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Autoimmune Exocrinopathy Presenting as Recurrent Parotitis of Childhood

Autoimmune Exocrinopathy Presenting as Recurrent Parotitis of Childhood Abstract • Objective. —To describe a case of autoimmune exocrinopathy in a child at the age of 3 months who presented with the origianl diagnosis of recurrent parotitis. Design. —This a case report of a 9-year-old girl with recurrent parotitis who was later found to have Sjögren's syndrome. The literature relating to primary Sjögren's syndrome in the pediatric population is reviewed. Setting. —The patient was seen in the rheumatology and otolaryngology clinics at a university medical center. Interventions. —This study did not address therapy. Main Outcome Measure. —Diagnosis of Sjögren's syndrome. Results. —The patient had xerostomia, an abnormal result of a salivary gland biopsy, SSA and SSB antibodies, and the histocompatibility antigens HLA-B8 and HLA-DR3 that are associated with Sjögren's syndrome. Conclusions. —Primary Sjögren's syndrome should be considered in cases of recurrent parotitis of childhood. More studies are needed to assess the natural history of autoimmune exocrinopathy in children.(Arch Otolaryngol Head Neck Surg. 1993;119:347-349) References 1. Geterud A, Lindvall A, Nylen O. Follow-up study of recurrent parotitis in childhood . Ann Otol Rhinol Laryngol . 1988;97:341-346. 2. Konno A, Ito E. A study on the pathogenesis of recurrent parotitis in childhood . Ann Otol Rhino Laryngol . 1979;88( (suppl 63) ):1-20. 3. Wilson RW, Provost TT, Bias WB, et al. Sjögren's syndrome: influence of multiple HLA-D region alloantigens on clinical and serologic expression . Arthritis Rheum . 1984;27:1245-1253.Crossref 4. Kraus A, Alarcon-Segovia D. Primary juvenile Sjögren's syndrome . J Rheumatol . 1988;15:803-806. 5. Chudwin SS, Daniels TE, Wara DW, et al. Spectrum of Sjögren's syndrome in children . J Pediatr . 1981;98:213-217.Crossref 6. Strand V, Talal N. Advances in the diagnosis and concept of Sjögren's syndrome (autoimmune exocrinopathy) . Bull Rheum Dis . 1980;30:1046-1052. 7. Vermylen C, Meurant A, Noel H, Claus D, Cornu G. Sjögren's syndrome in a child . Eur J Pediatr . 1985;144:266-269.Crossref 8. Dumas R, Guerand L, Jean R. Parotidite a rechute: forme de debut d'un syndrome de Gougerot Sjögren . Arch Fr Pediatr . 1975;32:461-470. 9. Franklin DJ, Smith RJH, Person DA, Pflugfelder SX. Sjögren's syndrome in children . Otolarygol Head Neck Surg . 1986;94:230-235. 10. Bartsocas S, Michail J. Sur un case rare de syndrome de Gougerot Houwers-Sjögren ou 'xerodermosteose' avec development precoce des organes genitaux et du corps, chez un infant . Arch Fr Pediatr . 1952;9:266-269. 11. Eliacher E, Perelman R, Hambourg M, Levavasseur C, Marie J. Syndrome de type 'Gourgerot-Sjögren' chez un infant . Ann Pediatr (Paris) . 1986;13:184-186. 12. Ipp M, Howard NJ, Tervo RC, Gelfand EW. Sicca syndrome and total lipodystrophy . Ann Intern Med . 1976;85:443-446.Crossref 13. Ludwiczak H, Tomaszewska E. Zespol Sjogrena n 13-letniej dziewczynki . Pediatr Pol .1970;45:585-587. 14. Siamopoulou-Mavridou A, Drosos AA, Andonopoulos AP. Sjögren's syndrome in childhood: report of two cases . Eur J Pediatr . 1989;148:523-524.Crossref 15. Basini JC, Acito P, Agostini U. Un caso di sindrome di Sjögren in eta pediatrica . Clin Pediatr . 1965;45:47-58. 16. Mizuno Y, Hara T, Hatae K. Recurrent parotid gland enlargement as an initial manifestation of Sjögren's syndrome in children . Eur J Pediatr . 1989;148:414-416.Crossref 17. Pittsley RA, Daniels TE, Fye KH, Michalski JP, Talal N. Autoimmune exocrinopathy (AE): a new definition of Sjögren's syndrome (SS) confirmed by labial salivary gland (LSG) biopsy . Arthritis Rheum . 1978;21:584. Abstract. http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Otolaryngology - Head & Neck Surgery American Medical Association

Autoimmune Exocrinopathy Presenting as Recurrent Parotitis of Childhood

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Publisher
American Medical Association
Copyright
Copyright © 1993 American Medical Association. All Rights Reserved.
ISSN
0886-4470
eISSN
1538-361X
DOI
10.1001/archotol.1993.01880150103015
Publisher site
See Article on Publisher Site

Abstract

Abstract • Objective. —To describe a case of autoimmune exocrinopathy in a child at the age of 3 months who presented with the origianl diagnosis of recurrent parotitis. Design. —This a case report of a 9-year-old girl with recurrent parotitis who was later found to have Sjögren's syndrome. The literature relating to primary Sjögren's syndrome in the pediatric population is reviewed. Setting. —The patient was seen in the rheumatology and otolaryngology clinics at a university medical center. Interventions. —This study did not address therapy. Main Outcome Measure. —Diagnosis of Sjögren's syndrome. Results. —The patient had xerostomia, an abnormal result of a salivary gland biopsy, SSA and SSB antibodies, and the histocompatibility antigens HLA-B8 and HLA-DR3 that are associated with Sjögren's syndrome. Conclusions. —Primary Sjögren's syndrome should be considered in cases of recurrent parotitis of childhood. More studies are needed to assess the natural history of autoimmune exocrinopathy in children.(Arch Otolaryngol Head Neck Surg. 1993;119:347-349) References 1. Geterud A, Lindvall A, Nylen O. Follow-up study of recurrent parotitis in childhood . Ann Otol Rhinol Laryngol . 1988;97:341-346. 2. Konno A, Ito E. A study on the pathogenesis of recurrent parotitis in childhood . Ann Otol Rhino Laryngol . 1979;88( (suppl 63) ):1-20. 3. Wilson RW, Provost TT, Bias WB, et al. Sjögren's syndrome: influence of multiple HLA-D region alloantigens on clinical and serologic expression . Arthritis Rheum . 1984;27:1245-1253.Crossref 4. Kraus A, Alarcon-Segovia D. Primary juvenile Sjögren's syndrome . J Rheumatol . 1988;15:803-806. 5. Chudwin SS, Daniels TE, Wara DW, et al. Spectrum of Sjögren's syndrome in children . J Pediatr . 1981;98:213-217.Crossref 6. Strand V, Talal N. Advances in the diagnosis and concept of Sjögren's syndrome (autoimmune exocrinopathy) . Bull Rheum Dis . 1980;30:1046-1052. 7. Vermylen C, Meurant A, Noel H, Claus D, Cornu G. Sjögren's syndrome in a child . Eur J Pediatr . 1985;144:266-269.Crossref 8. Dumas R, Guerand L, Jean R. Parotidite a rechute: forme de debut d'un syndrome de Gougerot Sjögren . Arch Fr Pediatr . 1975;32:461-470. 9. Franklin DJ, Smith RJH, Person DA, Pflugfelder SX. Sjögren's syndrome in children . Otolarygol Head Neck Surg . 1986;94:230-235. 10. Bartsocas S, Michail J. Sur un case rare de syndrome de Gougerot Houwers-Sjögren ou 'xerodermosteose' avec development precoce des organes genitaux et du corps, chez un infant . Arch Fr Pediatr . 1952;9:266-269. 11. Eliacher E, Perelman R, Hambourg M, Levavasseur C, Marie J. Syndrome de type 'Gourgerot-Sjögren' chez un infant . Ann Pediatr (Paris) . 1986;13:184-186. 12. Ipp M, Howard NJ, Tervo RC, Gelfand EW. Sicca syndrome and total lipodystrophy . Ann Intern Med . 1976;85:443-446.Crossref 13. Ludwiczak H, Tomaszewska E. Zespol Sjogrena n 13-letniej dziewczynki . Pediatr Pol .1970;45:585-587. 14. Siamopoulou-Mavridou A, Drosos AA, Andonopoulos AP. Sjögren's syndrome in childhood: report of two cases . Eur J Pediatr . 1989;148:523-524.Crossref 15. Basini JC, Acito P, Agostini U. Un caso di sindrome di Sjögren in eta pediatrica . Clin Pediatr . 1965;45:47-58. 16. Mizuno Y, Hara T, Hatae K. Recurrent parotid gland enlargement as an initial manifestation of Sjögren's syndrome in children . Eur J Pediatr . 1989;148:414-416.Crossref 17. Pittsley RA, Daniels TE, Fye KH, Michalski JP, Talal N. Autoimmune exocrinopathy (AE): a new definition of Sjögren's syndrome (SS) confirmed by labial salivary gland (LSG) biopsy . Arthritis Rheum . 1978;21:584. Abstract.

Journal

Archives of Otolaryngology - Head & Neck SurgeryAmerican Medical Association

Published: Mar 1, 1993

References