanimation developed good commissural excursion with symmetry at rest and when smiling (Fig. 2). None of the patients have shown any signs of recurrence on serial MRI 6, 12, and 36 months postoperatively.
Histological study confirmed the clinical diagnosis in all cases. Malformations were characterised by enlarged venous channels lined by a single flattened layer of endothelial cells surrounded by sparse, irregularly distributed, smooth muscle cells. Apparently no malformation affected the bones or lymphatics. Completeness of the excision was checked by serial MRI and then the size was compared with that of the excised piece. All 17 patients returned the questionnaire. All patients reported being satisfied with the treatment and would opt to undergo treatment again. Good clinical success and satisfaction of patients were closely related: all 17 patients who were clinical successes reported that they were satisfied with their treatment. Signs and symptoms before and after excision of the malformation are listed in Table 2. Swelling, pain, function, and cosmesis were satisfactory after excision. Bulk was reduced and the contour and function of the lip were improved in all patients (Fig. 1). One patient had
Fig. 2. (A) A 5-year-old girl with a venous malformation of the upper and lower lips and the chin. (B) The smiling patient 2 years postoperatively (published with the permission of the parents).
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Discussion Vascular malformations can be broadly classified into high and low flow, and the former includes capillary, venous, and lymphatic malformations. Unlike vascular tumours, they display no proliferative cellular activity and, given that they do not usually involute, they require treatment at some point. For extensive cervicofacial lesions the management is not easy because of the functional and aesthetic importance of the different parts of the face, and the presence of several delicate structures (such as the facial nerve) that are sometimes captured inside the mass. Currently, combined treatment is usually suggested for such cases, and several reports have described different treatments, although there is still a controversy about which is the most suitable.10–13 Options include sclerosing agents, resection, or laser, alone or in combination. Recently, some authors have advocated conservative treatment for such lesions, particularly when resection carries high morbidity. However, the results after conservative treatment in terms of recurrence have not been completely satisfactory for any kind of vascular malformation. For this reason, we add some clarifying indications to offer simple and effective management for large vascular malformations located in the lower face. However, some authors have reported that for very large tumours complete resolution cannot be obtained with any of the techniques currently available.14 Nowadays non-surgical management is preferred by several authors who have reported good to excellent results, with reduction in the size of the lesion and a low incidence of scarring or other complications. Derby and Low in 1997 proposed the use of laser treatment,15 and classified their patients according to the size of the tumour (<10 mm and 10 mm or more). They obtained good cosmetic results although they mentioned recurrences at 2–6 months. They did not achieve complete resolution in any of their cases. We think that their conclusions can be applicable only to small lesions based on the classification by size. Similarly, Sarig et al. reported greater success with laser treatment than with resection and sclerotherapy,6 also a lower incidence of complications. Although laser can shrink vascular malformations, it is not curative. As the malformations do not regress spontaneously, they must be completely removed for a definitive cure. Ueda et al.16 functionally reconstructed the upper and lower lips using 2 free flaps, a forearm flap and a functional free gracilis muscle transfer, after resection of an arteriovenous malformation. They obtained good functional eating and speaking. Sclerotherapy can be an alternative for an extensive venous malformation, either alone or combined with resection. Several studies have reported its effectiveness for reducing size and improving symptoms, but without achieving complete resolution.3,4 Ethanol was one of the main sclerosants used to reduce size. However, to reduce side effects different foams have been included, including sodium tetradecyl sulphate and polidocanol by ultrasound-guided sclerotherapy.17 Injection sclerotherapy using sodium tetradecyl sulphate foam
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offers an effective treatment option for the management of children who present with oral and pharyngeal venous malformations.18 However, malformations that were morphologically characterised by microcystic, septated vessels did not respond to foams, and these vascular malformations are best treated by resection.19 However, sclerotherapy combined with resection can be a useful way to prevent massive blood loss and provide a less aggressive resection. Reconstruction of labial defects remains a therapeutic challenge, as it has 2 fundamental requirements: achievement of good cosmesis and conservation of labial function.14 A malformation that affects the lip can severely affect a patient’s quality of life by causing cosmetic and functional impairment. In such cases, patients must be offered the most suitable and definitive approach. Conservative treatment of these cases in particular can be time-consuming and of doubtful efficacy. For large tumours, resection should be the first choice even though sclerotherapy first can be helpful to overcome complications such as haemorrhage and the mutilating effect of the procedure. We think that to attempt to treat these lesions solely non-surgically is not entirely correct, as neither sclerotherapy nor laser can eliminate the malformation completely, and although they can shrink the tumour and give symptomatic improvement, recurrence is often evident only a few months after treatment. In our study, all patients had had previous sclerotherapy, or laser, or both, in the hope of obtaining a good cosmetic result. However, recurrence was the rule, and so resection was offered to provide a definitive solution. After complete excision none of the patients included have so far had a recurrence. These results agree with those reported by Dubois et al.12 and Papadopoulos et al.20 in which the authors reported a 26% cure with sclerotherapy alone and a 50% cure when combined with resection. In our series the function of the oral sphincter and the overall appearance of the lip and face were satisfactory in the patients’ opinions. Because of the difficulty in managing venous malformations, the large interindividual variations in size and shape, and the variation in the effects of sclerotherapy and laser, it is difficult to inform patients about the realistic expectations of the different treatments. Given our results, we are able to tell patients that initial partial or complete relief of malformations over 3 cm in size in the lower face is expected after resection in all cases. However, in other studies there is a considerable chance of complications occurring during percutaneous treatment in 40% of patients, most of which are mild and temporary, whereas major complications occurred in 18% of patients.8
Conflict of interest We have no financial and personal relationships with other people or organisations that could inappropriately influence (bias) our work including employment, consultancies, stock ownership, honoraria,
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paid expert testimony, patent applications/registrations, and grants or other funding.
Ethics statement The work has been approved by the appropriate ethical committees related to the institution in which it was performed and that subjects gave informed consent to the work.
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