- Email: [email protected]
Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review
G Model
JORMAS-709; No. of Pages 5 J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Review
Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review M. Varadan a, A. Chopra
a,
*, A.D. Sanghavi a, K. Sivaraman b, K. Gupta c
a
Department of Periodontology, Manipal College of Dental Sciences, Manipal Academy of Higher Education, Madhav Nagar, 576104 Manipal, Karnataka, India b Department of Prosthodontics, Manipal College of Dental Sciences, Manipal Academy of Higher Education, Manipal, Karnataka, India c New Delhi, India
A R T I C L E I N F O
A B S T R A C T
Article history: Received 2 April 2019 Accepted 18 June 2019
Ankyloglossia, also known as tongue-tie, is an embryological anatomical malformation of the tongue, characterized by an abnormally short and a thick lingual frenum. Tongue-tie restricts the physiologic movements of the tongue and results in various functional, behavioral and speech abnormalities along with the development of frontal and lateral lisps. Ankyloglossia in infants is also linked with the difficulty in breastfeeding difficulty, gagging, choking or vomiting food, delayed development or deterioration of speech and behavioral issues. A lingual frenectomy is a common oral surgical procedure done to correct an ankylosed lingual frenum by severing the abnormal frenal attachment on the ventral surface of the tongue. However, lingual frenectomy is associated with few complications that should be addressed to achieve a good overall prognosis. Though a lot of research is available on the various techniques and rationale to correct ankyloglossia, no paper has yet highlighted the surgical complications associated with lingual frenectomy. Therefore, the present paper for the first time review and highlight the common intraoperative and postoperative complications following lingual frenectomy.
C 2019 Elsevier Masson SAS. All rights reserved.
Keywords: Ankyloglossia Tongue-tie Lingual frenum Frenectomy Complications Surgery
1. Introduction Ankyloglossia (ankylos – curved/tied; glossa – tongue), also known as tongue-tie, is an embryological anatomical malformation of the tongue, characterized by an abnormally short and thick lingual frenum with restricted tongue movement. The lingual frenum is a membranous fold of mucosal tissue, which connects the ventral surface of the tongue to the floor of the mouth on one side and the basal bone of mandible on the other side [1]. In some individuals, the fibers of the frenum are attached to the tip of the tongue thereby restricting its physiologic movements. Ankyloglossia is commonly observed in infants with an incidence rate of about 5%. Individuals with tongue-tie are unable to protrude the tip of the tongue beyond the mandibular incisor region or touch the palate. Other functional movements like tongue lateralization, tongue spread, tongue cupping, and tongue snapback are also restricted in individuals with ankyloglossia [1–5]. Furthermore, ankyloglossia in infants and toddlers can cause functional
* Corresponding author. E-mail address: [email protected] (A. Chopra).
problems such as breastfeeding difficulty, gagging, choking or vomiting food, and behavioral issues [5]. A short and fibrotic lingual frenum is also associated with delayed development or deterioration of speech. The pronunciation of consonants like ‘t’, ‘d’, ‘n’, and ‘l’ in the development of frontal and lateral lisps are commonly observed in toddlers with tongue-tie [7]. Altered chewing and swallowing pattern in individuals with ankyloglossia even affects the coordination of jaw muscles during speech and result in the development of habits such as mouth breathing, aerophagia, and forward tongue positioning [6–9]. The risk of developing class III malocclusions along with reduced maxillary growth and mandibular prognathism is increased in children with ankyloglossia. Therefore, surgical correction of aberrant lingual frenal attachment is essential to overcome these mechanical limitations and functional challenges [10–13]. The most common method to correct ankyloglossia is by the surgical excision of aberrant frenal attachments by the process known as frenotomy, frenectomy or frenuloplasty [14]. A surgical incision with a scalpel, electrocautery or soft tissue lasers is commonly used for lingual frenectomy. Even though the process of lingual frenectomy is simple, the anatomical location and topography of the lingual tissue make it vulnerable to various
https://doi.org/10.1016/j.jormas.2019.06.003 C 2019 Elsevier Masson SAS. All rights reserved. 2468-7855/
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003
G Model
JORMAS-709; No. of Pages 5 2
M. Varadan et al. / J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
postoperative and intraoperative complications. Although a lot of literature is available on the methods and techniques to correct ankyloglossia, no paper has yet highlighted and discussed the complications particularly associated with lingual frenectomy. Thus, the present paper aims for the first time aim to highlight the common etiology associated with various intraoperative and postoperative complications with lingual frenectomy. The paper also discusses the precautions and appropriates measures to prevent and manage complications associated with lingual frenectomy. 2. Material and method 2.1. Search strategy The following search terms were used for data collection: ‘‘Lingual Frenectomy AND Complications’’ and ‘‘Ankyloglossia AND Complications’’. Articles which were written only in the English language in PubMed and Cochrane Library database from 1930 till January 2019 were scrutinized. The following inclusion and exclusion criteria’s were assessed for selection of the article: 2.2. Inclusion/exclusion criteria The inclusion criteria for selecting articles include the type of article (randomized clinical trial comprising of longitudinal study design, cohort study, case-control study, and cross-sectional study), sample size, the statistical and clinical significance of the outcome, randomization and blinding. All in vivo, in vitro, animals’ studies, narrative review, systematic reviews, and meta-analysis analyzing the postoperative effects of lingual frenectomy were included. Case reports and case series were not included. 2.3. Data collection Around 154 articles were screened for review. Only 20 articles fulfilled the above-mentioned inclusion and exclusion criteria were included for review. 3. Results The complications following lingual frenectomy can be classified as follows: complications that arise immediately to few hours following lingual frenectomy (intraoperative or immediate complications), and complications that arise few days to weeks following frenectomy (postoperative or delayed complications) (Table 1). 3.1. Some complications that arises due to lingual frenectomy perse includes 3.1.1. Reattachment or recurrence of frenal attachment, scar tissue formation and restriction in tongue movement The most common complication associated with lingual frenectomy is the recurrence of frenal attachment and restriction in tongue movement. The recurrence of tongue-tie after frenectomy is attributed to the development of fibrous scar tissue at the site of the excision. Development of swelling and postoperative edema in the sublingual region can also restrict the movement of the tongue for a few days following a frenectomy. Scar formation has been observed in 14% of adults and 2% of children and infants following a lingual frenectomy [15–17]. The fibrous scar develops due to inadequate releasing incisions and incomplete removal of frenal attachment at the time of the surgery. Therefore, it is important to evaluate the depth of the incision and completely
remove all the fibrous attachments without inducing any injury to the anatomical structures. If incisions are given too superficially and complete removal of fibrous attachment is not done, the chances of recurrence are high. Therefore, both the anterior and posterior submucosal component of the frenum should be included in the incision. Snipping the attachments only in the anterior frenum should be avoided as it can cause the frenal fibers to reattach. Therefore, the frenum should be correctly excised so as to form a diamond-shaped wound. The incision should not be too deep as deep surgical incisions with infected instruments and suture materials increase the risk of sublingual space infection and development of gaping wound following a frenectomy [15,16]. Furthermore, to prevent the development of postoperative scarring following frenectomy, one must judiciously select the most appropriate method and technique for excising the aberrant frenal attachment. Lasers can be used as a promising alternative to scalpel and electrocautery for frenectomy as the incidence of recurrence, postoperative pain, and fibrous scar formation is less when compared to scalpel technique. The laser treated wound has less wound contraction and scarring compared to scalpel wounds as there is less number of myofibroblasts in the wound [18]. If scar tissue develops following lingual frenectomy, surgical excision of the scar tissue along with complete removal of the aberrant fibers should be attempted. 3.1.2. Development of new speech disorder or worsening of existing speech disorder A restricted movement of the lingual frenum along with scar tissue formation following frenectomy are some of the common causes for the occurrence of new speech abnormalities or worsening of existing speech after lingual frenectomy. It has been observed that lingual frenectomy can exaggerate the existing speech difficulties, induce articulation problems and lisping, especially for sounds that require tongue elevation such as ‘s’, ‘z’, ‘t’, ‘d’, ‘l’, ‘r’ [19,11,20]. Although some individuals can move their tongue postoperatively for the production of sounds, many individuals need postoperative exercises for protrusion, elevation, retraction and lateral movements of the tongue [19]. Speech therapy with oral kinesthesia (ability to feel the part and how they are moving) and diadochokinesis (ability to perform rapid, alternating movements) exercises should be mandatory done following lingual frenectomy so as to achieve good postoperative outcomes [11,20–23]. Speech exercises are generally recommended after assessing the vegetative skills and degree of speech production. Factors such as age, mother tongue, and any other languages the person uses for communication, familiarity with words, vocabulary, and accent, causes of speech impairment, the degree of inflammation of the tongue, presence of scar tissue determine the nature and duration of speech therapy following a frenectomy [24–29]. The postoperative speech exercises following frenectomy will help to develop new muscle movements, particularly those involving tongue-tip elevation and protrusion, inside and outside of the mouth. The lingual alveolar (t, d, n) and fricative sounds (s and z), that require minimal tongue elevation and mobility, can be produced without any difficulty following a frenectomy. However, the articulation of lingual alveolar sound (i), lingual palatal sound (r) and lingual dental sound (the), where the tongue-tip needs to be elevated to reach the alveolar ridge require extensive speech therapy after lingual frenectomy. Therefore, it is important to encourage patients to make tongue movements related to cleaning the oral cavity, such as sweeping the insides of the cheeks, fronts, and backs of the teeth, and licking right around both lips following frenectomy to prevent postoperative scar tissue formation [26]. Other exercises such as inward rolling of the tongue, whistling, rotating the tongue in the clockwise and anticlockwise direction while holding the fluid can be done to
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003
G Model
JORMAS-709; No. of Pages 5 M. Varadan et al. / J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
3
Table 1 Postoperative complications of lingual frenectomy. Complications
Etiology
Reference
Excessive bleeding or hemorrhage Formation of retention cyst or ranula
Injury to the submental sublingual artery and deep lingual veins Partial blockage of Wharton’s duct while suturing or injury while delivering local anesthesia Extravasation of blood into the tissue spaces between the muscles of the tongue and floor of the mouth Surgically induced trauma and tongue bite during or after surgical procedure Microbial contamination of the site due to faulty and deep surgical incisions Systemically immuno-compromised patients with poor wound healing capacity Infected suture material Development of a fibrous scar at the site of the excision Inadequate releasing incisions and incomplete removal of frenal attachment at the time of lingual frenectomy Restricted movement of the lingual frenum following surgery Scar tissue formation Lisping in mixed dentition stage Direct and indirect injury to the lingual nerve during incision, pressure from healing wound and postsurgical edema Movement of the needle itself, extra or intraneural hemorrhage from trauma to nearby blood vessels or the neurotoxicity of the used anesthetic site with anesthetic agents can induce nerve injury following lingual frenectomy
[3–7,15–18,23] [16,27,29–35]
Sublingual hematoma formation
Development of sublingual and submandibular space infection
Reattachment or recurrence of frenal attachment
Development of new speech disorder or worsening of existing speech disorder Numbness and paresthesia of the tongue and neighboring soft tissues
increase its to increase the flexibility, strength and improve the speech. Movement of the tongue with an opposing force can also be used while performing these exercises in order to increase the strength of the muscles. The pressure can be applied by using a tongue depressor or by pressing the tongue against the cheek by using the thumb from outside [5,7]. Additionally, if lingual frenectomy is planned for children in the mixed dentition stage, there is a high risk of developing frontal lisp during the speech. Frontal lisp occurs as the tongue positions itself in the space left by the loss of primary teeth and partially erupting permanent teeth. Additionally, a sudden increase in the movement of the tongue following frenectomy and the development of a tongue thrust swallow increase the risk of developing a frontal lisp. The static and new resting position of the tongue may result in the development of anterior open-bite. Therefore, it is crucial that children in the mixed dentition stage are mandatorily monitored by a speech therapist/orofacial myologist following lingual frenectomy [43]. 3.2. The common complications that arise due to injury to the important anatomic landmarks associated with lingual frenum 3.2.1. Excessive bleeding/hemorrhage during or immediately after the surgery The main etiology of excessive intraoperative bleeding following frenectomy is because of accidental injury to the major or minor blood vessels (submental or sublingual artery) while excising the aberrant frenal attachment. Around 3–8% of episodes of bleeding are observed in clinical practice during or after frenectomy [3–5,7]. The injury to the anastomosis of the sublingual artery with the terminal capillaries of the inferior alveolar artery on the other side of the lingual frenum is most often injured during lingual frenectomy [23]. The accidental injury to the superficial and deep lingual veins (lingual varicosities) can also induce bleeding during the surgery. Therefore, a deep and long incision that extends beyond the tongue into the gingival or mucosal tissue on the lingual aspect of the anterior mandible should be avoided to prevent injury to the branches of the inferior alveolar canal and its anastomosing plexus [16,17]. The intraoperative bleeding during lingual frenectomy should be managed by first identifying the source and type of bleeding (arterial, capillary or venous). Primary
[36–38]
[22,34]
[15,18]
[7,19,29]
[37–43]
closure and hemostasis should be immediately attempted by applying a pressure pack with local hemostatic agents such as an absorbable collagen sponge, oxidized cellulose, hemocoagulase, topical thrombin, etc. If a medium or large sized blood vessel is severed, surgical sutures on the ventral surface of the tongue should be immediately placed. The deep lingual vessels should be tied with a stick-tie mode of ligation to achieve hemostasis [5–7]. It is also recommended to pre-suture the base of the tongue with interrupted sutures at the most coronal and apical extension of the frenum in order to reduce the risk of bleeding. A novel pre-suturing technique in which two to four simple interrupted sutures are positioned in a vertical manner extending from the tip of the tongue to the base of the frenum followed by 2–3 simple interrupted sutures are recommended to achieve superior hemostasis [28]. Adjuncts like electrocautery, chemical cauterization with silver nitrate packs, and lasers can also be used to control the bleeding [14,22,23]. 3.2.2. Formation of mucus retention cyst or ranula Formation of a mucus retention cyst or ranula is another common finding during or after lingual frenectomy [16,27,29– 32]. Mucus retention cyst usually presents as a, slightly transparent bluish asymptomatic or painful swelling at the floor of the mouth after lingual frenectomy [32]. The swelling may arise after a few hours or days after frenectomy and make last for a week until complete resolution occurs. The retention cyst may even develop as a consequence of the focal containment of mucoid material due to rupture or blockage of the salivary duct (Wharton’s duct). Injury to the salivary duct may occur if the frenectomy incisions at the base of the tongue are too deep. The onset of infection, trauma or blockage of the salivary duct during suturing or while delivering local anesthesia can also result in the formation of retention cyst or ranula. Therefore, it is important to carefully suture the base of the tongue and avoid any injury to the duct of the sublingual salivary gland. In case an intraoral ranula or retention cyst develops, it can be treated by surgical excision, marsupialization, laser ablation or cryosurgery. After marsupialization, placement of a silk suture or seton into the dome of the cyst, intracystic injection therapy with OK-432 (Picibanil), botulinum toxin can also be done [29–31]. In children and newborn, an observation period of five months for spontaneous resolution of ranula should be given after frenectomy
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003
G Model
JORMAS-709; No. of Pages 5 4
M. Varadan et al. / J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
[27] https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3540233. In the young age group with minimal symptoms, aspiration of lesions along with periodic follow-up is suggested as an alternative to surgery [26–35] https://www.ncbi.nlm.nih.gov/pmc/articles/ PMC3540233/. 3.2.3. Sublingual hematoma formation The uncontrolled bleeding during lingual frenectomy or frenotomy often results in the extravasation of blood into the tissue spaces between the muscles of the tongue and floor of the mouth that result in the formation of a sublingual hematoma [36–38]. Additionally, surgically induced trauma and tongue bite during or after lingual frenectomy increase the risk of hematoma formation [39]. Trauma induced by the anesthetic needle can also result in intraneural hematoma formation following lingual frenectomy. It is important to immediately recognize the formation of the hematoma and manage it immediately. Bimanual palpation in the downward direction from the floor of the mouth towards the lingual surface of the mandible and upward from the submental skin along with the compression of the lesion should be done to prevent its spread. The local extravasation should be controlled by the use of various hemostatic agents either with or without surgical drainage along with intravenous administration of antibiotic prophylaxis and steroids [33–35]. The incisional drainage is contraindicated after frenectomy as it can result in severe hemorrhage and limit the self-tamponading action by hematoma itself [32–38]. If the bleeding fails to cease and turns into a dissecting hematoma, the source of bleeding must be topographically located using an angiography, angio-magnetic resonance imaging, or computed tomography scan images with contrast medium [41]. Once the source of bleeding is identified, the artery should be clipped through a full thickness mandibular flap of the lingual mucosa surface. If the bleeding cannot be controlled by a direct approach, angiographic embolization or external ligation of the carotid artery should be attempted [32,33]. It is also important to observe that a patent and secure airway is maintained in case a sublingual hematoma develops during or after lingual frenectomy. Tracheostomy or intubation is not necessary if the patient can breathe autonomously with good oxygen saturation. If patients develop dyspnea and difficulty in breathing either during or immediately after the procedure, all procedure should be stopped and the patient should be hospitalized. Intubation should be attempted and airway should be secured by a Guedel pattern airway. If intubation is not possible because of the large size of hematoma or progressive nature of the swelling, an emergency tracheostomy should be done [35]. 3.2.4. Numbness and paresthesia of the tongue and neighboring soft tissues Numbness and paresthesia of tongue following frenectomy are attributed to injury to the lingual nerve during frenectomy [42– 45]. The location of the lingual nerve in the floor of the mouth and the ventral surface of the tongue makes the site more susceptible to injury during lingual frenectomy. Although injury to the lingual nerve is transient, sensory disturbances in the form of paresthesia (abnormal sensation), hypoesthesia (reduced sensation), or dysesthesia (unpleasant abnormal sensation) may develop following lingual frenectomy. Although, the sensory loss following frenectomy is usually temporary and may last for not more than six months, continuous monitoring by the clinician is important. Additionally, injury to the lingual nerve during frenectomy may impair the speech, alter mastication, restrict the movement of the tongue, and induce loss of taste sensation from the ipsilateral anterior segment of the tongue [37]. In some individuals drooling of saliva, tongue biting, burning sensation of the tongue and anesthesia resulting in burns during eating and drinking following lingual frenectomy due to lingual nerve injury have also been observed [37].
A direct injury to the lingual nerve commonly occurs during the surgical removal of the aberrant frenal attachments. Therefore, it is crucial for clinicians to have comprehensive knowledge about the anatomical distributions and innervations of the neurovascular structures and muscles at the base of the tongue [38]. Morphological and anatomical variations in the terminal branches of the lingual nerve make the anterior two third and tip of the tongue more susceptible to injury [39–42]. Two variations in the terminal branches of the lingual nerve that should be borne in mind while performing lingual frenectomy are: single primary nerve trunk and two primary nerve trunks (one medial, distributed in the middle third of the tongue, and another lateral, distributed in the anterior third of the tongue) [41–43]. The common anatomical pattern of the lingual nerve such as types 1b or 2b are most susceptible to injury during lingual frenectomy. Therefore, surgical excision during frenectomy should be conservative and well planned to avoid untoward injury to lingual mucosa and aberrant branches of the lingual nerve [43–47]. Injury to the lingual nerve can even occur due to the pressure from the needle during local anesthesia, pressure from the healing granulation tissues and fluid collected at the surgical site due to postsurgical edema. Other causes such as the movement of the needle, extraneural or intraneural hemorrhage, neurotoxicity of the anesthetic site with anesthetic agents (4% articaine and 3%–4% prilocaine) can injure the nerve following lingual frenectomy [45]. If any injury to the lingual nerve is suspected, it is important to assess the severity and degree of injury. Minor injury to the lingual nerve with mild numbness, anesthesia, and dysesthesia usually resolves spontaneously with time and does not require any intervention. The minor lingual nerve injury should be managed conservatively, non-surgically and symptomatically. Vitamin B-complex administration can be prescribed to enhance recovery and fasten the healing of the injured nerve [48]. Low-level laser therapy can be used as an adjunct to conventional therapy to reduce the pain and edema following a frenectomy procedure. A meticulous evaluation of the degree of nerve injury by performing taste differential tests, twopoint discrimination tests, and mapping of the affected dermatomes for areas of sensational change with regular follow-up visits should be done if severe damage of the lingual nerve is suspected. Early surgical reconstruction and/or intervention should be attempted if the anesthesia has not resolved in 10–12 weeks after the surgical procedure. 3.2.5. Development of space infection The development of space infection following lingual frenectomy often manifests as a firm, painful, swelling at the floor of the mouth. The patient may also experience mild edema, the elevation of the tongue, drooling of saliva, difficulty in swallowing, fever, and dyspnea. In some individuals, a long-standing, untreated space infection may even result in abscess formation below the base of the tongue and cause severe airway obstruction [22]. An untreated sublingual space infection, if left untreated can develop into a severely life-threatening situation such as Ludwig’s angina. Therefore it is important to prevent the spread of infection by using sterile and non-infected instruments and suture material during the surgical procedure. In case the infection has spread into the sublingual space, postoperative antibiotics either orally or systemically along with warm saline gargles should be prescribed. A stab incision lateral to the sublingual plica is usually given to drain the pus and prevent its spread [34].
4. Conclusion Although lingual frenectomy is a common and technique sensitive surgical procedure, the anatomic and topographic lingual
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003
G Model
JORMAS-709; No. of Pages 5 M. Varadan et al. / J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
frenum makes the site vulnerable to various intraoperative and postoperative complications. Therefore, comprehensive knowledge and understanding of the various etiology of complications associated with lingual frenectomy are of paramount importance to provide optimal postoperative care and achieve good clinical outcomes and overall patient satisfaction. Disclosure of interest The authors declare that they have no competing interest. References [1] Johnson RV. Tongue-tie—exploding the myths. Infant 2006;2:96–9. [2] Hong P, Lago D, Seargeant J, Pellman L, Magit AE, Pransky SM. Defining ankyloglossia: a case series of anterior and posterior tongue-ties. Int J Pediatr Otorhinolaryngol 2001;74:1003–6. [3] Ballard J, Chantry C, Howard CR. Protocol Committee Academy of Breastfeeding Medicine0 Clinical Protocol Number 11: Mastitis. www.bfmed.org. Acad Breastfeed Med 2004 [https://abm.memberclicks.net/assets/DOCUMENTS/ PROTOCOLS/11-neonatal-ankyloglossia-protocol-english.pdf]. [4] Kotlow LA. Ankyloglossia (tongue-tie): a diagnostic and treatment quandary. Quintessence Int 1999;30:259–62. [5] Olivi G, Signore A, Olivi M, Genovese MD. Lingual frenectomy: functional evaluation and new therapeutical approach. Eur J Pediatr Dent 2012;13:101–6. [6] Kloars T. Familial ankyloglossia (tongue-tie). Int J Pediatr Otorhinolaryng 2007;71:1321–4. [7] Queiroz Marchesan I. Lingual frenulum: classification and speech interference. Int J Orofacial Myology 2004;30:31–8. [8] Bhattad MS, Baliga MS, Kriplani R. Clinical guidelines and management of ankyloglossia with 1year follow-up: report of 3 cases. Case Rep Dent 2013;2013:185803. http://dx.doi.org/10.1155/2013/185803 [Epub 2013 Jan 29]. [9] Tuli A, Singh A. Monopolar diathermy used for correction of ankyloglossia. J Ind Soc Pedodont Prev Dent 2010;28:130. [10] Defabianis P. Ankyloglossia and its influence on maxillary and mandibular development. (A seven-year follow-up case report). Funct Orthod 2000;17(4):25–33. [11] Marchesan I. Lingual Frenulum: classification and speech interference. IJOM 2004;30:31–8. [12] Yang HM, Woo YJ, Won SY, Kim DH, Hu KS, Kim HJ. Course and distribution of the lingual nerve in the ventral tongue region: anatomical considerations for frenectomy. J Craniofac Surg 2009;20(5):1359–63. [13] Campan P, Baron P, Duran D, Casteigt J. Lingual frenectomy: a therapeutic protocol. A technic for frenectomy with 2 incision lines combined with active postoperative kinesitherapy during and after healing. Schweiz Monatsschr Zahnmed 1996;106:45–54. [14] Suter VGA, Bornstein MM. Ankyloglossia: facts and myths in diagnosis and treatment. J Periodontol 2009;80:1204–19. [15] Messner AH, Lalakea ML. Ankyloglossia: controversies in management. Int J Pediatr Otorhinolaryngol 2000;54:123–31. [16] Klepacek I, Skulec R. Relation between lingual nerve, submandibular gland duct and mandibular body in the sublingual space. Acta Chir Plast 1993;36(1):26–7. [17] Rosano G, Taschieri S, Testori T, Del Fabbro M, El Haddioui A, Gaudy JF. Evaluation of anterior mandible anatomy in oral surgery. Ital Oral Surg 2008;7:7–14. [18] Zeinoun T, Nammour S, Dourov N, Aftimos G, Luomanen M. Myofibroblasts in healing laser excision wounds. Lasers Surg Med 2001;28:74–9. [19] Haytac MC, Ozcelik O. Evaluation of patient perceptions after frenectomy operations: a comparison of carbon dioxide laser and scalpel techniques. J Periodontol 2006;77:1815–9. [20] Martinelli RL, Marchesan IQ, Berretin-Felix G. Lingual frenulum protocol with scores for infants. Int J Orofacial Myology 2012;38:104–12.
5
[21] Manish A, Mala Dixit B, Lashika T, Shraddha K. Ankyloglossia – a review and case report. Euro J Pharmaceutical Med Res 2018;5(3):426–30. [22] Darshan HE, Pavithra PM. Tongue-tie: from confusion to clarity – a review. Int J Dent Clin 2011;3(1):48–51. [23] Shadab K, Shweta S, Vivek KS. Ankyloglossia: surgical management and functional rehabilitation of tongue. Ind J Dent Res 2017;28(5):585–7. [24] Seo YJ, Kim SJ, Munkhshur J, Chung KR, Ngan P, Kim SH. Treatment and retention of relapsed anterior open-bite with low tongue posture and tongue-tie: a 10-year follow-up. Korean J Orthod 2014;44(4):203–16. [25] Pick RM, Picaro BC, Silberman CJ. The laser gingivectomy: the use of CO2 laser for the removal of phenytoin hyperplasia. J Periodontol 1985;56:492–6. [26] Tanya D, Lele DP, Patil DP. Ankyloglossia: treatment with surgical lasers – a case report. IOSR J Dent Med Sci 2014;13:61–4. [27] Haberal I, Gocmen H, Samim E. Surgical management of pediatric ranula. Int J Pediatr Otorhinolaryngol 2004;68:161–3. [28] Khairnar M, Pawar B, Khairnar D. A novel surgical pre-suturing technique for the management of ankyloglossia. J Surg Tech Case Rep 2014;6(2):49–54. http://dx.doi.org/10.4103/2006-8808.147259. [29] Baurmash HD. Marsupialization for treatment of oral ranula a second look at the procedure. J Oral Maxillofac Surg 1992;50:1274–9. [30] Watanabe K, Tomiyama S, Jinnouchi K, Nakajima H, Yagi T. Local injection of OK-432 in the treatment of ranula: a case report. ENT J 2002;81:97–8. [31] Chow TL, Chan SW, Lam SH. Ranula successfully treated by botulinum toxin type A: report of 3 cases. Oral Surg Oral Med Oral Path Oral Radiol Endo 2008;105:41–2. [32] Stokes S, Gowrishankar S, Anand P. Mucus extravasation cyst as a complication of lingual frenectomy. Br J Oral Maxillofac Surg 2017;55:e86–189. [33] Pandit RT, Park AH. Management of pediatric ranula. Otolaryngol Head Neck Surg 2002;127:115–8. [34] Mihir RP, Allison MD, William WS. Oral and plunging ranulas: what is the most effective treatment? Laryngoscope 2009;119:1501–9. [35] Zhi K, Wen Y, Ren W, Zhang Y. Management of infant ranula. Int J Pediatr Otorhinolaryn 2008;72:823–6. [36] Hing NR, Bowler MD, Byth PL, Daly CG. Lingual hematoma leading to upper airway obstruction. Br J Oral Maxillofac Surg 1985;23:322–5. [37] Renehan A, Morton M. Acute enlargement of the tongue. Br J Oral Maxillofac Surg 1993;31:321–4. ˜ o-Garcı´a M. Hematoma of the floor of the mouth [38] Vera JD, Calleja JM, Burguen and airway obstruction during mandibular dental implant placement: a case report. Oral Maxillofac Surg 2008;12:223–36. [39] Loukas M, Kinsella CR, Kapos T, Tubbs RS, Ramachandra S. Anatomical variation in the arterial supply of the mandible with special regard to implant placement. Intern J Oral Maxillofac Surg 2008;37:367–71. [40] Smith MH, Lung KE. Nerve injuries after dental injection: a review of the literature. J Can Dent Assoc 2006;72:559–64. [41] Hwang HD, Kim JW, Kim YS, Kang DH, Kwon TJ. Angiographic embolization for hemorrhage control after dental implantation. J Korean Assoc Oral and Maxillofac Surgeon 2013;39:27–30. [42] Takei H, Caranza F. The periodontal flap. In: Newman M, Takei H, Klokkevold P, editors. Caranza’s clinical periodontology, ed. 10. Philadelphia: Saunder; 2009. p. 926–36. [43] Tan VL, Andrawos A, Ghabriel MN, Townsend GC. Applied anatomy of the lingual nerve: relevance to dental anaesthesia. Arch Oral Biol 2014;59:324–35. [44] Garlsto GA, Gaffen AS, Lawrence HP, Tenenbaum HC, Haas DA. Occurrence of paresthesia after dental local anesthetic administration in the United States. J Am Dent Assoc 2010;141:836–44. [45] Fielding AF, Rachiele DP, Frazier G. Lingual nerve paresthesia following third molar surgery. A retrospective clinical study. Oral Surg Oral Med Oral Path Oral Radiol Endodont 1997;84:345–8. [46] Piagkou M, Demesticha T, Skandalakis P, Johnson EO. Functional anatomy of the mandibular nerve: consequences of nerve injury and entrapment. Clin Anat 2011;24:143–50. [47] Rusu MC, Nimigean V, Podoleanu L, Ivas¸cu RV, Niculescu MC. Details of the intralingual topography and morphology of the lingual nerve. Int J Oral Maxillofac Surg 2008;37:835–9. [48] Sun H, Yang T, Li Q, et al. Dexamethasone and vitamin B12 synergistically promote peripheral nerve regeneration in rats by upregulating the expression of brain-derived neurotrophic factor. Arch Med Sci 2012;8:924–30.
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003
JORMAS-709; No. of Pages 5 J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Review
Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review M. Varadan a, A. Chopra
a,
*, A.D. Sanghavi a, K. Sivaraman b, K. Gupta c
a
Department of Periodontology, Manipal College of Dental Sciences, Manipal Academy of Higher Education, Madhav Nagar, 576104 Manipal, Karnataka, India b Department of Prosthodontics, Manipal College of Dental Sciences, Manipal Academy of Higher Education, Manipal, Karnataka, India c New Delhi, India
A R T I C L E I N F O
A B S T R A C T
Article history: Received 2 April 2019 Accepted 18 June 2019
Ankyloglossia, also known as tongue-tie, is an embryological anatomical malformation of the tongue, characterized by an abnormally short and a thick lingual frenum. Tongue-tie restricts the physiologic movements of the tongue and results in various functional, behavioral and speech abnormalities along with the development of frontal and lateral lisps. Ankyloglossia in infants is also linked with the difficulty in breastfeeding difficulty, gagging, choking or vomiting food, delayed development or deterioration of speech and behavioral issues. A lingual frenectomy is a common oral surgical procedure done to correct an ankylosed lingual frenum by severing the abnormal frenal attachment on the ventral surface of the tongue. However, lingual frenectomy is associated with few complications that should be addressed to achieve a good overall prognosis. Though a lot of research is available on the various techniques and rationale to correct ankyloglossia, no paper has yet highlighted the surgical complications associated with lingual frenectomy. Therefore, the present paper for the first time review and highlight the common intraoperative and postoperative complications following lingual frenectomy.
C 2019 Elsevier Masson SAS. All rights reserved.
Keywords: Ankyloglossia Tongue-tie Lingual frenum Frenectomy Complications Surgery
1. Introduction Ankyloglossia (ankylos – curved/tied; glossa – tongue), also known as tongue-tie, is an embryological anatomical malformation of the tongue, characterized by an abnormally short and thick lingual frenum with restricted tongue movement. The lingual frenum is a membranous fold of mucosal tissue, which connects the ventral surface of the tongue to the floor of the mouth on one side and the basal bone of mandible on the other side [1]. In some individuals, the fibers of the frenum are attached to the tip of the tongue thereby restricting its physiologic movements. Ankyloglossia is commonly observed in infants with an incidence rate of about 5%. Individuals with tongue-tie are unable to protrude the tip of the tongue beyond the mandibular incisor region or touch the palate. Other functional movements like tongue lateralization, tongue spread, tongue cupping, and tongue snapback are also restricted in individuals with ankyloglossia [1–5]. Furthermore, ankyloglossia in infants and toddlers can cause functional
* Corresponding author. E-mail address: [email protected] (A. Chopra).
problems such as breastfeeding difficulty, gagging, choking or vomiting food, and behavioral issues [5]. A short and fibrotic lingual frenum is also associated with delayed development or deterioration of speech. The pronunciation of consonants like ‘t’, ‘d’, ‘n’, and ‘l’ in the development of frontal and lateral lisps are commonly observed in toddlers with tongue-tie [7]. Altered chewing and swallowing pattern in individuals with ankyloglossia even affects the coordination of jaw muscles during speech and result in the development of habits such as mouth breathing, aerophagia, and forward tongue positioning [6–9]. The risk of developing class III malocclusions along with reduced maxillary growth and mandibular prognathism is increased in children with ankyloglossia. Therefore, surgical correction of aberrant lingual frenal attachment is essential to overcome these mechanical limitations and functional challenges [10–13]. The most common method to correct ankyloglossia is by the surgical excision of aberrant frenal attachments by the process known as frenotomy, frenectomy or frenuloplasty [14]. A surgical incision with a scalpel, electrocautery or soft tissue lasers is commonly used for lingual frenectomy. Even though the process of lingual frenectomy is simple, the anatomical location and topography of the lingual tissue make it vulnerable to various
https://doi.org/10.1016/j.jormas.2019.06.003 C 2019 Elsevier Masson SAS. All rights reserved. 2468-7855/
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003
G Model
JORMAS-709; No. of Pages 5 2
M. Varadan et al. / J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
postoperative and intraoperative complications. Although a lot of literature is available on the methods and techniques to correct ankyloglossia, no paper has yet highlighted and discussed the complications particularly associated with lingual frenectomy. Thus, the present paper aims for the first time aim to highlight the common etiology associated with various intraoperative and postoperative complications with lingual frenectomy. The paper also discusses the precautions and appropriates measures to prevent and manage complications associated with lingual frenectomy. 2. Material and method 2.1. Search strategy The following search terms were used for data collection: ‘‘Lingual Frenectomy AND Complications’’ and ‘‘Ankyloglossia AND Complications’’. Articles which were written only in the English language in PubMed and Cochrane Library database from 1930 till January 2019 were scrutinized. The following inclusion and exclusion criteria’s were assessed for selection of the article: 2.2. Inclusion/exclusion criteria The inclusion criteria for selecting articles include the type of article (randomized clinical trial comprising of longitudinal study design, cohort study, case-control study, and cross-sectional study), sample size, the statistical and clinical significance of the outcome, randomization and blinding. All in vivo, in vitro, animals’ studies, narrative review, systematic reviews, and meta-analysis analyzing the postoperative effects of lingual frenectomy were included. Case reports and case series were not included. 2.3. Data collection Around 154 articles were screened for review. Only 20 articles fulfilled the above-mentioned inclusion and exclusion criteria were included for review. 3. Results The complications following lingual frenectomy can be classified as follows: complications that arise immediately to few hours following lingual frenectomy (intraoperative or immediate complications), and complications that arise few days to weeks following frenectomy (postoperative or delayed complications) (Table 1). 3.1. Some complications that arises due to lingual frenectomy perse includes 3.1.1. Reattachment or recurrence of frenal attachment, scar tissue formation and restriction in tongue movement The most common complication associated with lingual frenectomy is the recurrence of frenal attachment and restriction in tongue movement. The recurrence of tongue-tie after frenectomy is attributed to the development of fibrous scar tissue at the site of the excision. Development of swelling and postoperative edema in the sublingual region can also restrict the movement of the tongue for a few days following a frenectomy. Scar formation has been observed in 14% of adults and 2% of children and infants following a lingual frenectomy [15–17]. The fibrous scar develops due to inadequate releasing incisions and incomplete removal of frenal attachment at the time of the surgery. Therefore, it is important to evaluate the depth of the incision and completely
remove all the fibrous attachments without inducing any injury to the anatomical structures. If incisions are given too superficially and complete removal of fibrous attachment is not done, the chances of recurrence are high. Therefore, both the anterior and posterior submucosal component of the frenum should be included in the incision. Snipping the attachments only in the anterior frenum should be avoided as it can cause the frenal fibers to reattach. Therefore, the frenum should be correctly excised so as to form a diamond-shaped wound. The incision should not be too deep as deep surgical incisions with infected instruments and suture materials increase the risk of sublingual space infection and development of gaping wound following a frenectomy [15,16]. Furthermore, to prevent the development of postoperative scarring following frenectomy, one must judiciously select the most appropriate method and technique for excising the aberrant frenal attachment. Lasers can be used as a promising alternative to scalpel and electrocautery for frenectomy as the incidence of recurrence, postoperative pain, and fibrous scar formation is less when compared to scalpel technique. The laser treated wound has less wound contraction and scarring compared to scalpel wounds as there is less number of myofibroblasts in the wound [18]. If scar tissue develops following lingual frenectomy, surgical excision of the scar tissue along with complete removal of the aberrant fibers should be attempted. 3.1.2. Development of new speech disorder or worsening of existing speech disorder A restricted movement of the lingual frenum along with scar tissue formation following frenectomy are some of the common causes for the occurrence of new speech abnormalities or worsening of existing speech after lingual frenectomy. It has been observed that lingual frenectomy can exaggerate the existing speech difficulties, induce articulation problems and lisping, especially for sounds that require tongue elevation such as ‘s’, ‘z’, ‘t’, ‘d’, ‘l’, ‘r’ [19,11,20]. Although some individuals can move their tongue postoperatively for the production of sounds, many individuals need postoperative exercises for protrusion, elevation, retraction and lateral movements of the tongue [19]. Speech therapy with oral kinesthesia (ability to feel the part and how they are moving) and diadochokinesis (ability to perform rapid, alternating movements) exercises should be mandatory done following lingual frenectomy so as to achieve good postoperative outcomes [11,20–23]. Speech exercises are generally recommended after assessing the vegetative skills and degree of speech production. Factors such as age, mother tongue, and any other languages the person uses for communication, familiarity with words, vocabulary, and accent, causes of speech impairment, the degree of inflammation of the tongue, presence of scar tissue determine the nature and duration of speech therapy following a frenectomy [24–29]. The postoperative speech exercises following frenectomy will help to develop new muscle movements, particularly those involving tongue-tip elevation and protrusion, inside and outside of the mouth. The lingual alveolar (t, d, n) and fricative sounds (s and z), that require minimal tongue elevation and mobility, can be produced without any difficulty following a frenectomy. However, the articulation of lingual alveolar sound (i), lingual palatal sound (r) and lingual dental sound (the), where the tongue-tip needs to be elevated to reach the alveolar ridge require extensive speech therapy after lingual frenectomy. Therefore, it is important to encourage patients to make tongue movements related to cleaning the oral cavity, such as sweeping the insides of the cheeks, fronts, and backs of the teeth, and licking right around both lips following frenectomy to prevent postoperative scar tissue formation [26]. Other exercises such as inward rolling of the tongue, whistling, rotating the tongue in the clockwise and anticlockwise direction while holding the fluid can be done to
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003
G Model
JORMAS-709; No. of Pages 5 M. Varadan et al. / J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
3
Table 1 Postoperative complications of lingual frenectomy. Complications
Etiology
Reference
Excessive bleeding or hemorrhage Formation of retention cyst or ranula
Injury to the submental sublingual artery and deep lingual veins Partial blockage of Wharton’s duct while suturing or injury while delivering local anesthesia Extravasation of blood into the tissue spaces between the muscles of the tongue and floor of the mouth Surgically induced trauma and tongue bite during or after surgical procedure Microbial contamination of the site due to faulty and deep surgical incisions Systemically immuno-compromised patients with poor wound healing capacity Infected suture material Development of a fibrous scar at the site of the excision Inadequate releasing incisions and incomplete removal of frenal attachment at the time of lingual frenectomy Restricted movement of the lingual frenum following surgery Scar tissue formation Lisping in mixed dentition stage Direct and indirect injury to the lingual nerve during incision, pressure from healing wound and postsurgical edema Movement of the needle itself, extra or intraneural hemorrhage from trauma to nearby blood vessels or the neurotoxicity of the used anesthetic site with anesthetic agents can induce nerve injury following lingual frenectomy
[3–7,15–18,23] [16,27,29–35]
Sublingual hematoma formation
Development of sublingual and submandibular space infection
Reattachment or recurrence of frenal attachment
Development of new speech disorder or worsening of existing speech disorder Numbness and paresthesia of the tongue and neighboring soft tissues
increase its to increase the flexibility, strength and improve the speech. Movement of the tongue with an opposing force can also be used while performing these exercises in order to increase the strength of the muscles. The pressure can be applied by using a tongue depressor or by pressing the tongue against the cheek by using the thumb from outside [5,7]. Additionally, if lingual frenectomy is planned for children in the mixed dentition stage, there is a high risk of developing frontal lisp during the speech. Frontal lisp occurs as the tongue positions itself in the space left by the loss of primary teeth and partially erupting permanent teeth. Additionally, a sudden increase in the movement of the tongue following frenectomy and the development of a tongue thrust swallow increase the risk of developing a frontal lisp. The static and new resting position of the tongue may result in the development of anterior open-bite. Therefore, it is crucial that children in the mixed dentition stage are mandatorily monitored by a speech therapist/orofacial myologist following lingual frenectomy [43]. 3.2. The common complications that arise due to injury to the important anatomic landmarks associated with lingual frenum 3.2.1. Excessive bleeding/hemorrhage during or immediately after the surgery The main etiology of excessive intraoperative bleeding following frenectomy is because of accidental injury to the major or minor blood vessels (submental or sublingual artery) while excising the aberrant frenal attachment. Around 3–8% of episodes of bleeding are observed in clinical practice during or after frenectomy [3–5,7]. The injury to the anastomosis of the sublingual artery with the terminal capillaries of the inferior alveolar artery on the other side of the lingual frenum is most often injured during lingual frenectomy [23]. The accidental injury to the superficial and deep lingual veins (lingual varicosities) can also induce bleeding during the surgery. Therefore, a deep and long incision that extends beyond the tongue into the gingival or mucosal tissue on the lingual aspect of the anterior mandible should be avoided to prevent injury to the branches of the inferior alveolar canal and its anastomosing plexus [16,17]. The intraoperative bleeding during lingual frenectomy should be managed by first identifying the source and type of bleeding (arterial, capillary or venous). Primary
[36–38]
[22,34]
[15,18]
[7,19,29]
[37–43]
closure and hemostasis should be immediately attempted by applying a pressure pack with local hemostatic agents such as an absorbable collagen sponge, oxidized cellulose, hemocoagulase, topical thrombin, etc. If a medium or large sized blood vessel is severed, surgical sutures on the ventral surface of the tongue should be immediately placed. The deep lingual vessels should be tied with a stick-tie mode of ligation to achieve hemostasis [5–7]. It is also recommended to pre-suture the base of the tongue with interrupted sutures at the most coronal and apical extension of the frenum in order to reduce the risk of bleeding. A novel pre-suturing technique in which two to four simple interrupted sutures are positioned in a vertical manner extending from the tip of the tongue to the base of the frenum followed by 2–3 simple interrupted sutures are recommended to achieve superior hemostasis [28]. Adjuncts like electrocautery, chemical cauterization with silver nitrate packs, and lasers can also be used to control the bleeding [14,22,23]. 3.2.2. Formation of mucus retention cyst or ranula Formation of a mucus retention cyst or ranula is another common finding during or after lingual frenectomy [16,27,29– 32]. Mucus retention cyst usually presents as a, slightly transparent bluish asymptomatic or painful swelling at the floor of the mouth after lingual frenectomy [32]. The swelling may arise after a few hours or days after frenectomy and make last for a week until complete resolution occurs. The retention cyst may even develop as a consequence of the focal containment of mucoid material due to rupture or blockage of the salivary duct (Wharton’s duct). Injury to the salivary duct may occur if the frenectomy incisions at the base of the tongue are too deep. The onset of infection, trauma or blockage of the salivary duct during suturing or while delivering local anesthesia can also result in the formation of retention cyst or ranula. Therefore, it is important to carefully suture the base of the tongue and avoid any injury to the duct of the sublingual salivary gland. In case an intraoral ranula or retention cyst develops, it can be treated by surgical excision, marsupialization, laser ablation or cryosurgery. After marsupialization, placement of a silk suture or seton into the dome of the cyst, intracystic injection therapy with OK-432 (Picibanil), botulinum toxin can also be done [29–31]. In children and newborn, an observation period of five months for spontaneous resolution of ranula should be given after frenectomy
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003
G Model
JORMAS-709; No. of Pages 5 4
M. Varadan et al. / J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
[27] https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3540233. In the young age group with minimal symptoms, aspiration of lesions along with periodic follow-up is suggested as an alternative to surgery [26–35] https://www.ncbi.nlm.nih.gov/pmc/articles/ PMC3540233/. 3.2.3. Sublingual hematoma formation The uncontrolled bleeding during lingual frenectomy or frenotomy often results in the extravasation of blood into the tissue spaces between the muscles of the tongue and floor of the mouth that result in the formation of a sublingual hematoma [36–38]. Additionally, surgically induced trauma and tongue bite during or after lingual frenectomy increase the risk of hematoma formation [39]. Trauma induced by the anesthetic needle can also result in intraneural hematoma formation following lingual frenectomy. It is important to immediately recognize the formation of the hematoma and manage it immediately. Bimanual palpation in the downward direction from the floor of the mouth towards the lingual surface of the mandible and upward from the submental skin along with the compression of the lesion should be done to prevent its spread. The local extravasation should be controlled by the use of various hemostatic agents either with or without surgical drainage along with intravenous administration of antibiotic prophylaxis and steroids [33–35]. The incisional drainage is contraindicated after frenectomy as it can result in severe hemorrhage and limit the self-tamponading action by hematoma itself [32–38]. If the bleeding fails to cease and turns into a dissecting hematoma, the source of bleeding must be topographically located using an angiography, angio-magnetic resonance imaging, or computed tomography scan images with contrast medium [41]. Once the source of bleeding is identified, the artery should be clipped through a full thickness mandibular flap of the lingual mucosa surface. If the bleeding cannot be controlled by a direct approach, angiographic embolization or external ligation of the carotid artery should be attempted [32,33]. It is also important to observe that a patent and secure airway is maintained in case a sublingual hematoma develops during or after lingual frenectomy. Tracheostomy or intubation is not necessary if the patient can breathe autonomously with good oxygen saturation. If patients develop dyspnea and difficulty in breathing either during or immediately after the procedure, all procedure should be stopped and the patient should be hospitalized. Intubation should be attempted and airway should be secured by a Guedel pattern airway. If intubation is not possible because of the large size of hematoma or progressive nature of the swelling, an emergency tracheostomy should be done [35]. 3.2.4. Numbness and paresthesia of the tongue and neighboring soft tissues Numbness and paresthesia of tongue following frenectomy are attributed to injury to the lingual nerve during frenectomy [42– 45]. The location of the lingual nerve in the floor of the mouth and the ventral surface of the tongue makes the site more susceptible to injury during lingual frenectomy. Although injury to the lingual nerve is transient, sensory disturbances in the form of paresthesia (abnormal sensation), hypoesthesia (reduced sensation), or dysesthesia (unpleasant abnormal sensation) may develop following lingual frenectomy. Although, the sensory loss following frenectomy is usually temporary and may last for not more than six months, continuous monitoring by the clinician is important. Additionally, injury to the lingual nerve during frenectomy may impair the speech, alter mastication, restrict the movement of the tongue, and induce loss of taste sensation from the ipsilateral anterior segment of the tongue [37]. In some individuals drooling of saliva, tongue biting, burning sensation of the tongue and anesthesia resulting in burns during eating and drinking following lingual frenectomy due to lingual nerve injury have also been observed [37].
A direct injury to the lingual nerve commonly occurs during the surgical removal of the aberrant frenal attachments. Therefore, it is crucial for clinicians to have comprehensive knowledge about the anatomical distributions and innervations of the neurovascular structures and muscles at the base of the tongue [38]. Morphological and anatomical variations in the terminal branches of the lingual nerve make the anterior two third and tip of the tongue more susceptible to injury [39–42]. Two variations in the terminal branches of the lingual nerve that should be borne in mind while performing lingual frenectomy are: single primary nerve trunk and two primary nerve trunks (one medial, distributed in the middle third of the tongue, and another lateral, distributed in the anterior third of the tongue) [41–43]. The common anatomical pattern of the lingual nerve such as types 1b or 2b are most susceptible to injury during lingual frenectomy. Therefore, surgical excision during frenectomy should be conservative and well planned to avoid untoward injury to lingual mucosa and aberrant branches of the lingual nerve [43–47]. Injury to the lingual nerve can even occur due to the pressure from the needle during local anesthesia, pressure from the healing granulation tissues and fluid collected at the surgical site due to postsurgical edema. Other causes such as the movement of the needle, extraneural or intraneural hemorrhage, neurotoxicity of the anesthetic site with anesthetic agents (4% articaine and 3%–4% prilocaine) can injure the nerve following lingual frenectomy [45]. If any injury to the lingual nerve is suspected, it is important to assess the severity and degree of injury. Minor injury to the lingual nerve with mild numbness, anesthesia, and dysesthesia usually resolves spontaneously with time and does not require any intervention. The minor lingual nerve injury should be managed conservatively, non-surgically and symptomatically. Vitamin B-complex administration can be prescribed to enhance recovery and fasten the healing of the injured nerve [48]. Low-level laser therapy can be used as an adjunct to conventional therapy to reduce the pain and edema following a frenectomy procedure. A meticulous evaluation of the degree of nerve injury by performing taste differential tests, twopoint discrimination tests, and mapping of the affected dermatomes for areas of sensational change with regular follow-up visits should be done if severe damage of the lingual nerve is suspected. Early surgical reconstruction and/or intervention should be attempted if the anesthesia has not resolved in 10–12 weeks after the surgical procedure. 3.2.5. Development of space infection The development of space infection following lingual frenectomy often manifests as a firm, painful, swelling at the floor of the mouth. The patient may also experience mild edema, the elevation of the tongue, drooling of saliva, difficulty in swallowing, fever, and dyspnea. In some individuals, a long-standing, untreated space infection may even result in abscess formation below the base of the tongue and cause severe airway obstruction [22]. An untreated sublingual space infection, if left untreated can develop into a severely life-threatening situation such as Ludwig’s angina. Therefore it is important to prevent the spread of infection by using sterile and non-infected instruments and suture material during the surgical procedure. In case the infection has spread into the sublingual space, postoperative antibiotics either orally or systemically along with warm saline gargles should be prescribed. A stab incision lateral to the sublingual plica is usually given to drain the pus and prevent its spread [34].
4. Conclusion Although lingual frenectomy is a common and technique sensitive surgical procedure, the anatomic and topographic lingual
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003
G Model
JORMAS-709; No. of Pages 5 M. Varadan et al. / J Stomatol Oral Maxillofac Surg xxx (2019) xxx–xxx
frenum makes the site vulnerable to various intraoperative and postoperative complications. Therefore, comprehensive knowledge and understanding of the various etiology of complications associated with lingual frenectomy are of paramount importance to provide optimal postoperative care and achieve good clinical outcomes and overall patient satisfaction. Disclosure of interest The authors declare that they have no competing interest. References [1] Johnson RV. Tongue-tie—exploding the myths. Infant 2006;2:96–9. [2] Hong P, Lago D, Seargeant J, Pellman L, Magit AE, Pransky SM. Defining ankyloglossia: a case series of anterior and posterior tongue-ties. Int J Pediatr Otorhinolaryngol 2001;74:1003–6. [3] Ballard J, Chantry C, Howard CR. Protocol Committee Academy of Breastfeeding Medicine0 Clinical Protocol Number 11: Mastitis. www.bfmed.org. Acad Breastfeed Med 2004 [https://abm.memberclicks.net/assets/DOCUMENTS/ PROTOCOLS/11-neonatal-ankyloglossia-protocol-english.pdf]. [4] Kotlow LA. Ankyloglossia (tongue-tie): a diagnostic and treatment quandary. Quintessence Int 1999;30:259–62. [5] Olivi G, Signore A, Olivi M, Genovese MD. Lingual frenectomy: functional evaluation and new therapeutical approach. Eur J Pediatr Dent 2012;13:101–6. [6] Kloars T. Familial ankyloglossia (tongue-tie). Int J Pediatr Otorhinolaryng 2007;71:1321–4. [7] Queiroz Marchesan I. Lingual frenulum: classification and speech interference. Int J Orofacial Myology 2004;30:31–8. [8] Bhattad MS, Baliga MS, Kriplani R. Clinical guidelines and management of ankyloglossia with 1year follow-up: report of 3 cases. Case Rep Dent 2013;2013:185803. http://dx.doi.org/10.1155/2013/185803 [Epub 2013 Jan 29]. [9] Tuli A, Singh A. Monopolar diathermy used for correction of ankyloglossia. J Ind Soc Pedodont Prev Dent 2010;28:130. [10] Defabianis P. Ankyloglossia and its influence on maxillary and mandibular development. (A seven-year follow-up case report). Funct Orthod 2000;17(4):25–33. [11] Marchesan I. Lingual Frenulum: classification and speech interference. IJOM 2004;30:31–8. [12] Yang HM, Woo YJ, Won SY, Kim DH, Hu KS, Kim HJ. Course and distribution of the lingual nerve in the ventral tongue region: anatomical considerations for frenectomy. J Craniofac Surg 2009;20(5):1359–63. [13] Campan P, Baron P, Duran D, Casteigt J. Lingual frenectomy: a therapeutic protocol. A technic for frenectomy with 2 incision lines combined with active postoperative kinesitherapy during and after healing. Schweiz Monatsschr Zahnmed 1996;106:45–54. [14] Suter VGA, Bornstein MM. Ankyloglossia: facts and myths in diagnosis and treatment. J Periodontol 2009;80:1204–19. [15] Messner AH, Lalakea ML. Ankyloglossia: controversies in management. Int J Pediatr Otorhinolaryngol 2000;54:123–31. [16] Klepacek I, Skulec R. Relation between lingual nerve, submandibular gland duct and mandibular body in the sublingual space. Acta Chir Plast 1993;36(1):26–7. [17] Rosano G, Taschieri S, Testori T, Del Fabbro M, El Haddioui A, Gaudy JF. Evaluation of anterior mandible anatomy in oral surgery. Ital Oral Surg 2008;7:7–14. [18] Zeinoun T, Nammour S, Dourov N, Aftimos G, Luomanen M. Myofibroblasts in healing laser excision wounds. Lasers Surg Med 2001;28:74–9. [19] Haytac MC, Ozcelik O. Evaluation of patient perceptions after frenectomy operations: a comparison of carbon dioxide laser and scalpel techniques. J Periodontol 2006;77:1815–9. [20] Martinelli RL, Marchesan IQ, Berretin-Felix G. Lingual frenulum protocol with scores for infants. Int J Orofacial Myology 2012;38:104–12.
5
[21] Manish A, Mala Dixit B, Lashika T, Shraddha K. Ankyloglossia – a review and case report. Euro J Pharmaceutical Med Res 2018;5(3):426–30. [22] Darshan HE, Pavithra PM. Tongue-tie: from confusion to clarity – a review. Int J Dent Clin 2011;3(1):48–51. [23] Shadab K, Shweta S, Vivek KS. Ankyloglossia: surgical management and functional rehabilitation of tongue. Ind J Dent Res 2017;28(5):585–7. [24] Seo YJ, Kim SJ, Munkhshur J, Chung KR, Ngan P, Kim SH. Treatment and retention of relapsed anterior open-bite with low tongue posture and tongue-tie: a 10-year follow-up. Korean J Orthod 2014;44(4):203–16. [25] Pick RM, Picaro BC, Silberman CJ. The laser gingivectomy: the use of CO2 laser for the removal of phenytoin hyperplasia. J Periodontol 1985;56:492–6. [26] Tanya D, Lele DP, Patil DP. Ankyloglossia: treatment with surgical lasers – a case report. IOSR J Dent Med Sci 2014;13:61–4. [27] Haberal I, Gocmen H, Samim E. Surgical management of pediatric ranula. Int J Pediatr Otorhinolaryngol 2004;68:161–3. [28] Khairnar M, Pawar B, Khairnar D. A novel surgical pre-suturing technique for the management of ankyloglossia. J Surg Tech Case Rep 2014;6(2):49–54. http://dx.doi.org/10.4103/2006-8808.147259. [29] Baurmash HD. Marsupialization for treatment of oral ranula a second look at the procedure. J Oral Maxillofac Surg 1992;50:1274–9. [30] Watanabe K, Tomiyama S, Jinnouchi K, Nakajima H, Yagi T. Local injection of OK-432 in the treatment of ranula: a case report. ENT J 2002;81:97–8. [31] Chow TL, Chan SW, Lam SH. Ranula successfully treated by botulinum toxin type A: report of 3 cases. Oral Surg Oral Med Oral Path Oral Radiol Endo 2008;105:41–2. [32] Stokes S, Gowrishankar S, Anand P. Mucus extravasation cyst as a complication of lingual frenectomy. Br J Oral Maxillofac Surg 2017;55:e86–189. [33] Pandit RT, Park AH. Management of pediatric ranula. Otolaryngol Head Neck Surg 2002;127:115–8. [34] Mihir RP, Allison MD, William WS. Oral and plunging ranulas: what is the most effective treatment? Laryngoscope 2009;119:1501–9. [35] Zhi K, Wen Y, Ren W, Zhang Y. Management of infant ranula. Int J Pediatr Otorhinolaryn 2008;72:823–6. [36] Hing NR, Bowler MD, Byth PL, Daly CG. Lingual hematoma leading to upper airway obstruction. Br J Oral Maxillofac Surg 1985;23:322–5. [37] Renehan A, Morton M. Acute enlargement of the tongue. Br J Oral Maxillofac Surg 1993;31:321–4. ˜ o-Garcı´a M. Hematoma of the floor of the mouth [38] Vera JD, Calleja JM, Burguen and airway obstruction during mandibular dental implant placement: a case report. Oral Maxillofac Surg 2008;12:223–36. [39] Loukas M, Kinsella CR, Kapos T, Tubbs RS, Ramachandra S. Anatomical variation in the arterial supply of the mandible with special regard to implant placement. Intern J Oral Maxillofac Surg 2008;37:367–71. [40] Smith MH, Lung KE. Nerve injuries after dental injection: a review of the literature. J Can Dent Assoc 2006;72:559–64. [41] Hwang HD, Kim JW, Kim YS, Kang DH, Kwon TJ. Angiographic embolization for hemorrhage control after dental implantation. J Korean Assoc Oral and Maxillofac Surgeon 2013;39:27–30. [42] Takei H, Caranza F. The periodontal flap. In: Newman M, Takei H, Klokkevold P, editors. Caranza’s clinical periodontology, ed. 10. Philadelphia: Saunder; 2009. p. 926–36. [43] Tan VL, Andrawos A, Ghabriel MN, Townsend GC. Applied anatomy of the lingual nerve: relevance to dental anaesthesia. Arch Oral Biol 2014;59:324–35. [44] Garlsto GA, Gaffen AS, Lawrence HP, Tenenbaum HC, Haas DA. Occurrence of paresthesia after dental local anesthetic administration in the United States. J Am Dent Assoc 2010;141:836–44. [45] Fielding AF, Rachiele DP, Frazier G. Lingual nerve paresthesia following third molar surgery. A retrospective clinical study. Oral Surg Oral Med Oral Path Oral Radiol Endodont 1997;84:345–8. [46] Piagkou M, Demesticha T, Skandalakis P, Johnson EO. Functional anatomy of the mandibular nerve: consequences of nerve injury and entrapment. Clin Anat 2011;24:143–50. [47] Rusu MC, Nimigean V, Podoleanu L, Ivas¸cu RV, Niculescu MC. Details of the intralingual topography and morphology of the lingual nerve. Int J Oral Maxillofac Surg 2008;37:835–9. [48] Sun H, Yang T, Li Q, et al. Dexamethasone and vitamin B12 synergistically promote peripheral nerve regeneration in rats by upregulating the expression of brain-derived neurotrophic factor. Arch Med Sci 2012;8:924–30.
Please cite this article in press as: Varadan M, et al. Etiology and clinical recommendations to manage the complications following lingual frenectomy: A critical review. J Stomatol Oral Maxillofac Surg (2019), https://doi.org/10.1016/j.jormas.2019.06.003