- Email: [email protected]
Histopathologic findings in a case of recurrent parotitis in adulthood
1332
RECURRENT
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-
1333
WANG ET AL
FIGURE I. Sialogram of right parotid gland showing punctate, globular sialectasis.
tures of pyogenic parotitis of childhood and adulthood were the same as those of Mikulicz-Sjiigren’s disease. All represent a spectrum of conditions in the family of connective tissue diseases7,’ Konno studied 13 cases of RPC with light and electron microscopy and found mild glandular destruction with proliferation of peripheral ductal epithelium and dilation of the peripheral portion of the ducts. * Our previous long-term followup study of RPA showed that the frequency of the swellings of the parotid gland decrease with age and finally disappear and the pun&ate sialectasis on the sialograms also is reduced or disappears completely. None of the patients developed SS.3
FIGURE 3. Electron micrograph of parotid gland showing numerous vacuoles of varying size and disappearance of most organelles within the cytoplasm (uranyl acetate and lead citrate stain, original magnification X7,000).
In our case, bilateral enlargement of parotid glands was found but there were no abnormalities in the laboratory tests, including the Schirmer test, total saliva flow rate, and serologic examinations. Histologically, there was marked vacuolar degeneration, which is different from the findings in SS. It is assumed that the degenerative changes associated with RPA result from longstanding inflammation of the parotid gland, and that this may be one of the factors resulting in recurrent parotitis continuing from childhood into adulthood. References
FIGURE 2. Histologic view of parotid gland showing enlargement and vacuolar degeneration of acinar ceIIs, dilation of intralobular ducts, and minimal inIiitration of inflammatory cells, (hematoxylineosin stain, original magnification X50).
1. Konno A, Ito E: A study on the pathogeneses of recurrent parotitis in childhood. Ann Otol Rhino1 Laryngol 88: 1, 1979 (suppl) 2. Galili D, Marmary Y: Juvenile recurrent parotitis: Clinicoradiologic follow-up study and the beneficial effect of sialography. Oral Surg Oral Med Oral Pathol61:550. 1986 3. Zou ZJ, Wang SL, Zhu JR, et al: Recurrent parotitis in childhood. A report of 102 cases. Chin Med J [En@] 103:576, 1990 4. Mandel L: Inflammatory diseases, in Rankow RM, Polayes IM (eds): Diseases of Salivary Glands (ed 1). Philadelphia, PA, Saunders, 1976, p 202 5. Zou ZJ, Wang SL, Wu QG, et al: Chronic obstructive parotitis: A report of 92 cases. Oral Surg Oral Med Oral Path01 73:434, 1992 6. Wang SL, Zou ZJ, Yu SF, et al: Does recurrent swellings of the parotids in adults lead to Sj&ren’s syndrome? Int J Oral Maxillofac Surg (Submitted) 7. Blatt IM: On sialectasis and benign lymphosialadenopathy. Laryngoscope 74: 1684, 1964 8. Blatt IM: Chronic and recurrent inflammations about the salivary glands with special reference to children: A report of’25 cases. Laryngoscope 76~917, 1966 9. Work WP, Balsakis IG: Classification of salivary gland diseases, Otolaryngol Clin North Am 10:287, 1977
RECURRENT
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-
1333
WANG ET AL
FIGURE I. Sialogram of right parotid gland showing punctate, globular sialectasis.
tures of pyogenic parotitis of childhood and adulthood were the same as those of Mikulicz-Sjiigren’s disease. All represent a spectrum of conditions in the family of connective tissue diseases7,’ Konno studied 13 cases of RPC with light and electron microscopy and found mild glandular destruction with proliferation of peripheral ductal epithelium and dilation of the peripheral portion of the ducts. * Our previous long-term followup study of RPA showed that the frequency of the swellings of the parotid gland decrease with age and finally disappear and the pun&ate sialectasis on the sialograms also is reduced or disappears completely. None of the patients developed SS.3
FIGURE 3. Electron micrograph of parotid gland showing numerous vacuoles of varying size and disappearance of most organelles within the cytoplasm (uranyl acetate and lead citrate stain, original magnification X7,000).
In our case, bilateral enlargement of parotid glands was found but there were no abnormalities in the laboratory tests, including the Schirmer test, total saliva flow rate, and serologic examinations. Histologically, there was marked vacuolar degeneration, which is different from the findings in SS. It is assumed that the degenerative changes associated with RPA result from longstanding inflammation of the parotid gland, and that this may be one of the factors resulting in recurrent parotitis continuing from childhood into adulthood. References
FIGURE 2. Histologic view of parotid gland showing enlargement and vacuolar degeneration of acinar ceIIs, dilation of intralobular ducts, and minimal inIiitration of inflammatory cells, (hematoxylineosin stain, original magnification X50).
1. Konno A, Ito E: A study on the pathogeneses of recurrent parotitis in childhood. Ann Otol Rhino1 Laryngol 88: 1, 1979 (suppl) 2. Galili D, Marmary Y: Juvenile recurrent parotitis: Clinicoradiologic follow-up study and the beneficial effect of sialography. Oral Surg Oral Med Oral Pathol61:550. 1986 3. Zou ZJ, Wang SL, Zhu JR, et al: Recurrent parotitis in childhood. A report of 102 cases. Chin Med J [En@] 103:576, 1990 4. Mandel L: Inflammatory diseases, in Rankow RM, Polayes IM (eds): Diseases of Salivary Glands (ed 1). Philadelphia, PA, Saunders, 1976, p 202 5. Zou ZJ, Wang SL, Wu QG, et al: Chronic obstructive parotitis: A report of 92 cases. Oral Surg Oral Med Oral Path01 73:434, 1992 6. Wang SL, Zou ZJ, Yu SF, et al: Does recurrent swellings of the parotids in adults lead to Sj&ren’s syndrome? Int J Oral Maxillofac Surg (Submitted) 7. Blatt IM: On sialectasis and benign lymphosialadenopathy. Laryngoscope 74: 1684, 1964 8. Blatt IM: Chronic and recurrent inflammations about the salivary glands with special reference to children: A report of’25 cases. Laryngoscope 76~917, 1966 9. Work WP, Balsakis IG: Classification of salivary gland diseases, Otolaryngol Clin North Am 10:287, 1977