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Intravenous Caffeine Rescue for Postoperative Hypoventilation in a 16-Year-Old With Trisomy 10: A Case Report

Evans, Michael MD*; Lam, Humphrey MD*†; Austin, Thomas MD*†

doi: 10.1213/XAA.0000000000000523
Case Reports: Case Report
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Trisomy 10 is a rare disorder, with only 35 cases reported in the literature. Anesthetic management may be challenging in this patient population because of craniofacial, cardiac, and renal abnormalities commonly seen in the disorder. We describe a 16-year-old male with an anesthetic history notable for prolonged emergence, postoperative hypoxia, postoperative reintubation, and unexpected hospital admission presenting for dental extraction of impacted teeth. We utilized intravenous caffeine, a respiratory stimulant used in preterm infants, to facilitate recovery from anesthesia.

From the *Emory University School of Medicine; and Children’s Healthcare of Atlanta at Egleston, Atlanta, Georgia.

Accepted for publication January 23, 2017.

Funding: None.

The authors declare no conflicts of interest.

Address correspondence to Humphrey Lam, MD, Emory University School of Medicine, Children’s Healthcare of Atlanta at Egleston, 1405 Clifton Rd NE, Atlanta, GA 30322. Address e-mail to humphrey.vo.lam@emory.edu.

The literature is sparse with regard to trisomy 10 as a whole. Only 35 cases have been recognized since the disorder was first described in 1974.1 Very few of these articles address the anesthetic management of children with trisomy 10, and most discuss the phenotypic presentation for a certain cytogenetic abnormality. The most common abnormalities seen in trisomy 10 include craniofacial abnormalities, mental retardation along a wide spectrum, growth retardation, psychomotor retardation, hypotonia, and cardiac and renal abnormalities.

We present the case of a 75-kg 16-year-old male with trisomy 10, autism, and partial 18q deletion who presented for dental extraction of impacted teeth. His anesthetic history was notable for prolonged emergence, postoperative desaturations, and masseter muscle spasm. His surgical history included an adenotonsillectomy, multiple gastric foreign body removals, forearm fracture repair, and Nissen fundoplication. Written approval for publication of this manuscript was obtained from the parents.

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CASE DESCRIPTION

Our patient manifested many of the traits noted in trisomy 10, including mild mental retardation, autism, hypotonia, psychomotor retardation, a double inferior vena cava, and a bicuspid aortic valve. A sleep study performed at the age of 3 years was normal with the exception of frequent arousals, which are often associated with paradoxical breathing. He had a subsequent tonsillectomy and adenoidectomy, but his delayed emergence and postoperative hypopneas persisted after all subsequent anesthetics. His parents stated that he required overnight stays after all outpatient surgeries because of prolonged postoperative somnolence.

Before proceeding to the operating room for his dental procedure, the patient was premedicated with 15 mg of oral midazolam 51 minutes before induction. An uneventful inhalational induction with nitrous oxide and sevoflurane was performed. An intravenous (IV) catheter was placed. A nasal endotracheal tube was placed atraumatically. General anesthesia was maintained with sevoflurane. The patient received ketorolac 30 mg IV and fentanyl 25 µg IV for intraoperative analgesia. He was extubated awake uneventfully at an end-tidal sevoflurane concentration of 0.3% and brought to the postanesthesia care unit (PACU). In the recovery room, the patient had frequent episodes of desaturation and hypopnea that required continued reassessments by an anesthesiologist. Over time, the patient’s alertness decreased secondary to his hypoventilation. Support with a Jackson-Rees circuit improved his ventilation and alertness; however, the patient was unable to maintain his airway and alertness without support. After 90 minutes in the PACU, the parents were informed of the patient’s poor respiratory status.

Shortly after, caffeine citrate 60 mg was administered IV over 30 minutes. Within 15 minutes of administration, the patient’s alertness (response to pain/verbal stimuli) and O2 saturations (93% to 98%) improved. Respirations remained in the 16 to 22 per minute range but were no longer shallow. His blood pressure and heart rate remained stable. After 85 minutes, the patient was discharged in stable condition, with no further oxygen desaturations after the administration of caffeine.

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DISCUSSION

Caffeine can suppress and abort postoperative apneas in preterm infants. A loading dose of caffeine 10 mg/kg IV leads to a sustained increase in diaphragmatic activity with an associated increase in tidal volume in preterm infants.2–4 Caffeine may enhance diaphragmatic contractility via adrenal release of epinephrine, an increase sarcoplasmic reticulum calcium concentration, or central stimulation.4 Caffeine also has a potential role in antagonizing the ventilator depressive effects of opioid analgesics. It is a nonselective phosphodiesterase inhibitor with adenosine antagonistic activity.

Kimura et al5 demonstrated the reversal of opioid-induced respiratory depression by caffeine in anesthetized rats. Wang et al6 performed an animal study that showed caffeine significantly accelerating recovery from general anesthesia from both isoflurane and propofol. Additionally, a randomized, double-blind, placebo-controlled trial investigated the use of IV caffeine in adults with obstructive sleep apnea undergoing uvulopalatopharyngoplasty. Sixty patients were blindly grouped to receive 500 mg of caffeine benzoate or normal saline after surgery. Response to verbal commands and PACU duration were significantly shorter in the caffeine arm. The number of patients with adverse postextubation events in recovery (desaturation, supraglottic obstruction, breath holding, laryngospasm, and reintubation) was significantly decreased in the caffeine-treated group.7 Khalil et al8 studied the administration of caffeine benzoate 20 mg/kg IV in children with obstructive sleep apnea undergoing adenotonsillectomy. The children treated with caffeine had fewer adverse postextubation respiratory events.

In our patient, we used a substantially smaller dose of caffeine (60 mg) than that suggested in the literature. The patient could have received 500 mg of caffeine if dosed like an adult. However, the patient was caffeine-naïve per his parents. Despite the wide therapeutic window of caffeine, we were unsure how a patient with a unique genetic history was going to respond to a large loading dose. Thus, we decided to administer an amount of caffeine that is equivalent to a can of soda or cup of coffee, with the thought that we would administer a larger dose if the initial dose was ineffective.

The patient’s improvement in alertness and ventilation may have occurred temporally with administration of caffeine being mere coincidence. However, given the patient’s previous postanesthetic history, his prolonged sedation is most likely an idiosyncratic effect due to his genetic disorder rather than a standard anesthetic effect. This is shown in the pharmacokinetics of inhaled anesthetics and midazolam. Picard et al9 studied the recovery of 24 patients aged 3 to 10 years from sevoflurane anesthesia. In the study, the mean duration of anesthesia under sevoflurane 2% to 3% was 54 minutes. The mean time to extubation (from the end of anesthesia) was 14 minutes; time to response to verbal command was 21 minutes; and time to discharge from the recovery room was 45 minutes, with a range of 24 to 75 minutes (no standard deviation was noted). Criteria for discharge were being fully awake, able to cough or breathe deeply, moving all limbs voluntarily and maintaining oxygen saturation of >93% on room air. In an adult study, Dogru et al10 reported 9 minutes to extubation, and time to discharge was 38 minutes under sevoflurane anesthesia, with a standard deviation of 8 minutes. Our patient was exposed to sevoflurane (1.1%–2.3%) for 52 minutes with minimal opioid administration. However, our patient was unarousable after 90 minutes in the PACU, beyond the upper range of the Picard study and larger than 3 standard deviations greater than the mean of the Dogru study. Thus, the normal sedative effect of sevoflurane should have been minimal before caffeine administration.

Another argument for delayed discharge was that the patient received preoperative midazolam 0.5 mg/kg orally. Oral midazolam has been shown to delay recovery in anesthetics <30 minutes in children.11 Our 75-kg patient received 0.2 mg/kg 51 minutes before induction. The maximal sedative effect after oral administration is 30 minutes, and the serum concentration peaks at 50 to 60 minutes.11 The serum concentration can stay at therapeutic levels for light sedation until 2 hours after administration for oral doses of 0.45 mg/kg and above.11 Before the patient received a lower midazolam dose and entered the PACU 2 hours after administration, the normal sedative effect of midazolam should have been eliminated before caffeine administration.

All in all, our patient had a better postoperative course than his previous surgeries, which were plagued with reintubation and overnight admissions. All his previous anesthetics were similar to the one we provided except for IV caffeine in the recovery room. We believe that in this particular case, caffeine achieved our desired goals—increased alertness and adequate ventilation. Caffeine could be a useful tool for a hypoventilating patient who is at an increased risk for postoperative respiratory complications.

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DISCLOSURES

Name: Michael Evans, MD.

Contribution: This author helped design the work, and write and revise the manuscript.

Name: Humphrey Lam, MD.

Contribution: This author helped design the work, and write and revise the manuscript.

Name: Thomas Austin, MD.

Contribution: This author helped design the work, and write and revise the manuscript.

This manuscript was handled by: Raymond C. Roy, MD.

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REFERENCES

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