Paraneoplastic visceral neuropathy as a cause of severe gastrointestinal motor dysfunction

Chinn, Janet; Schuffler, Michael D.

doi:10.1016/0016-5085(88)90362-9

Cited by 136 publications

(71 citation statements)

References 26 publications

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“…Aetiologies include disease processes localised to the colon, such as Hirschsprung’s disease, or generalised motility disorders that affect multiple regions of the gastrointestinal tract [5]. CIPO can be a paraneoplastic disorder which appears to be secondary to an autoimmune process triggered by a cross-reaction between antigens shared by the tumour cells and myenteric plexus neurons, and several cases have been reported in association with thymoma or small-cell lung cancer [6,7,8]. CIPO has also been described during the course of idiopathic autonomic neuropathy characterised by the high frequency of circulating ganglionic AChR autoantibodies [9].…”

Section: Introductionmentioning

confidence: 99%

Thymoma-Associated Neuromyotonia with Antibodies against Voltage-Gated Potassium Channels Presenting as Chronic Intestinal Pseudo-Obstruction

Viallard¹,

Vincent

Moreau

et al. 2005

Eur Neurol

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Chronic intestinal pseudo-obstruction can occur as a paraneoplastic disorder, and several cases have been reported in association with thymoma or small-cell lung cancer. Autoantibodies against voltage-gated potassium channels (VGKCs) are found in acquiredneuromyotonia (Isaac’s syndrome), and have been reported in one case of slow transit constipation without apparent neurological disease. We describe a patient with VGKC antibodies, acquired neuromyotonia and thymoma, who first presented with a severe slow-transit constipation and in whom the gastrointestinal symptoms responded well to plasmapheresis. We suggest that VGKC antibodies might be helpful in patients with possible paraneoplastic chronic intestinal pseudo-obstruction, and a positive result should stimulate the search for a thymoma or other tumour and raise the possibility of immunotherapy.

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Section: Introductionmentioning

confidence: 99%

Thymoma-Associated Neuromyotonia with Antibodies against Voltage-Gated Potassium Channels Presenting as Chronic Intestinal Pseudo-Obstruction

Viallard¹,

Vincent

Moreau

et al. 2005

Eur Neurol

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“…Such secondary GINMDs include intestinal pseudo-obstruction occurring as a paraneoplastic syndrome, most commonly in association with small cell lung carcinoma (SCLC) (Schuffler et al 1983;Chinn et al 1988;Sodhi et al 1989;Lucchinetti et al 1998), but also in cases of bronchial carcinoid (Gerl et al 1992), thymoma (Pande et al 1999), ganglioneuroblastoma and neuroblastoma (Schobinger-Clement et al 1999;Martin et al 2008) and ovarian carcinoma . GINMDs may also occur in association with infective systemic diseases such as Chagas' disease (South American trypanosomiasis) (Ferreira-Santos 1961), connective tissue disorders such as scleroderma (Treacy et al 1963;Cohen et al 1972;Battle et al 1981) and some endocrine and metabolic disorders e.g.…”

Section: Gastrointestinal Neuromuscular Diseasesmentioning

confidence: 99%

“…These include lymphocytic and plasmacellular infiltration, enteroglial cell proliferation and neuronal degeneration and loss, progressing to aganglionosis in the most severe cases (Schuffler et al 1983;Chinn et al 1988;Schobinger-Clement et al 1999;Lee et al 2001); (2) patients with SCLC and GI dysmotility often have co-existing neurological disorders including peripheral neuropathies, cerebellar degeneration and limbic encephalitis, which are thought to have an autoimmune origin (Chinn et al 1988;Lucchinetti et al 1998;Vernino et al 1998); (3) several potentially pathogenic 'paraneoplastic autoantibodies' reactive against antigens expressed by tumour cells and neurons have been identified, which in combination with neurological disorders, including GI symptoms, strongly suggest the presence of an underlying malignancy (Lucchinetti et al 1998;Pande et al 1999;Schobinger-Clement et al 1999;Lee et al 2001). …”

Section: Paraneoplasiamentioning

confidence: 99%

The role of humoral autoimmunity in gastrointestinal neuromuscular diseases

Hubball

Martin

Lang

et al. 2009

Progress in Neurobiology

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Dysfunction of the gastrointestinal neuromuscular apparatus (including interstitial cells ofCajal) is presumed to underlie a heterogeneous group of disorders collectively termed gastrointestinal neuromuscular diseases (GINMDs). Humoral (antibody)-mediated autoimmunity directed against ligand-or voltage-gated ion channels or associated proteins involved in neuromuscular transmission is associated with several acquired neuromuscular diseases of the periphery and is now implicated in an increasing number of less well-characterised diseases. Anti-channel antibodies have been reported in small numbers of patients with GINMD, particularly in those with GI dysfunction secondary to an underlying disease such as neoplasia or infection. However, little is known of humoral autoimmunity in primary GINMDs.This thesis investigated the association between anti-channel antibodies and GINMD, and the human GI tract as a potential target of anti-channel antibodies. The presence of antivoltage-and ligand-gated antibodies was investigated in the serum of patients with primary achalasia, enteric dysmotility and intestinal pseudo-obstruction, and in those with GINMD secondary to Chagas' disease. The functional effect of sera from some of these patients on colonic smooth muscle contractility was also characterised. Finally, the distribution of six voltage-gated potassium channels (VGKCs), which serve essential roles in the peripheral and central nervous systems and are known targets of pathological autoantibodies, were investigated in all layers of the human GI tract.Our findings suggest that currently recognised anti-channel antibodies are not a common pathogenic factor in primary GINMDs. Anti-channel antibodies, in particular anti-VGKC antibodies, were however found in a significant number of individuals with Chagas' disease. Furthermore, circulating factors in patients with chagasic GI disease altered GI smooth muscle contractility in vitro which may have pathological relevance to GI

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“…A gastroparesia pode ser observada nos carcinomas mamário, brônquico ou esofágico quando cursam com invasão do nervo vago 8 . Além disso, é descrita como integrante da síndrome paraneoplásica nos tumores carcinóide e carcinoma pulmonar de pequenas células 29 .…”

Section: Diagnóstico Diferencial Das Gastroparesias No Diabetesunclassified

Gastroparesia diabética

Cesarini¹,

Ferreira²,

Dib³

1997

Rev. Assoc. Med. Bras.

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INTRODUÇÃOAlterações na motilidade gastrointestinal decorrentes da neuropatia autonômica são freqüentes nos pacientes diabéticos, e se constituem em importante causa de morbidade. Os distúrbios da motilidade esofágica, gástrica ou intestinal levam a sintomas muitas vezes severos e podem, também, dificultar a obtenção de um bom controle glicê-mico 1 . Abordaremos, aqui, o comprometimento da função da motilidade do estômago próprio do diabetes melito, que determina retardo no esvaziamento gástrico, caracterizando o quadro da gastroparesia diabética estudada pela primeira vez por Kassander, em 1954 2 . EPIDEMIOLOGIAOs achados, quanto à prevalência da gastroparesia diabética (GPD), são discordantes. Um estudo retrospectivo envolvendo 43.900 prontuários de pacientes do Ambulatório da Clínica Joslin, em Boston (EUA), mostrou que apenas 0,08% dos indivíduos tinha o diagnóstico de GPD 3 . No entanto, em estudos prospectivos radiológicos, o retardo do esvaziamento gástrico foi observado em 22% dos indivíduos diabéticos do tipo I 2 . Num grupo de pacientes com evidência clínica de neuropatia, que tiveram o esvaziamento gástrico estudado por método radioisotópico, foi observado retardo do esvaziamento em 27% dos casos 4 . A GPD manifesta-se, geralmente, após oito a dez anos do diagnóstico do diabetes, como ocorre com a maioria das outras complicações da doença 5,6 . PATOGÊNESEOs fatores etiopatogênicos mais comumente implicados na GPD são a neuropatia autonômica e alterações na secreção de hormônios envolvidos na regulação da função digestiva 7 . O componente neurológico seria o fator mais importante, uma vez que, na maioria dos portadores da GPD, comprova-se a presença de neuropatia autonô-mica e neuropatia periférica 1,8 . A melhora dos sintomas com o uso de agentes colinérgicos evidencia o provável papel patogênico do sistema nervoso autonômico.No entanto, discute-se a importância da perda da função vagal na patogênese dessa complicação, uma vez que a vagotomia cirúrgica promove o esvaziamento gástrico para líquidos, fato não observado na GPD. Isso sugere a implicação de outros mecanismos na gênese da GPD 9 . Nos estudos de esvaziamento gástrico com tecné-cio, observa-se que há baixa correlação entre a intensidade dos sintomas e o grau de gastroparesia documentada pelos métodos de avaliação de esvaziamento gástrico 6 . Acometimento heterogêneo da inervação aferente explicaria, ao menos em parte, a grande variabilidade de sintomas em indivíduos com distúrbio da motilidade gástrica 6 . Rathmann 10 demonstrou associação entre a neuropatia aferente visceral e presença de GPD oligossintomática. Outras evidências sugerem a participação do sistema nervoso autonômico na patogênese da GPD mediante alterações na liberação de hormônios enté-ricos. Tais distúrbios parecem ter relação com a diminuição da atividade vagal 11,12 . Devido ao tônus vagal diminuído, a gastrina encontra-se elevada, causando o retardo do esvaziamento gástrico. Em adição, o pH gástrico aumentado, devido à deficiência de secreção ácida, também serve de es...

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Paraneoplastic visceral neuropathy as a cause of severe gastrointestinal motor dysfunction

Cited by 136 publications

References 26 publications

Thymoma-Associated Neuromyotonia with Antibodies against Voltage-Gated Potassium Channels Presenting as Chronic Intestinal Pseudo-Obstruction

Thymoma-Associated Neuromyotonia with Antibodies against Voltage-Gated Potassium Channels Presenting as Chronic Intestinal Pseudo-Obstruction

The role of humoral autoimmunity in gastrointestinal neuromuscular diseases

Gastroparesia diabética

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