Sinonasal Hemangioma: Diagnosis,
Treatment, and Follow-Up of 37
Patients at a Single Center
Jong Seung Kim, MD,* and Sam Hyun Kwon, MD, PhDy
Purpose: Hemangioma is a common benign vascular lesion of the head and neck region. It rarely in-
volves the mucous membranes of the nasal cavity and paranasal sinuses. It should be added to the differ-
ential diagnosis of nasal cavity masses when the presenting symptoms are epistaxis or nasal obstruction.
Materials and Methods: A retrospective chart review was conducted of a histologic diagnosis of
lobular capillary hemangioma or cavernous hemangioma of the sinonasal mucous membranes treated at
the Chonbuk National University Hospital from January 1995 through December 2015. There were
1,479 patients diagnosed with hemangiomas in the total body area. Medical records were reviewed to
gather data on clinical symptoms, demographic characteristics, site of tumor, imaging and histologic fea-
tures, and treatment. A review of the literature on previously diagnosed cases of sinonasal hemangioma
was performed.
Results: Three hundred patients had hemangiomas in the head and neck region and only 37 patients
(12.5%) had hemangiomas of the sinonasal mucous membranes. The most common complaint was nasal
obstruction (59.5%), followed by epistaxis (51.4%). There were 18 male (48.6%) and 19 female (51.4%)
patients. The predominant subsites were the nasal septum (40.5%), followed by the inferior turbinate
(29.7%), the maxillary sinus (8.1%), and the uncinate process (8.1%). Lobular capillary hemangioma
(24 of 37) was more common than cavernous hemangioma (13 of 37). There was a meaningful correlation
between the histologic type and tumor site of the hemangioma.
Conclusion: Sinonasal hemangiomas are relatively uncommon. Clinicians should be aware of the clin-
ical, radiologic, and histologic features of sinonasal hemangiomas to avoid a misdiagnosis of a malignant
tumor, angiofibroma, or other benign mass.
Ó 2017 Published by Elsevier Inc on behalf of the American Association of Oral and Maxillofacial
Surgeons
J Oral Maxillofac Surg 75:1775-1783, 2017
Hemangioma is the most common vascular lesion of approach. Given the complexities of hemangiomas,
the head and neck; however, it seldom occurs in the the authors summarize their 21 years of experience
sinonasal area.1 Proper diagnosis of this tumor is espe- of clinical symptoms, medical history, and endoscopic
cially important because biopsy sampling in an outpa- and radiologic findings.
tient clinic can lead to massive bleeding. Treatment of Although there are several published case reports of
this disease ranges from feeding vessel embolization sinonasal hemangiomas, there is a paucity of literature
to surgical removal by an external or endoscopic reviewing the clinical presentation, diagnosis, and
Received from Department of Otorhinolaryngology–Head and Neck Address correspondence and reprint requests to Dr Kwon: 2-20
Surgery, College of Medicine, Chonbuk National University, Jeonju, Keum-Am-Dong Department of Otorhinolaryngology–Head and
Korea; and Research Institute of Clinical Medicine of Chonbuk Neck Surgery, Chonbuk National University Medical School, Jeonju
National University and Biomedical Research Institute of Chonbuk 561-180, Republic of Korea; e-mail: [email protected]
National University Hospital, Jeonju, Korea. Received August 1 2016
*Professor. Accepted December 28 2016
yProfessor. Ó 2017 Published by Elsevier Inc on behalf of the American Association of Oral
This work was supported by the Fund of Biomedical Research and Maxillofacial Surgeons
Institute, Chonbuk National University Hospital. 0278-2391/16/31332-5
Conflict of Interest Disclosures: None of the authors have any http://dx.doi.org/10.1016/j.joms.2016.12.044
relevant financial relationship(s) with a commercial interest.
1775
1776 SINONASAL HEMANGIOMA
management of the 2 histologic types, lobular capillary who were treated from 1995 through 2015 at the
hemangioma (LCH) and cavernous hemangioma (CH). Department of Otolaryngology, Chonbuk National
This report presents a summary of the presenting symp- University Hospital (tertiary care teaching hospital)
toms and signs, radiologic and endoscopic findings, and were identified. From 1995 through 2015, 1,479 pa-
the authors’ 21 years of experience in the diagnosis and tients were diagnosed with hemangiomas in the total
management of 37 patients with sinonasal hemangiomas. body area. The clinical data of all patients with these
histologically confirmed diagnoses of hemangioma of
Materials and Methods the sinonasal mucosa were retrospectively reviewed
(Table 1). Also collected were data on preoperative im-
ETHICAL CONSIDERATIONS
aging, including nasal endoscopic evaluation and
This study was reviewed and approved by the insti- radiologic assessment by computed tomography
tutional ethical review board of the Chonbuk National (CT) and magnetic resonance imaging (MRI).
University Hospital (Jeonju, Korea) and conducted in Histologic criteria for LCH included the presence of
compliance with the Declaration of Helsinki. circumscribed ‘‘lobules’’ of capillaries arranged around
angular dilated feeding vessels, each with cytologically
STUDY DESIGN bland endothelium, and the criteria for CH included
All patients with a confirmed histologic diagnosis of larger dilated vessels with sinusoidal blood-filled inter-
LCH or pyogenic granuloma, CH, and mixed types communicating cavities arranged in a lobular or
Table 1. CHARACTERISTICS OF PATIENTS WITH SINONASAL HEMANGIOMA
Histologic Type
LCH CH P Value
Adult/pediatric sample 20/4 11/2 .920
Age (yr), mean (SD) 47.08 (22.06) 38.85 (22.07) .391
Male/female 11/13 8/5 .371
Right/left laterality 11/13 7/6 .746
Size 1.64 (1.04) 1.49 (0.76) .476
Duration 7.79 (13.00) 4.12 (6.25) .204
History .464
HTN 7 2
Nasal trauma 1
HBV 1
DM 2
Appendectomy 1
HCMP 1
Angina 2
Conchotomy 1
Hormonal replacement 1
therapy
Origin site .009
Nasal septum 10 5
Inferior turbinate 9 1
Maxillary sinus 3
Uncinated process 3
Middle turbinate 2
Posterior ethmoid sinus 1 1
Symptom .939
Nasal obstruction 13 9
Epistaxis 12 7
Rhinorrhea 4 4
Postnasal drip 3 2
Recurrence 0 2 .117
Abbreviations: CH, cavernous hemangioma; DM, diabetes mellitus; HBV, hepatitis B virus infection; HCMP, hypertrophic cardio-
myopathy; HTN, hypertension; LCH, lobular capillary hemangioma; SD, standard deviation.
Kim and Kwon. Sinonasal Hemangioma. J Oral Maxillofac Surg 2017.
KIM AND KWON 1777
diffuse pattern and lined by a single layer of Table 2. SITE OF ORIGIN OF SINONASAL
flattened endothelium. Only patients who had a HEMANGIOMA ACCORDING TO HISTOLOGIC TYPE
confirmed histopathologic diagnosis as described
earlier were included. Posterior
Maxillary Ethmoid
IT Septum UP Sinus MT Sinus Total
DATA ANALYSES
LCH 9 10 3 0 0 1 23
Patient data were analyzed according to age, gender, CH 1 5 0 3 2 1 12
laterality, size of hemangioma, clinical manifestations,
medical history, tumor site, radiologic findings, histo- Note: P < .01.
pathologic findings, and recurrence. Multivariate logis- Abbreviations: CH, cavernous hemangioma; IT, inferior
turbinate; LCH, lobular capillary hemangioma; MT, middle
tic regression tests were conducted to evaluate the turbinate; UP, uncinated process.
correlation among age, size, gender, laterality, medical
Kim and Kwon. Sinonasal Hemangioma. J Oral Maxillofac Surg
history, symptom duration, and histologic type. The c2 2017.
test was used to evaluate the correlation among
gender, laterality, origin, and histologic type. The inde-
CLINICAL SYMPTOMS
pendent t test was used to analyze differences in age,
size, and symptom duration according to histologic Of the 37 patients diagnosed with hemangioma of
type. IBM SPSS 20.0 (IBM Corp, Armonk, NY) was the sinonasal mucous membranes, the most common
used for all statistical descriptive analyses. presenting symptom at the initial visit was nasal
obstruction (59.5%), followed by epistaxis (51.4%),
rhinorrhea (24.3%), and postnasal drip (16.2%).
Results
CONCOMITANT DISEASES
DEMOGRAPHIC CHARACTERISTICS
Of 15 patients (40.5%) with underlying diseases, hy-
The patients’ average age was 44.1 years (standard
pertension was the most common (35.1%), followed
deviation [SD], 20.9 yr; range, 4 to 80 yr) and the dis-
by diabetes mellitus (5.4%), angina pectoris (5.4%),
tribution was relatively even across ages. Nineteen
and trauma (2.7%). The mean age of patients with hy-
male patients (51.4%) and 18 female patients (48.6%)
pertension was 64.89 years (SD, 14.46 yr) and the
were diagnosed with hemangioma during this period,
mean age of patients with normal blood pressure
showing no gender preponderance (Table 1). Heman-
was 37.53 years (SD, 20.04 yr), with a significant age
giomas occurred on the right (48.6%) and left (51.4%)
difference between the 2 age groups (P = .001).
sides, showing no left-versus-right preponderance.
The follow-up period after surgery ranged from 1 to
80 months (average follow-up, 11.4 months). Based TUMOR SITE
on histologic findings, 24 cases were diagnosed as The most frequently involved site was the nasal
LCH and 13 were diagnosed as CH. Of patients with septum (40.5%), closely followed by the inferior turbi-
LCH, 11 were male and 13 were female. Twenty nate (29.7%), maxillary sinus (8.1%), and uncinated
were adults older than 18 years and 4 were children process (8.1%). There were 2 cases each that occurred
younger than 18 years. Of patients with CH, 8 were at the middle turbinate and the posterior ethmoid
male and 5 were female; 11 were adults and 2 were sinus (5.4%). One case occurred at the nasal
children. There was no significant difference between dorsum (2.7%).
the adult and pediatric populations (P = .920). There
was a difference in tumor site according to histologic
type (Table 2). Mean size of the hemangiomas was Table 3. NUMBER OF PATIENTS WITH A DIAGNOSIS
OF HEMANGIOMA SEEN AT AUTHORS’ INSTITUTION
1.59 cm (SD, 0.94 cm) Surgery was performed under FROM 1995 THROUGH 2015
local anesthesia in 25 cases and under general anes-
thesia in 12 cases. Region Patients, n Proportion %
Total 1,479 HN/total 20.3
PREVALENCE HN 300 SR/HN 12.3
SR 37 SR/total 2.5
Of 1,479 patients who had hemangiomas in the total
body area, 300 patients had hemangiomas in the head Abbreviations: HN, head and neck region; SR, sinonasal
and neck region and only 37 of 300 patients (12.5%) region.
had hemangiomas of the sinonasal mucous mem- Kim and Kwon. Sinonasal Hemangioma. J Oral Maxillofac Surg
branes (Table 3). 2017.
1778 SINONASAL HEMANGIOMA
FIGURE 1. Axial contrast-enhanced computed tomogram of patient 24 shows heterogenous mottled enhancement of the mass in the left maxillary sinus.
Kim and Kwon. Sinonasal Hemangioma. J Oral Maxillofac Surg 2017.
RADIOLOGIC FINDINGS SURGICAL TECHNIQUE
Sinonasal CT was performed on 27 patients (73%). None of the patients underwent preoperative biopsy
Nineteen of these 27 patients were assessed using sampling in the outpatient clinic. All patients were
nonenhanced CT and 8 patients underwent contrast- treated by functional endoscopic sinus surgery under
enhanced CT and 6 of these patients showed enhance- local or general anesthesia, depending on the location
ment after contrast medium injection. The enhance- and size of the tumor. No known external approach
ment pattern was heterogeneous in all cases (canine fossa approach, Caldwell-Luc approach, or
(septum, 2 of 6; inferior turbinate, 2 of 6; maxillary si- lateral rhinotomy) was used. The tumor was treated
nus, 2 of 6; Fig 1). CT displayed bony destruction in 7 by en bloc resection in the sub-mucoperiosteal plane.
patients and no evidence of destruction in 20. Of pa- If this was not possible, then electrocauterization or
tients showing bony destruction, most cases occurred drilling with a diamond burr was used on the origin
at the inferior turbinate (4 of 7), whereas 2 of 7 pa- of the tumor. Total removal of the lesion was achieved
tients had destruction of the medial maxilla and 1 pa- in all cases. The wound site was left to heal by second-
tient had destruction at the posterior ethmoid sinus. ary healing with frequent saline douching. There were
Only 2 patients were assessed using MRI. A T1- no complications with this technique.
weighted image showed iso-signal intensity with an inter-
nal low signal line (Fig 2). A T2-weighted image showed
high signal intensity with an internal low signal line RECURRENCE
(Fig 3). The periphery rim was depicted with relatively Two patients with CH had a recurrence, 1 at
higher signal intensity than the center of the mass on 4 months and the other at 60 months after surgery.
the T1-weighted image and as a low signal on the T2- One patient had a diffuse tumor on the right side of
weighted image. There were heterogeneously enhanced the nasal septum, and the other patient, who was
lesions in the 2 cases (Fig 4). None of the patients under- younger, had an origin of the maxillary sinus (Fig 5).
went preoperative angiography or embolization. No recurrence was observed in patients who had LCH.
KIM AND KWON 1779
FIGURE 2. Axial T1-weighted magnetic resonance image of patient 24 shows iso-signal intensity with an internal low signal line. The margin of
the mass shows higher signal intensity than the center of the mass.
Kim and Kwon. Sinonasal Hemangioma. J Oral Maxillofac Surg 2017.
STATISTICAL ANALYSES Among the symptoms of hemangioma, there was a
A Pearson correlation test of age, size, and duration similarity between the 2 histologic types of hemangi-
of symptoms showed a positive correlation between omas (LCH and CH; P = .939; Table 1). The most com-
tumor size and duration of symptoms (P = .006; mon symptoms were nasal obstruction, followed by
R2 = 0.198; Fig 6). When patients were divided into epistaxis, rhinorrhea, and postnasal drip. Rhinorrhea
2 groups according to histologic tumor type (LCH and postnasal drip are nonspecific symptoms also seen
or CH), multivariate logistic regression analysis in allergic rhinitis and rhinosinusitis. The differential
showed no significant differences in age, gender, lat- diagnosis of nasal masses presenting with nasal obstruc-
erality, size, duration, and history of hypertension be- tion and epistaxis includes organizing hematoma, nasal
tween the 2 histologic tumor types (P > .05). polyps, angiofibroma, fungal balls, and malignancy.
However, there was a significant difference between Organizing hematoma usually damages the medial
the histologic tumor type and the tumor site (P = maxilla and ethmoid cavity, and angiofibroma usually af-
.009; Table 2). fects the pterygopalatine fossa extending to the nasal
cavity, nasopharynx, and sphenoid sinus.2,3 Fungal
balls usually show calcifications on nonenhanced CT,
Discussion
and sinonasal malignancy usually exhibits invasive and
Hemangiomas of the sinonasal mucosa are much destructive characteristics on CT and MRI, which
rarer than cutaneous hemangiomas. In this retrospec- differentiate them from hemangiomas.4
tive study, sinonasal hemangiomas composed 12.2% of There are 2 subtypes of hemangioma, determined
all head and neck hemangiomas and 2.5% of all heman- histologically.1 The 2 subtypes are differentiated by
giomas treated at the authors’ institution. dominant microscopic vessels, which contain
1780 SINONASAL HEMANGIOMA
FIGURE 3. Axial T2-weighted magnetic resonance image of patient 24 shows heterogenous strong high signal intensity in the left maxillary
sinus. The margin of the mass exhibits a low signal.
Kim and Kwon. Sinonasal Hemangioma. J Oral Maxillofac Surg 2017.
capillary-size vessels and larger endothelium-lined tion. Change of airflow can aggravate bleeding from
vascular spaces. In LCH, the hemangioma is character- the tumor. These clinical symptoms of hemangiomas
ized by submucosal vascular proliferation and capil- do not differ from those reported in previous studies.7
lary lobules; it is the most common type of pediatric However, this retrospective study showed that hyper-
hemangioma in the head and neck region.5 LCH ex- tension was the most common medical history of
hibits lobular capillary proliferation; however, LCH adults with hemangiomas (35.1%). Moreover, mean
and capillary hemangioma can be interspersed.6,7 CH age of patients with hypertension (64.9 14.5 yr)
is a less common type of hemangioma and shows a was significantly older than that of patients with
higher incidence in adults and women.5 normal blood pressure (37.7 20.3 yr; P = .001). In
In the present study, there were 6 hemangiomas in older individuals, blood pressure is usually higher
the pediatric population (4 LCHs and 2 CHs). Howev- because of loss of elasticity in blood vessels.8,9
er, analysis showed no correlations between age and Previous studies have reported that renal or
gender according to histologic subtype of hemangio- pulmonary hemangiomas can be caused by
ma (LCH vs CH; P > .05). Rather, a positive correlation hypertension.10,11 Although the exact relation
was found between symptom duration and tumor size. between systemic hypertension and hemangioma
A diagnosis of hemangioma delayed as briefly as remains unknown, a potential connection between
1 month can increase tumor by 0.38 mm (Fig 6). hypertension and hemangioma can be inferred
More than half the patients complained of nasal from the literature. Based on the findings of
obstruction and epistaxis. Thirty-two of 37 patients this retrospective study, the authors propose
had the hemangioma located in the nasal cavities hypertension as a possible common coexisting risk
(86.5%) and the mass effect results in nasal obstruc- factor in older patients. Angioma and venous
KIM AND KWON 1781
FIGURE 4. Axial T1-weighted contrast-enhanced magnetic resonance image of patient 24 shows heterogenous strong high signal intensity in
the left maxillary sinus.
Kim and Kwon. Sinonasal Hemangioma. J Oral Maxillofac Surg 2017.
‘‘malformations’’ in Klippel-Trenaunay and Cobb syn- important role in the pathogenesis of LCH, possibly in
dromes are manifestations of focal venous hyperten- association with injury.14 This theory has implications
sion in different parts of the body.12 As the present in middle-age women who have received hormonal
results suggest, hypertension could induce venous replacement therapy. Future study of hormonal recep-
malformation or dilatation of the vein or local obstruc- tor expression in such specimens is needed.
tion of vessels, which could become prominent and Sinonasal hemangiomas usually arise from the nasal
distinct over time. This theory might be another clue septum and inferior turbinate.13,14 The present study
in addition to other potential causes, such as local supports previous data showing a marked
trauma, congenital malformation, and hormon- preponderance at the inferior turbinate and septum.
al therapy.13 When the hemangioma occurred at these sites
In the present study, 1 patient received hormone (inferior turbinate and nasal septum), the tumor was
replacement therapy at middle age and another patient removed under local anesthesia regardless of
had a history of nasal mass removal. The tumor sites of histologic type. In this study, the authors also
these patients were the inferior turbinate and nasal observed relatively unusual sites of origin, including
septum, respectively. Among traumatic factors, habitual the maxillary sinus (3 of 37), posterior ethmoid sinus
picking and nasal packing are the most common etio- (2 of 37), and uncinated process (3 of 37). The
logic risk factors of LCH.13,14 In the patient with a operation was performed under general anesthesia
history of nasal mass removal, nasal packing might when the tumor was located at these sites. Statistical
have caused injury to the nasal septum and inferior analyses showed that the tumor site differed
turbinate, leading to LCH on the nasal septum. according to histologic type (Table 2). LCH occurred
Among hormonal factors, progesterone could play an more commonly at the inferior turbinate and nasal
1782 SINONASAL HEMANGIOMA
subperichondrial or subperiosteal dissection. These
techniques obviate embolization and lower the risk
of substantial bleeding. In addition, the endoscopic
technique has the benefit of precise handling and bet-
ter visualization of the tumor. Its ability to magnify al-
lows visualization of very small vessels and thus
obviates surgical loupes. The endoscopic technique
also can be applied in a deeper origin site, such as
the maxillary sinus or posterior ethmoid sinus,
without concern about an external scar (Fig 5). How-
ever, if the tumor involves the anterior or lateral
portion of the maxillary sinus, then surgery cannot
be performed only with an endoscopic technique; an
external approach, such as the canine fossa or the
Caldwell-Luc approach, might be needed.
The availability of contrast-enhanced CT enables cli-
nicians to better discern vascular tumors from other
benign masses. CT and MR images visualize expansile
remodeling, erosion, and less frequently destructive
changes of bone. Internal amorphous or curvilinear
FIGURE 5. Endoscopic image of patient 24 shows a representa-
tive clinical image of a cavernous hemangioma. Asterisk, cavernous
calcifications also might be visible.15 Enhanced CT
hemangioma of the left maxillary sinus; arrow, middle turbinate. can be used to differentiate hemangiomas from malig-
Kim and Kwon. Sinonasal Hemangioma. J Oral Maxillofac Surg nant tumors, which show increased vascularity. If the
2017. mass was larger than 2 cm and the mass showed dam-
age of adjacent structures, then MRI was performed in
the present patients.
septum, whereas CH more often affected the maxillary
MRI of sinonasal hemangiomas shows heteroge-
sinus and middle turbinate. Hemangiomas from
neously high and low signals on T1- and T2-weighted
these unusual sites should be distinguished from
images. Kim et al7 reported that post-enhanced CT im-
other tumors.
ages of hemangiomas showed early strong enhance-
Preoperative angiography and embolization are not
ment and delayed washout and that MRI displayed
usually necessary, because the main vessel of the hem-
marked gadolinium enhancement with heterogeneous-
angioma is a capillary or small vein rather than an ar-
ly high signals on T1- and T2-weighted images. The pre-
tery. In the present patients, excision or primary
sent results support these findings. In addition, based
removal of the main stalk of the hemangioma was per-
on the present results, the authors suggest that the pe-
formed using monopolar or bipolar cautery after
riphery of hemangiomas shows a high signal on T1 im-
ages and low signals on T1 enhanced and T2-weighted
images. Although a small number of MRIs was per-
formed in this series, the additional radiologic charac-
teristics noted could help differentiate hemangiomas
from other vessel-rich tumors. The differential diagno-
ses of sinonasal hemangiomas should include sinonasal
polyps, mucocele and inverted papillomas, malignant
tumors, and when there is bone destruction.
One limitation of this study is the retrospective
methodology. However, conducting prospective
cohort studies and reporting experiences of rare clin-
ical conditions at a single institution is difficult.
Clinicians should be aware of the presenting symp-
toms and imaging characteristics of hemangiomas of
the sinonasal mucosa, so that they are not misdiag-
nosed as malignant tumors, angiofibroma, or other
FIGURE 6. Graphic representation of the linear regression be-
tween symptom duration and size of hemangioma (size = 0.038
benign masses. There was a meaningful correlation be-
duration + 1.341). tween histologic type and tumor site, with LCHs
Kim and Kwon. Sinonasal Hemangioma. J Oral Maxillofac Surg occurring more often at the nasal septum and inferior
2017. turbinate than CHs.
KIM AND KWON 1783
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