<div>High-flow Nasal Cannula Oxygen Therapy for Respiratory Management after
Postoperative Re-intubation/Re-extubation in Patients with Trisomy 18
and Trisomy 13: Two Case Reports</div><div>Running head: High-flow Nasal Cannula Therapy after Re-intubation</div><div>Hirofumi Hirano, M.D.<sup>1</sup>, Yoshie Taniguchi, M.D.,
Ph.D.<sup>1</sup>, Masato Kato, M.D., Ph.D.<sup>2</sup></div><div><sup>1</sup>International University of Health and Welfare
Hospital</div><div><sup>2</sup>International University of Health and Welfare Shioya
Hospital</div><div>Corresponding author: Hirofumi Hirano</div><div>537-3 Iguchi, Nasushiobara-shi, Tochigi 329-2763, Japan</div><div>Tel: +81 287-37-2221; Fax: +81 287-39-3001</div><div>Email: hirofumi@iuhw.ac.jp</div><div>Funding: None.</div><div>Conflicts of interest: None.</div><div>Ethics approval:</div><div>Ethical approval was not obtained because our institution does not
require this for case reports.</div><div>Patient consent:</div><div>Our institution requires the consent of the patient for case reports,
the consent of a person with parental authority was obtained.</div><div>Counts:</div><div>Key Clinical Message: 32 words</div><div>Abstract: 49 words</div><div>Introduction: 122 words</div><div>Case Description: 730 words</div><div>Discussion: 543 words</div><div>Acknowledgments: 30 words</div><div>Author contributions:25 words</div><div>Overall: 1531 words</div><div>Key Clinical Message:</div><div>The use of high-flow nasal cannula oxygen therapy for respiratory
disorders in children with severe mental and physical disabilities in
the perioperative period is applicable to prevent re-intubation and
additional invasive procedures.</div><div>Abstract:</div><div>We present two cases of general anesthesia in children with 18, 13
trisomy. One patient had difficulty with intubation and had to be
reintubated postoperatively, another developed postoperative acute
respiratory distress syndrome. The use of postoperative high-flow nasal
cannula oxygen therapy to avoid reintubation is considered a feasible
strategy.</div><div>Glossary of Terms:</div><div>HFNC: high-flow nasal cannula</div><div>ARDS: acute respiratory distress syndrome</div><div>ICU: intensive care unit</div><div>POD: postoperative day</div><div>FIO2: fraction of inspired oxygen</div><div>CT: computed tomography</div><div>SaO2: arterial oxygen saturation</div><div>PaO2: partial pressure of arterial oxygen</div><div>Introduction:</div><div>Trisomy 18 and Trisomy 13 are associated with a variety of
complications, including severe mental retardation and congenital heart
disease, as well as extremely poor prognosis, which has generally
precluded aggressive medical intervention in these patients. However,
recent reports<sup>1, 2</sup> that various aggressive medical
interventions such as cardiovascular surgery can improve prognosis have
led to increasing opportunities for these patients to undergo general
anesthesia and intensive care. We report our experience with high-flow
nasal canula (HFNC) oxygen therapy for respiratory management after
postoperative re-intubation/re-extubation in two patients with trisomy
18 and trisomy 13.</div><div>Written informed consent was obtained from guardians of the patients to
be used for presentation and publication in a journal.</div><div>This article adheres to the Case Report guidelines.</div><div>Case Description:</div><div>Case 1</div><div>The female patient was aged 2 years and 3 months with a height of 71.8
cm and a weight of 5.35 kg. She had trisomy 18 with mental retardation,
interventricular septum defect, interatrial septum defect, patent ductus
arteriosus, and pulmonary hypertension. No surgery was performed for
these anomalies and only laparoscopic gastrostomy was performed this
time.</div><div>General anesthesia was induced slowly with sevoflurane and maintained
with air, oxygen, sevoflurane, and remifentanil. Anticipating difficulty
in intubation due to micrognathia, cervical spinal deformity, limited
mandibular elevation, and leftward deviation of the head and neck,
instead of trying to intubate with a laryngoscope, we decided to
intubate with a video laryngoscope from the beginning. An endotracheal
tube with a microcuff diameter of 3.5 mm was inserted into the trachea
using a video laryngoscope and a bougie. The field of view with the
video laryngoscope was not good. The axis was off and the vocal cords
were seen in an oblique position. Intubation was done once, and a leak
was noted after intubation. The operation time was 51 min, there was no
significant blood loss, and the infusion volume was 150 mL. The patient
was extubated in the operating room after the surgery was completed.
However, due to markedly labored breathing, she was re-intubated about 5
min later and admitted to the intensive care unit (ICU).</div><div>A planned extubation was performed in the operating room on
postoperative day (POD) 5.
Hydrocortisone (4 mg/kg 3 times a day) was administered in advance. The
patient remained on continuous intravenous fentanyl at 0.3 μg/kg/h until
2 h before ICU discharge, and then was transferred to the operating room
while being administered dexmedetomidine at about 0.3 μg/kg/h.</div><div>She was extubated with the bougie left in the trachea in anticipation of
re-intubation. Immediately after extubation, HFNC (MR290, Optiflow
Junior; Fisher &amp; Paykel Healthcare, Auckland, New Zealand) was started
at a fraction of inspired oxygen (FIO2) of 0.9 and flow rate of 6 L/min.
After the patient was observed in the operating room for about 75 min,
the bougie was removed, FIO2 was lowered to 0.70, and the patient was
returned to the ICU. Blood gas tests were not performed, and the HFNC
settings were evaluated and managed by arterial oxygen saturation.</div><div>She was discharged from the ICU on POD 6, continued on HFNC (FIO2: 0.2
L, flow rate: 3 L/min) in the general ward until POD 8, and was
discharged from the hospital on POD 16 with no further issues or
complications.</div><div>Case 2</div><div>The female patient was 18 years old with a height of 154 cm and a weight
of 29 kg. She had trisomy 13 with severe mental retardation and
epilepsy. Other medical history or medical concerns included
dextrocardia, cleft palate, scoliosis, hearing loss, and left
microphthalmia. The patient underwent laparoscopic gastrostomy for
difficulty in oral intake and dysphagia.</div><div>General anesthesia was induced rapidly with propofol, fentanyl, and
rocuronium and was maintained with air, oxygen, sevoflurane, and
remifentanil. No problem occurred during surgery or immediately after
extubation.</div><div>On POD 2, the patient’s respiratory condition worsened, and laboratory
and abdominal computed tomography (CT) findings were suggestive of acute
pancreatitis. She was admitted to the ICU, intubated and ventilated, and
subsequently presented with symptoms of acute respiratory distress
syndrome.</div><div>On POD 14, the patient was extubated with improvement in the findings of
pancreatitis and respiratory function. Sedation during ventilation had
been maintained with continuous intravenous propofol alone for 2 days,
with a dose of 0.5 mg/kg/h at the time of extubation. Immediately before
extubation, blood gas analysis showed a partial pressure of arterial
oxygen (PaO2) of 83.2 mmHg and arterial oxygen saturation (SaO2) of 96.9
% under spontaneous breathing (FIO2: 0.3, positive end-expiratory
pressure: 6 mmHg, pressure support: 6 mmHg).</div><div>After extubation, respiratory support with HFNC was started at a FIO2 of
0.40 and flow rate of 50 L/min. Three hours after the start of HFNC,
blood gas analysis showed PaO2 77.0 mmHg and SaO2 95.2 %. FIO2 and flow
rate were tapered according to blood gas analysis results. Subsequently,
the patient’s body movements became more vigorous, and the day after
extubation, she was switched from HFNC (FIO2: 0.35, flow rate: 20 L/min;
blood gas analysis: PaO2 64.1 mmHg, SaO2: 92.8 %) to nasal cannula
oxygen therapy (flow rate: 4 L/min).</div><div>She was discharged from the ICU without problems with oxygenation or
respiratory status.</div><div>Discussion</div><div>The commonality in the two cases presented here is that both patients
received active intervention with gastrostomy, but were then
re-intubated, admitted to the ICU, and placed under respiratory
management with HFNC after extubation.</div><div>Case 1 was a difficult-to-intubate case which, despite sufficient
preparation and care taken during anesthesia induction and intubation,
required re-intubation probably due to residual expiratory sevoflurane
concentration during extubation, airway narrowing due to extubation, and
glottis and laryngeal edema despite one-try intubation and a short
operative time. Colleti et al. reported a case of a patient intubated
with acute laryngitis in which the use of HFNC after planned extubation
avoided re-intubation.<sup>3</sup> Thus, planned extubation and
subsequent HFNC use may be one way to prevent re-intubation.</div><div>Respiratory problems are common in children, especially those with
severe mental and physical disabilities. These patients should be
observed with extreme caution immediately after awakening from general
anesthesia and extubation, and after re-intubation. To avoid the need
for additional invasive procedures such as tracheostomy triggered by
general anesthesia or surgery, careful consideration must be given to
respiratory management after extubation and, if re-intubation results,
to respiratory management after re-extubation.</div><div>In case 1, the patient was re-intubated postoperatively, but the use of
HFNC immediately after extubation yielded good results. Based on this
experience, in case 2, in which postoperative acute respiratory distress
syndrome occurred resulting in re-intubation, we decided to use HFNC
after weaning from mechanical ventilation.</div><div>HFNC requires essentially no sedatives and can be readily used in
children with severe mental and physical disabilities, who are prone to
tongue swallowing and respiratory depression even with the slightest use
of sedatives. In case 2, although the patient expressed refusal of HFNC
during its use and had to be weaned in the middle of tapering the dose
of oxygen, the use of sedatives could have been considered to continue
HFNC. The use of HFNC with sedatives has been reported in both children
and adults and is particularly common in dental treatment of children
with severe mental and physical disabilities.<sup>4-7</sup></div><div>Reported cases of HFNC use include patients with respiratory disorder or
exacerbation of bronchitis or pneumonia due to chronic
disease,<sup>8</sup> airway narrowing after surgery for laryngeal
granuloma,<sup>9</sup> postoperative management of congenital
heart disease,<sup>10</sup> and use for low-invasive procedures
and tests performed under sedation.<sup>4-6,11</sup> HFNC is
indicated for a wide range of respiratory disorders, and based on these
reports, it may be useful in the perioperative respiratory management of
children, especially those with severe mental and physical disabilities.</div><div>These patients with trisomy 13 and trisomy 18 who were re-intubated
postoperatively, the use of HFNC after extubation enabled maintenance of
a good respiratory status. The results of the present two cases suggest
that the use of HFNC for respiratory disorders in children in the
perioperative period and children with severe mental and physical
disabilities, taking advantage of its features, is applicable to a wide
range of cases as one strategy to prevent re-intubation.</div><div>In conclusion, the results of the present case series suggest that the
use of HFNC for respiratory disorders in children in the perioperative
period and children with severe mental and physical disabilities, taking
advantage of its features, is applicable to a wide range of cases as one
strategy to prevent re-intubation and additional invasive procedures.</div><div>Acknowledgments:</div><div>We thank pediatric surgeons Keisuke Fukui, Shun Watanabe and
pediatrician Hideo Shimoizumi for management of critical care and
collaboration obtained the informed consent from the guardians of the
patients.</div><div>Author contributions:</div><div>Hirofumi Hirano: This author drafted the manuscript.</div><div>Yoshie Taniguchi: This author helped prepare the manuscript.</div><div>Masato Kato: This author helped prepare the manuscript.</div><div>References:</div><div>1. Tsutsumi, M., Ohta, E., Morii, M., et al. 2010.A retrospective review
of trisomy 18 infants hospitalized from 2000 to 2009 at Fukuoka
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