Primary Localized Nodular Tongue Amyloidosis Associated with Sjögren’s Syndrome

Ohashi, Kenichi; Yamada, Masahito; Hasegawa, Makoto; Maeda, Soukei; Komatsuzaki, Atsushi

doi:10.1159/000276907

Cited by 27 publications

(13 citation statements)

References 6 publications

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“…Systemic AL amyloidosis itself can affect the lacrimal and salivary glands and is therefore one of the causes of sicca syndrome (7). SS has also been associated with the presence of localized amyloid in sites such as the lungs (8), the breast (9), the tongue (10), and the skin (11). Three types of localized cutaneous amyloidosis can be recognized: macular, lichen, and nodular types, of which the nodular type is the rarest (12).…”

mentioning

confidence: 99%

Sjögren's syndrome and localized nodular cutaneous amyloidosis: Coincidence or a distinct clinical entity?

Meijer¹,

Schönland²,

Palladini³

et al. 2008

Arthritis & Rheumatism

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Objective. To report 8 patients with Sjögren's syndrome (SS) and localized nodular cutaneous amyloidosis and to examine serologic and immunohistologic findings that may link the 2 diseases.Methods. The databases for 3 amyloidosis centers were searched for patients with localized nodular cutaneous amyloidosis and SS. Eight patients with this combination were identified, and clinical, serologic, and histologic parameters were retrospectively evaluated.Results. Among the 8 patients with a clinical diagnosis of SS, 6 fulfilled the American-European Consensus Group criteria for SS. All of the patients were women in whom SS had been diagnosed at a median age of 47 years (range 30-61 years) and amyloid had been diagnosed at a median age of 60 years (range 42-79 years). The presence of the immunoglobulin light chain type of amyloid (AL amyloid) was confirmed in 4 patients. In 3 of these 4 patients as well as 2 other patients, a light chain-restricted plasma cell population was observed near the amyloid deposits. Progression to systemic amyloidosis was not observed in any patient during a median followup of 3.5 years.

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mentioning

confidence: 99%

Sjögren's syndrome and localized nodular cutaneous amyloidosis: Coincidence or a distinct clinical entity?

Meijer¹,

Schönland²,

Palladini³

et al. 2008

Arthritis & Rheumatism

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“…Only eight cases have been previously reported in the English literature. In all these published cases, amyloidosis was localised or limited to dermis,7-9 lung,10 11 or tongue 12. In some cases, plasma cells were found surrounding the amyloid deposits.…”

Section: Discussionmentioning

confidence: 95%

Concomitant diagnosis of primary Sjogren's syndrome and systemic AL amyloidosis

Delévaux

2001

Annals of the Rheumatic Diseases

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A 48 year old woman was referred to hospital for buccal discomfort. Physical examination showed a macroglossia and features of xerostomia. She was diagnosed as having primary Sjögren's syndrome according to the criteria proposed by the European Community study group in 1993. Furthermore, a lower lip salivary gland biopsy showed amyloid deposits that were also seen in the stomach and in the bone marrow. Echocardiography was consistent with cardiac amyloidosis. Serum immunofixation identified a monoclonal IgG . As far as is known, this is the first report of systemic primary amyloidosis associated with Sjögren's syndrome. The relation between these two disorders is discussed.

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“…While it is well accepted that the localized form of lingual amyloidosis can be reasonably treated with surgery [2, 9, 12, 13], no established treatment exists for the management of macroglossia due to systemic amyloidosis. Cases reported in the literature are rare, and scientific evidence is lacking.…”

Section: Discussionmentioning

confidence: 99%

Macroglossia due to Systemic Amyloidosis: Is There a Role for Radiotherapy

Thibault

Vallières²

2011

Case Rep Oncol

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Background: Macroglossia due to amyloid depositions can cause cosmetic problems and functional disability, and can lead to life-threatening airway obstruction. Management of macroglossia in systemic amyloidosis is controversial, and the role of surgery is unclear. Case Description: We present a case of a 66-year-old woman affected by macroglossia due to light chain amyloidosis who presented with eating and breathing difficulties. Because of prior successful results of radiotherapy for localized amyloid disease, our patient was treated with external beam radiation therapy (20 Gy in 10 fractions). The treatment was well tolerated by the patient. However, her systemic amyloidosis progressed, with a subclinical increase in tongue width. Conclusions: This is the first reported use of radiotherapy for amyloidosis of the tongue. There was no evidence of benefit using a total dose of 20 Gy. This therapeutic modality is not recommended for the routine management of macroglossia.

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Primary Localized Nodular Tongue Amyloidosis Associated with Sjögren’s Syndrome

Cited by 27 publications

References 6 publications

Sjögren's syndrome and localized nodular cutaneous amyloidosis: Coincidence or a distinct clinical entity?

Sjögren's syndrome and localized nodular cutaneous amyloidosis: Coincidence or a distinct clinical entity?

Concomitant diagnosis of primary Sjogren's syndrome and systemic AL amyloidosis

Macroglossia due to Systemic Amyloidosis: Is There a Role for Radiotherapy

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