Literature DB >> 1915519

Chronic bilateral dacryo-adenitis in identical twins: a possible incomplete form of Sjögren syndrome.

C Besana1, C Salmaggi, C Pellegrino, L Pierro, S Vergani, A Faravelli, C Rugarli.   

Abstract

We report an unusual case of chronic bilateral dacryo-adenitis in 10-year-old identical twin sisters. Both girls presented with bilateral lacrimal gland enlargement and developed moderate xerophthalmia and keratitis. Both the lacrimal and minor salivary gland biopsies showed a non-granulomatous inflammatory infiltration of mono-nuclear cells. All granulomatous diseases and neoplasms could therefore be ruled out and only Sjögren syndrome and very few other forms of chronic dacryo-adenitis remained as possible diagnoses. Both patients and their parents were evaluated for auto-antibodies. Very low titres of smooth muscle antibodies were found in one, antinuclear antibodies in two and anti-dsDNA antibodies in all four members of the family. Even though the titres of antinuclear and anti-dsDNA antibodies increased in one of the sisters, both patients did not develop any sign or symptom of a systemic connective tissue disease. During the 6 years' follow up, both patients showed persistent tarsal gland enlargement but no other symptoms apart from a moderate xerophthalmia and occasional mild keratitis.

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Year:  1991        PMID: 1915519     DOI: 10.1007/bf02072627

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  24 in total

1.  ANTINUCLEAR AND PRECIPITATING AUTO-ANTIBODIES IN SJOEGREN'S SYNDROME.

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Journal:  Ann Rheum Dis       Date:  1965-01       Impact factor: 19.103

2.  SOME UNUSUAL CASES OF SJOGREN'S SYNDROME.

Authors:  H Coverdale
Journal:  Br J Ophthalmol       Date:  1948-09       Impact factor: 4.638

3.  Familial CRST syndrome with sicca complex.

Authors:  R A Frayha; K F Tabbara; R S Geha
Journal:  J Rheumatol       Date:  1977       Impact factor: 4.666

4.  [Familial scleroderma with Sjogren's syndrome and lymphocyte and chromosome abnormalities].

Authors:  J P Camus; I Emerit; P Reinert; P Guillien; J Crouzet; J Fourot
Journal:  Ann Med Interne (Paris)       Date:  1970-02

5.  Achalasia sicca--juvenile Sjögren's syndrome with achalasia and gastric hyposecretion.

Authors:  S Similä; J Kokkonen; M Kaski
Journal:  Eur J Pediatr       Date:  1978-10-12       Impact factor: 3.183

6.  Primary Sjogren's syndrome and autoimmune hemolytic anemia in sisters. A family study.

Authors:  E P Boling; J Wen; J D Reveille; W B Bias; T M Chused; F C Arnett
Journal:  Am J Med       Date:  1983-06       Impact factor: 4.965

7.  Preliminary diagnostic criteria for Sjögren's syndrome.

Authors:  F N Skopouli; A A Drosos; T Papaioannou; H M Moutsopoulos
Journal:  Scand J Rheumatol Suppl       Date:  1986

8.  HLA DR alloantigens in different subsets of patients with Sjögren's syndrome and in family members.

Authors:  D L Mann; H M Moutsopoulos
Journal:  Ann Rheum Dis       Date:  1983-10       Impact factor: 19.103

9.  Recurrent parotid gland enlargement as an initial manifestation of Sjögren syndrome in children.

Authors:  Y Mizuno; T Hara; K Hatae; T Hirano; K Ueda; N Nakamura; Y Mizoguchi
Journal:  Eur J Pediatr       Date:  1989-02       Impact factor: 3.183

10.  Sjögren's syndrome. Influence of multiple HLA-D region alloantigens on clinical and serologic expression.

Authors:  R W Wilson; T T Provost; W B Bias; E L Alexander; D W Edlow; M C Hochberg; M B Stevens; F C Arnett
Journal:  Arthritis Rheum       Date:  1984-11
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  5 in total

Review 1.  Genetics of Sjögren's syndrome in the genome-wide association era.

Authors:  John A Ice; He Li; Indra Adrianto; Paul Chee Lin; Jennifer A Kelly; Courtney G Montgomery; Christopher J Lessard; Kathy L Moser
Journal:  J Autoimmun       Date:  2012-01-29       Impact factor: 7.094

Review 2.  Genes and Sjögren's syndrome.

Authors:  Beth L Cobb; Christopher J Lessard; John B Harley; Kathy L Moser
Journal:  Rheum Dis Clin North Am       Date:  2008-11       Impact factor: 2.670

Review 3.  Primary Sjögren's syndrome in children: is a family approach indicated?

Authors:  Barbara S Longhi; Simone Appenzeller; Maraisa Centeville; Reinaldo J Gusmão; Roberto Marini
Journal:  Clinics (Sao Paulo)       Date:  2011       Impact factor: 2.365

Review 4.  Genetic aspects of Sjögren's syndrome.

Authors:  Anne I Bolstad; Roland Jonsson
Journal:  Arthritis Res       Date:  2002-09-24

5.  Familial Risk of Sjögren's Syndrome and Co-aggregation of Autoimmune Diseases in Affected Families: A Nationwide Population Study.

Authors:  Chang-Fu Kuo; Matthew J Grainge; Ana M Valdes; Lai-Chu See; Shue-Fen Luo; Kuang-Hui Yu; Weiya Zhang; Michael Doherty
Journal:  Arthritis Rheumatol       Date:  2015-07       Impact factor: 10.995

  5 in total

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