Journal of Cutaneous and Aesthetic Surgery
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CORRESPONDENCE  
Year : 2020  |  Volume : 13  |  Issue : 1  |  Page : 73-75
Venolymphatic malformation of the tongue and response to sclerotherapy using sodium tetradecyl sulfate


1 Department of Dermatology & STDRadiodiagnosis University College of Medical Sciences & GTB Hospital (University of Delhi), Delhi – 110095, India
2 Radiodiagnosis University College of Medical Sciences & GTB Hospital (University of Delhi), Delhi – 110095, India

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Date of Web Publication17-Mar-2020
 

How to cite this article:
Singal A, Bhatt S. Venolymphatic malformation of the tongue and response to sclerotherapy using sodium tetradecyl sulfate. J Cutan Aesthet Surg 2020;13:73-5

How to cite this URL:
Singal A, Bhatt S. Venolymphatic malformation of the tongue and response to sclerotherapy using sodium tetradecyl sulfate. J Cutan Aesthet Surg [serial online] 2020 [cited 2020 Apr 1];13:73-5. Available from: http://www.jcasonline.com/text.asp?2020/13/1/73/280798




Dear Editor

A 28-years-old healthy woman presented with a bluish swelling on the dorsum of tongue, which was present for almost two decades. Her parents noticed bluish discoloration of tongue when she was approximately 8 years old, which enlarged at puberty to a pea-sized swelling. The lesion increased progressively to the present size in the last 2–3 years. Examination revealed a bilobed swelling, 3 × 4cm in size [Figure 1]A, present over the dorsum of middle one-third of the left half of the tongue. The swelling was soft, partially compressible with no thrill or bruit. The patient was majorly asymptomatic with no pain or difficulty in mastication but gave a history of occasional bleeding while chewing hard food. Rest of the mucocutaneous examination and physical examination was normal. Her hematological investigations and blood biochemistry revealed no abnormality. High-resolution ultrasonography (USG) showed an anechoic lesion on the dorsum of the tongue on the left side. The superficial, irregular, anechoic, cystic component reached up to the tongue surface. Lesion had a vaguely echogenic deeper component, which showed mild vascularity on color Doppler imaging [Figure 2]A. Contrast-enhanced computed tomography (CECT) of the face showed a non-enhancing, low attenuation mass of water density (Houndsfield Unit was 10-12 HU) with a deeper well-defined, irregular, homogenously enhancing component [Figure 2]B with enhancement persisting in the delayed phases as well. These findings were consistent with combined venolymphatic malformation (VLM) of tongue. The patient was treated by sclerotherapy using intralesional injections of 3% sodium tetradecyl sulfate (STS). The lesion was injected with insulin syringe at multiple sites, using approximately 0.3–0.4mL of solution. Postinjection, manual compression using gauze piece was given for 3min. She reported transient pain and burning sensation immediately after the injection that resolved spontaneously in the next 15min. No delayed adverse effects were noted. Two such injections at 3-week interval led to complete resolution of the lesion [Figure 1]B with no recurrence after 1 year.
Figure 1: A 28-year-old woman with VLM of the tongue. (A) Pre-op. (B) Post-op after two doses of STS

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Figure 2: (A) Longitudinal colour Doppler scan shows a superficial, irregular, anechoic and a deeper, vaguely, echogenic component demonstrating mild vascularity shown by the red and blue color in the USG image representing flow of blood in different directions in the lesion. (B) Coronal CECT image shows a non-enhancing, predominantly water density mass with few enhancing septae and a homogenously enhancing vascular component within the tongue.

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Vascular malformations are structural malformations of vascular development. Depending on their predominant vessel type, they have been categorized as arterial, venous, lymphatic, or mixed types.[1] On the basis of flow characteristic, vascular malformations are generally subdivided into slow-flow lesions (venous, capillary, and lymphatic malformations) and fast-flow lesions (arterial malformations as aneurysm and arteriovenous malformation).[2] VLMs are tumorlike lesions combining dysplastic lymphatic and venous vessel structures.[3] Of the slow-flow lesions, venous malformations (VMs) are the most common ones accounting for two-third of all such cases. VMs and VLMs are generally present at birth and grow in size proportionately to the body growth. Approximately 40% of the lesions occupy head and neck region. Symptoms may range from asymptomatic to mild discomfort and pain, bleeding, disfigurement, and functional impairment. Clinically, it may not be possible to differentiate VMs and VLMs. Diagnosis of VLM is generally based on USG and color Doppler imaging as was apparent in this case. CECT or magnetic resonance imaging is useful to correctly delineate the extent of the lesion, which is required to plan the treatment.[4] A specific feature of VM is the presence of phleboliths that was not observed in this case. It is possible that many cases of VLMs are treated as VMs in the absence of radioimaging.

VLMs do not involute spontaneously and require surgical intervention or treatment with sclerosing agents. Various treatment modalities include supportive therapy such as compression, sclerotherapy, lasers, and surgery or combination of the two modalities.[3] Sclerotherapy is economical and efficacious and forms the first-line nonsurgical intervention, it is not widely used. The treatment has to be tailored for individual lesion based on the size, location, symptoms, and proximity to the vital structures.[5] Multiple sessions may be required. A variety of sclerosing agents have been used, namely 95% ethanol, polidocanol, and STS.[5] Other agents include sodium morrhuate, Picibanil (OK-432), bleomycin, and intravenous doxycycline. We report this case to document the following: (1) occurrence of VLM of the tongue and (2) excellent response of sclerotherapy with STS.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Khatib Y, Dande M, Patel RD, Gite V. Venolymphatic vascular malformation of the parotid gland extending into the parapharyngeal space: a rare presentation. J Oral Maxillofac Pathol 2016;20:308-11.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Shah KM, Karagir A, Adaki S. Slow-flow-type venous malformation of tongue. BMJ Case Rep 2013;2013:bcr2013008945.  Back to cited text no. 2
    
3.
Kadota Y, Utsumi T, Kawamura T, Inoue M, Sawabata N, Minami M, et al. Lymphatic and venous malformation or “lymphangiohemangioma” of the anterior mediastinum: case report and literature review. Gen Thorac Cardiovasc Surg 2011;59:575-8.  Back to cited text no. 3
    
4.
Yakes WF. Diagnosis and management of low-flow veno-lymphatic vascular malformations. Ces Radiol 2008;62:131-45.  Back to cited text no. 4
    
5.
Alakailly X, Kummoona R, Quereshy FA, Baur DA, González AE. The use of sodium tetradecyl sulphate for the treatment of venous malformations of the head and neck. J Maxillofac Oral Surg 2015;14:332-8.  Back to cited text no. 5
    

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Correspondence Address:
Archana Singal
Department of Dermatology and STD, University College of Medical Sciences and Guru Teg Bahadur Hospital, University of Delhi, Delhi 110095.
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JCAS.JCAS_70_19

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