IVIG for Thymoma-Associated Pseudo-Obstruction: Report of Successful Treatment

Greenburg, David; Mo, Clifton C.; Hemmer, Paul A.

doi:10.1159/000103648

Cited by 3 publications

(3 citation statements)

References 9 publications

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“…Intravenous methylprednisolone (IVMP), intravenous immunoglobulin (IVIg), and plasmapheresis (PP) are considered safe and appropriate therapies. Several case reports have demonstrated the effect of these first-line immunotherapies on patients with achalasia or IPO [78][79][80][81]. Flanagan et al assessed a retrospective case series consisting of 23 patients with AGID who completed a 6-12-week therapeutic trial of IVMP and/or IVIg [4].…”

Section: Therapy and Therapeutic Targets In Agidmentioning

confidence: 99%

Autoimmune gastrointestinal dysmotility: the interface between clinical immunology and neurogastroenterology

Nakane

Mukaino

Ihara

et al. 2020

Immunological Medicine

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Autoimmune gastrointestinal dysmotility (AGID), an idiopathic or paraneoplastic phenomenon, is a clinical form of limited autoimmune dysautonomia. The symptoms of AGID and gastrointestinal manifestations in patients with autoimmune rheumatic diseases are overlapping. Antineuronal autoantibodies are often detected in patients with AGID. Autoantibodies play a key role in GI dysmotility; however, whether they cause neuronal destruction is unknown. Hence, the connection between the presence of these autoantibodies and the specific interference in synaptic transmission in the plexus ganglia of the enteric nervous system has to be determined. The treatment options for AGID are not well-defined. However, theoretically, immunomodulatory therapies have been shown to be effective and are therefore used as the first line of treatment. Nonetheless, diverse combined immunomodulatory therapies should be considered for intractable cases of AGID. We recommend comprehensive autoimmune evaluation and cancer screening for clinical diagnosis of AGID. Univocal diagnostic criteria, treatment protocols, and outcome definitions for AGID are required for prompt diagnosis and treatment and appropriate management of immunotherapy, which will circumvent the need for surgeries and improve patient outcome. In conclusion, AGID, a disease at the interface of clinical immunology and neurogastroenterology, requires further investigations and warrants cooperation among specialists, especially clinical immunologists, gastroenterologists, and neurologists.

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Section: Therapy and Therapeutic Targets In Agidmentioning

confidence: 99%

Autoimmune gastrointestinal dysmotility: the interface between clinical immunology and neurogastroenterology

Nakane

Mukaino

Ihara

et al. 2020

Immunological Medicine

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“…Several therapy options have proven efficacious including corticosteroids, intravenous immunoglobulin (IVIg), and plasmapheresis. 4,[8][9][10] Although pyridostigmine provides symptomatic relief and does not modify the underlying immune process, we chose to avoid immunomodulator therapy given the reassuring safety profile and cost of pyridostigmine as compared with other treatment options.…”

Section: Discussionmentioning

confidence: 99%

Clinical Improvement With Pyridostigmine in a Patient With Acetylcholine Receptor Antibody-Associated Autoimmune Gastrointestinal Dysmotility

2022

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Autoimmune gastrointestinal dysmotility (AGID) is a rare form of limited autoimmune dysautonomia caused by autoantibodies against the enteric nervous system. Our patient was a 53-year-old man with 1 year of bloating, intolerance of oral intake, and recurrent ileus. Esophageal manometry showed aperistalsis and hypotensive lower sphincter, consistent with scleroderma esophagus. However, because the patient had no other sequelae of this disease, AGID was considered. Serologic evaluation revealed ganglionic acetylcholine receptor autoantibodies. Treatment with pyridostigmine led to resolution of symptoms. Early recognition of AGID should be considered when manometry shows scleroderma esophagus in patients without other evidence of systemic sclerosis.

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“…Autoantibodypatterns include VGKC as well as neuronal AChR as targets. Prokinetic agents (i.e., pyridostigmine) and immunosuppressive therapies (corticosteroids, intravenous immunoglobulins) were utilized (85)(86)(87)(88)(89).…”

Section: Chronic Gastrointestinal Dysmotility/pseudoobstructionmentioning

confidence: 99%

Autoimmune disorders and paraneoplastic syndromes in thymoma

Blum¹,

Misch²,

Kollmeier³

et al. 2020

J Thorac Dis

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Thymomas are counted among the rare tumour entities which are associated with autoimmune disorders (AIDs) and paraneoplastic syndromes (PNS) far more often than other malignancies. Through its complex immunological function in the context of the selection and maturation of T cells, the thymus is at the same time highly susceptible to disruptive factors caused by the development and growth of thymic tumours. These T cells, which are thought to develop to competent immune cells in the thymus, can instead adopt autoreactive behaviour due to the uncontrolled interplay of thymomas and become the trigger for AID or PNS affecting numerous organs and tissues within the human body. While myasthenia gravis is the most prevalent PNS in thymoma, numerous others have been described, be they related to neurological, cardiovascular, gastrointestinal, haematological, dermatological, endocrine or systemic disorders. This review article sheds light on the pathophysiology, epidemiology, specific clinical features and therapeutic options of the various forms as well as courses and outcomes of AID/PNS in association with thymomas.Whenever suitable and backed by the limited available evidence, the perspectives from both the thymoma and the affected organ/tissue will be highlighted. Specific issues addressed are the prognostic significance of thymectomy on myasthenia gravis and other thymoma-associated AID/PND and further the impact and safety of immunotherapies on AID and PND relating to thymomas.

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IVIG for Thymoma-Associated Pseudo-Obstruction: Report of Successful Treatment

Cited by 3 publications

References 9 publications

Autoimmune gastrointestinal dysmotility: the interface between clinical immunology and neurogastroenterology

Autoimmune gastrointestinal dysmotility: the interface between clinical immunology and neurogastroenterology

Clinical Improvement With Pyridostigmine in a Patient With Acetylcholine Receptor Antibody-Associated Autoimmune Gastrointestinal Dysmotility

Autoimmune disorders and paraneoplastic syndromes in thymoma

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