Involvement of oral tissues by AL amyloidosis: a literature review and report of eight new cases

Matsuo, Flávia Sayuri; Paulo, Luiz Fernando Barbosa de; Servato, João Paulo Silva; Faria, Paulo Rogério de; Cardoso, Sérgio Vitorino; Loyola, Adriano Mota

doi:10.1007/s00784-015-1649-3

Cited by 27 publications

(38 citation statements)

References 26 publications

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“…We observed that women were more commonly affected than men and that the tongue was the most common location of amyloidosis, unlike the findings of Stoopler et al, who reported that intraoral amyloid deposits were most commonly found in the buccal mucosa [3]. However, our findings are consistent with those of Matsuo et al, who found that oral amyloidosis occurred most commonly in the tongue and among women in their study [16]. The immunophenotyping results of two patients in the present study showed an association with multiple myeloma and arthritis-related senile amyloidosis, whereas those of other patients showed localized amyloidosis of the oral cavity, with no systemic correlation.…”

Section: Discussionsupporting

confidence: 87%

Immunophenotyping Oral Amyloidosis for the Precise Identification of the Biochemical Forms: A Retrospective Study

Binmadi¹,

Intapa²,

Chaisuparat³

et al. 2018

TODENTJ

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Background: Amyloidosis refers to a group of systemic and localized disorders associated with the accumulation of misfolded protein aggregates called amyloids in different parts of the body. Owing to the existence of multiple forms of amyloids with similar tertiary structures, precise identification of their biochemical form is critical for correct therapy. Objective: This retrospective study aimed to determine whether typing of oral amyloid deposits can help diagnose a serious systemic condition in the early phase of the disease Methods: All histopathologically confirmed cases of amyloidosis managed over a 14-year period (January 1, 1997 to December 31, 2011) were retrieved for analysis. Two board-certified oral and maxillofacial pathologists reviewed the histopathological findings of amyloidosis on the basis of its classic Congo red staining characteristics. This was followed by immunohistochemical analysis of biopsy samples using a panel of antibodies specific for different forms of amyloidosis. Results: The most common location of amyloidosis was the tongue, and women were more commonly affected than men. The patient age ranged from 11 to 83 years (average 59.3 years). In patient 9, light-chain and pre-albumin (transthyretin) antibodies were related to arthritis and senile amyloidosis, respectively. The biopsy sample of patient 10, who was reported to have multiple myeloma, was positive for light chains and β2 microglobulin. All other samples exhibited localized (solitary) amyloidosis. Conclusion: Histological analysis coupled with immunostaining with a panel of specific antibodies might assist in identifying early systemic amyloidosis in patients with localized oral forms of the disease.

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Section: Discussionsupporting

confidence: 87%

Immunophenotyping Oral Amyloidosis for the Precise Identification of the Biochemical Forms: A Retrospective Study

Binmadi¹,

Intapa²,

Chaisuparat³

et al. 2018

TODENTJ

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“…Diagnosis of amyloidosis is usually made associating the clinical features with histopathological exam, which reveal the presence of acellular eosinophilic deposits. These hyaline deposits typically show a perivascular distribution and they are positive for the Congo red staining and apple‐green birefringent under polarized light …”

Section: Introductionmentioning

confidence: 99%

“…These hyaline deposits typically show a perivascular distribution and they are positive for the Congo red staining and apple-green birefringent under polarized light. 1,[5][6][7] The aim of this report is to describe a clinical case of an elderly patient with dysphagia related to macroglossia and caused by primary amyloidosis associated with multiple myeloma. In this case, the organs predominantly involved were tongue, leading to macroglossia causing difficulty in swallowing.…”

Section: Introductionmentioning

confidence: 99%

Dysphagia due to macroglossia in a patient with amyloidosis associated with multiple myeloma: A case report

Costa

Ribeiro

Ferreira

et al. 2018

Special Care in Dentistry

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Amyloidosis is a group of heterogeneous conditions characterized by the deposition of extracellular proteinaceous substances called amyloids. The diagnosis is usually confirmed after a particular body organ has been affected. However, oral manifestations have been identified in 39% of affected patients. Systemic amyloidosis may be primary or in association with multiple myeloma and typically affecting elder adults. The diagnosis of amyloidosis is usually associated with clinical features with histopathological findings, which reveal the presence of acellular eosinophilic deposits. The aim of this report is to describe a clinical case of an elderly patient with dysphagia related to macroglossia and enlargement caused by primary amyloidosis associated with multiple myeloma.

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“…Clinically, amyloidosis can be localized or systemic [3]. Systemic amyloidosis can further be divided into: AL amyloidosis (associated with lymphoid or plasma cell neoplasms), AA amyloidosis (associated with chronic inflammatory diseases) and hereditary amyloidosis [3,4].…”

Section: Introductionmentioning

confidence: 99%

Intraoral manifestation of systemic AL amyloidosis with unique microscopic presentation of intracellular amyloid deposition in striated muscles

Oruba¹,

Kaczmarzyk²,

Urbańczyk³

et al. 2018

pjp

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We report the history of a 59-year old patient with systemic AL amyloidosis of intraoral manifestation. The patient first presented with complaints about dysphagia and remarkable enlargement of the tongue with highly reduced mobility, as well as bilateral submucosal thickenings on the cheeks. Histopathological examination of the incisional biopsy of the buccal mucosa and underlying tissues revealed AL amyloidosis. The microscopic presentation was, however, unique, as the amyloid deposits were present intracellularly in the striated muscles. The subsequent bone marrow biopsy confirmed the diagnosis of primary amyloidosis/multiple myeloma-associated amyloidosis.

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Involvement of oral tissues by AL amyloidosis: a literature review and report of eight new cases

Abstract: This article highlights the clinicopathological data of patients with amyloidosis affecting oral tissues and compare these new findings with other worldwide descriptions. Because of its rarity, such data are often unfamiliar to most clinicians and pathologists.

Cited by 27 publications

References 26 publications

Immunophenotyping Oral Amyloidosis for the Precise Identification of the Biochemical Forms: A Retrospective Study

Immunophenotyping Oral Amyloidosis for the Precise Identification of the Biochemical Forms: A Retrospective Study

Dysphagia due to macroglossia in a patient with amyloidosis associated with multiple myeloma: A case report

Intraoral manifestation of systemic AL amyloidosis with unique microscopic presentation of intracellular amyloid deposition in striated muscles

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