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Journal of Advanced Clinical & Research Insights (2015), 1, 1–2

CASE REPORT

Parotid gland hemangioma with cytomegalovirus K. R. Chatura1, Mohammad Shahid Iqbal2

1Department of , Jagadguru Jayadeva Murugarajendra Medical College, Davangere, Karnataka, India, 2Department of Pathology, Kamineni Institute of Medical Sciences, Narketpally, Nalgonda, Andhra Pradesh, India

Keywords Abstract Cytomegalovirus, hemangioma, parotid Salivary glands are a common site for hemangiomas in the head and neck region. Surgical pathologists are unfamiliar with hemangioma in salivary glands. We report a rare case of Correspondence arteriovenous hemangioma of the parotid gland associated with cytomegalovirus. K. R. Chatura, Professor, Department of Pathology, Jagadguru Jayadeva Murugarajendra Medical College, Davangere - 577 004, Karnataka, India. Email: [email protected]

Received 05 September 2014; Accepted 25 October 2014 doi: ***

Introduction Pathological fi ndings

Hemangiomas are the most common benign vasoformative Macroscopically, a lobulated, soft tumor measuring tumors of infancy and childhood. According to Enzinger and 4 cm × 3 cm was received. It was unencapsulated. There were Weiss, hemangiomas are broadly classifi ed into capillary, areas of hemorrhage but no necrosis. Histological examination cavernous and miscellaneous forms such as verrucous, venous, did not demonstrate a border between the tumor and parotid arteriovenous hemangiomas and so forth.[1] Cytomegalovirus gland. Large thick and thin walled vessels fi lled with (CMV) infection has been observed in the parotid gland of were seen to intermingle with salivary lobules [Figure 1]. Large infants.[2] To the best of our knowledge, however, only two cases eosinophilic intranuclear inclusions with halos (owl’s eye of parotid juvenile hemangioma associated with CMV infection inclusions) and also basophilic cytoplasmic inclusions were seen have been described.[3,4] Therefore, we present a rare case of in many ductal epithelial cells [Figure 2]. Scant scattered chronic arteriovenous hemangioma with CMV infection in the salivary infl ammatory cells were seen in some lobules. Based on these gland, hitherto not reported. light microscopic fi ndings, lesion was diagnosed as arteriovenous hemangioma associated with parotid CMV infection. Case Report Discussion A 5-month-old male baby presented with a swelling on the right side of his face for the past 2 months. On examination, a mass Hemangioma is considered to be the most common was noted extending from the right preauricular region to the of childhood, and is one of the most common salivary gland middle of the neck. Sonography showed a large lobulated tumor tumors of childhood and infancy, but is rarely biopsied and in the right parotid region. The left parotid gland was normal. is, therefore, often unfamiliar to the surgical pathologist.[5] The parotid tumor was resected for a suspected clinical diagnosis Hemangiomas account for approximately 0.4% salivary gland of cystic hygroma and sent for histopathological examination. tumors and occur almost exclusively in the parotid (90%) No other clinical abnormalities had been detected. Mother and representing approximately 50% of parotid tumors in the 1st child were not immunocompromised. year of life.[6]

Journal of Advanced Clinical & Research Insights ● Vol. 1:4 ● Jan-Feb 2015 1 Parotid hemangioma with CMV Chatura and Iqbal

surprisingly high incidence of these inclusion-bearing or cytomegalic cells in the duct epithelium of salivary glands in infants at necropsy, 12% of 183 necropsies.[7] In our case, ductal epithelium showed viral cytopathic eff ect of CMV infection. Mattes et al. showed signifi cant association between the detection of owl’s eye inclusion bodies and positive CMV polymerase chain reaction, and hence they concluded that histological detection of owl’s eye inclusion bodies is an insensitive but highly specifi c method of detecting CMV organ involvement.[8] In our case of arteriovenous hemangioma, an incidental association or causation by CMV is not clear. Such an association has been reported in Juvenile hemangiomas.[3,4]

Conclusion

Hemangiomas of the parotid gland are frequent and the most Figure 1: Large thick and thin walled vessels fi lled with blood were common tumor in children. The association with CMV in non- seen to intermingle with salivary lobules (H and E, ×100) immunocompromised living infants is extremely rare in resected specimens of salivary gland hemangioma.

References

1. Weiss SR, Goldblum JR. Benign tumors and tumor like lesions of blood vessels. In: Enzinger and Weiss’s Soft Tumors. 5th ed. China: Mosby Elsevier; 2007. 2. Gnepp DR, El-Moft y SK. Viral sialadenitis. In: Damjanov I, Linder J, editors. Anderson’s Pathology. 10th ed. St Louis: Mosby; 1996. p. 1620. 3. Ayadi L, Khabir A, Boudawara T, Kharrat M, Makni S, Jlidi R. Cellular hemangioma of the parotid gland associated with an infection by cytomegalovirus. Rev Stomatol Chir Maxillofac 2003;104:290-3. 4. Horie Y, Kato M. Juvenile hemangioma (infantile hemangioendothelioma) of the parotid gland associated with cytomegalovirus infection. Pathol Int 1999;49:668-71. 5. Childers EL, Furlong MA, Fanburg-Smith JC. Hemangioma Figure 2: Numerous ductal epithelial cells showed large eosinophilic of the salivary gland: A study of ten cases of a rarely biopsied/ intranuclear inclusions with halos (owl’s eye inclusions) and also excised lesion. Ann Diagn Pathol 2002;6:339-44. basophilic cytoplasmic inclusions (H and E, ×400) 6. Barnes L, Eveson JW, Reichart P, Sidransky D, editors. World Health Organization Classifi cation of Tumours: Pathology and Genetics of Head and Neck Tumours. Lyon, France: IARC Press; 2005. Salivary gland hemangioma is relatively rare in surgical 7. Dudgeon JA. Cytomegalovirus infection. Arch Dis Child pathology fi les of Armed Forces Institute of Pathology, includes 1971;46:581-3. usual hemangioma subtypes, mainly in females, and juvenile 8. Mattes FM, McLaughlin JE, Emery VC, Clark DA, Griffi ths hemangioma, displaying distinctive histology predominately PD. Histopathological detection of owl’s eye inclusions is still in males. Coincidental viral cytopathic eff ect of CMV infection specifi c for cytomegalovirus in the era of human herpesviruses was identifi ed in only one but not in the case of arteriovenous 6 and 7. J Clin Pathol 2000;53:612-4. hemangioma.[5] Long before the advent of diagnostic virology, pathologists have been aware of the presence of large How to cite this article: Chatura KR, Iqbal MS. Parotid gland inclusion-bearing cells in the ducts of salivary glands and hemangioma with cytomegalovirus infection. J Adv Clin Res in other organs. Contemporary studies have revealed a Insights 2015;1:1-2.

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