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CASE REPORT
Year : 2021  |  Volume : 39  |  Issue : 3  |  Page : 329-335
 

Use of Foley's catheter as a tourniquet for the management of vascular lesion of lip in Mowat-Wilson syndrome


Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences, SGT University, Gurugram, Haryana, India

Date of Submission24-Sep-2020
Date of Decision12-Sep-2021
Date of Acceptance13-Sep-2021
Date of Web Publication22-Nov-2021

Correspondence Address:
Dr. Rajeev Pandey
Department of Oral and Maxillofacial Surgery, Faculty of Dental Sciences, SGT University, Gurugram, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jisppd.jisppd_414_20

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   Abstract 


Background: Vascular malformation of lower lip is a very rare anomaly. The lesion leads to facial asymmetry, difficulty in speech and eating and drooling of saliva. Treatment goals include symmetrical reconstruction of the lip with minimal scarring, provide adequate bulk for the reconstruction of vermillion, in toto removal of the lesion and prevent recurrence. The most common complication during surgical removal of these lesions includes blood loss and profuse bleeding which leads to poor visibility, increased operation time and postoperative requirement of blood transfusion. Therefore, the use of sclerosing agent is recommended before surgical removal. This may help in decreasing bleeding during surgery but not in all cases. Case Report: Here, we report the use of Foley's catheter for the management of a high flow lesion of lower lip in a 12-year-old patient diagnosed with Mowat-Wilson syndrome. This technique helped in providing bloodless field which lead to minimal blood loss and good visibility intraoperatively.


Keywords: Catheters, cleft palate, lip, syndrome, tourniquets, vascular malformations


How to cite this article:
Pandey R, Kumar S, Kumar J, Arya V, Thakker R, Singh M. Use of Foley's catheter as a tourniquet for the management of vascular lesion of lip in Mowat-Wilson syndrome. J Indian Soc Pedod Prev Dent 2021;39:329-35

How to cite this URL:
Pandey R, Kumar S, Kumar J, Arya V, Thakker R, Singh M. Use of Foley's catheter as a tourniquet for the management of vascular lesion of lip in Mowat-Wilson syndrome. J Indian Soc Pedod Prev Dent [serial online] 2021 [cited 2021 Nov 29];39:329-35. Available from: https://www.jisppd.com/text.asp?2021/39/3/329/330710





   Introduction Top


Vascular malformation (VM) is a group of tumors that are vascular in origin. It is caused by lymphatic or angiovascular proliferation.[1] They can occur at any part of the body but are usually seen in the craniofacial and neck region, frequently involving upper lip. Vascular anomalies involving lips not only affect facial anatomy but can also result in facial distortion as they become large. These lesions are mostly seen at birth, whereas few of them become obvious by adolescence or adulthood.[2] The enlargement is believed to be a result of pressure or flow change, shunting, ectasia and collateral proliferation.[3] These types of lesions should be diagnosed and treated promptly as they keep on growing and later on management become more challenging. In craniofacial region, it is rare to find the involvement of lower lip. Mowat Wilson syndrome (MWS) is a rare multiple anomaly congenital syndrome with distinct facial phenotype, and here we report a case of such a syndrome, in which Foley's catheter was used as novelty in the form of a tourniquet for the management of vascular lesion of lower lip.


   Case Report Top


A 12-year-old male reported to the Department of Oral and Maxillofacial Surgery with chief complaint of swelling on the right side of lower lip and cleft in the soft palate region. On extraoral examination, the patient appeared to be syndromic with a distinct facial phenotype: hypertelorism was present along with high forehead, frontal bossing, large eyebrows, large and deep set eyes, saddle nose, open mouth, large and lifted ear lobes, M-shaped upper lip, frequent smiling and severe intellectual deficiency [Figure 1] and [Figure 2]. The swelling over the lower lip was first seen 1½ year after birth which was initially very small (pea size) and had been growing in size for the past 10 years. At present, the swelling was approximately 3 cm × 3 cm in size, extending from half centimeter short of the right corner of mouth till midline and about 1 cm short of labial sulcus to white roll of lower lip. It was well defined, spherical in shape, bluish red and sessile. No discharge was noticed and there was thinning of overlying mucosa [Figure 3]. On palpation it was soft, non tender, pulsatile, smooth surfaced, non fixed, non indurated, non fluctuant, and slightly mobile with normal temperature. On intraoral examination, there was cleft of soft palate (Veau Group I Defect) [Figure 4]. Medical history of patient revealed that the patient had epilepsy since birth and was under medication for the same. Based on the clinical examination, the patient was diagnosed with MWS with VM of lower lip and cleft of soft palate. The patient was advised contrast magnetic resonance imaging (MRI) to identify the feeding vessel, which came out to be a branch of the right facial artery and lesion was a high-flow arteriovenous malformation [Figure 5]. The patient was planned for surgical excision of the lesion along with palatoplasty. All routine investigations were advised along with electroencephalogram, ultrasonography of abdomen, and 2-dimensional electrocardiography to rule out cranial, abdominal and cardiac abnormalities.
Figure 1: Frontal view

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Figure 2: Profile view

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Figure 3: Vascular lesion associated with the lower lip

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Figure 4: Cleft Palate

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Figure 5: Contrast magnetic resonance imaging showing the extent of the lesion along with draining vessels

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Surgical treatment

Under general anesthesia, endotracheal intubation was done using RAE south pole tube and throat pack was placed. Intraoral and extraoral painting was done using 5% povidone-iodine. Cleft palate was repaired first using Veau-Wardill-Kilner V-Y Plasty along with veloplasty with the help of Dingman mouth gag. A marking for V-Y closure of lesion was done [Figure 6]. Local infiltration was done using 2% lignocaine with 1:150,000 adrenaline after which a stab incision was made on skin below the lesion so that it divides it into two parts about 2 cm away from the lesion. This incision was used to insert Foley's catheter. Two (number 12) Foley catheters were inserted through the incision using a Crile curved hemostat and brought through the other side (intraoral). A ring-shaped rubber band-like piece of the Foley's catheter was cut and both ends of the catheter are inserted in this ring and both ends of the catheter were pulled through this ring to stop the blood flow from both lateral and medial sides of lesion [Figure 7], [Figure 8], [Figure 9]. After achieving desired action of tourniquet, mucosal incision was made over the lesion following the planned marking, submucosal dissection was performed following which, the feeder vessel was isolated and then ligated. The lesion was identified and removed in toto. The tourniquet was released gradually and bleeders were cauterized using bipolar cautery followed by the removal of the tourniquet. Layered closure was done using 3-0 vicryl suture, starting with the muscle layer and then mucosal closure was done in V-Y manner [Figure 10], [Figure 11], [Figure 12]. Lesion was sent for histopathological examination, which confirmed arteriovenous malformation (AVM). Follow-up of 3 months showed no recurrence of the vascular lesion [Figure 13] and adequate healing of the palate [Figure 14].
Figure 6: Marking for closure

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Figure 7: Crile hemostat for the insertion of the Foley's

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Figure 8: Positioning of the Foley's

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Figure 9: Blanching of the lesion due to torniquet action of the Foley's

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Figure 10: Complete resected lesion

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Figure 11: Esthetic closure of the deformity

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Figure 12: Resected lesion

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Figure 13: Appearance of the lip after 3 months

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Figure 14: Healing of the palate

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   Discussion Top


MWS is a rare genetic disorder characterized by moderate-to-severe intellectual disability, multiple congenital anomalies, and a typical facial phenotype. It was first described by Mowat et al. in 1998. It is estimated to occur in 1 in 50,000–100,000 births. [Table 1] shows the various features associated with MWS syndrome and features which were present in our case [Figure 1], [Figure 2], [Figure 4], [Figure 15], [Figure 16], [Figure 17], [Figure 18]. The main etiological factor is heterozygous mutation of the ZEB2 gene.[4] The mutation leads to the loss of function or decreased function of the gene. The treatment of MWS is based on the needs of each individual. Most patients require the intervention of pediatrician, neurosurgeon, cardiologist, neurologist, gastroenterologist and general surgeon. Physical therapy, speech therapy, and occupational therapy help an individual with development delay to reach their full potential. Genetic counseling is also recommended for family and patients of the affected individuals.
Table 1: Features of Mowat-Wilson syndrome that were present and absent in our patient

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Figure 15: Craniofacial deformities

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Figure 16: Deformity of the feet

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Figure 17: Deformity of the fingers

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Figure 18: Deformity of the genital

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Vascular anomalies are group of disorders that affect any segment of the vascular tree: veins, arteries, capillaries, lymphatics, or a combination of these. These lesions are difficult to diagnose and treat because of uncertain behavior. Confusing nomenclature has been historically hampering the classification and identification of vascular anomalies. An updated classification given by the International Society for the Study of Vascular Anomalies/Biologic classification divided vascular birthmarks into vascular tumors and VMs.[5],[6] VMs are caused by abnormal development of vascular elements during embryogenesis and in fetal life.

VM can be either slow-flow which is of capillary, lymphatic, or venous origin or fast-flow which is of arterial origin, but if there are combinations of these elements, the malformation is called lymphaticovenous malformation,capillary-lymphaticovenous malformation, or AVM.[3],[7] AVMs are the result of errors of vascular development between the 4th and 6th weeks of gestation.[1] AVM is fast-flow and most aggressive form of VM. They cause shunting of blood due to anomalous capillary beds between the arterial and venous system. They lead to significant deformity and functional impairment. AVM is usually noted at birth but are commonly misdiagnosed as other vascular anomalies, leading to delay in diagnosis. AVM keeps growing in synchronous with the growth of the child and puberty, and trauma may trigger rapid growth of the lesion. Enlargement of AVM is due to changes in the pressure and flow within the malformation, ectasia, shunting and collateral proliferation. Although the pathogenesis of AVM is not clear, there are theories such as defects in transforming growth factor-beta signaling, and genetic two-hit hypothesis causing the AVM.

AVMs can be of soft or hard tissue; intraosseous AVMs commonly manifest clinically as painful pulsating mass, bone overgrowth at the lesion site, skin ulceration, osteolytic bone lesion, and enlargement of the draining vein. Patients with AVM of soft tissue usually present with a localized swelling with bleeding tendency. These lesions are a pulsatile mass with palpable thrill and occasionally some of these are able to be auscultated for a bruit.[7] The patient is often able to detect a “whirring sound” or will claim to hear their heartbeat within the lesion. Occasionally, functional impairment due to arterial steal, local hyperthermia, and ischemia may be observed. Shunting of blood diminishes nutritive flow due to which skin necrosis, ulceration and bleeding can be appreciated.

History and clinical examination can distinguish vascular tumors and VMs in over 90% of cases. Diagnosis of AVM is based on clinical features and the use of diagnostic aids such as ultrasound with color Doppler, contrast-enhanced computed tomography (CT), contrast-enhanced MRI, and magnetic resonance angiography. Plain radiography has limited role. In hard tissue VMs, lesion may appear as poorly defined radiolucent image, sometimes honeycomb or soap bubble appearance may be present. In CT scan with iodinated contrast, AVMs appear as a highly enhancing lesion. CT with contrast demonstrates soft-tissue enhancement along with dilated feeding and draining vessels. MRI helps in the diagnosis of VMs either by itself or before angiography. It can easily differentiate between high-flow and low-flow lesions. MRI of an AVM demonstrates a collection of vascular flow voids corresponding to fast-flow vessels, and it can be considered the investigation of choice due to its ability to delineate the extent of the lesion, provide multiplane images. Even on MRI, small vessel-rich AVMs often appear as a well-circumscribed tumor such as vascular mass.

Angiography demonstrates variably dilated or tortuous feeding arteries, arteriovenous shunting occasionally with visualization of discrete fistulae and dilated draining veins. Biopsy may be considered for histological diagnosis when the imaging findings conflict with clinical findings.

Over the past years, there has been use of different treatment modalities for the management of head-and-neck AVMs.[8] Considering the benign cellular nature of AVMs, as less aggressive methods as possible were tried for the management of these lesions, but unfortunately in many cases, they were unsuccessful. Sclerotherapy and embolization techniques are based on using various materials to control bleeding. These materials consist of embolizing agent; their uses were accepted on preoperative adjuvant methods for reducing excessive hemorrhage, reducing surgical operative time and palliative modalities for extensive, deep, or inaccessible lesions.[3] Frequently, proximal ligation of feeding arteries to the AVMs results in the recruitment of collateral vessels and recurrence. Furthermore, this ligation and subsequent collateralization (even more than previous) makes future embolization impossible and aggravates AVM's growth. Yet, ligation as an intraoperative or emergency accessory tool to control unwanted hemorrhage can be used. Furthermore, other less aggressive surgical methods such as curettage and partial resection cause reestablishment of the lesion and lead to rapid recurrence too, and may be accompanied with dangerous intraoperative bleeding. Hence, the complete resection is the most curative surgical method for AVMs and is often performed after presurgical selective embolization.[8],[9] This combination method has the greatest success rate and is nowadays considered a gold standard treatment modality. However, this total resection particularly in head-and-neck region has some intrinsic drawbacks including remaining surgical defect that may produce significant esthetic and functional problems and proximity to vital structures that can restrict proper resection. In our technique, Foley's catheter was used as a tourniquet, this novel use of Foley's catheter came out to be fruitful as it provided adequate tourniquet action, i.e. providing a bloodless field. Stopper for tourniquet was also made by cutting a ring out of catheter only, which also served its purpose well and provided a strong grip without slipping. Authors recommend the use of Foley's catheter as a tourniquet in maxillofacial region for VMs where the lesion is well demarcated.


   Conclusion Top


MWS is a rare entity and may require early maxillofacial surgery for the management of craniofacial lesions in many cases. The use of Foley's catheter is a simple technique, saves time, and provide adequate hemostasis during surgical excision of vascular lesion of lip.

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.

Acknowledgment

The authors would like to thank the parents of the child for their kind permission in allowing us to present a case report of their child with his photographs.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Pandhare MN, Jyoti DB, Mandale MS, Suresh RB. Acquired arteriovenous malformation of lip occurring as an occupational hazard: A case report with review of literature. J Oral Maxillofac Pathol 2018;22:287.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Candamourty R, Venkatachalam S, Babu MR, Reddy VK. Low flow vascular malformation of the buccal mucosa treated conservatively by sclerotherapy (3% sodium tetradecyl sulfate). J Nat Sci Biol Med 2012;3:195-8.  Back to cited text no. 2
    
3.
Marler JJ, Mulliken JB. Current management of hemangiomas and vascular malformations. Clin Plast Surg 2005;32:99-116.  Back to cited text no. 3
    
4.
Garavelli L, Mainardi PC. Mowat-Wilson syndrome. Orphanet J Rare Dis 2007;2:42.  Back to cited text no. 4
    
5.
International Society for the Study of Vascular Anomalies. ISSVA Classification for vascular anomalies [Internet]. Approved at the 20th ISSVA workshop, Melbourne, April 2014, last revision May 2018. Available from: 130356_ISSVA-Classification-2018.pdf. [Last cited on 2019 Sep 01].  Back to cited text no. 5
    
6.
Steiner JE, Drolet BA. Classification of vascular anomalies: An update. Semin Intervent Radiol 2017;34:225-32.  Back to cited text no. 6
    
7.
Buckmiller LM, Richter GT, Suen JY. Diagnosis and management of hemangiomas and vascular malformations of the head and neck. Oral Dis 2010;16:405-18.  Back to cited text no. 7
    
8.
Kesavadas C, Joseph S, Gupta AK, Nair R, Rao VR, Mandalam R, et al. Craniofacial vascular malformation: Preoperative embolization. Indian J Plast Surg 2000;33:58-64.  Back to cited text no. 8
    
9.
Seccia A, Salgarello M, Farallo E, Falappa PG. Combined radiological and surgical treatment of arteriovenous malformations of the head and neck. Ann Plast Surg 1999;43:359-66.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14], [Figure 15], [Figure 16], [Figure 17], [Figure 18]
 
 
    Tables

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