Otolaryngology

An unusual case of epistaxis in a four month old

a b s t r a c t

Epistaxis is a routine complaint in Emergency Medicine and presents most commonly in adults and children and its incidence increases with age. It is rare in infants and neonates. We discuss a case of epistaxis in a four-month- old male who presented to a critical access hospital. What initially appeared to be routine brisk epistaxis was later discovered to be a large, complex, epiglottic hemangioma. The patient was stabilized using topical Tranexamic acid, Nasal packing with ketamine sedation, and sent to a tertiary care center for definitive management. He re- quired advanced airway management in the OR for definitive airway management for airway-obstructing hem- angioma excision.

Introduction

Epistaxis is a routine complaint in Emergency Medicine and its inci- dence increases with age [1]. It is rare in infants and neonates [2]. This report discusses a case of epistaxis in a four-month-old male.

1. Case report

A four-month-old Native American male presented to a rural, critical access emergency department with his foster mother for epistaxis. Chart review noted his biological mother suffered from substance use disorder and the child was in foster care due to prior nonaccidental trauma. There was concern of congenital syphilis upon birth, but he completed antibiotic treatment. The patient also had a history of admis- sion for RSV Bronchiolitis and hypoxemia that required supplemental oxygen via high flow nasal cannula but not endotracheal intubation. He had a normal growth curve and reassuring primary care check-ups. He was examined immediately upon arrival to the ED. He had brisk bleeding from his left nare and was being propped up by his foster mother, but was able to cry and cough to clear the bloody secretions ap- propriately. He weighed 7.8 kg. His initial vital signs were: HR 160, RR

60, O2 95% on room air.

Upon difficult visualization of his left anterior nare, brisk bleeding was noted without an easily identifiable source. He was administered oxymetazoline spray with slowed bleeding. The oropharynx was bloody

* Corresponding author.

E-mail address: [email protected] (A.E. Robinson).

without an obvious source of bleeding. The initial exam was concerning for severe epistaxis with a possible posterior bleed. Given the age, the differential diagnosis included arteriovenous malformation (AVM), hemangioma, retained foreign body, trauma, or coagulopathy as a pos- sible source. He continued to cry and protect his airway. He was placed on end tidal CO2, oximetry, and cardiac monitoring. Airway equipment was prepared, including an endotracheal tube, two supraglottic devices (iGel(TM), King LTSD(TM)) and a transtracheal jet ventilator. His labs returned with a hemoglobin of 10.7 and a venous blood gas with a pH of 7.44 and a pCO2 of 31 mmHg, diagnostic of respiratory alkalosis sec- ondary to tachypnea. His platelet count was 650,000 per mm³ and his international ratio (INR) was not resulted due to lab error.

His nose continued to bleed when pressure was released. Transfer to a pediatric ICU bed at a tertiary care facility was 25 min away by air transport or 90 min by ground transport. Given the airway concerns, the provider elected flight. The patient was administered ketamine 3 mg/kg intramuscular for nasal packing. The patient’s nasal passage was packed with a 2.5 cm Rhino Rocket(TM), the smallest available, soaked with Tranexamic acid , and the balloon was filled with 2 mL of air until the confirmatory balloon was sufficiently inflated. Packing notably reduced the bleeding, however the patient continued to cough, albeit less frequently. A peripheral IV was established and the flight crew ar- rived at that time. He was observed for approximately 25 min and was administered an additional dose of ketamine 1 mg/kg IV for contin- ued sedation. He continued to protect his airway and tolerate secretions without evidence of hypersalivation. His transfer was without incident. Upon arrival at the tertiary care facility, his repeat labs 4 h after ini- tial presentation were notable for a hemoglobin of 8.5 g/dL. The nasal

https://doi.org/10.1016/j.ajem.2021.04.035

Fig. 1. Saggital plane MRI image of epiglottic hemangioma.

packing was left in place overnight after evaluation by the otolaryngol- ogy (ENT) service, and was removed the following morning. The bleed- ing had stopped. An MRI was obtained and was noted to have a small abnormality in the left nare that was initially thought to be the source of bleeding. ENT performed nasopharyngoscopy and noticed a small amount of soft tissue swelling of the superior posterior nare that was suspected to be the source of bleeding. They planned to go to the oper- ating room for a biopsy. Per the mother, the patient had a history of “odd breathing.” Due to this, ENT performed a sedation-facilitated direct lar- yngoscopy prior to intubation for the biopsy. During laryngoscopy, they noticed a large, vascular mass on the epiglottis that was very friable and showed stigmata of recent bleeding. The procedure was aborted and the patient was emergently transferred to a quaternary pediatric medical center for subspecialist ENT consultation. The source of epistaxis was determined to be brisk bleeding from the epiglottic mass. He underwent an MRI of the face and neck, which suggested that the mass was a hem- angioma (more favorably) or a teratoma and was causing mild tracheal deviation (Fig. 1). In the operating room, endotracheal intubation was difficult because the mass completely obstructed the airway. With a fiberoptic scope, providers were able to circumnavigate the mass and confirm endotracheal tube placement. Next, a biopsy confirmed a hem- angioma. The patient required a transfusion, steroids, and propranolol administration. The patient had prolonged intubation secondary to bi- lateral pneumonia versus pneumonitis. On postoperative day 5, bedside scope demonstrated no change in the mass despite propranolol, and due to the airway-impeding location, he was taken to the operating room a second time for resection. He was successfully extubated on post-operative day 7. On six-month follow-up, the patient is doing well. There were no apparent complications from his hospitalization.

Discussion

Epistaxis is a common emergency department complaint. It is most commonly caused by digital trauma, but epistaxis is not usually seen in young infants because of their poor dexterity [2]. Literature on supra- glottic or epiglottic hemangiomas remains rare [3], and there is a paucity of case reports describing even nasopharyngeal hemangiomas [4].

It is important for the emergency physician to be aware that epi- staxis may be caused by airway hemangioma, in order to be wary of any airway manipulation, and to be prepared for severe hemorrhage if manipulation becomes necessary. To the unaware, the patient’s initial presentation, though uncommon, might seem to be another routine

epistaxis; however, this case illustrates that epistaxis, which is rare in infants, is unlikely to be benign in this age, and indeed may be due to a life-threatening hemangioma or AVM. In cases like this, especially in extremes of age, it is important to have a provider present who is skilled at emergency airway and sedation. It is probable that the infant’s hem- angioma was bleeding so briskly he was coughing and expelling blood through the nostrils, though this was difficult to discern from the ex- pected airway reflexes that would have occurred due to posterior epi- staxis. In addition, bleeding stopped with TXA-soaked nasal packing. TXA is a lysine-based medication that is an anti-fibrinolytic with many possible topical applications [5]. It is possible that the TXA may have dripped posteriorly and topically coated the hemangioma after nasal packing, slowing the acute hemorrhage.

Procedural sedation with Intramuscular ketamine was used to facilitate both nasal packing, IV placement, and safe transport. There is a lack of literature on ketamine in this age demographic [6-9]. There is concern that airway compromise, including laryngospasm and apnea, are more common in infants due to their upper airway anatomy; how- ever, these claims are largely based on case reports [9-11]. Multiple air- way adjuncts were prepared as described above, however, the critical access facility did not have video laryngoscopy available. This likely would have made intubation more challenging, especially in the setting of severe bleeding [12]. Ketamine was used due to familiarity with the drug and its relative safety profile. [6,13,14]. There was no preme- dication for secretions, though preparations were made to manage them with glycopyrrolate and bedside equipment [13,14]. Generally, children less than 6 months of age are not given ketamine [10]. How- ever, the provider was most comfortable with ketamine in this setting given its more stable (but not absent) effects of respiratory rate comp- ared to a benzodiazepine such as midazolam. The patient tolerated ketamine very well with no signs of respiratory failure and maintained his cough reflex.

Rapid Sequence Intubation was considered for airway pro- tection prior to transport and the need for sedation to maintain nasal packing. In retrospect, with the knowledge of an airway-obstructing hemangioma, RSI might have resulted in an airway disaster. The airway was able to be successfully managed in the operating room at the qua- ternary center with more sophisticated equipment and resources.

Propranolol is first line therapy for complicated infantile hemangi- omas [15]. It helps to stop further growth and promotes regression of the mass [15,16]. The mechanism of action is not clear but it may cause vasoconstriction, induce apoptosis, and modify angiogenesis via modulation of growth factors [16]. It is usually given for 6-12 months. However, in this patient, prolonged treatment was aborted in favor of emergent resection of the mass to avoid airway compromise or life- threatening hemorrhage into the airway.

Conclusion

Emergency providers must keep a high index of suspicion for un- usual and dangerous pathology when presented with unusual cases. While epistaxis is often benign, especially in the pre-school and older population, broader differentials must be considered when presenting in the extremes of age. Having airway adjuncts like supraglottic devices, video laryngoscopy, and disposable fiberoptic scopes are valuable in all emergency settings, even the austere, can be life-saving The decision to intubate a tenuous airway, and understanding the risks and benefits in limited resource settings is crucial.

Declaration of Competing Interest

None of the authors have conflicts of interest to disclose.

References

AJEM

An unusual case of epistaxis in a four month old

a b s t r a c t

Leave a Reply Cancel reply