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| CASE REPORT |
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| Year : 2019 | Volume
: 2
| Issue : 3 | Page : 148-150 |
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Persistent buccopharyngeal membrane: Anaesthetic management
Reena, Shweta Agarwal, Kanika Gupta, Pradeepika Gangwar
Department of Anaesthesiology, Heritage Institute of Medical Sciences, Varanasi, Uttar Pradesh, India
| Date of Submission | 25-Jun-2019 |
| Date of Acceptance | 13-Sep-2019 |
| Date of Web Publication | 30-Jan-2020 |
Correspondence Address:
Dr. Reena
Department of Anaesthesiology, Heritage Institute of Medical Sciences, NH-2, Bhadwar, Varanasi - 221 311, Uttar Pradesh
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/ARWY.ARWY_21_19
Persistent buccopharyngeal membrane (PBM) is a rare congenital anomaly. Very few case reports have been published to date. We report the case of a 5-year-old girl with PBM with a central 1 cm diameter perforation posted for excision of the membrane. We have briefly reviewed the embryology of PBM with a focus on perioperative anaesthetic concerns and airway management options.
Keywords: Embryology, oropharyngeal membrane, persistent buccopharyngeal membrane
How to cite this article:
Reena, Agarwal S, Gupta K, Gangwar P. Persistent buccopharyngeal membrane: Anaesthetic management. Airway 2019;2:148-50 |
| Introduction | |  |
The buccopharyngeal membrane is a bilayer membrane covering the embryonic mouth, perforation of which is essential for oral cavity formation. In humans, persistence of the buccopharyngeal membrane can lead to orofacial defects such as choanal atresia, oral synechiae and cleft palate.[1] Persistent buccopharyngeal membrane (PBM) is almost always associated with a difficult airway requiring emergent or planned tracheostomy or fibreoptic intubation. We describe the case of a 5-year-old girl with PBM who was successfully intubated following direct laryngoscopy through a 1 cm wide central perforation on the membrane.
| Case Report | | |
A 5-year-old girl was posted for the excision of PBM. Her chief complaints were difficulty in swallowing, difficulty in breathing during sleep and nasal twang in speech for the past 1 year. On opening her mouth, the membrane was visible with a central 1 cm diameter perforation through which only the uvula was visible [Figure 1]. Her mouth opening was adequate and one upper central incisor was loose. Nasal patency was checked to rule out choanal atresia. Systemic examination also did not reveal any associated abnormality. Past history, obtained from her grandfather and father, was also not significant enough to point towards any other abnormality. Her routine blood investigations were normal. Chest X-ray and electrocardiogram were done to rule out any associated cardiopulmonary abnormalities.
The child was kept fasted for 6 h for light snacks and 2 h for clear fluids. A 22 gauge intravenous cannula was sited in her left hand, and Ringer lactate was started using a 100 mL burette set. As we did not have a paediatric fibreoptic bronchoscope, our plan was to check if we could pass the tip of a Miller blade beyond the hole in the buccopharyngeal membrane and negotiate the endotracheal tube (ETT). We had taken consent for tracheostomy in case we were not able to intubate. The child was preoxygenated for 5 min, and anaesthesia was induced intravenously with 0.5 mg midazolam, 30 μg fentanyl and 30 mg propofol. As mask ventilation was adequate, we gave her succinylcholine 20 mg IV. Direct laryngoscopy was performed by a senior anaesthesiologist using a size 1 Miller blade. An attempt was made to negotiate the tip of the blade through the perforation in the PBM. Initially, no part of the larynx could be seen. Application of backward, upward and rightward pressure (BURP) by a second anaesthesiologist resulted in visualisation of only the tip of the epiglottis. An unsuccessful attempt was made to slip a 4.0 mm ID uncuffed ETT just behind the epiglottis. The child started breathing spontaneously. As we were able to ventilate the child adequately, we administered vecuronium 1 mg IV and mask ventilated her for 3 min before the next attempt at intubation. The ETT was given a 'J' shape with the help of a stylet. While maintaining the same view with laryngoscopy and BURP, the tube was successfully introduced this time into the larynx. The ETT was taped securely and connected to a paediatric circuit after removing the stylet. The position of the ETT was confirmed with capnography and bilaterally equal breath sounds on auscultation. Anaesthesia was maintained with sevoflurane 1%–2% in oxygen and nitrous oxide in a 1:1 ratio and muscle relaxation with vecuronium 0.5 mg top-ups. Following the removal of one side of the membrane, the surgeons were requested to put a gauze pack around the ETT as this was not possible while the membrane was intact. The tonsil that was embedded under the right side of the membrane was removed. At the end of the surgery, we injected 0.5 mL of a mixture (2 mL each of 0.5% bupivacaine and 2% lignocaine with 1:200,000 adrenaline) at each of the sites to block greater and lesser palatine and nasopalatine nerves to provide postoperative analgesia. The duration of surgery was 1.5 h. The throat pack was removed, and the patient was extubated at the end of the surgery after confirming haemostasis and adequate antagonism of residual neuromuscular blockade.
| Discussion | | |
During embryonal development, the stomodeum or primordial mouth is separated from the primitive pharynx and foregut by a two-cell layered membrane known as oral or buccopharyngeal membrane.[1] This membrane ruptures around the 26th day of intrauterine life to establish a communication between the mouth and the future pharynx.[2] The rupture of the membrane is believed to be due to stress caused by rapid growth and differentiation of the pharynx, nose, face and tongue in the embryo.[1] The failure of the membrane to break down results in PBM which can be either complete or partial.[3],[4] Complete PBM with no communication to the pharynx presents with respiratory distress and dysphagia and is an immediate threat to life.[4] Emergency tracheostomy may be needed to establish the airway at birth.[5] Total PBM with a central slit-like perforation or partial PBM may remain undiagnosed until the child complains of dysphagia or breathing difficulty. It may also be diagnosed as an incidental finding.[3] In this child, the attachments of the membrane were superiorly to the soft palate, laterally along the anterior tonsillar pillars and inferiorly at the junction of the anterior two-thirds and posterior one-third of the tongue. This presentation matches with that which is reported in literature.[6] Due to the presence of a perforation, our case remained undiagnosed till the child presented at the age of 5 years.
The rarity of this condition is evident from the fact that only 27 cases of PBM have been reported to date. Verma andGeller reviewed 23 reported cases of PBM in 2009.[4] We reviewed more recent literature and found four more reported cases of PBM.[3],[7],[8],[9] Persistence of buccopharyngeal membrane has also been implicated as the causative factor for other congenital orofacial defects such as choanal atresia, intraoral bands or synechiae. Gartlan et al. suggested that oral bands consisting of soft tissue only should be termed as synechiae and bony involvement should be called syngnathia.[10] They also divided oral synechiae into two categories as follows: posterior intraoral bands near the base of the tongue representing the remnants of the buccopharyngeal membrane and anterior intraoral bands which are ectopic membranes.[10] Another theory suggests penetration of the buccopharyngeal membrane by mesodermal tissue from the posterior third of the growing tongue, leading to its persistence by strengthening it.[2]
These defects can occur either in isolation or in combination with other congenital anomalies including various cardiac, vertebral and orofacial defects.[4],[7] Teratogenic drug effects during early embryonic development have also been postulated when intraoral synechiae have been found in association with limb deformities.[10],[11] Our patient, however, did not have any other associated congenital defects.
PBM cases reported in literature so far were managed either by tracheostomy or fibreoptic intubation.[5],[9] In our case, we chose direct laryngoscopy for the first two attempts while reserving tracheostomy as our last option to secure the patient's airway. Pirat et al. have described retrograde intubation to secure the airway in an infant with Pierre Robin syndrome and glossopharyngeal membrane even after the excision of the membrane.[8]
The authors routinely practice palatine nerve blocks for palate surgeries to provide excellent analgesia in the postoperative period.[12] In this child, blockade of the greater and lesser palatine and nasopalatine nerves provided optimum postoperative analgesia. We, along with our otorhinolaryngologists, followed up the patient for the next 6 months to identify any subsequent development of fibrosis or stenosis. The child did not develop any such problems till the time of writing this article.
| Conclusion | | |
PBM is a rare condition which may or may not be associated with other congenital anomalies. A thorough preoperative workup with meticulously planned airway management is required to handle such difficult cases.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the parents of the child have given their consent for the image and other clinical information to be reported in the journal. The parents understand that the name and initials of the child will not be published and due efforts will be made to conceal the identity of the child, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
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| 2. | Agarwal R, Kumar P, Kalra GS, Bhushan S, Chandra R. Persistent buccopharyngeal membrane: A report of two cases. Plast Reconstr Surg 1996;98:866-8. |
| 3. | Ramachandran S, Mamatha GP, Annigeri RG, Shettar SS. Persistent buccopharyngeal membrane in an adult an incidental finding – A case report. Int J Dent Clin 2010;2:74-6. |
| 4. | Verma SP, Geller K. Persistent buccopharyngeal membrane: Report of a case and review of the literature. Int J Pediatr Otorhinolaryngol 2009;73:877-80. |
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| 8. | Pirat A, Candan S, Unlükaplan A, Kömürcü O, Kuşlu S, Arslan G. Percutaneous dilational tracheotomy for airway management in a newborn with Pierre-Robin syndrome and a glossopharyngeal web. Respir Care 2012;57:627-30. |
| 9. | Kuppswamy B, Rajaleelan W, Jacob NS, Ponniah M. Anesthetic management of an adolescent with congenital glossopharyngeal web. Saudi J Anaesth 2016;10:243-5. |
| 10. | Gartlan MG, Davies J, Smith RJ. Congenital oral synechiae. Ann Otol Rhinol Laryngol 1993;102:186-97. |
| 11. | Ooi EH, Khouri Z, Hilton M. Persistent buccopharyngeal membrane in an adult. Int J Oral Maxillofac Surg 2005;34:446-8. |
| 12. | Reena, Bandyopadhyay KH, Paul A. Postoperative analgesia for cleft lip and palate repair in children. J Anaesthesiol Clin Pharmacol 2016;32:5-11. |
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