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Hypovolemic shock after labial and lingual frenulectomy: A report of two cases
International Journal of Pediatric Otorhinolaryngology 100 (2017) 223e224
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: http://www.ijporlonline.com/
Case Report
Hypovolemic shock after labial and lingual frenulectomy: A report of two cases Lauren F. Tracy a, *, Gabriel Gomez b, Lewis J. Overton a, Wade G. McClain b a b
University of North Carolina, Department of Otolaryngology/Head and Neck Surgery, 101 Manning Drive, Chapel Hill, NC 27599, USA University of North Carolina, Department of Pediatric Otolaryngology, 101 Manning Drive, Chapel Hill, NC 27599, USA
a r t i c l e i n f o
a b s t r a c t
Article history: Received 19 April 2017 Received in revised form 5 July 2017 Accepted 12 July 2017 Available online 14 July 2017
Lingual and labial frenulectomy are commonly performed as an outpatient procedure, either in an office setting or under general anesthesia. Frenulectomy is generally regarded by both otolaryngologists and dentists as a straightforward and low-risk procedure with limited evidence-based indications and similarly few contraindications. We describe two cases of hypovolemic shock occurring after outpatient frenulectomy requiring emergent interventions of cardiopulmonary resuscitation and blood transfusion. These rare, but life-threatening outcomes warrant recognition as potential complications for the presumed benign labial and lingual frenulectomy. We additionally briefly review indications for upper labial and lingual frenulectomy. © 2017 Published by Elsevier Ireland Ltd.
Keywords: Frenulum Ankyloglossia Frenulectomy
1. Introduction We report two cases of large volume blood loss causing hypovolemic shock after outpatient labial and lingual frenulectomy surgery. One patient required cardiopulmonary resuscitation and the other required emergent operative intervention to control bleeding. Both patients required blood transfusions due to large volume blood loss. It is important to recognize this potentially life-threatening complication resulting from labial or lingual frenulectomy. 2. Materials and methods Retrospective report of two cases. 3. Results 3.1. Case 1 description A healthy 13-month-old female underwent both lingual and upper labial frenulectomy for ankyloglossia and prominent labial frenulum. The procedure was performed under general anesthesia at an outpatient otolaryngology surgery center. The surgical sites
* Corresponding author. E-mail address: [email protected] (L.F. Tracy). http://dx.doi.org/10.1016/j.ijporl.2017.07.013 0165-5876/© 2017 Published by Elsevier Ireland Ltd.
were without bleeding and patient was able tolerate oral intake without difficulty in the immediate post-operative period. Approximately 19 hours after surgery, the patient was overheard coughing while asleep. The patient's mother noted the child to be limp and unresponsive with blood-stained bed linens. Cardiopulmonary resuscitation was performed, spontaneous respirations resumed, and EMS was called. On initial evaluation the EMS providers observed tachycardia, tachypnea, hypotension, pallor and lethargy. The patient was emergently transported to the Emergency Department with continued resuscitative efforts. In the Emergency Department, 1 unit of PRBCs were given which improved the patient's hematocrit to 28%. The otolaryngology service was consulted and observed clot over labial frenulectomy site and healing lingual frenulectomy site. The clot was removed to reveal an erythematous wound base which was cauterized with silver nitrate. The patient was monitored overnight and no further bleeding occurred. Patient had no previous personal or family history of bleeding diatheses and work-up for these abnormalities was negative. 3.2. Case 2 description A 4-year-old male with history of autism and neurofibromatosis type 2 underwent anterior lingual frenulectomy for ankyloglossia and dysphagia after evaluation by an otolaryngologist and speech pathologist. This procedure was performed immediately after routine surveillance MRI of his brain under procedural sedation.
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Scissors were used to cut frenulum and silver nitrate was applied to achieve hemostasis. No sutures were applied and no bleeding was noted at conclusion of the case. Two days following the procedure, the patient experienced an episode of bleeding from the mouth which resolved at home without intervention. On post-procedure day 8, the patient bled again and evaluation in the emergency department revealed a small blood clot on floor of mouth without active bleeding. The patient was released home then represented two days later with reports of three additional small-volume oral bleeding episodes. He was tachycardic and blood hemoglobin was 7.8 gm/dL. Intravenous fluids and 1 unit of packed red blood cells were administered and the patient was urgently taken to the operating room for control of hemorrhage. Intra-operatively, arterial bleeding was noted at prior frenulectomy site and this was controlled with suction electrocautery and oversewing of prior surgical site. Patient was discharged the next day in stable condition and follow up clinical evaluation demonstrated excellent healing. 4. Discussion The labial maxillary frenulum is a membranous fold comprised of muscle and connective tissue that attaches the lip to gingiva, alveolar mucosa, and underlying periosteum [1]. It is a normal anatomical structure and is considered pathogenic only in the setting of aberrant attachment with associated midline diastema, gingival recession, or shallow vestibule [2]. Broad indications for labial frenulum removal include release of gingival tension and facilitation of orthodontic treatment [3]. Additionally, in the scenario of plaque accumulation and difficulty with dental hygiene, upper labial frenulectomy has been suggested as a beneficial intervention [4]. Maxillary labial frenulum has been implicated in breast feeding difficulty with anecdotal evidence suggesting restricted upper lip movement may interfere with adequate attachment during feeding [5]. Shah et al. endeavored to outline specific goals for labial frenulectomy. These included closure of midline diastema by orthodontic appliance, hypertrophic frenulum, presence of midline maxillary suture with complete eruption of upper size permanent anterior teeth [6]. Additionally, they recommended against upper labial frenulectomy in the absence of anterior tooth eruption. A pathogenic frenulum can be removed by either frenectomy or frenotomy surgery. A frenectomy involves complete removal of frenulum in addition to the bony attachment while frenotomy is the incision of only the mucosal and muscular frenal attachment. A number of techniques exist for frenulectomy and frenotomy including “conventional” scalpel technique, electrocautery, or CO2 laser [2]. Closure techniques include secondary healing, Z-plasty closure and primary closure with absorbable suture. These procedures are performed by otolaryngologists and dentists, however, there is an extreme paucity of data about labial frenulectomy in the field of otolaryngology. A PubMed database review of “otolaryngology, frenectomy, frenum, frenulotomy” revealed only 1 article in the otolaryngology literature about labial frenulectomy. Pransky et al. reported a 2% incidence of upper lip tie in a review of 618 newborn patients and conjectured a relationship
between upper lip tie and breast feeding difficulty. For this review, the definition of upper lip tie was based on visualization of frenulum and mother subjective report of limited upper lip movement during breastfeeding [5]. The lingual frenulum is a thin strip of tissue attaching the floor of the mouth to the midline ventral surface of the tongue. Ankyloglossia was first defined as inability to protrude the tongue beyond the lower incisors secondary to a short fibrous lingual frenulum [8]. Since this description, others have offered classifications based on specific measurements but clinical history and inspection continue as the most common method of diagnosis (9). Incidence of ankyloglossia by examination alone has been cited between 1e10% of all infants in various studies [9]. Neonatal feeding difficulties, as well as speech problems and social issues are amongst the most cited indications for surgical intervention. However, there is no consensus regarding the significance of ankyloglossia in these scenarios. Additionally, timing and optimal method of intervention remains controversial with mixed outcomes after intervention for ankylgossia [10]. Literature on complications after lingual frenulectomy has been limited to case reports describing airway obstruction in Pierre Robin Sequence patients and infection [11,12]. To our knowledge this the first article describing bleeding as a significant event after lingual frenulectomy. 5. Conclusion In conclusion, we aim to present a rare and life-threatening complication of these routine procedures. These are commonly performed in the pediatric population as an outpatient procedure by otolaryngologists and dentists. There is a paucity of data in otolaryngology literature regarding upper labial frenulectomy or complications of lingual frenulectomy. Practioners should be aware of indications for this procedure as well as the potential for lifethreatening complications in the postoperative period. References [1] H. Jhaveri, The Aberrant Frenum. Dr. PD Miller, the Father of Periodontal Plastic Surgery, 2006, pp. 29e34. [2] S.K. Devishree, Gujjari, P.V. Shubbashini, Frenectomy: a review with the reports of surgical techniques, Clin. Diagn Res. 6 (2012) 1587e1592. [3] A.G. Moghe, et al., Frenectomy review: comparison of conventional techniques with diode laser, Laser 3 (2010) 15e18. [4] M.C. Haytac, O. Ozcelik, Evaluation of patient perceptions after frenectomy operations: a comparison of carbon dioxide laser and scalpel techniques, J Periodontol. 11 (2006) 1815e1819. [5] S.M. Pransky, D. Lago, P. Hong, Breastfeeding difficulties and oral cavity anomalies: the influence of posterior anklyglossia and upper-lip ties, Int. J Ped Otorhinolaryngol. 79 (2015) 1714e1717. [6] J. Shah, F. Dyer, Current practice for performing labial frenectomies a service evaluation, Br. Orthod. Soc. Clin. Eff. Bull. 21 (2008) 4. [8] A.F. Wallace, Tongue tie, Lancet 2 (1963) 377e378. [9] V.G. Suter, M.M. Bornstein, Ankyloglossia: facts and myths in diagnosis and treatment, J. Periodontol. 80 (2009) 1204e1219. [10] A.H. Messner, M.L. Lalakea, Ankyloglossia: controversies in management, Int. J. Pediatr. Otorhinolaryngol. 54 (2000) 123e131. [11] D.J.D. Genther, Airway obstruction after lingual frenulectomy in two infants with Pierre-Robin Sequence, Int. J Pediatr. otorhinolaryngol. 79 (2015) 1592. [12] A.A. Isaiah, Infected sublingual hematoma: a rare complication of frenulectomy, ENT J. 92 (2013) 296.
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: http://www.ijporlonline.com/
Case Report
Hypovolemic shock after labial and lingual frenulectomy: A report of two cases Lauren F. Tracy a, *, Gabriel Gomez b, Lewis J. Overton a, Wade G. McClain b a b
University of North Carolina, Department of Otolaryngology/Head and Neck Surgery, 101 Manning Drive, Chapel Hill, NC 27599, USA University of North Carolina, Department of Pediatric Otolaryngology, 101 Manning Drive, Chapel Hill, NC 27599, USA
a r t i c l e i n f o
a b s t r a c t
Article history: Received 19 April 2017 Received in revised form 5 July 2017 Accepted 12 July 2017 Available online 14 July 2017
Lingual and labial frenulectomy are commonly performed as an outpatient procedure, either in an office setting or under general anesthesia. Frenulectomy is generally regarded by both otolaryngologists and dentists as a straightforward and low-risk procedure with limited evidence-based indications and similarly few contraindications. We describe two cases of hypovolemic shock occurring after outpatient frenulectomy requiring emergent interventions of cardiopulmonary resuscitation and blood transfusion. These rare, but life-threatening outcomes warrant recognition as potential complications for the presumed benign labial and lingual frenulectomy. We additionally briefly review indications for upper labial and lingual frenulectomy. © 2017 Published by Elsevier Ireland Ltd.
Keywords: Frenulum Ankyloglossia Frenulectomy
1. Introduction We report two cases of large volume blood loss causing hypovolemic shock after outpatient labial and lingual frenulectomy surgery. One patient required cardiopulmonary resuscitation and the other required emergent operative intervention to control bleeding. Both patients required blood transfusions due to large volume blood loss. It is important to recognize this potentially life-threatening complication resulting from labial or lingual frenulectomy. 2. Materials and methods Retrospective report of two cases. 3. Results 3.1. Case 1 description A healthy 13-month-old female underwent both lingual and upper labial frenulectomy for ankyloglossia and prominent labial frenulum. The procedure was performed under general anesthesia at an outpatient otolaryngology surgery center. The surgical sites
* Corresponding author. E-mail address: [email protected] (L.F. Tracy). http://dx.doi.org/10.1016/j.ijporl.2017.07.013 0165-5876/© 2017 Published by Elsevier Ireland Ltd.
were without bleeding and patient was able tolerate oral intake without difficulty in the immediate post-operative period. Approximately 19 hours after surgery, the patient was overheard coughing while asleep. The patient's mother noted the child to be limp and unresponsive with blood-stained bed linens. Cardiopulmonary resuscitation was performed, spontaneous respirations resumed, and EMS was called. On initial evaluation the EMS providers observed tachycardia, tachypnea, hypotension, pallor and lethargy. The patient was emergently transported to the Emergency Department with continued resuscitative efforts. In the Emergency Department, 1 unit of PRBCs were given which improved the patient's hematocrit to 28%. The otolaryngology service was consulted and observed clot over labial frenulectomy site and healing lingual frenulectomy site. The clot was removed to reveal an erythematous wound base which was cauterized with silver nitrate. The patient was monitored overnight and no further bleeding occurred. Patient had no previous personal or family history of bleeding diatheses and work-up for these abnormalities was negative. 3.2. Case 2 description A 4-year-old male with history of autism and neurofibromatosis type 2 underwent anterior lingual frenulectomy for ankyloglossia and dysphagia after evaluation by an otolaryngologist and speech pathologist. This procedure was performed immediately after routine surveillance MRI of his brain under procedural sedation.
224
L.F. Tracy et al. / International Journal of Pediatric Otorhinolaryngology 100 (2017) 223e224
Scissors were used to cut frenulum and silver nitrate was applied to achieve hemostasis. No sutures were applied and no bleeding was noted at conclusion of the case. Two days following the procedure, the patient experienced an episode of bleeding from the mouth which resolved at home without intervention. On post-procedure day 8, the patient bled again and evaluation in the emergency department revealed a small blood clot on floor of mouth without active bleeding. The patient was released home then represented two days later with reports of three additional small-volume oral bleeding episodes. He was tachycardic and blood hemoglobin was 7.8 gm/dL. Intravenous fluids and 1 unit of packed red blood cells were administered and the patient was urgently taken to the operating room for control of hemorrhage. Intra-operatively, arterial bleeding was noted at prior frenulectomy site and this was controlled with suction electrocautery and oversewing of prior surgical site. Patient was discharged the next day in stable condition and follow up clinical evaluation demonstrated excellent healing. 4. Discussion The labial maxillary frenulum is a membranous fold comprised of muscle and connective tissue that attaches the lip to gingiva, alveolar mucosa, and underlying periosteum [1]. It is a normal anatomical structure and is considered pathogenic only in the setting of aberrant attachment with associated midline diastema, gingival recession, or shallow vestibule [2]. Broad indications for labial frenulum removal include release of gingival tension and facilitation of orthodontic treatment [3]. Additionally, in the scenario of plaque accumulation and difficulty with dental hygiene, upper labial frenulectomy has been suggested as a beneficial intervention [4]. Maxillary labial frenulum has been implicated in breast feeding difficulty with anecdotal evidence suggesting restricted upper lip movement may interfere with adequate attachment during feeding [5]. Shah et al. endeavored to outline specific goals for labial frenulectomy. These included closure of midline diastema by orthodontic appliance, hypertrophic frenulum, presence of midline maxillary suture with complete eruption of upper size permanent anterior teeth [6]. Additionally, they recommended against upper labial frenulectomy in the absence of anterior tooth eruption. A pathogenic frenulum can be removed by either frenectomy or frenotomy surgery. A frenectomy involves complete removal of frenulum in addition to the bony attachment while frenotomy is the incision of only the mucosal and muscular frenal attachment. A number of techniques exist for frenulectomy and frenotomy including “conventional” scalpel technique, electrocautery, or CO2 laser [2]. Closure techniques include secondary healing, Z-plasty closure and primary closure with absorbable suture. These procedures are performed by otolaryngologists and dentists, however, there is an extreme paucity of data about labial frenulectomy in the field of otolaryngology. A PubMed database review of “otolaryngology, frenectomy, frenum, frenulotomy” revealed only 1 article in the otolaryngology literature about labial frenulectomy. Pransky et al. reported a 2% incidence of upper lip tie in a review of 618 newborn patients and conjectured a relationship
between upper lip tie and breast feeding difficulty. For this review, the definition of upper lip tie was based on visualization of frenulum and mother subjective report of limited upper lip movement during breastfeeding [5]. The lingual frenulum is a thin strip of tissue attaching the floor of the mouth to the midline ventral surface of the tongue. Ankyloglossia was first defined as inability to protrude the tongue beyond the lower incisors secondary to a short fibrous lingual frenulum [8]. Since this description, others have offered classifications based on specific measurements but clinical history and inspection continue as the most common method of diagnosis (9). Incidence of ankyloglossia by examination alone has been cited between 1e10% of all infants in various studies [9]. Neonatal feeding difficulties, as well as speech problems and social issues are amongst the most cited indications for surgical intervention. However, there is no consensus regarding the significance of ankyloglossia in these scenarios. Additionally, timing and optimal method of intervention remains controversial with mixed outcomes after intervention for ankylgossia [10]. Literature on complications after lingual frenulectomy has been limited to case reports describing airway obstruction in Pierre Robin Sequence patients and infection [11,12]. To our knowledge this the first article describing bleeding as a significant event after lingual frenulectomy. 5. Conclusion In conclusion, we aim to present a rare and life-threatening complication of these routine procedures. These are commonly performed in the pediatric population as an outpatient procedure by otolaryngologists and dentists. There is a paucity of data in otolaryngology literature regarding upper labial frenulectomy or complications of lingual frenulectomy. Practioners should be aware of indications for this procedure as well as the potential for lifethreatening complications in the postoperative period. References [1] H. Jhaveri, The Aberrant Frenum. Dr. PD Miller, the Father of Periodontal Plastic Surgery, 2006, pp. 29e34. [2] S.K. Devishree, Gujjari, P.V. Shubbashini, Frenectomy: a review with the reports of surgical techniques, Clin. Diagn Res. 6 (2012) 1587e1592. [3] A.G. Moghe, et al., Frenectomy review: comparison of conventional techniques with diode laser, Laser 3 (2010) 15e18. [4] M.C. Haytac, O. Ozcelik, Evaluation of patient perceptions after frenectomy operations: a comparison of carbon dioxide laser and scalpel techniques, J Periodontol. 11 (2006) 1815e1819. [5] S.M. Pransky, D. Lago, P. Hong, Breastfeeding difficulties and oral cavity anomalies: the influence of posterior anklyglossia and upper-lip ties, Int. J Ped Otorhinolaryngol. 79 (2015) 1714e1717. [6] J. Shah, F. Dyer, Current practice for performing labial frenectomies a service evaluation, Br. Orthod. Soc. Clin. Eff. Bull. 21 (2008) 4. [8] A.F. Wallace, Tongue tie, Lancet 2 (1963) 377e378. [9] V.G. Suter, M.M. Bornstein, Ankyloglossia: facts and myths in diagnosis and treatment, J. Periodontol. 80 (2009) 1204e1219. [10] A.H. Messner, M.L. Lalakea, Ankyloglossia: controversies in management, Int. J. Pediatr. Otorhinolaryngol. 54 (2000) 123e131. [11] D.J.D. Genther, Airway obstruction after lingual frenulectomy in two infants with Pierre-Robin Sequence, Int. J Pediatr. otorhinolaryngol. 79 (2015) 1592. [12] A.A. Isaiah, Infected sublingual hematoma: a rare complication of frenulectomy, ENT J. 92 (2013) 296.