A novel case of TIF1 gamma autoantibody positive dermatomyositis associated with a non-functional pancreatic neuroendocrine tumor

Varedi, Danny; Frigerio, Alice; Scaife, Courtney L.; Hull, Christopher M.

doi:10.5070/d3253043339

Cited by 4 publications

(2 citation statements)

References 7 publications

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“…A retrospective analysis by Harada et al analyzed 14 patients with anti-TIF1𝛄 positivity out of a pool of 85 patients with DM over a prolonged 18-year course and identified dermatologic manifestations such as erythema, V neck sign, heliotrope rash, and nail fold telangiectasias more frequently present, whereas no patients developed features of ILD on HRCT ( 77 ). Intriguingly, patients with anti-TIF1𝛄 positivity have been found to have an increased incidence of developing malignant tumors ( 78 , 79 ). Patients with anti-TIF1𝛄 and pulmonary involvement should be followed closely for the development of malignancy.…”

Section: Dermatomyositismentioning

confidence: 99%

Myositis interstitial lung disease and autoantibodies

Chaudhry

Christopher‐Stine

2023

Front. Med.

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The aim of this review is to examine and evaluate published literature associated with idiopathic inflammatory myopathies (IIM) and interstitial lung disease (ILD) based on myositis specific autoantibodies (MSA) and the potential clinical significance of each autoantibody subtype for the practicing clinician. The review is a comprehensive search of literature published in PubMed from the year 2005 and onward coinciding with the surge in the discovery of new MSAs. Additionally, we comment on recommended multidisciplinary longitudinal care practices for patients with IIM-ILD with regard to imaging and other testing. Treatment is not covered in this review.

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Section: Dermatomyositismentioning

confidence: 99%

Myositis interstitial lung disease and autoantibodies

Chaudhry

Christopher‐Stine

2023

Front. Med.

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“…The onset of neurological manifestations usually precedes cancer diagnosis and may lead to earlier cancer detection and treatment [1,2]. Although most studies focus on small-cell lung cancer (SCLC)-associated paraneoplastic neurological syndromes [3,4], diverse neurological manifestations have been described accompanying other neuroendocrine neoplasms (NENs), including "high-risk" paraneoplastic neurological phenotypes (e.g., Lambert-Eaton myasthenic syndrome, limbic encephalitis or rapidly progressive cerebellar syndrome) [5][6][7][8][9][10][11] and other classical presentations of neurological autoimmunity (e.g., brainstem encephalitis, myelopathy) [12][13][14][15][16][17][18][19][20]. A single patient may have multiple neural autoantibodies, the patient's serological profile reflects the antigenic makeup of the underlying tumor [21] and may herald tumor recurrence or predict better tumor survival [3,[21][22][23][24][25][26][27].…”

Section: Introductionmentioning

confidence: 99%

Neurological autoimmunity in patients with non‐pulmonary neuroendocrine neoplasms: clinical manifestations and neural autoantibody profiles

Mangioris,

Halfdanarson,

Lennon

et al. 2024

Euro J of Neurology

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Background and purposeParaneoplastic neurological autoimmunity is well described with small‐cell lung cancer, but information is limited for other neuroendocrine neoplasms (NENs).MethodsAdult patients with histopathologically confirmed non‐pulmonary NENs, neurological autoimmunity within 5 years of NEN diagnosis, and neural antibody testing performed at the Mayo Clinic Neuroimmunology Laboratory (January 2008 to March 2023) were retrospectively identified. Control sera were available from patients with NENs without neurological autoimmunity (116).ResultsThirty‐four patients were identified (median age 68 years, range 31–87). The most common primary tumor sites were pancreas (nine), skin (Merkel cell, eight), small bowel/duodenum (seven), and unknown (seven). Five patients received immune checkpoint inhibitor (ICI) therapy before symptom onset; symptoms preceded cancer diagnosis in 62.1% of non‐ICI‐treated patients. The most frequent neurological phenotypes (non‐ICI‐treated) were movement disorders (12; cerebellar ataxia in 10), dysautonomia (six), peripheral neuropathy (eight), encephalitis (four), and neuromuscular junction disorders (four). Neural antibodies were detected in 55.9% of patients studied (most common specificities: P/Q‐type voltage‐gated calcium channel [seven], muscle‐type acetylcholine receptor [three], anti‐neuronal nuclear antibody type 1 [three], and neuronal intermediate filaments [two]), but in only 6.9% of controls. Amongst patients receiving cancer or immunosuppressive therapy, 51.6% had partial or complete recovery. Outcomes were unfavorable in 48.3% (non‐ICI‐treated) and neural autoantibody positivity was associated with poor neurological outcome.DiscussionNeurological autoimmunity associated with non‐pulmonary NENs is often multifocal and can be treatment responsive, underscoring the importance of rapid recognition and early treatment.

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Anti-TIF1 gamma-positive IPAF patient developed stage IVB lung squamous carcinoma in 1 year: a case report

Xie

Li²,

et al. 2021

BMC Pulm Med

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Background Patients with connective tissue disease, such as dermatomyositis (DM), and positive anti-TIF1γ self-antibodies are commonly diagnosed with malignant tumors as a comorbidity. The relationship between anti-TIF1γ self-antibodies and existing malignant tumors has been confirmed by several reports. However, interstitial pneumonia with autoimmune features (IPAF) cases with a positive anti-TIF1γ self-antibody developing to solid malignant tumors are rarely reported now. Case presentation Herein, we presented an IPAF patient with anti-TIF1γ self-antibodies. No evidence of malignant tumors was found at the initial visit. However, the patient had developed stage IVB lung squamous cell carcinoma at the 1-year follow-up review. Conclusions Altogether, this report described a rare case of IPAF patient with anti-TIF1γ self-antibodies developed to advanced lung squamous cell carcinoma in 1 year. The present case highlights more frequent imaging examinations to identify the occurrence of malignant tumors as early as possible in IPAF patients with positive anti-TIF1γ self-antibodies.

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A novel case of TIF1 gamma autoantibody positive dermatomyositis associated with a non-functional pancreatic neuroendocrine tumor

Cited by 4 publications

References 7 publications

Myositis interstitial lung disease and autoantibodies

Myositis interstitial lung disease and autoantibodies

Neurological autoimmunity in patients with non‐pulmonary neuroendocrine neoplasms: clinical manifestations and neural autoantibody profiles

Anti-TIF1 gamma-positive IPAF patient developed stage IVB lung squamous carcinoma in 1 year: a case report

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