vascular enlargement of lower lip - a case report · regional flaps are available. this paper...

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U n ive rs ity J D en t S c ie 20 15 ; 1(2) : 80 -82 ABSTRACT: Hemangiomas are benign skin lesions of infancy and childhood, occurring in 10- 12% of children at 1 year of age. Commonly located in head and neck region (60% of cases) and occur more frequently in the lips, tongue, and palate. 50% of hemangiomas have complete resolution up to 1 year of age. Complications occur only in 20% of the cases; most common is ulceration. Many treatment modalities exist including conservative and surgical methods. When non-operative modalities fail, surgery is usually needed. Small lesions can be treated by horizontal and vertical wedge resections, elliptical excisions and for large vascular lesions; different local and regional flaps are available. This paper reports a case of cavernous hemangioma in a 40 year-old male patient, who underwent simple excision (Reduction cheiloplasty) of the hemangioma of lower lip. One year follow-up shows successful treatment with no recurrence. 1 2 3 4 Himanshu P Singh, Sunil K Mall, Anuj Garg, Saad Ahmed 1,2 Sr. Lecturer, Department of Oral & Maxillofacial Surgery, 3,4 Institute of Dental Sciences, Bareilly Senior Lecturer, Department of Oral Pathology & Microbiology, Dental College, Azamgarh INTRODUCTION : In 1982, Mulliken and Glowacki, classified vascular lesions as haemangiomas or vascular malformations.1,2 The haemangioma is a benign proliferation of endothelial cells common in the head and neck. These lesions are three to five times more common in females, with an even higher female preponderance in hemangiomas that are problematic or associated with structural abnormalities. There is an increased frequency of hemangiomas in premature infants with a reported incidence of 23% in neonates who weigh less than 1200 g. Hemangiomas are unusual in dark-skinned infants.3 The most frequent location for oral haemangiomas is the lip. Treatment is needed to prevent complications such as ulceration, infection, bleeding or obstruction of the airway.1 Historically, hemangiomas have been classified in a variety of ways. An important descriptive classification is related to the depth of soft tissue involvement i.e. superficial, deep, and mixed.4 The term cavernous hemangioma has traditionally been applied when lesional vascular channels are considerably enlarged.4 The large forms of infantile cavernous hemangiomas frequently have accelerated growth and may cause significant functional disturbances and existential complications.5 The Head & Neck region is more commonly affected especially the face, oral mucosa, lips, tongue and trunk. Size can vary from few millimeters to several centimeters (0.25 to 200 cm3). The skin is the organ of most frequent occurrence.6 The clinical appearances of the lesions vary from an anemic spot, at times erythematous, to a small cluster of deep red papules.7 The initiation of lesion occurs with a rapid growth stage, and at the end of the first month of life approximately 90% of the cases become evident. Followed, a stagnation stage is initiated and continues for several months. Then, they involute slowly showing a regression rate of 90% up to 10 years of age. An unbalance in the angiogenesis seems to be main reason for the development of hemangioma, which causes an uncontrolled proliferation of vascular elements,8 associated with substances such as vascular endothelial growth factor (VEGF), basic fibroblast growth factor (BFGF) and indoleamine 2,3-dioxygenase (IDO), which are found in large amount during proliferative stages, although reduced throughout involution stage.5,9 Clinical findings and semiotic maneuvers, such as diascopy or glass-slide pressure are very helpful in differentiating vascular from nonvascular lesions.8 Diagnostic imaging has also a great role in revealing size, extension and location, as well as for follow up of lesions treated under a systemic therapy.10 VASCULAR ENLARGEMENT OF LOWER LIP - A CASE REPORT Journal of Dental Sciences University Key Words: Hemangioma, intramuscular hemangioma, cavernous hemangioma. Source of support : Nil Conflict of interest : None Case Report 80

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Page 1: VASCULAR ENLARGEMENT OF LOWER LIP - A CASE REPORT · regional flaps are available. This paper reports a case of cavernous hemangioma in a 40 year-old male patient, who underwent simple

University J Dent Scie 2015; 1(2) : 80-82

ABSTRACT: Hemangiomas are benign skin lesions of infancy and childhood, occurring in 10-12% of children at 1 year of age. Commonly located in head and neck region (60% of cases) and occur more frequently in the lips, tongue, and palate. 50% of hemangiomas have complete resolution up to 1 year of age. Complications occur only in 20% of the cases; most common is ulceration. Many treatment modalities exist including conservative and surgical methods. When non-operative modalities fail, surgery is usually needed. Small lesions can be treated by horizontal and vertical wedge resections, elliptical excisions and for large vascular lesions; different local and regional flaps are available. This paper reports a case of cavernous hemangioma in a 40 year-old male patient, who underwent simple excision (Reduction cheiloplasty) of the hemangioma of lower lip. One year follow-up shows successful treatment with no recurrence.

1 2 3 4Himanshu P Singh, Sunil K Mall, Anuj Garg, Saad Ahmed 1,2 Sr. Lecturer, Department of Oral & Maxillofacial Surgery,

3,4Institute of Dental Sciences, Bareilly Senior Lecturer, Department of Oral Pathology & Microbiology, Dental College, Azamgarh

INTRODUCTION : In 1982, Mulliken and Glowacki,

classified vascular lesions as haemangiomas or vascular

malformations.1,2 The haemangioma is a benign

proliferation of endothelial cells common in the head and

neck. These lesions are three to five times more common in

females, with an even higher female preponderance in

hemangiomas that are problematic or associated with

structural abnormalities. There is an increased frequency of

hemangiomas in premature infants with a reported incidence

of 23% in neonates who weigh less than 1200 g.

Hemangiomas are unusual in dark-skinned infants.3 The

most frequent location for oral haemangiomas is the lip.

Treatment is needed to prevent complications such as

ulceration, infection, bleeding or obstruction of the airway.1

Historically, hemangiomas have been classified in a variety of

ways. An important descriptive classification is related to the

depth of soft tissue involvement i.e. superficial, deep, and

mixed.4 The term cavernous hemangioma has traditionally

been applied when lesional vascular channels are

considerably enlarged.4 The large forms of infantile

cavernous hemangiomas frequently have accelerated growth

and may cause significant functional disturbances and

existential complications.5

The Head & Neck region is more commonly affected

especially the face, oral mucosa, lips, tongue and trunk. Size

can vary from few millimeters to several centimeters (0.25 to

200 cm3). The skin is the organ of most frequent occurrence.6

The clinical appearances of the lesions vary from an anemic

spot, at times erythematous, to a small cluster of deep red

papules.7 The initiation of lesion occurs with a rapid growth

stage, and at the end of the first month of life approximately

90% of the cases become evident. Followed, a stagnation

stage is initiated and continues for several months. Then, they

involute slowly showing a regression rate of 90% up to 10

years of age. An unbalance in the angiogenesis seems to be

main reason for the development of hemangioma, which

causes an uncontrolled proliferation of vascular elements,8

associated with substances such as vascular endothelial

growth factor (VEGF), basic fibroblast growth factor (BFGF)

and indoleamine 2,3-dioxygenase (IDO), which are found in

large amount during proliferative stages, although reduced

throughout involution stage.5,9 Clinical findings and

semiotic maneuvers, such as diascopy or glass-slide pressure

are very helpful in differentiating vascular from nonvascular

lesions.8 Diagnostic imaging has also a great role in revealing

size, extension and location, as well as for follow up of lesions

treated under a systemic therapy.10

VASCULAR ENLARGEMENT OF LOWER LIP - A CASE REPORT

Journal of Dental Sciences

University

Key Words: Hemangioma, intramuscular hemangioma, cavernous hemangioma.

Source of support : NilConflict of interest : None

CaseReport

80

Page 2: VASCULAR ENLARGEMENT OF LOWER LIP - A CASE REPORT · regional flaps are available. This paper reports a case of cavernous hemangioma in a 40 year-old male patient, who underwent simple

University J Dent Scie 2015; 1(2) : 80-82

CASE REPORT : A 40 year-old male patient presented with

a volume increase in the lower lip since long time. The lesion

was compromising his facial esthetics. History revealed the

presence of a red spot on lower lip at the age of 6 months,

which had increased considerably ever since. Clinically, the

lesion was pale red, fluctuant, and had quite elevated areas

[Fig.1]. Lesion was painless and extended over the entire

lower lip, changing color under compression (diascopy).

Radiographically, there were no alterations and the bone

structure of jaws was normal. Fine needle aspiration biopsy

confirmed the diagnosis of hemangioma. Reduction

Cheiloplasty was performed as treatment was mainly of

esthetic concern for patient.

The surgery was carried out under general anesthesia in a

private hospital (Mukherjee Hospital, Ayub khan-Chaupla

road, Bareilly, U.P). Marking was followed by injection of

dilute adrenaline 1:200,000 and isolation of the area. The

lesion is removed in a rectangular fashion. Excess amount is

excised both intraoraly & extraoraly [Fig.2,3]. Healing was

uneventful without necrosis or infection except for a

superficial dehiscence of the vermilion border that healed

with conservative treatment. Muscle function returned

immediately after disappearance of edema. The lower lip was

in normal balance with intact commisures and adequate

buccal sulcus. No drooling was observed or reported, and the

nasolabial and labiomental folds were preserved. Lip

movements including opening the mouth, pouting the lips,

blowing up the cheeks and other movements were restored to

normal. One year after treatment, the esthetic result was

remarkable [Fig.4].

Fig.1- Preoperative photo

Fig.2

Fig.3

Excess tissue excised intraorally (Fig.2) & extraorally (Fig.3)

Fig.4- Postoperative photo

DISCUSSION : Hemangiomas are the benign tumors. They

have different life cycle, which includes 3 stages: 1) the

proliferating phase (0–1 year), 2) the involuting phase (1–5

years), and 3) the involuted phase (>5 years). They are generally

noted with in first 6 months of postnatal life as an erythematous

spot.7,11,15 These lesions are three to five times more common

in females, and the superficial type is the most frequent one,1,2,3

unlike the present case. According to the cases reported in the

literature, approximately 80% of the patients present with a

single lesion, and the head and neck sites are more commonly

affected.6 Hemangiomas of the lip were found to regress less

completely than elsewhere in the body. Surgeons are often asked

to intervene not only when hemangiomas cause life threatening

conditions but also for cosmetic concerns.

In the present case, although the lesion was very old but the

patient sought treatment only when the lesion causes esthetic and

social impairments, as observed in previously reported cases.16

Treatment plan for hemangiomas must consider the size,

location, lesion hemodynamics & patients age.14

Systemic corticosteroid therapy is considered as the most

efficient treatment for infantile hemangiomas if started early in

its first phase of life cycle. It is the first choice of therapy to treat

hemangiomas of infancy. Triamcinolone (25 mg/mL), at a

dosage of 3 to 5 mg/kg, injected slowly at a low pressure is most

commonly used. Main adverse-effects in use of long-term

systemic corticosteroids includes: cushingoid features,

disturbed growth pattern, susceptibility to serious infections,

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Page 3: VASCULAR ENLARGEMENT OF LOWER LIP - A CASE REPORT · regional flaps are available. This paper reports a case of cavernous hemangioma in a 40 year-old male patient, who underwent simple

University J Dent Scie 2015; 1(2) : 80-82

loss of appetite, behavioral changes, polyuria, polyphagia,

thrush, and gastrointestinal discomfort.17 If systemic

corticosteroids don't respond, surgery is indicated, especially

for esthetic corrections. Some authors advice sclerotherapy

before surgery because endothelial cell fibrosis facilitates

lesion removal, thus avoiding hemorrhage risk.8,13 Amongst

different sclerosing agents available in the market, excellent

results have been reported with sodium morrhuate, sodium

sulfate tetradecyl, polydocanol and ethanolamine oleate, and

hypertonic glucose solution.12,13,18

In present case, the size and location of the lesion, and

systemic condition of the patient allowed to perform the

surgical procedure with favorable prognosis. Postoperative

period was uneventful and shows satisfactory healing of the

operated area. In conclusion, surgical excision is a treatment

option to be considered as it provides good esthetic and

functional outcomes.

REFERENCES :

1. Bonet-Coloma C, et al. Clinical characteristics, treatment

and outcome of 28 oral haemangiomas in pediatric

patients Med Oral Patol Oral Cir Bucal. 2011;16:19-22.

2. El-sayed Ibrahim El-shafey, Ali Abdel-wahab. Vascular

Lower Lip Enlargement:Reconstruction Using the Step

Technique. Egypt, J. Plast. Reconstr. Surg. 2005;29:97-

104.

3. Jennifer J. Marler, John B. Mulliken. Current

management of hemangiomas and vascular

malformations. Clin Plastic Surg. 2005;32:99 – 116.

4. Sobrinho FPG, Félix MA, Valle AC, Lessa HA.

Hemangioma de úvula: relato de um caso. Rev Bras

Otorrinolaringol 2000;69:571- 574.

5. Frieden IJ. Management of hemangiomas. Special

symposium. Ped Dermatol 1997;14:57-83.

6. Mulliken JB, Fishman SJ, Burrows PE. Vascular

anomalies. Curr Probl Surg 2000;37:518-584.

7. E sterly NB. Hemangiomas. In: Harper J, Oranje A, Prose

N, eds. Textbook of pediatric dermatology. Oxford:

Blackwell Science. 2000:997-1016.

8. Matsumoto K, Nakanishi H, Koizumi Y, Seike T, Kanda

I, Kubo Y. Sclerotherapy of hemangioma with late

involution. Dermatologic Surgery 2003;29:668-671.

9. N ishida R, Inoue R, Takimoto Y, Kita T. A sclerosant

with astringent properties developed in China for

oesophageal varices: comparason with ethanolamine

oleate and polidocanol. J Gastroenteral Hepatol

1999;14:481-488.

10. E ichenfield LF. Evolving knowledge of hemangiomas and

vascular malformations. Arch Dermatol 1998;134:740-742.

11. Dourmishev LA, Dourmishev AL. Craniofacial cavernous

h e m a n g i o m a : s u c c e s s f u l t r e a t m e n t w i t h

methylprednisolone. Acta Dermatoven APA 2005;14:49-

52.

12. Marchuk DA. Pathogenesis of hemangioma. J Clin Invest

2001;107:665-666.

13. Johann ACBR, Aguiar MCF, Carmo MAV, Gomez RS,

Castro WH, Mesquita RA. Sclerotherapy of begin oral

vascular lesion with ethanolamine oleate: an open clinical

trial with 30 lesions. Oral Surg Med Pathol Oral Radiol

Endod 2005;100:579-584.

14. B aurmash H, Mandel L. The nonsurgical treatment of

hemangioma with sotradecol. Oral Surg Oral Med Oral

Pathol 1963;16:777-782.

15. Zhao Y, Glesne D, Huberman E. A human peripheral blood

monocyte - derived subset acts as pluripotent stem cells.

Proc Nati Acad Sci USA 2003;100:2426-2431.

16. Linda C, Chang LC, Haggstrom AN, Drolet BA, Baselga E,

Chamlin SL, et al. Growth characteristics of infantile

hemangiomas: implications for management. Ped Dermatol

2008;22:360-367.

17. Zhou Q, Yang XJ, Zheng JW, Wang YA, Zhang ZY. Short-

term high-dose oral prednisone on alternate days is safe and

effective for treatment of infantile hemangiomas. J Oral

Maxillofac Surg 2010;109:166-167.

18. U n ZJ, Zhang L, Zhanh WF, Alsharif MJ, Chen XM, Zhao

YF. Epithelioid hemangioma in the oral mucosa: a

clinicopathological study of seven cases and review of the

literature. Oral Oncology 2006;42:441-447.

CORRESPONDANCE :

Dr. Himanshu Pratap Singh, MDS

Oral & Maxillofacial Surgeon,

Institute of Dental Sciences, Bareilly (U.P.)-INDIA.

Email : [email protected]

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