J Oral Maxillofac 50:1329-1332.

Surg 1992

Hemangiopericytoma of the Parotid Gland: Report of Case GUO LEE, DDS,* DANIEL YONG-KIE WONG, DDS, MSJ AND RICHARD CHE-SHOA CHANG, DDS, MS+

Hemangiopericytoma is a rare vascular tumor arising from the proliferation of pericytes that surround capillaries. This tumor can occur in almost every region of the body, but most often occurs in the soft tissues of the extremities.le3 The term hemangiopericytoma was first coined by Stout and Murray4 in 1942. It represents 1% of all vasoformative neoplasms, 15% to 25% of which occur in the head and neck region.5 Review of the literature by Pagliaro et al6 in 1988 revealed 18 cases located in the parotid gland. No additional cases have been reported since that time. This article presents a primary hemangiopericytoma in the parotid gland of a Chinese boy. Report of a Case In February 1990, a 1Cyear-old boy was first seen at Taipei Veterans General Hospital in Taiwan with a complaint of a bulging mass over the right infra-auricular region that he had noticed about 3 months earlier. The lesion was asymptomatic except for a progressive increase in size. His past medical history and systemic review did not reveal any abnormalities. Examination of the right infra-auricular region showed a movable, oval mass about 3 X 4 cm in diameter, firm in consistency, covered by nonadherent normal skin. There was no cervical lymph node enlargement. A panoramic radiograph of the region was taken and revealed no evidence of

With a diagnosis of parotid gland tumor, the patient was admitted to the hospital and surgical excision of the mass was performed on February 26 1990. During the procedure, an oval, well encapsulated mass was found just under the five branches of the facial nerve. It was embedded in the deep lobe of the parotid gland in the right retromandibular fossa and extended to the superficial lobe anteriorly. The mass measured about 3 X 4 cm and was characterized by a homogeneous tan-colored surface. It did not appear to infiltrate the attached parotid gland and the capsule appeared intact. It was removed completely with preservation of the facial nerve, and the wound was closed primarily. Subsequent microscopic examination showed the resected margins to be free of tumor. The pathology report described the gross specimen as a greyish white, firm mass measuring about 4 X 3 X 2 cm. The microscopic sections were composed of ovoid or spindleshaped tumor cells arranged in nest patterns and abundant vascular channels. Cellular pleomorphism was minimal and no mitotic figures were present (Fig 2). A fibrous pseudocap sule, with hemorrhage, was visualized and the parotid gland tissue outside the capsule was normal (Fig 3). A silver reticulin stain showed reticulin fibers that enmeshed the tumor cells outside the vascular spaces (Fig 4). A Masson’s trichome stain did not reveal a fibrous or muscular origin of the tumor (Fig 5). The UEAl stain failed to show endothelial cell prolifer-

bony destruction. A postcontrast computed tomographic (CT) scan showed an enhanced mass over the right parotid gland, mainly involving the superficial and deep lobes (Fig 1).

* Chief Resident and Clinical Instructor, Oral and Maxillofacial Surgery, Dental Department, Veterans General Hospital, and School of Dentistry, National Yang Ming Medical College, Taipei, Taiwan. t Associate Professor and Attending Physician, Oral and Maxillofacial Surgery, Dental Department, Veterans General Hospital; School of Dentistry, National Yang Ming Medical College, Taipei, Taiwan. $ Associate Professor and Chairman, Oral and Maxillofacial Surgery, Dental Department, Veterans General Hospital, and School of Dentistry, National Yang Ming Medical College, Taipei, Taiwan. Address correspondence and reprint requests to Dr Lee: Division of Oral and Maxillofacial Surgery, Dental Department, Veterans General Hospital, VACRS, Taipei, Taiwan 11217, Republic of China. 0 1992 American

Association

of Oral and Maxillofacial

Surgeons

FIGURE 1. Computed tomography scan with contrast showing extent of tumor in the right parotid gland.

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FIGURE 2. Photomicrograph of hemangiopericytoma composed of ovoid and spindle-shaped cells with illdefined cytoplasmic borders

arranged in nest patterns. No mitotic figures are identified (hema-

HEMANGIOPERICYTOMA

FIGURE 3.

OF THE PAROTID GLAND

Photomicrograph of the pseudoencapsulated heman-

giopericytoma. Nonnal parotid gland tissue can be noted outside the tumor capsule (hematoxylin-eosin stain, original magnification X40).

toxylin-eosin stain, original magnification Xl 00).

FIGURE 4. Silver reticulin stain showing vascular spaces with sharply demarcated reticulin sheaths. Each pericyte is enclosed by reticulin fibers (original magnification X 100).

FIGURE 5. Masson’s trichome stain showing that the tumor was not of fibrous or myogenous origin (original magnification x400).

ation in the tumor (Fig 6). The final diagnosis was benign hemangiopericytoma of the parotid gland. The postoperative course was uneventful and the patient was discharged on the fifth postoperative day. There was no evidence of local recurrence or distant metastases at a a-year follow-up examination.

Discussion Hemangiopericytoma is an uncommon mesenchyma1 tumor of blood vessel origin that can be found in

FIGURE 6. Factor VIII (UEAl) stain showing uptake by the endothelial cells, but the tumor cells are not stained (original magnification X400).

any part of the body. Although Rouget had discovered the “cellules adventices” in 1873, it was not until 1923 that Zimmermann comprehensively described the pericytes as modified muscle cells with branching cytoplasmic processes encircling small blood vessels.’ The first description of tumors of the pericytes was made in 1942, when a review of 691 blood vessel tumors

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revealed that 1.3% could be morphologically distinguished as arising from pericytes.4 The cause of hemangiopericytoma is not known, although trauma, steroid therapy, and hormonal imbalance have been implicated in the past8 The tumor may occur in any age group, with a peak incidence in the middle decades.‘-” No appreciable difference in occurrence between the sexes has been found. In elderly patients, the tumor more frequently pursues a malignant course,” whereas the congenital and infantile forms are almost invariably superficial in site and benign in behavior. A slowly expanding, asymptomatic mass is the general mode of presentation. Pain can result when the tumor is confined within an unyielding space, such as a sinus, or if there is visceral or neural compression by the tumor mass.” Other less common signs include telangiectasia, pulsation, or audible bruit,‘3 and nonmetastatic systemic effects include hypoglycemia, hypotension, hypokalemia secondary to hypereninemia, musculinization, and cardiac failure resulting from arteriovenous shunting.14 Hemangiopericytoma is a tumor that does not have sufficient arresting gross features to enable it to be recognized clinically, and the plain radiograph is nonspecific. While angiography may be characteristic, it is not of unequivocal diagnostic value in all casesI The CT scan and magnetic resonance imaging may be useful, but there are too few reports to evaluate their application. Thus, histologic examination remains the only definitive means to confirm the presence of this tumor. Histologically, a proliferation of pericytes is found between normal capillaries, occurring as sheets of ovoid or spindle-shaped cells with indistinct cytoplasm. A capsule is usually present, although tumor cells may be seen outside the capsule. A staghorn appearance of the vessels can often be found due to compression by proliferating tumor cells. A silver reticulin stain is helpful in diagnosis by demonstrating both the extravascular location of the pericytes and the surrounding reticulin network.‘5-‘7 In this case, a Masson’s trichome stain was used, which did not suggest a fibrous or leiomyomatous origin of the tumor. A factor VIII antigen stain (UEAl) was used and hemangioendothelioma was excluded by negative reactivity. From a histologic viewpoint, these tumors are classified as benign, borderline, or malignant, according to the degree of cellularity, nuclear atypia, number of mitotic figures and evidence of necrotic changes.” However, histologically benign tumors have been reported to metastasize.16 Stout, in 1949, pointed out the difficulty in establishing any exact histologic criteria for malignancy and stressed that all hemangiopericytoma should be considered potentially malignant until proved otherwise.

Although slow growing and locally malignant, these tumors, in general, are known to metastasize by lymphatic and hematogenous routes to the lungs, liver, bone, and local lymph nodes.” Recurrence rates range from 20% to 57%, and may vary with the particular organ system involved. l9 The overall mortality from hemangiopericytoma may be as high as 50%.9 At present, 18 cases of hemangiopericytoma of the parotid gland have been reported.6 A sufficient follow-up is lacking in 8 of these cases. Eight patients died after intervals varying from a minimum of 16 months to a maximum of 11 years and 5 months. Many had local and distant recurrence, especially in the lungs and bone. Two were free of disease at 12 and 16 years, respectively. There have been no local recurrence or distant metastases in our case during a 2-year postoperative follow-up. Wide surgical excision is the treatment of choice. There is no place in the surgical management for enucleation alone, as nodules of tumor cells may be outside the capsule.16 The radioresistant nature of hemangiopericytoma has been proved. Radiation as a primary modality is not indicated, although incomplete excision or a large local recurrence may be treated by irradiation as a palliative measure in inoperable cases.” Chemotherapy has been suggested as adjuvant therapy,*” but is not recommended as part of the first-line treatment of hemangiopericytoma. *’ In view of the relatively common local recurrence and very late appearance of metastases, wide surgical excision and lifelong followup are essential. References I. Enzinger FM, Smith BH: Hemangiopericytoma: An analysis of 106 cases. Hum Pathol 7:61, 1976 2. Fischer JH: Hemangiopericytoma: A review of twenty cases. Can Med Assoc J 83:1136, 1960 3. McMaster MJ, Some EH, Ivins JC: Hemangiopericytoma: A clinicopathologic study and long-term follow-up of 60 patients. Cancer 36:2232, 1975 4. Stout AP, Murray MR: Hemangiopericytoma: Vascular tumor featuring Zimmermann’s pericytes. Ann Surg 116:26, 1942 5. Bat&is JG, Rice DH: The pathology of head and neck tumors: Vasoformative tumors, part 9B. Head Neck Surg 3:326, 198 I 6. Pagliaro G, Poli P, Ralza G, et al: Hemangiopericytoma of the submandibular gland (A case report). J Laryngol Otol 102: 97, 1988 7. Das AK, Gans BJ: Hemangiopericytoma of oral cavity: Review of literature and report of a case. J Oral Surg 23:456, 1965 8. Chawla OP, Oswal VH: Hemangiopericytoma of the nose and paranasal sinuses. J Laryngol Otol 101:729, 1987 9. O’Brain P, Brasfield RD: Hemangiopericytoma. Cancer 18:249,

1965 10. Gudrun R: Hemangiopericytoma in otolaryngology. J Laryngol oto193:477, 1979 1 I. Cole HN, Reagan JW, Lund HZ: Hemangiopericytoma. Arch Dermato172:328, 1955 12. Backwinkel KD, Diddams JA: Hemangiopericytoma: Report of a case and comprehensive review of the literature. Cancer 25: 896, 1970

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13. Enzinger FM, Weiss SW: Soft Tissue Tumors. St Louis, MO, Mosby, 1983, p 463 14. Gemer RE, Moore GE, Pickren JW: Hemangiopericytoma. Ann Surg 179:128, 1974 15. Stout AP: Hemangiopericytoma: A study of 25 new cases. Cancer 2:1027, 1949 16. Walike JW, Bailey BJ: Head and neck hemangiopericytoma. Arch Otolaryngol 93:345, 197 1 17. Suit HD, Russell WO, Martin RG: Management ofpatients with sarcoma of soft tissue of an extremity. Cancer 3 1: 1247, 1973

PAROTITIS IN ADULTHOOD

18. Croxatto JO, Font L: Hemangiopericytoma Pathol 13:210, 1982

of the orbit. Hum

19. Goldwasser MS, Daw JL: Hemangiopericytoma of the palate: Case report. J Oral Maxillofac Surg 48:2 11, 1990 20. Wong P, Yagoda A: Chemotherapy of malignant hemangiopericytoma. Cancer 4 1: 1256, 1978 21. Robb PJ, Singh S, Hartley RB, et al: Malignant hemangioperi-

cytoma of the parapharyngeal space. Head Neck Surg 9: 179, 1987

J Oral MaxillofacSurg 50:1332-1333,

1992

Histopathologic Findings in a Case of Recurrent Parotitis in Adulthood SONGLIN WANG,* DDS, PHD, ZHAOJU ZOU, DDS, FICDJ QIQUANG WU, DDS,* AND KAIHUA SUN, DDS§

Most cases with recurrent parotitis in childhood (RPC) can be cured before puberty.‘” A few of them, however, may continue to adulthood. l-4To distinguish this condition from other types of chronic parotitis in adults, such as chronic obstructive parotitiq5 we suggest that the diagnosis of recurrent parotitis in adults (RPA) be reserved for the cases derived from RPC.6 The pathophysiology and development of RPA is still not well known. To our knowledge, there are few reports of RPA in the literature, especially concerning its histopathology. This report describes a case of RPA studied with sialography and light and electron microscopy. Report of Case A 29-year-old man came for treatment with bilateral recurrent swellings of parotid glands occurring three to six times a year from the ages of 8 to 14 years old. These swellings occurred mainly after catching a cold. In recent years, bilateral enlargement of the parotid glands was noticed, especially of the right side.

Examination showed that both the right and left parotid glands were enlarged, but salivary flow was clear. The results of the Schirmer test were normal (wetting of 3 cm in the right, 1.5 cm in the left). The total saliva flow rate was 10 mL/6 min (less than 6 mL of saliva produced by chewing 5 g of medical paraffin for 6 minutes after fasting in the morning is considered abnormal). Laboratory tests, including blood counts, protein electrophoresis, and analysis for antinuclear antibody, were normal. Both parotid glands exhibited diffise, punctate, globular sialectasis on the sialograms (Fig I). Superficial parotidectomy of the right parotid gland was performed. Gross enlargement of the gland was noticed. The prominent changes seen with light microscopy were enlarged acini filled with vacuoles, dilation of the intralobular ducts and some interlobular ducts, and a slight infiltration of inflammatory cells (Fig 2). Electron microscopy showed irregular enlargement of the intercellular spaces in the acini and cytoplasm filled with vacuoles of various sizes. Some vacuoles were fused to other vacuoles. Some vacuoles were of moderate electron density and some of low density. Most of the organelles within the cytoplasm had disappeared (Fig 3). The epithelial cells of the intralobular ducts had became flattened, and there was a reduced number of microvilli facing the duct lumen.

Discussion Received from the School of Stomatology, Beijing Medical University, China. * Lecturer, Department of Oral Radiology. t Professor, Department of Oral and Maxillofacial Surgery and Radiology. $ Professor, Department of Oral Pathology. $ Professor, Division of Electron Microscopy, Department of Oral Pathology. Address correspondence and reprint requests to Dr Wang: Department of Oral Radiology, Beijing Hospital for Stomatology, Tian Tan Xi Li No. 4, Beijing 100050, China. 0 1992 American

Association

0278-2391/92/5012-5012-0017$3.00/O

of Oral and Maxillofacial

Surgeons

Recurrent parotitis in adults is a chronic inflammatory disease of the parotid gland. Some authors combine it with other types of diseases under the heading of nonobstrucive parotitis.7-9 Clinically, RPA is easily confused with Sjogren’s syndrome (SS) associated with retrograde infection, especially the subclinical type of SS. Patients with SS experience recurrent swelling of the parotid gland before the onset of dry mouth and dry eyes, and the sialograms also show punctate, globular sialectasis.6 Blatt reported that the histologic fea-

Hemangiopericytoma of the parotid gland: report of case.

J Oral Maxillofac 50:1329-1332. Surg 1992 Hemangiopericytoma of the Parotid Gland: Report of Case GUO LEE, DDS,* DANIEL YONG-KIE WONG, DDS, MSJ AND...
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