Lacrimal Gland Enlargement in Familial Sarcoidosis

Nowinski, Thaddeus S.; Flanagan, Joseph C.; Ruchman, Mark C.

doi:10.1016/s0161-6420(83)80015-3

Cited by 13 publications

(5 citation statements)

References 19 publications

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“…Notably, most sarcoidosis cases in that study were female [15]. Others reported rare clinical presentations in familial sarcoidosis, including neuro-ophthalmic sarcoidosis occurring in a woman, whose paternal-uncle also had sarcoidosis [4], and sarcoidosis of the lacrimal gland in two sisters [7]. By means of phenotypic expression modeling in African American sibling pairs using logistic regression, increased risks for ocular (OR 3; CI 1.7-5.4) and liver involvement (OR 3.3; CI 1.5-7.4) were found in the affected sibs [38].…”

Section: Clinical Findings In Familial Sarcoidosismentioning

confidence: 67%

“…Since the beginning of the 20th century, there are mainly case reports and surveys on the familial aggregation of sarcoidosis [4][5][6][7][8][9][10][11][12][13][14][15][16][17][18][19]. In 2001, Rybicki and colleagues were the first to confirm the familial aggregation of sarcoidosis in the ACCESS study, including 355 European American and 291 African American case-control pairs [2].…”

Section: Prevalence Of Familial Sarcoidosis Among Sarcoidosis Patientsmentioning

confidence: 99%

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Clinical epidemiology of familial sarcoidosis: A systematic literature review

Terwiel

Moorsel

2019

Respiratory Medicine

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Introduction: Although the presence of familial sarcoidosis has been confirmed, clinical and epidemiological data on its characteristics are scattered and sometimes paradoxical. The objective of this review is to assess what is known on the clinical epidemiology of familial sarcoidosis, by combining data from early case reports with recent population based data; aiming to support in clinical decision making and providing information to patients. Method: A systematic review of the literature in PubMed was done and 27 studies with clinical or epidemiological data on familial sarcoidosis, published between 1947 and 2017, were included. Results: The pooled prevalence proportion of familial sarcoidosis, based on twelve study populations, was 9.5% (CI 4.6-16.1), highest in French, African American, Dutch and Irish patients. A heritability of 60-70% was estimated in diverse studies. Relative types and relationships most often reported in familial sarcoidosis were siblings and mother-child relationships. Familial risk is heterogeneous. In African Americans specific environmental factors have been associated with familial sarcoidosis (OR between 1.5 and 3.2). European American and African American subjects had different relative risks for first degree familial relationships (OR of 16.6 vs 3.1) and relative risk differed between relative types. Clinical findings in familial sarcoidosis are still obscure. Conclusions: Prevalence of familial sarcoidosis is high in specific study populations from countries worldwide. The estimated heritability of 60-70%, suggests a shared determinant, and the heterogeneous familial risk, associated with both genetic and environmental factors. Familial relative risks and clinical phenotypes may differ between ethnic groups and relative types, but require further study.

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Section: Clinical Findings In Familial Sarcoidosismentioning

confidence: 67%

Section: Prevalence Of Familial Sarcoidosis Among Sarcoidosis Patientsmentioning

confidence: 99%

Clinical epidemiology of familial sarcoidosis: A systematic literature review

Terwiel

Moorsel

2019

Respiratory Medicine

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“…This is an extremely rare entity, and only one sarcoidosis case with simultaneous anterior uveitis and dacryoadenitis has been reported so far 2. Sarcoidosis was excluded in our case on the results of the examinations, although biopsy was not performed.…”

Section: Discussionmentioning

confidence: 86%

Severe anterior uveitis associated with idiopathic dacryoadenitis in diabetes mellitus patient

Takahashi

Kakizaki²,

Ichinose³

et al. 2011

OPTH

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A 38-year-old woman with diabetes mellitus complained of acute visual loss in the left eye (20/200) and swollen left upper eyelid. Slit lamp examination of the left eye revealed ciliary injection, posterior synechia iritis, numerous inflammatory cells, and fibrin exudates in the anterior chamber. T1-weighted enhanced magnetic resonance imaging demonstrated left lacrimal gland enhancement with inflammatory spread to the left anterior ocular segment. Blood examination showed increased blood sugar but the other components were within normal limits. The patient was treated with steroid pulse therapy (methylprednisolone 1 g/day × 3 days) under a blood sugar control regimen in consultation with an endocrinologist, after which additional peribulbar injection of triamcinolone acetonide (40 mg) was performed. Resolution of the anterior uveitis and the dacryoadenitis was obtained after 2 months and there was no recurrence 1 year after the therapy. This is a rare case of severe anterior uveitis caused by idiopathic dacryoadenitis in a patient with diabetes mellitus.

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“…Decreased size of the supraclavicular lymph node after anti-tuberculosis therapy also supported reactivation of tuberculosis. Several reports are available about lacrimal gland granuloma in patients with sarcoidosis [9] or Crohn's disease [10]. Tuberculous dacryoadenitis is also known as a disease with lacrimal gland granuloma, though it is a very rare condition [11].…”

Section: Discussionmentioning

confidence: 99%

A case of immunoglobulin G4-related chronic sclerosing sialadenitis and dacryoadenitis associated with tuberculosis

et al. 2008

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We describe a 64-year-old woman with chronic sclerosing sialadenitis and dacryoadenitis, which developed during treatment for cervical lymph node tuberculosis. Anti-tuberculosis treatment did not improve the swelling in the lacrimal and submandibular glands, and a biopsy specimen of the lacrimal gland showed inflammation, with abundant lymphoid follicles with fibrosis and granuloma without caseous necrosis. Immunohistological examination of a repeat biopsy specimen showed abundant immunoglobulin (Ig) G4-positive plasma cell infiltration. Corticosteroid therapy improved the salivary gland swelling without reactivation of the tuberculosis. This case suggests that an abnormal immunological reaction to tuberculosis may be one of the etiological candidates for IgG4-related disease.

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Lacrimal Gland Enlargement in Familial Sarcoidosis

Cited by 13 publications

References 19 publications

Clinical epidemiology of familial sarcoidosis: A systematic literature review

Clinical epidemiology of familial sarcoidosis: A systematic literature review

Severe anterior uveitis associated with idiopathic dacryoadenitis in diabetes mellitus patient

A case of immunoglobulin G4-related chronic sclerosing sialadenitis and dacryoadenitis associated with tuberculosis

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