p ISSN
2723-6927-e ISSN 2723-4339
Characteristics
of Oral Hemangioma and Vascular Malformation Cases: A Study at Hasan Sadikin
General Hospital, 2019-2023
Yashinta Rachmavita1*, Harmas Yazid Yusuf2, Melita Sylvyana3
1Oral and Maxillofacial Surgery Department,
Faculty of Dentistry Padjadjaran University, West Java, Indonesia
2,3Oral and Maxillofacial Surgery Department, Dr.
Hasan Sadikin General Hospital, Padjadjaran Univeristy, West Java, Indonesia
Email: yashinta21001@mail.unpad.ac.id1*
This
observational retrospective study conducted at Hasan Sadikin General Hospital
(RSHS) in Bandung from January 2019 to February 2023 aimed to characterise oral
hemangiomas and vascular malformations. Out of 52 medical records analysed,
infantile hemangioma was the most common type (17%), typically presenting
between ages 13 to 24 years. Predominantly located around the lip, tongue, and
buccal mucosa, it seldom caused bleeding or pain, with women being more
affected. Treatment often involved sclerotic agent injections. Venous
malformations accounted for 58% of vascular malformations, showing similar age
of presentation and distribution as hemangiomas, and were also treated with
sclerotic agents. Accurate diagnosis and tailored treatment are crucial due to the
diverse nature of these anomalies. Early detection using imaging techniques
like ultrasound and CT angiography is imperative, given the potential impact on
patients, including pain, swelling, and cosmetic concerns, particularly in the
head and neck area. However, treatment approaches vary based on factors such as
type and size, necessitating further research to refine diagnostic and
therapeutic strategies and optimise patient outcomes.
Keywords: Hemangioma,
Vascular Malformations, Vascular Anomalies, Infantile Hemangioma, Venous
Malformation
INTRODUCTION
Vascular anomalies are abnormalities that happen because of
dysfunction in vasculogenesis and angiogenesis, as well as congenital
abnormalities and dysmorphogenesis neonatal in every area, mostly in the head
and neck (60%). The term hemangioma has long been used to describe various
anomalies of vascular development in infancy and childhood, which increase the
number of blood vessels. Hemangioma appears in neonates at the age of 6-8
months (proliferative phase) and gets slowed down at the age of 5-10 years
(involution phase). Vascular malformations appear at birth and develop along
with the process of growth and regeneration of endothelial cells in children.
The incidence of vascular malformation is 31% found in the head and neck
region. Vascular malformations can be categorised based on the type of vessel
involved (capillary, vein or arteriovenous) and based on hemodynamic properties
(low flow or high flow) (Steiner
& Drolet, 2017).
Pain and swelling are the main complaints in cases of vascular
malformations. Aesthetic factors are the main aspect that influences
psychological stress and anti-social behaviour (Lee
& Chen, 2005). Recurrent bleeding, secondary
infections, and ulceration have been found in several reports. The gold
standards for supporting the examination are Ultrasound and CT Angiography.
Appropriate and regular follow-up is necessary to determine appropriate
therapy. Laser therapy, sclerotherapy, embolisation, electrochemical therapy,
cooper needle treatment, and resection are options for treating vascular
anomalies depending on the type, size, location, patient status, and
availability of facilities in the health centres (Tasker, Acerini, Holloway, Shah, &
Lillitos, 2021).
The urgency of researching
vascular anomalies lies in their significant impact on affected individuals,
particularly in terms of health outcomes and quality of life. Vascular
anomalies, characterised by abnormalities in vasculogenesis and angiogenesis,
pose considerable challenges due to their diverse manifestations and potential
complications. These anomalies, including hemangiomas and vascular
malformations, predominantly affect the head and neck region, with a
considerable incidence rate.
Individuals with vascular
anomalies often experience symptoms such as pain, swelling, recurrent bleeding,
and secondary infections, which can lead to significant discomfort and
complications. Moreover, the aesthetic concerns associated with vascular
anomalies can cause psychological stress and anti-social behaviour, further
exacerbating the burden on affected individuals.
Early detection and
accurate diagnosis of vascular anomalies are crucial for appropriate management
and timely intervention. Gold-standard diagnostic tools such as ultrasound and
CT angiography are essential for effectively assessing the extent and
characteristics of these anomalies. Furthermore, the availability and
accessibility of appropriate treatment modalities are paramount for optimising
patient outcomes. However, the choice of treatment options for vascular
anomalies depends on various factors, including the type, size, and location of
the anomaly, as well as the patient's overall health status and the resources
available in healthcare facilities.
Therefore, conducting
research on vascular anomalies is imperative to enhance our understanding of
their pathogenesis, clinical features, diagnostic approaches, and treatment
strategies. Such research endeavours can ultimately contribute to improving the
management and outcomes of individuals affected by vascular anomalies, thus fulfilling
an essential need within the medical community and benefiting patient care.
RESEARCHED METHODS
This research is an observational study with a retrospective
descriptive approach. This research was conducted at RSHS Bandung from January
2019 – February 2023. The population and sample of this study were all medical
records of a patient diagnosed with Hemangioma and Vascular Malformations at
the Oral and Maxillofacial Surgery Department RSHS. The research uses inclusion
and exclusion criteria. Inclusion was the patient diagnosed with Hemangioma and
Vascular Malformations which in anamnesis patients including type, onset, age,
region, gender, main complaint, therapy, history of reference, supporting
examination, bleeding, and pain at the Inpatient Installation of Dr. Hasan
Sadikin in the period January 2019 – February 2023. The exclusion criteria in
this study were incomplete patient medical record data in anamnesis, including the
onset of the occurrence. The variables studied include type, onset, age,
region, gender, primary complaint, therapy, history of reference, supporting
examination, bleeding, and pain. The categorical data processing was done using
a computer program, and the research data were grouped and expressed in
proportions. This study has received an ethical approval number from the Health
Research Ethics Committee of Dr. Hasan Sadikin General Hospital Bandung with
the ethical committee approval number 973/UN6.KEP/EC/2023.
RESULTS AND DISCUSSION
The results of data collection on the Characteristics of Oral
Hemangioma and Vascular Malformations at Hasan Sadikin General Hospital Oral
and Maxillofacial Surgery Department on January
2019 – February 2023, there were 52 patients, nine patients with Infantile
Hemangioma, three patients with Congenital Hemangioma, four patients with
Arteri-Venous Malformations, 30 patients with Venous Malformations, and five
patients with Lymphatic Malformations.
Table 1. Characteristic Based on Type, Therapy,
Age, Gender, Region, Bleeding, and Pain
|
Criteria |
Infantile Hemangioma |
Congenital Hemangioma |
Arteri Venous Malformation |
Venous Malformation |
Arteri Malformation i |
Lymphatic Malformation |
Capillary Malformation |
|
|
Age |
0-12 y.o |
1 |
3 |
0 |
7 |
0 |
4 |
0 |
|
13-24 y.o |
4 |
0 |
3 |
13 |
0 |
1 |
0 |
|
|
25-36 y.o |
2 |
0 |
1 |
8 |
0 |
1 |
0 |
|
|
37-48 y.o |
0 |
0 |
0 |
1 |
0 |
0 |
0 |
|
|
>48 y.o |
2 |
0 |
0 |
1 |
0 |
0 |
0 |
|
|
Region |
Tongue |
2 |
0 |
0 |
5 |
0 |
2 |
0 |
|
Palate |
0 |
0 |
0 |
0 |
0 |
0 |
0 |
|
|
Gingiva |
0 |
0 |
0 |
0 |
0 |
1 |
0 |
|
|
Buccal Mucous |
0 |
0 |
0 |
7 |
0 |
1 |
0 |
|
|
Lip |
6 |
1 |
2 |
12 |
0 |
2 |
0 |
|
|
Tongue + Buccal Mucous |
0 |
1 |
0 |
0 |
0 |
0 |
0 |
|
|
Lip + Buccal Mucous |
1 |
1 |
0 |
3 |
0 |
0 |
0 |
|
|
Lip + Mentale |
0 |
0 |
1 |
0 |
0 |
0 |
0 |
|
|
Lip + Tongue |
0 |
0 |
1 |
0 |
0 |
0 |
0 |
|
|
Lip + Tongue + Buccal Mucous |
0 |
0 |
0 |
1 |
0 |
0 |
0 |
|
|
Palate + Sublingual |
0 |
0 |
0 |
1 |
0 |
0 |
0 |
|
|
Buccal + Sublingual + Submandible + Submental |
0 |
0 |
0 |
1 |
0 |
0 |
0 |
|
|
Bleeding |
Yes |
0 |
1 |
1 |
4 |
0 |
0 |
0 |
|
No. |
9 |
2 |
3 |
26 |
0 |
6 |
0 |
|
|
Pain |
Yes |
0 |
0 |
1 |
4 |
0 |
4 |
0 |
|
No. |
9 |
3 |
3 |
26 |
0 |
2 |
0 |
|
|
Gender |
Man |
1 |
1 |
2 |
11 |
0 |
4 |
0 |
|
Woman |
8 |
2 |
2 |
19 |
0 |
2 |
0 |
|
|
Therapy |
Excision |
1 |
0 |
0 |
1 |
0 |
1 |
0 |
|
Ligation |
0 |
0 |
0 |
0 |
0 |
0 |
0 |
|
|
Sclerosing Agent |
7 |
3 |
4 |
23 |
0 |
4 |
0 |
|
|
Sclerosing Agent + Excision |
1 |
0 |
0 |
2 |
0 |
1 |
0 |
|
|
Endovaskular |
0 |
0 |
0 |
0 |
0 |
0 |
0 |
|
|
Sclerosing Agent + Phlebolitectomy |
0 |
0 |
0 |
3 |
0 |
0 |
0 |
|
|
Ligation + Biopsy Extirpation + Glossoplasty |
0 |
0 |
0 |
1 |
0 |
0 |
0 |
|
From the
results of research using medical records based on the criteria of type, age,
region, gender, therapy, complaints of bleeding and pain, it was found that nine
patients were diagnosed as infantile hemangioma or 17% with most ages at
arrival for treatment 13-24 years occurred in the lip area, tongue followed by
lips and buccal mucosa without complaints of bleeding and pain, mainly occurs
in women and was treated by sclerosing agent in the form of bleomycin
injection. Congenital hemangioma in 3 people, or 6% with most ages at arrival
for treatment 0-12 years, occurred in the area of the lips, tongue, and buccal
mucosa followed by the lips and buccal mucosa without complaints of bleeding
and pain mainly occurs in women and was treated by a sclerosing agent in the
form of injection bleomycin.
Arteriovenous
malformations in 4 people or 8%, most ages at arrival for treatment 13-24
years, occurred in the lip area without complaints of bleeding and pain; the
incidence of women was the same as men and was treated by a sclerosing agent in
the form of bleomycin injection. Venous malformations in 30 people (or 58%),
most ages at arrival for treatment (13-24 years old), occur in the area of the
lips and tongue, followed by the buccal mucosa, without complaints of bleeding.
Some were accompanied by pain, mainly occurring in women and were treated by a
sclerosing agent in the form of bleomycin injection. Lymphatic malformations in
6 people or 11%, most ages at arrival for treatment 0-12 years old, occurred in
the tongue area without complaints of bleeding, and some were accompanied by
pain, mostly occurred in men and were treated with a sclerosing agent in the
form of bleomycin injection.
Table 2. Characteristic
Based on History of Reference
|
Characteristic |
Frequency |
Proportion (%) |
|
History of Reference |
||
|
Public Health Center, Public Hospital |
50 |
96% |
|
Own desire |
2 |
4% |
From the
results of research using medical records based on the criteria of history of reference,
it was found that 50 patients were referred from public health centres and
public hospitals (96%), and those who came with their own desire were two
people (4%).
Table 3. Characteristics Based on Main Complaints
|
Characteristic |
Frequency |
Proportion (%) |
|
Main complaints |
||
|
Lump |
23 |
44% |
|
Bleeding Aesthetic Lump + Pain Bleeding + Pain |
9 18 1 1 |
17% 35% 2% 2% |
From the results of research using medical records based on the main
complaint, it was found that patients came for treatment at KSM Oral and
Maxillofacial Surgery RSHS Bandung due to lumps in 23 people (44%), bleeding in
9 people (17%), aesthetic complaints in 18 people (35%), lumps. Accompanied by
pain in 1 person (2%) and bleeding accompanied by pain in 1 person (2%).
Table 4. Characteristics Based on Onset
|
Characteristic |
Frequency |
Proportion (%) |
|
Onset |
||
|
From birth |
12 |
23% |
|
Adulthood |
40 |
77% |
From the results of research using medical records based on the onset of
events, it was found that 12 patients (23%) complained of hemangiomas and
vascular malformations since birth, and 40 people (77%) complained in adulthood.
Table 5. Characteristics Based on Support Examination
|
Characteristic |
Frequency |
Proportion (%) |
|
Support Examination |
||
|
USG + CT Angiography 3 6% CT Angiography 3
6% USG 12
23% No support examination 34 65% |
||
From the results of research using medical records based on supporting
examinations, it was found that three patients (6%) underwent ultrasound and CT
angiography, three patients (6%) of CT angiography, 12 patients (23%) underwent
ultrasound, and 34 patients (65%) did not undergo support examination.
Discussion
From 99 patients, 34 patients experiencing hemangiomas and 65 other
patients experiencing vascular malformations, this research found that the
number of sufferers of hemangiomas and vascular malformations in the oral
cavity at KSM Oral and Maxillofacial Surgery at Dr Hasan Sadikin General
Hospital is 52 patients with 12 patients experiencing hemangiomas and 40
patients experiencing vascular malformations from January 2019 to February
2023. This study follows the ISSVA classification, which divides vascular
anomalies into the proliferation of vascular tumours and non-proliferation of
vascular malformations, which can be subclassified based on their potential to
develop, leading to malignancy (tumour) or involvement of vascular vessels
(malformation).
Standard therapies for vascular anomalies at Hasan Sadikin General Hospital
are sclerosing agent injection and lesion excision. It differed from the study
by Leonard et al., which stated that therapy for hemangioma was by
intralesional injection of corticosteroids (triamcinolone, dexamethasone, or
methylprednisolone). This difference can occur because most hemangioma patients
who come for treatment have already passed the proliferation phase and vascular
malformation ((Steiner & Drolet, 2017); (Mukhopadhyay, Galui, Biswas, Saha, &
Sarkar, 2020)).
Managing small vascular anomalies in the maxillofacial area remains
controversial and must be reviewed, especially for long-term aesthetic results
and sequelae. The main purpose of administering intralesional injections and
sclerotic agents for vascular anomalies include (1) preventing or avoiding
complications that threaten life or body function, (2) wound care, (3)
preventing permanent disability, (4) reducing the psychosocial burden for
patients and their families, and (5) prevent lesions from developing
aggressively, potentially resulting in scars that have a strong possibility of
involution without significant residual (Smith et al., 2022).
Infantile
hemangiomas and venous malformations were the most common types of vascular
anomalies found in this study. Infantile hemangioma was found in 9 patients,
with the main complaint being a lump; based on anamnesis, it was found at birth
and was slowly undergoing an involution phase. This is suitable for an
epidemiological study conducted by Lauren et al., which found that infantile
hemangiomas develop during the first two months of life and show rapid
proliferation between 6 and 12 months of age, followed by a prolonged
involution period. Most infantile hemangiomas will shrink spontaneously at the
age of 6 and 9 years. However, in some cases, they do not regress completely,
so lump defects are still found and are the main complaint of patients when
they go to Hasan Sadikin General Hospital (Zhang, Zhou, & Shen, 2020).
An
observational retrospective study in Spain showed that 82% of hemangioma
samples had a predilection in the lips. However, other epidemiological studies
showed a predilection in the ventral tongue in the first place, followed by the
lips and buccal mucosa. It was the same with research at Hasan Sadikin General
Hospital that the lips are the most predilection location, as it is known that
hemangiomas in the lower lip region show a lower level of involution compared
to lesions located elsewhere (Zhang et al., 2020).
The results of the comparison between genders
regarding the incidence of oral hemangioma cases in this study show that the
incidence rate in women is higher than in men, with a ratio of 2:1 and a
percentage of 63% in women and 37% in men. The results of this study are suitable
with epidemiological research conducted by Correa et al. regarding the
prevalence of oral hemangiomas and vascular malformations in the Brazilian
population which states that hemangioma cases occur more frequently in women
than men with a ratio of 4:1. Hemangiomas are more common in women due to
estrogen in the female hormone being the target of vascular lesions and also
the levels of the estrogen receptor (ER), progesterone receptor (PR), and
androgen receptor (AR) ((Chinyama, 2020);(Frongia, Byeon, Mehrabi, & Günther, 2021)). Differences in the ratio of women to men
between the results of our research and Correa et al.'s research may be caused
by differences in sample size and ethnicity.
Congenital hemangioma in this study was found in 3 patients and was
reported in the age range 0-12 years, with the main complaint being a lump, and
based on the anamnesis, it was found at birth. This result is suitable for
epidemiological research, which shows that congenital hemangioma is seen at
birth, passes through a proliferation phase, and involutes quickly or not at
all (El Zein et al., 2020).
In his epidemiological study, Mulliken stated that the most significant
predilection for congenital hemangioma was on the lips, followed by the
combination of the tongue and buccal mucosa and the lips and buccal mucosa.
This is suitable with research conducted at Hasan Sadikin Hospital that 100% of
congenital hemangioma patients have a predilection on the lip area (Jorge, Nascimento, Fernandes, & Soares,
2022).
Based on research, hemangiomas that occur in Caucasian babies have a
prevalence of 3-5 times higher in women. This condition is suitable with what
was found in babies who had congenital hemangioma at Hasan Sadikin General
Hospital, which was more dominated by women than men, with a prevalence of
women two times higher. This difference in ratio could be caused by a smaller
number of samples and differences in ethnicity/race at Hasan Sadikin General
Hospital (CAGLIARI, 2021).
Research conducted by England et al. shows that most hemangiomas do not
cause pain, show no symptoms, and tend to bleed if the lesion is traumatised;
this is suitable with the results of our study where all hemangioma patients,
both congenital and infantile, did not complain of bleeding or pain (Aziz, Brown, Baghdadi, Kamel, & Pawlik, 2022).
The treatment that can be given to patients with hemangiomas with a
diameter of less than two cm is intralesional injection of triamcinolone 3-5
mg/kg (10-15mg/ml) using a 25 gauge needle. Each injection is carried out at
intervals of 6-8 weeks to evaluate the peak phase of involution. Corticosteroid
drugs can be given for lesions that are large, destructive, and endanger vital
organs. Prednisolone can be given at a dose of 2-3 mg/kg/day once every two
weeks, and the dose is reduced every 10-11 months. Excision is indicated for tumours
that have ulcerated, bleeding, causing airway obstruction, and disrupting
vision.
This is different from the treatment carried out at Hasan Sadikin General
Hospital in that hemangiomas are often treated with sclerotic agent injections,
and this happens because patients who experience hemangiomas at Hasan Sadikin
General Hospital are over 12 months old when the proliferation phase has ended.
Herdata et al. stated that hemangiomas in patients less than 12 months old are
treated with intralesional injection of corticosteroids (triamcinolone) to
inhibit the proliferation phase (2 to 12 months), and over 12 months, therapy
is recommended with bleomycin injection which plays a role in the angiogenesis
process. Although corticosteroid therapy is easy and effective, some cases do
not show a response and improvement in lesions after a maximum of 3 doses.
Long-term use of corticosteroids is avoided to prevent adverse side effects
such as growth disorders, Cushingoid effects, and infections. Another effect of
corticosteroids that have been reported is suppressing the immune system by
reducing the migration of neutrophils to sites of inflammation.
Intralesional corticosteroids are only given to small hemangiomas (2-3 cm
in diameter) for six weeks at a dose of no more than 3 mg per kilogram of body
weight. The mechanism of corticosteroids against hemangiomas is not yet known
with certainty, but it is known that steroids can inhibit the proliferation of
immature pericytes, stimulate the vasoconstrictive effects of epinephrine and
norepinephrine on smooth muscle, inhibit estradiol receptors and angiogenesis.
The use of systemic corticosteroids at the proliferation stage can inhibit
growth and cause hemangioma regression by around 30% -90%. It can also prevent
ulceration and thus speed up healing. However, in some cases, the use of
corticosteroids is found to be resistant, so other types of alternative therapy
need to be used, such as bleomycin or vincristine injections ((Sebaratnam, Wong, & Wargon, 2021) (Yajun, Shan, & Qihong, n.d.))
Vascular malformations in this
study were found in 40 patients, with the main complaints being lumps,
aesthetics, pain, ulcers, and, in some cases, bleeding. Based on the anamnesis,
the vascular malformation was discovered after birth and grew with age. This is
suitable with epidemiological research conducted by Munden and Behravesh that
vascular malformations grow along with the child's development and, without
spontaneous regression, can cause morbidity, pain, and discomfort and can cause
local and systemic complications ((Behravesh et al., 2016);(Munden et al., 2014)).
Venous malformations consist of ectatic venous channels, which are usually
found in the head, neck, legs, and other parts of the body and are considered
sporadic and influenced by genetic patterns. In this study, venous
malformations were found in 30 patients, with the main complaints being lumps
in 30% (9 cases), aesthetic 50% (15 cases), bleeding in 16% (5 cases), and some
phleboliths appeared in 3% (1 case) in several areas. This is suitable with the
epidemiological study conducted by Dubois et al. that the main complaints of
venous malformations are lumps with a percentage of 44% (23 cases), aesthetics
with a percentage of 35% (18 cases), and bleeding with a percentage of 17% (9
cases) ((Soman et al., 2020);(Hussein & Malguria, 2020)).
Venous malformations involve areas with lots of blood vessels, so they
generally rarely occur in areas with few blood vessels, for example, in the
palate area. (17) Based on epidemiological studies conducted by Correa et al.,
the predilection for venous malformations occurs mostly on the lips (45%),
followed by the tongue (32 %) and buccal mucosa (18%). This is suitable with
research at Hasan Sadikin Hospital, which found that lips are the main
predilection (40%) for venous malformations, ns followed by the buccal mucosa
(23%) (Habibie, 2021).
Research in Mexico shows that the prevalence ratio of men to women in
venous malformations is 1:1.2 (Rendón Elías, Hernández Sánchez, Albores
Figueroa, Montes Tapia, & Gómez Danés, 2014), and this value is
suitable with the results of research at Hasan Sadikin General Hospital, which
states that women dominate cases of venous malformations with a ratio of 1:1.7.
The next most common type of malformation is arteriovenous malformation.
This is suitable for research in Mexico by Felipe et al., who found that
arteriovenous malformations rank second after venous malformations, followed by
lymphatic malformations. Arteriovenous malformations in this study were found
in 4 patients, with the main complaints being lumps and aesthetics. Based on
anamnesis, it is found after adulthood and develops with increasing age.
The predilection area in cases of arteriovenous malformations in this study
was mostly in the lip area; most did not complain of bleeding, although some of
them complained of bleeding accompanied by pain if the lesion was traumatised.
This is suitable with studies conducted by Leonard, which found that the lips
and cheeks are the prominent predilection for arteriovenous malformations in
the maxillofacial area ((Zhou & Chen, 2020);(Tantray, Iram Shafi, Chauhan, &
Muzaffar, 2020))
The gender prevalence of
arteriovenous malformations, according to studies, is dominated by women; this
is different from research at Hasan Sadikin General Hospital, which states the
ratio is the same between men and women. This sex ratio, which is not
significantly different, is probably because arteriovenous malformations are
sporadic but can be inherited in families as an autosomal dominant trait, and
the number of samples and time in this study was smaller compared to other
studies. Men and women are equally affected, and gene expression has great variability. Arteriovenous malformations are
one manifestation of many different genetic syndromes that have varying
inheritance patterns, and the chance of recurrence depends on the specific
syndrome present. ((Lazaris, 2019);(Thiele & Nemergut, 2020)).
The next most common type of vascular malformation is lymphatic
malformation. This study found lymphatic malformations in 6 patients, with main
complaints in the form of lumps, bleeding, pain, and aesthetics. This is
suitable for a study conducted by Leonard, which found that complaints of
lymphatic malformations can include swelling, pain, and even fever. Based on
anamnesis is found after birth and develops with increasing age (Kilich, Perelygina, & Sullivan, 2023).
Studies state that lymphatic malformations can occur in all parts of the
body, and from 4 patients studied at Hasan Sadikin General Hospital, 3 of them
had a predilection that spread to the tongue, buccal mucosa, and lips, while one
patient experienced malformation in the tongue area. This is suitable for a
study conducted by Leonard, which found that lymphatic malformations occur
spread in the area of the tongue, floor of the mouth, mandible, submandibular,
and neck (Jiang et al., 2023).
The lymphatic malformations in this study were not accompanied by bleeding.
However, the patient complained of pain; this is suitable with what Elise et
al. stated, that lymphatic malformations tend to swell when infection or trauma
occurs. Prodromal symptoms may accompany complaints of infection. Tertiary
lymphoid organs in lymphatic malformations can trigger inflammation of the
airway mucosa and interfere with masticatory function (Bonilla‐Velez et al., 2021).
The standard therapy carried out at RSHS for
all vascular anomalous lesions is by using sclerotic agent injection with a
total of 40 cases, with the injection of choice being bleomycin. According to
Kaban et al., hemangioma lesions can be observed first until they reach the
involution phase. Subsequently, if the lip lesion ulcerates, regular cleaning
is required, and topical antibiotic use is recommended. However, if bleeding
occurs, pressure can be used to control bleeding. Pharmacological therapy is
given when local therapy is inadequate to heal ulceration. Lesions that cause
necrosis, damage to vital structures, and airway obstruction can be treated
with pharmacological therapy in the form of intralesional injections to speed
up the involution phase. Surgical management, such as excision, is indicated
for ulcerated lesions that bleed, causing respiratory and visual impairment. In
small venous malformations, injection of 1% sodium tetradecyl sulfate is
indicated. If symptoms are accompanied by phleboliths, then anticoagulant
therapy with aspirin is recommended (Cooke-Barber,
Kreimer, Patel, Dasgupta, & Jeng, 2020).
In a study by Berenger et al., in 40 patients
treated with large doses of ethanol, 30 patients (75%) showed marked
improvement or complete recovery. In comparison, ten patients (25%) experienced
slight improvement or did not respond to treatment. The main complications
reported include acute blistering (50%), hemoglobinuria (28%), deep ulceration
(13%), and nerve injury (7.5%). Temporary facial paresis was reported in two
patients, and permanent unilateral vocal cord paralysis in one patient. (30) We
have received several reports from the use of ethanol on venous malformation
lesions outside the oral cavity, causing tissue necrosis; this is our basis for
using bleomycin injection as therapy for malformations of oral cavity veins at
Hasan Sadikin General Hospital.
Lee et al. looked at 87 patients who had undergone
sclerotherapy with large doses of ethanol (total 305 sessions; mean 3.5). The
results obtained were excellent in 23 patients (32.4%), good in 37 patients
(52.1%) and poor in 11 patients (15.5%). Patients who experience swelling and
pain at the injury site are treated with intravenous or intramuscular analgesic
drugs. Other complications reported (four patients; 4.6%) were respiratory
distress (two cases), tongue hypoesthesia (one case), and temporary facial
nerve paralysis (one case). In a study by Liu et al., 23 patients were treated
with low-dose ethanol and followed up for an average of 20 months. All patients
experienced remission or reduction of symptoms. Improved clinical outcomes were
obtained in 9 and 14 patients, respectively. Patients who exhibit mild to
moderate swelling and pain are treated conservatively and recover within a few
days. Skin necrosis or nerve damage was not reported (Lim et al., 2021).
In the current study, injection sclerotherapy was
considered an effective treatment for vascular malformations. However, based on
other research sources, therapy with different types of sclerotic agents, such
as ethanol, can be carried out at low doses to prevent tissue necrosis and
fibrosis. Besides that, even though the hemangioma lesion will involute and
undergo a regression process, treatment with intralesional injection into the
oral cavity is needed as early as possible to prevent bleeding during the
process of eating and chewing and prevent interference with other vital organs.
CONCLUSION
REFERENCES
Aziz,
H., Brown, Z. J., Baghdadi, A., Kamel, I. R., & Pawlik, T. M. (2022). A
comprehensive review of hepatic hemangioma management. Journal of
Gastrointestinal Surgery, 26(9), 1998–2007.
Behravesh,
S., Yakes, W., Gupta, N., Naidu, S., Chong, B. W., Khademhosseini, A., &
Oklu, R. (2016). Venous malformations: clinical diagnosis and treatment. Cardiovascular
Diagnosis and Therapy, 6(6), 557.
Bonilla‐Velez,
J., Whitlock, K. B., Ganti, S., Theeuwen, H. A., Manning, S. C., Bly, R. A., …
Perkins, J. A. (2021). Active observation as an alternative to invasive
treatments for pediatric head and neck lymphatic malformations. The
Laryngoscope, 131(6), 1392–1397.
CAGLIARI,
S. S. O. F. (2021). Selected Abstracts of the 17th International Workshop on
Neonatology and Pediatrics• Virtual Edition. Journal of Pediatric and
Neonatal Individualized Medicine, 10(2), e100229.
Chinyama,
C. N. (2020). Hamartoma. Breast Pathology, 135–143.
Cooke-Barber,
J., Kreimer, S., Patel, M., Dasgupta, R., & Jeng, M. (2020). Venous
malformations. Seminars in Pediatric Surgery, 29(5), 150976.
Elsevier.
El
Zein, S., Boccara, O., Soupre, V., Vieira, A. F., Bodemer, C., Coulomb, A., …
Fraitag, S. (2020). The histopathology of congenital haemangioma and its
clinical correlations: a long‐term follow‐up study of 55 cases. Histopathology,
77(2), 275–283.
Frongia,
G., Byeon, J.-O., Mehrabi, A., & Günther, P. (2021). Recurrence rate of
infantile hemangioma after oral propranolol therapy. European Journal of
Pediatrics, 180, 585–590.
Habibie,
Y. A. (2021). The 4th Aceh Surgery Update 2021. Bali Medical Journal, 10(1),
1–65.
Hussein,
A., & Malguria, N. (2020). Imaging of vascular malformations. Radiologic
Clinics, 58(4), 815–830.
Jiang,
Y., Liu, Y., Wang, X., Chen, Z., Shi, W., & Yu, G. (2023). Classification
specific lymphatic malformations management on head and neck in children. Oral
Surgery, Oral Medicine, Oral Pathology and Oral Radiology, 135(4),
e74–e82.
Jorge,
A. F. F., Nascimento, L., Fernandes, L., & Soares, E. (2022). Rapidly
involuting congenital haemangioma: diagnostic and therapeutic approach
regarding two case reports. BMJ Case Reports CP, 15(2), e247689.
Kilich,
G., Perelygina, L., & Sullivan, K. E. (2023). Rubella virus chronic
inflammatory disease and other unusual viral phenotypes in inborn errors of
immunity. Immunological Reviews.
Lazaris,
A. M. (2019). Regarding “The Society for Vascular Surgery practice guidelines
on the care of patients with an abdominal aortic aneurysm.” Journal of
Vascular Surgery, 69(3), 975.
Lee,
C.-H., & Chen, S.-G. (2005). Direct percutaneous ethanol instillation for
treatment of venous malformation in the face and neck. British Journal of
Plastic Surgery, 58(8), 1073–1078.
Lim,
C. S., Evans, N., Kaur, I., Papadopoulou, A., Khalifa, M., Tsui, J., … Brookes,
J. (2021). Incidence of major complication following embolo-sclerotherapy for
upper and lower extremity vascular malformations. Vascular, 29(1),
69–77.
Mukhopadhyay,
S., Galui, S., Biswas, R., Saha, S., & Sarkar, S. (2020). Oral and
maxillofacial injuries in children: a retrospective study. Journal of the
Korean Association of Oral and Maxillofacial Surgeons, 46(3), 183.
Munden,
A., Butschek, R., Tom, W. L., Marshall, J. S., Poeltler, D. M., Krohne, S. E.,
… Catanzarite, V. (2014). Prospective study of infantile haemangiomas:
incidence, clinical characteristics and association with placental anomalies. British
Journal of Dermatology, 170(4), 907–913.
Rendón
Elías, F. G., Hernández Sánchez, M., Albores Figueroa, R., Montes Tapia, F. F.,
& Gómez Danés, L. H. (2014). Congenital vascular malformations update. Medicina
Universitaria, 16(65), 184–198.
Sebaratnam,
D. F., Wong, L.-C. F., & Wargon, O. (2021). Infantile hemangioma. Part 2:
management. Journal of the American Academy of Dermatology, 85(6),
1395–1404.
Smith,
H., Lim, C. S., Evans, N., Papadopoulou, A., Khalifa, M., Tsui, J., … Brookes,
J. (2022). Incidence of major complications from embolo-sclerotherapy of head
and neck vascular malformations in a single specialist centre. Vascular,
30(5), 952–959.
Soman,
S., Das, T. A., Kalathil, L. S. M., Aslam, S., Thomas, T., & Depesh, V.
(2020). Hemangiomas of the oral and maxillofacial region: a series of five
cases with literature review. International Journal of Oral Care and
Research, 8(4), 97–102.
Steiner,
J. E., & Drolet, B. A. (2017). Classification of vascular anomalies: an
update. Seminars in Interventional Radiology, 34(03), 225–232.
Thieme Medical Publishers.
Tantray,
S., Iram Shafi, R., Chauhan, K., & Muzaffar, S. (2020). Oral pathology in
paediatric patients-mini-systematic review. IP International Journal of
Medical Paediatrics and Oncology, 6(3), 96–102.
Tasker,
R. C., Acerini, C. L., Holloway, E., Shah, A., & Lillitos, P. (2021). Oxford
Handbook of Paediatrics 3e. Oxford University Press.
Thiele,
E. L., & Nemergut, E. C. (2020). Miller’s anesthesia. Anesthesia &
Analgesia, 130(6), e175–e176.
Yajun,
M., Shan, W., & Qihong, F. (n.d.). New Progress in Clinical Treatment of Infantile
Hemangioma. Vol, 10, 355–365.
Zhang,
H., Zhou, Q., & Shen, W. (2020). Clinical Features and Management of
Oral Nonodontogenic Masses in Children.
Zhou,
J., & Chen, Z. (2020). Rare adult pancreatic hemangioma: review of the
literature with a case report. Gland Surgery, 9(5), 1596.
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