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The Laryngoscope

C 2016 The American Laryngological,


V

Rhinological and Otological Society, Inc.

Outcomes of Recalcitrant Idiopathic Epistaxis in Children:


Septoplasty as a Surgical Treatment
Jessica M. Levi, MD; Katherine M. McKee-Cole, MD; Patrick C. Barth, MD; Robert M. Brody, MD;
James S. Reilly, MD
Our objective was to measure short- and long-term outcomes of children presenting with recalcitrant idiopathic epistaxis. The study was an 11-year (20002011) retrospective chart review of children evaluated and treated for epistaxis. A
retrospective review of patients with diagnostic International Classification of Diseases, Ninth Revision code 784.7 (epistaxis) and 21.5/21.88 (septoplasty) was completed reviewing age at presentation, type of surgery, and number of bleeding
events prior to and after surgery. In our cohort, almost 100% of children with idiopathic recurrent epistaxis responded to
topical treatments. About 0.2% were refractory (20/9239), and 90% of those (18/20) resolved with either a traditional
septoplasty (14/20) or modified septoplasty (6/20) without cartilage excision, with a mean follow-up of 35 months. One of
the patients who did not show resolution was found to have Von Willebrand disease, which likely contributed to this outcome. Septoplasty surgery, with or without cartilage removal, appears beneficial for refractory idiopathic epistaxis in
children.
Key Words: Epistaxis, septoplasty.
Level of Evidence: 4
Laryngoscope, 00:000000, 2016

INTRODUCTION
Fifteen percent of the US population experiences at
least one episode of epistaxis yearly, with a total of 400
million incidents per year.1 Though rare in infants, the
incidence of epistaxis increases with age and peaks in the
elderly.2 Episodic idiopathic epistaxis is a common complaint in children. Although many episodes are minor
and are treated at home without significant blood loss, a
small subset of children experience continued recurrent
idiopathic epistaxis. Without effective treatment, these
children may develop anemia, have decreased quality of
life, and have increased absence from school and daycare.
The ideal treatment for this subset of children has
not been elucidated. A Cochrane review in 2004 found
only three studies examining different treatment options
for otherwise healthy children with recurrent epistaxis,
including petroleum jelly, silver nitrate cautery, and
antiseptic ointment. Taken together, these studies did

From the Nemours/Alfred I. duPont Hospital for Children (J.M.L.,


Wilmington, Delaware; Boston University,
Boston Medical Center (J.M.L.), Boston, Massachusetts; Thomas Jefferson
University (K.M.M.-C., P.A.B., R.M.B., J.S.R.), Philadelphia, Pennsylvania,
U.S.A.
Editors Note: This Manuscript was accepted for publication
March 7, 2016.
Presented as a poster at the American Society of Pediatric Otolaryngology, Las Vegas, Nevada, May 1618, 2014.
The study was supported by a Nemours Foundation Study Grant.
The authors have no other funding, financial relationships, or conflicts of interest to disclose.
Send correspondence to Katherine McKee-Cole, MD, Nemours/
Alfred I. duPont Hospital for Children, PO Box 269, Wilmington, DE
19899. E-mail: katiemckeecole@gmail.com
K.M.M.-C., P.A.B., R.M.B., J.S.R.),

DOI: 10.1002/lary.26010

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not provide enough evidence to make any treatment


recommendations.3
In our study, we examined the outcomes of children
with idiopathic recurrent epistaxis recalcitrant to medical management and operative nasal cautery treated
with traditional septoplasty and a modified version of
this procedure (involving elevation of mucoperichondrial
septal flaps without removal of septal cartilage) used in
those without a septal deviation.

MATERIALS AND METHODS


The Nemours institutional review board approved this retrospective electronic medical record review. We searched the
Nemours Childrens Health medical records from facilities in
Orlando, Florida and Wilmington, Delaware from January 2000
to October 2011 for International Classification of Diseases,
Ninth Revision codes 784.7 (epistaxis) and 21.5/21.88 (septoplasty). All children who received a septoplasty without epistaxis as an indication were excluded. The resulting cohort of
children was analyzed into two groups: those who underwent
traditional septoplasty with removal of septal cartilage and
those with no cartilage removal. Cartilage was removed if a septal deviation was present. The children have been followed for 3
years postprocedure.
The modified septoplasty (MSP) involves a caudal incision
made along the anterior portion of the septum (Fig. 1). Either a
hemitransfixion incision or Killian incision is used based on surgeon preference, but we believe a hemitransfixion is superior
because it is located more anteriorly and therefore is more
likely to include all of the problematic blood vessels. A subperichondrial flap is then raised posteriorly to the bony cartilaginous junction down to the bony nasal floor using a Cottle or
Freer elevator. In the case of a Killian incision, an incision is
made through the cartilage to gain access to the subperichondrium on the opposite side. For the hemitransfixion incision,

Levi et al.: Recalcitrant Idiopathic Epistaxis in Children

Fig. 1. Illustrations showing elevation of the


mucoperichondrial flap. A Killian, full transfixion, or hemitransfixion incision may be
performed. A hemitransfixion or full transfixion incision may allow for greater inclusion
of anterior venous plexus vessels. [Color
figure can be viewed in the online issue,
which is available at www.laryngoscope.
com.]

the contralateral mucoperichondrium is identified anteriorly.


This is then dissected free from the septal cartilage using a Cottle or Freer elevator. Once the mucoperichondrial flaps are
raised on each side, the initial incision is closed and a mattress
stitch with chromic suture is placed, including both mucoperichondrial layers as well as the intervening septal cartilage. No
packing is placed. In the traditional septoplasty, the above procedure is performed with some portion of the septal cartilage
and/or bone removed.

RESULTS
During the study period, 4,392,548 pediatric visits
were seen within our pediatric healthcare system, and
14,607 (0.33%) of these were for epistaxis as a chief complaint. There was an average of 1.6 visits per child, and
the total number of children with epistaxis identified
was 9,239. Of these, 61% were male and 39% were
female.
From this subset of 9,239 children, 6,718 were successfully managed medically with bacitracin to the bilateral anterior nares at bedtime and nasal saline spray
three times per day. The duration of these treatments
ranged from 2 weeks to 6 months based upon the lack of
recurrent epistaxis with treatment cessation. Additionally, environmental modifications, such as a humidifier
in the bedroom, and refraining from digital or other
trauma, were recommended to all patients. In the initial
9,239 children, there were 2,521 procedures completed,
primarily nasal cauterization undertaken in the operating room for those patients who had persistent epistaxis
despite maximal medical management. This subset of
patients had a laboratory evaluation with a complete
blood count and coagulation factors. Twenty-seven percent of the children had surgical intervention. From this
group, 100 encounters were identified involving septoplasty in children with a previous diagnosis of epistaxis.
These 100 encounters involved 35 distinct patients.
Upon review, 15 children had septoplasty performed for
reasons other than epistaxis and were excluded from the
study. Among the 20/9239 (0.02%) remaining patients
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who had septoplasty performed for epistaxis, 14 underwent a traditional septoplasty, and six underwent the
modified procedure described.
This patient population consisted of 14 males and
six females, with a mean and median age at presentation of 11.1 and 10.5 years, respectively (standard deviation [SD] 5 4.6 years; range, 417 years). The MSP
group consisted of four males and two females, with a
mean and median age of 9.0 and 8 years respectively
(SD 5 4.4 years; range, 417 years). The traditional septoplasty group consisted of 10 males and four females,
with a mean and median age at presentation of 12.1 and
14 years, respectively (SD 5 4.5 years; range, 417
years).
Each child receiving an MSP had experienced
recurrent weekly or daily nosebleeds for a duration
ranging from 3 to 120 months. Every patient in the MSP
group failed medical management and underwent nasal
cautery in the operating room at least once prior to
definitive surgery (mean 5 1.3 nasal cautery procedures;
SD 5 1.03). Children receiving traditional septoplasty
were a more heterogeneous group; frequency of epistaxis
ranged from nightly to weekly and duration ranged from
5 to 132 months. One of the patients in this group had
undergone multiple prior office nasal cauterizations, one
had a prior septoplasty, one had a prior open-reduction
internal fixation of nasal bone fractures, and one had
prior nasal surgery of unknown type outside of the
United States.
Among the 20 patients treated with traditional or
MSP for epistaxis, 18 experienced complete resolution of
their symptoms at their follow-up 1 month after surgery.
Thirteen of the 14 patients in the traditional septoplasty
group were free of epistaxis postoperatively, with one
patient lost to follow-up. Five of the six patients in the
MSP group were free of epistaxis postoperatively. Two of
these five patients experienced minor episodes of epistaxis in the immediate postoperative period, with ultimate resolution 1 week postoperatively. The patient in
the MSP group who continued to experience epistaxis
Levi et al.: Recalcitrant Idiopathic Epistaxis in Children

TABLE I.
Surgeries Attempted Prior to Modified Septoplasty/Septoplasty, Surgeries Performed, and Outcomes Achieved.
Age at
Presentation, yr

Sex

Surgeries Attempted Prior


to TS/MSP

Surgery
Performed

Cautery x 3

MSP

Complete resolution

8
10

M
F

Cautery x 1
Cautery x 1

MSP
MSP

Found to have Von Willebrand disease


Complete resolution

Cautery x 2

MSP

Complete resolution

Cautery x 1

MSP

2 episodes 1 week immediately postoperatively


then resolution of epistaxis

17

Cautery x 1

MSP

16

Nasal surgery in Jamaica

TS

2 episodes 1 week immediately postoperatively


then resolution of epistaxis
Complete resolution

16

Cautery x 1

TS

Complete resolution

15
17

M
M

Cautery x 2
Cautery x 1

TS
TS

Complete resolution
Complete resolution

Cautery x 1

TS

Complete resolution

16
16

M
F

Prior septoplasty
Cautery x 2

TS
TS

Complete resolution
Complete resolution

15

Cautery x 2

TS

Complete resolution

13
11

F
M

Cautery x 1
Cautery x 1

TS
TS

Complete resolution
Complete resolution

Cautery x 2

TS

Complete resolution

5
4

M
M

Multiple cautery procedures in office


Cautery x 1

TS
TS

Complete resolution
Complete resolution

ORIF nasal fx

TS

Complete resolution

Epistaxis Postsurgery

F 5 female; fx 5 fracture; M 5 male; MSP 5 modified septoplasty; ORIF 5 open reduction internal fixation; TS 5 traditional septoplasty.

postoperatively was found to have Von Willebrand disease, the likely cause of his recurrent epistaxis and continued need for repeated cautery (Table I).
This cohort as a whole had been followed with office
visits and phone calls from parents if resumed trouble
with epistaxis developed for a mean period of 35 months
postoperatively (SD 5 16 months). The MSP group had
been followed for a mean 35 months (SD 5 6.11 months).
The traditional septoplasty group had also been followed
for a mean of 35.8 months (SD 5 19.4 months).

DISCUSSION
Recurrent idiopathic epistaxis is a common childhood complaint. Bleeding most frequently originates from
Kiesselbachs plexus, located in the anteroinferior portion
of the nasal septum, which is formed by the anastomosis
of the anterior ethmoidal, sphenopalatine, greater palatine, and superior labial arteries. Drying environmental
factors or nose picking (digital manipulation) can lead
to trauma of this delicate area and promote bleeding.
Though nasal cautery can staunch bleeding, recurrent
bleeding may still occur. In such cases, many practitioners simply cauterize any offending sites multiple
times and eventually find success, either by inducing
enough scarring to prevent the vessels from bleeding, or
perhaps more likely, from the children simply outgrowing
the episodes of epistaxis. Multiple cauterizations, however, can require multiple administrations of general
anesthesia and carry a small risk of septal perforation.
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Some practitioners only cauterize one side of the nose at


a time to avoid this potential risk, so patients with bilateral bleeding can have both delayed treatment and
delayed symptom resolution.
Although small in number, some children continue
to suffer from epistaxis despite interventions with conservative measures and nasal cautery. Currently, there
is no precise definition on what constitutes failure to
respond to nasal cautery after medical management,
and the number of attempts differs among physicians.
However, for this set of patients showing recalcitrant
epistaxis after one or more attempts at nasal cautery, a
more definitive procedure can certainly be appealing as
an alternative to continued administrations of nasal
cautery.
In a single case report from 1987, Guarisco et al.4
demonstrated efficacy of a limited septoplasty as a therapy for a child with recurrent epistaxis secondary to
Glanzmann thrombasthenia. There have been no followup studies of this nasal septoplasty procedure in larger
patient populations. To our knowledge, there are no publications regarding septoplasty for treatment of epistaxis
in adults or in children with idiopathic recurrent epistaxis as studied here.
In 1998, an animal study performed by Dano et al.5
showed that supraperichondrial dissection of the septum
leads to reduced vascularity of the septal mucosa on histologic inspection. The author performed unilateral dissections on the septal mucosa of 16 laboratory rabbits
and analyzed the number of blood vessels as well as the
Levi et al.: Recalcitrant Idiopathic Epistaxis in Children

area occupied by those blood vessels compared to the


opposite side, which functioned as a control. This study
lends further credence to the theory that by raising and
replacing a mucoperichondrial flap, the fibrosis and
reduced vascularity of the nasal septal mucosa prevents
future episodes of epistaxis. A study of this type, evaluating the area occupied by blood vessels on each side of
the septum after a unilateral mucoperichondrial flap has
been raised and replaced, and mattress sutures have
been placed through the septum and mucoperichondrium on each side multiple times, could help delineate
if there is an histologic benefit to bilateral over unilateral mucoperichondrial flaps. If there is not a difference
on each side, the potential risk for perforation by raising
bilateral mucoperichondrial flaps may be avoided. Additionally, this study by Dano et al, has not been repeated,
and this could be done to evaluate whether the results
are indeed reproducible.
A stepwise approach to treatment, beginning with
simpler measures and advancing to more complex medications and surgical interventions, is employed with this
common event. Prevention begins with avoidance of finger scratching or manipulation into the nasal cavity and
is bolstered by the application of topical creams or petroleum jelly. The optimal treatments for acute episodes
include external nasal pressure and oxymetazolinesoaked pledgets. Significant epistaxis uncontrolled by
medical management ultimately requires embolization
or surgical treatment. Embolization occludes the arterial
supply of the nasal mucosa to staunch bleeding though
ischemia; however, a low risk of facial tissue infarction
is associated with this intervention.6,7 Surgery is aimed
at locating and controlling the current offending vessels.
For children with idiopathic recurrent recalcitrant epistaxis, chemical cautery with silver nitrate is often performed first; if this intervention does not sufficiently
stop any bleeding electrocautery may be necessary.
Unfortunately, there is a small subset of this population
that fails medical management and nasal cautery, and
for such recalcitrant patients the next step is often
unclear.
In entities associated with potentially frequent and
catastrophic hemorrhage, such as hereditary hemorrhagic telangiectasia, more extensive ablation of nasal
mucosa can be accomplished with lasers, locoregional
nasolabial and forehead flaps, or even free flaps such as
the radial forearm free flap.812 The potassium titanyl
phosphate laser has been used to more broadly coagulate
the nasal mucosa.8 Coblation has also been studied, as
have argon plasma coagulation and even intensitymodulated radiotherapy.911 There are significant complications and morbidity associated with the flap procedures listed, such as scarring at the donor split
thickness skin graft site, as well as common complaints
of nasal crusting and foul odor. Variations of Youngs
procedure,12 which closes off the nasal cavity entirely by
means of mucocutaneous flaps, could also rarely be performed. Though these extensive surgeries could be utilized in patients with recalcitrant bleeding, more
conservative measures with less morbidity should be
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attempted first and generally are successful in pediatric


patients with recurrent idiopathic epistaxis.
We propose that septoplasty and MSP are efficacious in treating recurrent recalcitrant pediatric epistaxis because of fibrosis and scarring of causative
vasculature on the mucosal flaps and, in the case of septoplasty removal, of deviated cartilage responsible for
turbulent airflow. Additionally, MSP provides a treatment modality for idiopathic recurrent recalcitrant epistaxis in those without septal deviation, a feature
previously used to justify the septoplasty technique.
Without removal of septal cartilage, MSP has a
decreased risk for morbidity compared to traditional septoplasty, which can include complications such as septal
perforation and, rarely, cerebrospinal fluid leak. Additionally, reports exist of alteration of facial growth with
removal of the cartilaginous septum in young children.
In consideration of its high efficacy, decreased risk of
morbidity, and minimal alteration to the anatomic structure of the nose, we prefer MSP to traditional septoplasty in the specific patient subset without septal
deviations as a treatment for recurrent idiopathic epistaxis resistant to cautery, though both are efficacious.
Although this procedure was greatly successful in
89% patients, two patients had a small amount of epistaxis immediately postoperatively. This may be because
of a lag period prior to the establishment of scarring and
fibrosis. The failure of this procedure to control epistaxis
in one patient with Von Willebrand disease suggests
that, although the procedure may be used in conjunction
with other treatment types, the patient and family
should be counseled that recurrent epistaxis after the
procedure can occur with acquired coagulopathies,
potentially requiring further intervention.
Idiopathic recurrent recalcitrant pediatric epistaxis
lacks published evidence, limiting the literature available for presentation. This study was limited by its retrospective nature as well as variable follow-up times. A
future prospective multicenter trial with a standardized
operative technique could increase the patient volume
available for analysis and improve the ability to draw
conclusions regarding the effect of this intervention.
Potentially, using the rabbit model discussed above with
histopathologic evaluation, modifications of this procedure to increase fibrosis may be investigated as well,
such as application of topical iodine, tetracycline,
increased absorbable suturing, use of a medical grade
adhesive, or mechanical abrasion of the medial flap
surface.

CONCLUSION
Many conservative topical treatments have been
recommended for epistaxis and are effective for more
than 99.8% of children. Ninety percent (18/20) of children with persistent idiopathic recurrent epistaxis experienced complete remission after septoplasty or MSP
with a mean follow-up of 35 months. Our evidence suggests that septoplasty with or without cartilage removal
is a potentially effective means of treatment for children
with idiopathic refractory epistaxis.
Levi et al.: Recalcitrant Idiopathic Epistaxis in Children

Acknowledgments
The authors thank Erin Field, PA, for providing the original figure.

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