| 20 | Smile Dental Journal | Volume 5, Issue 2 - 2010 Congenital Hemangioma of Parotid Gland in Infancy A Case Report Saydé Sokhn Challita CD, DUA, DUB Department of Oral and Maxillofacial Radiology, Lebanese University, School of Dentistry, Beirut, Lebanon [email protected]Abstract Hemangiomas of the parotid gland represent about 1-5% of all salivary gland tumors. Hemangiomas are common neoplasms in childhood but capillary hemangiomas are rare in parotid glands. We present a case of congenital capillary hemangioma of the parotid gland. Clinical history and physical examination are usually enough to deduce that the lesion is a hemangioma. The CT and ultra-sonographic (US) findings confirmed the vascular nature of the lesion and showed the extension of this latest that involved the whole parotid gland. The most appropriate management for such neoplasm is conservative therapy. Key Words: Parotid gland; Vascular lesion; Hemangioma. Introduction The classification of vascular lesions of the head and neck has been somewhat confusing. In 1982, Mulliken and Glowacki proposed a classification for these vascular lesions that was based on their natural history as well as their cellular structure. This classification provides insight into the natural history and management of vascular lesions. These authors identified two main categories of lesions: hemangioma (HA) and vascular malformations. 1 Utilizing the classification proposed by Mulliken and Glowacki, the term hemangioma should be reserved for vascular masses that are observed in early infancy, rapidly enlarge, and ultimately regress during adolescence. This classification has been adopted and reported by many authors. 1,2 Unlike hemangiomas, vascular malformations are not tumors. Rather, they are true congenital vascular anomalies that may not be clinically observed until late infancy or early childhood. This paper reports a case of capillary parotid hemangioma in infancy. Case Presentation The baby girl was born in March 2006. At birth, she had a cutaneous vascular mark on the right side of her lower lip. At the age of 4 months, this stain started to grow and a swelling was noted in her right parotid region. The swelling continued to grow until she had 15 months and remained stable till the age of 30 months (Figure 1). No remarkable medical history until this day was noticed. At 4 months age, the physical exam demonstrated swelling that extended from the lobe of the right ear to the inferior lip. The overlying skin was normal and smooth with no signs of redness; on palpation the lesion was compressible; and a change in size during crying or straining was observed. At this time, her doctor recommended an ultra-sonographic (US) exam to evaluate the lesion content. The images showed an auricular structure indistinguishable from the parotid- masseterin region, lobular in contour and measuring 3.5 x 2.5cm approximately. On US, this structure did not present a homogenous glandular architecture but was hypoechogenic, multilocular and appeared to be well vascularised. This aspect of the lesion and its evolution evoked a neoplasm of the parotid gland. The left parotid gland was normal (Figure 2). Hitaf Nasrallah Nasseh CD, DUA, DUB Department of Pediatric Dentistry, Lebanese University, School of Dentistry, Beirut, Lebanon [email protected]
AbstractHemangiomas of the parotid gland represent about 1-5% of all salivary gland tumors. Hemangiomas are common neoplasms in childhood but capillary hemangiomas are rare in parotid glands. We present a case of congenital capillary hemangioma of the parotid gland. Clinical history and physical examination are usually enough to deduce that the lesion is a hemangioma.
The CT and ultra-sonographic (US) findings confirmed the vascular nature of the lesion and showed the extension of this latest that involved the whole parotid gland. The most appropriate management for such neoplasm is conservative therapy.
IntroductionThe classification of vascular lesions of the head and neck has been somewhat confusing. In 1982, Mulliken and Glowacki proposed a classification for these vascular lesions that was based on their natural history as well as their cellular structure. This classification provides insight into the natural history and management of vascular lesions. These authors identified two main categories of lesions: hemangioma (HA) and vascular malformations.1 Utilizing the classification proposed by Mulliken and Glowacki, the term hemangioma should be reserved for vascular masses that are observed in early infancy, rapidly enlarge, and ultimately regress during adolescence. This classification has been adopted and reported by many authors.1,2
Unlike hemangiomas, vascular malformations are not tumors. Rather, they are true congenital vascular anomalies that may not be clinically observed until late infancy or early childhood.This paper reports a case of capillary parotid hemangioma in infancy. Case PresentationThe baby girl was born in March 2006. At birth, she had a cutaneous vascular mark on the right side of her lower lip. At the age of 4 months, this stain started to grow and a swelling was noted in her right parotid region. The swelling continued to grow until she had 15 months and remained stable till the age of 30 months (Figure 1).
No remarkable medical history until this day was noticed.
At 4 months age, the physical exam demonstrated swelling that extended from the lobe of the right ear to the inferior lip. The overlying skin was normal and smooth with no signs of redness; on palpation the lesion was compressible; and a change in size during crying or straining was observed.
At this time, her doctor recommended an ultra-sonographic (US) exam to evaluate the lesion content. The images showed an auricular structure indistinguishable from the parotid-masseterin region, lobular in contour and measuring 3.5 x 2.5cm approximately. On US, this structure did not present a homogenous glandular architecture but was hypoechogenic, multilocular and appeared to be well vascularised. This aspect of the lesion and its evolution evoked a neoplasm of the parotid gland. The left parotid gland was normal (Figure 2).
Hitaf Nasrallah NassehCD, DUA, DUB
Department of Pediatric Dentistry, Lebanese University, School of Dentistry, Beirut, Lebanon
To evaluate the medial extension of the lesion, a CT scan with contrast was recommended. The CT images showed a mass in the right parotid gland that appeared to be multilocular and nodular and was measuring 2.5 x 4cm. This mass was enhanced with contrast agent. High attenuation areas appeared to be related to a vascular malformation.
The posterior fossa did not present any visible vascular malformation and abnormalities. The pterygoid fossa was intact (Figure 3 (a-b)).
The findings were suggestive of a HA of the right parotid gland overtaking the whole gland, extended to the lip and associated with vascular turgescence ahead. No other abnormalities were found.
Medical treatment such as steroids and interferon therapy has been proposed by some doctors but the
parents refused. After many opinions, a treatment was not recommended at this age but a follow-up schedule was set-up to evaluate the progression of the lesion.
DiscussionNon-epithelial tumors overall constitute less than 5% of all salivary gland neoplasms, but in children they represent more than 50% of all tumors. These include lymphangiomas, hemangioma, lymphoid lesions, neurogenic tumors, lipomas and sarcomas.3 In addition; salivary gland neoplasms are rare and account for less than 1% of head and neck neoplasms. Less than 4% of salivary gland neoplasms occur before the age of sixteen.4
Capillary hemangiomas are rare in the parotid gland. Submandibular gland involvement is also rare. However in this case it may be difficult to distinguish between tumors arising within the gland or those arising within the soft tissue adjacent to the gland.
The normal parotid glands can be easily visualized on US as homogenous echogenic structures.4 On US, the echogenicity of a HA is variable and depends on the size of the cystic component.3 The gland is usually enlarged and appears hypoechoic or more or less isoechoic, but may be echogenic compared to the surrounding cervical soft tissues. They are highly compressible. Color doppler study helps to detect the perfusion in the HA and confirm the vascular nature of the lesion and helps to differentiate it from a lymphangioma.
On CT, these tumors are often lobular, may extend to the overlying skin or may have phleboliths. The mass effect of the tumor on the surrounding tissues is relatively rare reflecting their soft nature, but our patient present an evident labial extension.
In general, HA typically appear several weeks after birth, and undergo rapid enlargement out-of-proportion to the child’s growth. Sometime, a slow involution phase can be observed5,6 whereas 45% were first observed as a neonatal stain like our case.
The involution phase is characterized by a slow diminution in tumor size, can take many years to complete and ultimately may leave residual stigmata. No treatment is indicated because of likely spontaneous involution, but treatment may be needed in cases of “alarming” HA or when there is cosmetic disfigurement.1,4 It has been reported in literature that female-to-male ratio was 4.5:1.1,3,5
40% of parotid HAs were on the right side, 36% were on the left, and 24% were bilateral.Whereas ulceration has been reported to occur in 5% of all cutaneous hemangiomas, some authors found that 59% of parotid hemangiomas ulcerated during the early proliferative phase.6
(Fig. 1) Fifteen month’s old baby with a swelling on her right parotid gland region and the lower lip.
(Fig. 2) Conventional sonographic exam, taken at the age of four months, demonstrates a hypoechogenic and multilocular glandular architecture. This structure appeared to be well vascularised.
Because of the benign nature of this lesion, accurate clinical and radiologic diagnosis is important so that unnecessary biopsy may be avoided. Any large, rapidly growing parotid mass in an infant, particularly if it is small or unnoticed at birth, is most likely to be HA.
Significant systemic complications such as cardiac failure or Kasabach-Merritt syndrome are rare. When they occur, therapy with oral corticosteroids or interferon α2a or both may be used, although these may be less successful for parotid lesions than for HA arising at other sites. Surgical resection is not recommended because of the risk of damage to the facial nerve and favorable prognosis with
expectant management. Reinisch et al. suggests that early resection is a reasonable treatment option when performed by an experienced surgeon familiar with the anatomy of the facial nerve and parotid region.7,8
Parotid hemangiomas are usually evident on physical examination. These tumors can be confirmed by various diagnostic tools, such as US, computed tomography, and magnetic resonance imaging. US may be appropriate for initial evaluation of pediatric salivary gland lesions because of its low cost and noninvasive nature. Magnetic resonance imaging may be the preferred method of study for vascular lesions involving the parotid gland
(Fig. 3-a) A CT exam. There is an augmentation of the right parotid gland that appears to be multilocular and nodular and measuring 2.5 x 4cm.
(Fig. 3-b) A CT exam enhancing with contrast agent: there is a high attenuation area that appears to be related to a vascular turgescence.
because of excellent soft tissue resolution. This diagnostic study is expensive and may require sedation or anesthesia in young children. US and MRI both demonstrate blood �ow and the parenchyma without using ionizing radiation. 2,6,7,8
In the present case, parotid HA was diagnosed by clinical history, physical examination, ultrasonography and computed tomography. In our patient, US at 10 MHz showed a non-homogeneous mass enlarging and replacing most of the parotid gland, a lobular structure with �ne echogenic internal septations, and numerous large intratumoral vessels. Previously published cases showed similar sonographic �ndings to those found in our patient but with homogeneous mass. 8
The di�erential diagnosis includes cystic lymphangioma and the rare malignant hemangioendothelioma, both of which can be distinguished histologically. There are no known instances of malignant transformation of this lesion. Typically these capillary HA of early childhood regress by adulthood. 1,3,8
Complete natural regression is usually seen in over 50% of children by 5 years of age and 70% in children 7 years of age. 2,4
In our case, the regression was 70% at the age of 40 months without any treatment. However, most hemangioma requires no treatment unless life threatening complications occur.
ConclusionWe have shown a case of parotid HA with inferior lip extension with non-homogeneous mass and the conservative therapy was the treatment of choice. Many previous studies said:” once the natural regression of infantile hemangioma is fully appreciated, observation becomes the standard treatment”. 1,5,9
References1. Peter M. Som, M.D., Hugh D. Curtin, M.D. Head and Neck
Imaging. Fourth edition. Mosby 2003.2. Savvas Andronikou, Kieran McHugh and Jane Linward. MRI
features of bilateral parotid haemangiomas of infancy. Eur Radiol. 2003;13:711-6.
3. Bhatt CJ, Jagrit NN. Congenital hemangioma of parotid gland: a case report. Indian J Radiol Imaging. 2003;13:15-6.
4. John F. Reinisch, Raymond J. Harshbarger, M.D., and John G. Meara. Surgical Management of Parotid Hemangioma. Plast. Reconstr. Surg., June 2004.
5. Arin K. Greene and John B. Mulliken, M.D. Management of Parotid Hemangioma in 100 Children. Plast. Reconstr. Surg., January 2004.
6. Gammper, T. J., and Morgan, R. F. Vascular anomalies: Hemangiomas. Plast. Reconstr. Surg. 2002;110:572.
7. Boye, E., Yu, Y., Paranya, G., Mulliken, J. B., Olsen, B. R., and Bischo�, J. Clonality and altered behavior of endothelial cells from hemangiomas. J. Clin. Invest. 2001;107:745.
8. Derek J. Roebuck and Anil T. Ahuja. Hemangioendothelioma of the Parotid Gland in Infants:Sonography and Correlative MR Imaging. Am J Neuroradiol. January 2000;21:219-23.
9. Hani Hashem Sinno, MD, Sachin A. Chitte, MD, and H. Bruce Williams, MD. Management of the Parotid Gland Hemangioma in Infants. Plast.reconstruct. Surg. 2006.
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