736
Anesth Analg 56:736-738,1977
Induction of Anesthesia in Infant with Frontonasal Dysplasia and Meningoencephalocele: A Case Report XAVIER MARQUEZ, M D * RENATO S. ROXAS, M D I Detroit, Michigan!:
A
must have full control of the patient’s airway a t all times. In pediatric anesthesia, intubation can prove very challenging in the presence of some rare congenital abnormalities of the maxillofacial structures. This case report deals with two approaches in an infant with frontonasal dysplasia and meningoencephalocele. N ANESTHFSIOLOGIST
CASE REPORT A full-term, 3.5-kg girl was delivered a t another hospital after an uneventful parturition. The Apgar score was 1 a t the 1st minute and Apgar 6 at 5 minutes. No resuscitative measures were required but the patient had a severe congenital anomaly of the facial structures, consisting of a 27 x 8-cm tumor in the midline of the face and a median cleft midline syndrome ( frontonasal dysplasia) (fig 1). The nostrils were completely separated, the mouth was 6.6 cm wide surrounding the lower edge of the mass, and the child also had a large cleft palate (fig 2 ) . The patient was transferred to our hospital for evaluation and possible surgical excision of the tumor and for cosmetic repairs. At 11 days of age, the girl was scheduled for angiographic studies to determine the characteristics of the tumor. T o assure a safe and adequate airway, endotracheal intubation was considered necessary. In view of
the technical difficulty of ventilating this infant by the conventional method (bag and mask), awake intubation was attempted. This task was difficult not only because the baby was awake and active but also because of the large tumor which might easily be traumatized and which obstructed the view of the operator. After intubation, a cerebral arteriogram was taken under 0.2 to 0.3 percent halothane-N,O-0, (50:50) anesthesia. Both the course of the anesthesia and the immediate postoperative course were uneventful.
At age 1 month, the patient was scheduled for removal of a cystic, probably meningoencephalocele type, tumor. On a preoperative visit, the patency of the right nostril was established by auscultation. The left nostril appeared to be stenotic. The rest of the examination showed no abnormalities. Growth and development for her age were likewise within normal limits. Scopolamine (0.1 mg I M ) , 1 hour preoperatively, was given as preanesthetic medication. In the operating room, a 3.5 mm Portex nasotracheal tube was passed through the right nostril down to the posterior pharynx and anesthesia induced by 0.75 to 1 percent halothane in N,O-0,. Ventilation, using a modified Ayre’s T-piece through the nasopharyngeal airway proved easy, especially upon closure of the mouth (fig 3 ) . Succinyl-
*Clinical Fellow, Pediatric Anesthesia. ?Program Director, Department of Anesthesia. $Children’s Hospital of Michigan, Detroit, Michigan 48201 Paper received: December 17, 1976 Accepted for publication: January 3, 1977
737
Clinical Reports
FIG1. Shows the large meningoencephalic tumor.
FIG2. Shows the wide nostril separation and large cleft palate.
choline chloride (4 mg I M ) was given and oral intubation was accomplished without difficulty. Uneventful excision of the tumor was completed in 4 hours. The nasopharyngeal airway was kept in place for the first 24 postoperative hours, to prevent any possible obstruction, and the subsequent course was satisfactory. The kind of problem just presented is extremely rare.
COMMENTS Sedano and associates,1.4 reviewing the literature to 1970, found reports of about 70 cases of frontonasal dysplasia. We could not find any previous report of both frontonasal dysplasia and meningoencephalocele. Sometimes the anesthesiologist may find himself in such a challenging and dangerous situation, where control of the airway is in question, but we have demonstrated that such a
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Current Researches VOL.56, No. 5, SEPT.-OCT., 1977
patient can be ventilated without the bagand-mask technic.
The preoperative assessment of the patency of the nostril and clinical experience in ventilating babies with respiratory distress syndrome via nasal cannula led to the second approach to the handling of this infant’s airway.:%** Inflation of the stomach was not a problem in this case, but in the event that it occurs, intermittent placement of an orogastric catheter, as necessary, should help. We believe that, at least for small children and probably also in some adults, this method of induction of anesthesia gives good control of the airway, is safe and helpful, and can resolve unusual problems of ventilation prior to intubation.
REFERENCES 1. Sedano H, Cohen MM, Jirasek J, et al: Frontonasal dysplasia. J Pediatr 76:906-913, 1970 2. Cohen MM, Sedano H, Gorlin RT: Frontonasal dysplasia (Median cleft face syndrome) : Comments on etioloev and Dathoeenesis. Birth Defects: Original articl;? series, k o l GII, No. 7:117119, 1971
3. Kattwinkel J, Fleming D, Cha CH, et al: A device for administration of continuous Dositive airway pressure by the nasal route. J Pediatr 52: 131-134, 1973
FIG3. Shows the nasopharyngeal airway in place postoperatively.
4. Agostino R, Orzalesi M, Nodari S, et al: Continuous positive airway pressure (CPAP) by nasal cannula in the respiratory distress syndrome (RDS) of the newborn. Ped Research 7:50, 1973
PLASMA LEVELS OF LIDOCAINE. Venous (plasma) levels of lidocaine were measured in 5 subjects following nebulization of either a 280-mg dose using intermittent positive pressure breathing (IPPB) or a 400-mg dose using ultrasound. Even though a lower dose of lidocaine was given by IPPB, this system of delivery produced higher plasma concentrations of the drug than ultrasound but was more effective i n eliminating t h e gag reflex. I n no subject did plasma concentrations of lidocaine exceed 1.1 pg/ml, which is f a r below toxic levels (>5 pg/ml). Aerosolized lidocaine, administered by IPPB, i s a safe and effective topical anesthetic agent which may be useful for instrumentation of t h e upper airway. (Chinn WM, Zavala DC, Ambre J : Plasma levels of lidocaine following nebulized aerosol administration. Chest 71 :346-348,1977)
Anesth Analg 56:736-738,1977
Induction of Anesthesia in Infant with Frontonasal Dysplasia and Meningoencephalocele: A Case Report XAVIER MARQUEZ, M D * RENATO S. ROXAS, M D I Detroit, Michigan!:
A
must have full control of the patient’s airway a t all times. In pediatric anesthesia, intubation can prove very challenging in the presence of some rare congenital abnormalities of the maxillofacial structures. This case report deals with two approaches in an infant with frontonasal dysplasia and meningoencephalocele. N ANESTHFSIOLOGIST
CASE REPORT A full-term, 3.5-kg girl was delivered a t another hospital after an uneventful parturition. The Apgar score was 1 a t the 1st minute and Apgar 6 at 5 minutes. No resuscitative measures were required but the patient had a severe congenital anomaly of the facial structures, consisting of a 27 x 8-cm tumor in the midline of the face and a median cleft midline syndrome ( frontonasal dysplasia) (fig 1). The nostrils were completely separated, the mouth was 6.6 cm wide surrounding the lower edge of the mass, and the child also had a large cleft palate (fig 2 ) . The patient was transferred to our hospital for evaluation and possible surgical excision of the tumor and for cosmetic repairs. At 11 days of age, the girl was scheduled for angiographic studies to determine the characteristics of the tumor. T o assure a safe and adequate airway, endotracheal intubation was considered necessary. In view of
the technical difficulty of ventilating this infant by the conventional method (bag and mask), awake intubation was attempted. This task was difficult not only because the baby was awake and active but also because of the large tumor which might easily be traumatized and which obstructed the view of the operator. After intubation, a cerebral arteriogram was taken under 0.2 to 0.3 percent halothane-N,O-0, (50:50) anesthesia. Both the course of the anesthesia and the immediate postoperative course were uneventful.
At age 1 month, the patient was scheduled for removal of a cystic, probably meningoencephalocele type, tumor. On a preoperative visit, the patency of the right nostril was established by auscultation. The left nostril appeared to be stenotic. The rest of the examination showed no abnormalities. Growth and development for her age were likewise within normal limits. Scopolamine (0.1 mg I M ) , 1 hour preoperatively, was given as preanesthetic medication. In the operating room, a 3.5 mm Portex nasotracheal tube was passed through the right nostril down to the posterior pharynx and anesthesia induced by 0.75 to 1 percent halothane in N,O-0,. Ventilation, using a modified Ayre’s T-piece through the nasopharyngeal airway proved easy, especially upon closure of the mouth (fig 3 ) . Succinyl-
*Clinical Fellow, Pediatric Anesthesia. ?Program Director, Department of Anesthesia. $Children’s Hospital of Michigan, Detroit, Michigan 48201 Paper received: December 17, 1976 Accepted for publication: January 3, 1977
737
Clinical Reports
FIG1. Shows the large meningoencephalic tumor.
FIG2. Shows the wide nostril separation and large cleft palate.
choline chloride (4 mg I M ) was given and oral intubation was accomplished without difficulty. Uneventful excision of the tumor was completed in 4 hours. The nasopharyngeal airway was kept in place for the first 24 postoperative hours, to prevent any possible obstruction, and the subsequent course was satisfactory. The kind of problem just presented is extremely rare.
COMMENTS Sedano and associates,1.4 reviewing the literature to 1970, found reports of about 70 cases of frontonasal dysplasia. We could not find any previous report of both frontonasal dysplasia and meningoencephalocele. Sometimes the anesthesiologist may find himself in such a challenging and dangerous situation, where control of the airway is in question, but we have demonstrated that such a
738
ANESTHESIA AND ANALGESIA ...
Current Researches VOL.56, No. 5, SEPT.-OCT., 1977
patient can be ventilated without the bagand-mask technic.
The preoperative assessment of the patency of the nostril and clinical experience in ventilating babies with respiratory distress syndrome via nasal cannula led to the second approach to the handling of this infant’s airway.:%** Inflation of the stomach was not a problem in this case, but in the event that it occurs, intermittent placement of an orogastric catheter, as necessary, should help. We believe that, at least for small children and probably also in some adults, this method of induction of anesthesia gives good control of the airway, is safe and helpful, and can resolve unusual problems of ventilation prior to intubation.
REFERENCES 1. Sedano H, Cohen MM, Jirasek J, et al: Frontonasal dysplasia. J Pediatr 76:906-913, 1970 2. Cohen MM, Sedano H, Gorlin RT: Frontonasal dysplasia (Median cleft face syndrome) : Comments on etioloev and Dathoeenesis. Birth Defects: Original articl;? series, k o l GII, No. 7:117119, 1971
3. Kattwinkel J, Fleming D, Cha CH, et al: A device for administration of continuous Dositive airway pressure by the nasal route. J Pediatr 52: 131-134, 1973
FIG3. Shows the nasopharyngeal airway in place postoperatively.
4. Agostino R, Orzalesi M, Nodari S, et al: Continuous positive airway pressure (CPAP) by nasal cannula in the respiratory distress syndrome (RDS) of the newborn. Ped Research 7:50, 1973
PLASMA LEVELS OF LIDOCAINE. Venous (plasma) levels of lidocaine were measured in 5 subjects following nebulization of either a 280-mg dose using intermittent positive pressure breathing (IPPB) or a 400-mg dose using ultrasound. Even though a lower dose of lidocaine was given by IPPB, this system of delivery produced higher plasma concentrations of the drug than ultrasound but was more effective i n eliminating t h e gag reflex. I n no subject did plasma concentrations of lidocaine exceed 1.1 pg/ml, which is f a r below toxic levels (>5 pg/ml). Aerosolized lidocaine, administered by IPPB, i s a safe and effective topical anesthetic agent which may be useful for instrumentation of t h e upper airway. (Chinn WM, Zavala DC, Ambre J : Plasma levels of lidocaine following nebulized aerosol administration. Chest 71 :346-348,1977)
