Oral and Maxillofacial Surgery Cases 6 (2020) 100147

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Oral and Maxillofacial Surgery Cases

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Recurrence of intraosseous cavernous hemangioma in the maxilla:

A case report
Sung-Min Park, Jae-Hoon Lee *
Department of Oral and Maxillofacial Surgery, College of Dentistry, Dankook University, 119 Dandae-ro, Dongnam-gu, Cheonan, 31116, South
Korea

A R T I C L E I N F O A B S T R A C T

Keywords: A 12-year-old girl was referred to the Department of Oral and Maxillofacial Surgery for a large
Recurrence cystic lesion, which was gradually increasing in size. We diagnosed her with intraosseous hem­
Cavernous hemangioma angioma based on the magnetic resonance imaging, computed tomography, and angiography
Intraosseous
findings and performed surgical curettage.
Curettage
Sclerotherapy
Three months later, there were no recurrences or cosmetic deformities. However, 3 years later,
she revisited the Department of Oral and Maxillofacial Surgery with the complaint of gingival
hardness. Recurrent cavernous hemangioma was diagnosed based on the imaging features, and
the tumor was removed via the same incision site. The histopathological diagnosis of the spec­
imen was recurrent cavernous hemangioma. Herein, we report a rare case of recurrent cavernous
hemangioma in the maxilla that was successfully treated. To the best of our knowledge, this is the
first report of a recurrent intraosseous hemangioma of maxilla containing multiple teeth has been
completely cured without tooth extraction or surgical resection.

1. Introduction

Hemangiomas are benign vascular lesions, mostly arising from soft tissues. Intraosseous hemangiomas are rare, accounting for only
1% of cases of benign bone tumors [1]. They are mainly located in the vertebral column and calvarium and rarely in the facial bones
[2]. The maxilla is infrequently affected, and cavernous hemangiomas in the maxilla usually involve the teeth and alveolar processes.
Herein, we report a case of recurrence of cavernous hemangioma in the right maxilla after its surgical removal. All the teeth were
preserved in the treatment of recurrent hemangioma.

2. Case report

A 12-year-old girl presented to the oral and maxillofacial surgery clinic at the Dankook University Dental Hospital with a painless,
radiolucent lesion in the right maxilla, with progressive enlargement and hardening. She had no history of systemic diseases or trauma.
Plain radiography showed a well-defined radiolucent cystic lesion in the right maxillary sinus that extended from the canine to the first
molar and had caused root resorption (Fig. 1A). Coronal cone-beam computed tomography (CT) of the lesion involving the teeth and
alveolar processes showed significant expansion of the right maxilla with some periosteal bone formation, which suggested a slow rate
of expansion of the lesion (Fig. 1B). Magnetic resonance imaging (MRI) revealed a well-defined homogenous mass measuring 3� 2.9 �

* Corresponding author.
E-mail addresses: pak2692@gmail.com (S.-M. Park), lee201@dankook.ac.kr (J.-H. Lee).

https://doi.org/10.1016/j.omsc.2020.100147
Received 6 January 2020; Received in revised form 23 February 2020; Accepted 26 March 2020
Available online 6 April 2020
2214-5419/© 2020 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
S.-M. Park and J.-H. Lee Oral and Maxillofacial Surgery Cases 6 (2020) 100147

Fig. 1. A. A panoramic view of the lesion shows involvement of the right side of the maxilla with root resorption from the maxillary right canine to
the maxillary right first molar. B. Coronal cone-beam computed tomography shows a well-defined radiolucent mass in the right maxilla, perforating
the maxillary sinus. C. T2-weighted coronal magnetic resonance imaging shows a well-defined, lobular, hyperintense mass in the right maxilla
measuring 3.0 � 2.9 � 2.7 cm. D. A right external carotid arteriogram shows subtle tumor staining in the right maxilla.

Fig. 2. A. Intraoperative image showing exposure of the lesion with honeycomb appearance. B. Histopathologic photograph show bone trabeculae
with cavernous endothelial lined blood vessels (Hematoxylin and eosin; X40).

2.7 cm which was hypointense on T1-weighted MRI and markedly hyperintense on T2-weighted MRI (Fig. 1C).
Transfemoral cerebral angiography (TFCA) was performed with the suspicion of vascular malformation (Fig. 1D). The main feeding
vessel of the tumor was the internal maxillary artery, but embolization was not performed, as there were no enlarged feeding vessels or
rapid arteriovenous shunting.
Complete excision involving the surgical margin was required to prevent tumor recurrence. However, considering the age of the
patient, we decided to perform curettage to preserve the teeth and facial bone as much as possible.

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S.-M. Park and J.-H. Lee Oral and Maxillofacial Surgery Cases 6 (2020) 100147

Fig. 3. A. Coronal cone-beam computed tomography shows a honeycomb-shaped bony lesion in the right maxillary sinus. B. T2-weighted coronal
magnetic resonance imaging shows a well-defined, lobular, hyperintense mass in right maxilla measuring 4.1 � 2.4 � 3.4 cm.

Fig. 4. After the first surgery, postoperative coronal cone-beam computed tomography shows the possibility of incomplete removal of the tumor
(red arrow) because of a difficult access during the surgery.

First, an aspiration of the sinus, attempted under general anesthesia, revealed only a limited amount of hematic material with
minimal bleeding. Secondly, the anterior maxillary wall was exposed with the Caldwell-Luc approach. We observed tumors protruding
into the anterior maxillary wall and achieved curettage while preserving the stability of the teeth and alveolar processes. The tumors
were removed from the maxilla and teeth, and subsequently, the residual bones and roots of the exposed teeth were thoroughly scraped
using surgical curettes.
We removed all tumors from the palatal mucosa, and the palatal region was curetted and cauterized, with caution to avoid
perforation. There was no bleeding tendency at the time of surgery, and nasal filling was performed to prevent postoperative bleeding.
The histopathologic diagnosis was intraosseous cavernous hemangioma of the maxilla (Fig. 2).
Three months after the surgery, there were no recurrences or cosmetic deformities. We recommended root canal treatment because
of the possibility of devitalized teeth after the curettage. The patient was lost to follow-up.
However, 3 years later, she revisited the Department of Oral and Maxillofacial Surgery with the complaint of gingival hardness.
Recurrent cavernous hemangioma was diagnosed based on the imaging features, and the tumor extended to the maxillary right central
incisor (Fig. 3). Surgical curettage was performed again, with preservation of the teeth and alveolar processes, via the previous incision
site. At this time, a sclerosing agent, 3% sodium tetradecyl sulfate (STS), was injected at the surgical site to reduce the risk of
recurrence.
The histopathological diagnosis of the specimen was recurrent intraosseous cavernous hemangioma of the maxilla. There were no
recurrences of symptoms for 4 postoperative years. The root canal treatment was performed for all teeth from the central incisor to the
second molar. The most recent imaging study revealed new bone formation in the right maxilla and no tooth mobility. Clinically, we
observed no facial deformities.

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S.-M. Park and J.-H. Lee Oral and Maxillofacial Surgery Cases 6 (2020) 100147

Fig. 5. A. panoramic view shows new bone formation surrounding the teeth. B. Coronal cone-beam computed tomography shows new bone for­
mation at the operated site.

3. Discussion

Complete excision including a surgical margin of sound tissue is the preferred treatment of intraosseous hemangioma, and re­
currences are rare [3,8]. However, in this case, curettage was the treatment of choice, considering the age of the patient, as preserving
the teeth and minimizing facial deformities were important.
At the first operation, the tumor could have been removed easily, without excessive bleeding. However, access to the area between
the palatal root and the palatal alveolar process was hindered by the teeth. Therefore, an additional curettage and cauterization were
performed in that area, but complete removal of the tumor may not have been achieved.
It was concluded that residual tumor at the site could have caused the recurrence (Fig. 4). A palatal approach could have more
reliably removed the tumor. However, the involved teeth were mobile, so a palatal approach could not be made.
The recurrent tumor was bigger than the initial tumor. And it seemed a that surgical resection was needed. But for the removal of
the recurred hemangioma, we selected conservative curettage again. Furthermore, sclerotherapy with 3% STS was performed.
Sclerotherapy is an alternative treatment method for hemangioma and can also be used as an adjunct to surgery or a postoperative
complement [4].
Sclerotherapy is more useful for soft-tissue hemangiomas. However, it can be used as an adjunct therapeutic method in cases of
intraosseous hemangioma [5]. Many sclerosing agents have been used in the treatment of hemangioma. Particularly, STS has been
used for years in the treatment of varicose vein, hemorrhoids, and hemangioma [6]. The mechanism of action of STS is induction of
extra-vascular inflammation and thrombosis, resulting in ischemic necrosis of the blood vessels and fibrosis of the lesion [7].
In our patient, as residual tumor was suspected on the palatal side, STS was injected intraoperatively at the palatal mucosa after the
curettage. After 4 years, there were no evidences of recurrence, and new bone formation was observed surrounding the teeth (Fig. 5).
Intraosseous hemangiomas of the maxilla present with pain, dental anomalies, gum bleeding, tooth mobility, and sinusitis-like
symptoms [8]. Total surgical excision is the preferred treatment method for intraosseous hemangiomas. However, the oral and

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S.-M. Park and J.-H. Lee Oral and Maxillofacial Surgery Cases 6 (2020) 100147

maxillofacial surgeons should consider the negative impacts of dentofacial disfigurement on the quality of life and other possible social
and psychological implications, especially for younger patients.
In conclusion, to avoid these complications that may arise from hemangiomas, a multidisciplinary plan including the precise
diagnosis and appropriate therapeutic approaches are necessary.
Although surgical excision remains the preferred treatment, curettage, sclerotherapy, embolization, or a combination of these
methods can be effective. Furthermore, routine follow-ups should be performed because of the potential risk of complications and
recurrence.

Ethics statement/confirmation of patient’s permission

The patient has given permission to report this case.

Declaration of competing interest

None.

References

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