Autoimmune sclerosing cholangitis: An atypical association with kawasaki disease

Rosencrantz, Richard; Huang, Tiangui; Sonke, Pierre-Yves; Tewari, Deepali; Chander, Praveen N.

doi:10.1002/hep.28694

Cited by 6 publications

(2 citation statements)

References 6 publications

(11 reference statements)

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“…KD is a childhood systemic vasculitis which still remains without a well-defined etiology. The epidemiology and clinical presentation suggest a possible underlying viral/bacterial infection or an abnormal immunologic response to an infection in genetically predisposed children; autoimmune disease is a possibility as well [ 13 , 14 ]. After exhaustive investigation, we were not able to find any of the above-mentioned causative triggers of KD in our patient and therefore we are not aware if the agent triggering KD was also hepatotropic and responsible for both the vasculitis and this atypically prolonged liver involvement.…”

Section: Discussionmentioning

confidence: 99%

Atypically Protracted Course of Liver Involvement in Kawasaki Disease. Case Report and Literature Review

et al. 2021

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Hypertransaminasemia in patients with Kawasaki disease (KD) is reported to be transient. Here, we describe a child with an atypically protracted course of liver tests abnormalities and review the inherent literature. The patient was hospitalized at age 7-months for isolated hypertransaminasemia detected during a classical KD diagnosed 3 months before, and persistent since then. KD clinical evolution had been favorable, with rapid response to acetylsalicylic acid and intravenous immunoglobulins. Liver enzymes however remained persistently elevated with a fluctuating pattern (ALT > AST levels; peak of AST 186 IU/L and ALT 240 IU/L). During follow-up, the main causes of liver dysfunction had to be excluded through appropriate and extensive laboratory investigations. Transaminases values become steadily normal only 7 months after the acute presentation of KD. Conclusions: Our report shows that an atypically protracted courses of KD-related hypertransaminasemia above the previously reported temporal limits should be taken into account during the stepwise diagnostic approach to the patient’s liver dysfunction. Insidious acetylsalycilic acid-hepatotoxicity warrants consideration in the differential diagnosis.

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Section: Discussionmentioning

confidence: 99%

Atypically Protracted Course of Liver Involvement in Kawasaki Disease. Case Report and Literature Review

et al. 2021

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“…Ohnishi et al reported a 4-year-old boy with KD presenting as sigmoid colitis and Rosencrantz et al reported a 2.5-year-old boy with KD presenting as sclerosing cholangitis. 18,19 Chen et al have demonstrated that sonographic gallbladder abnormalities are associated with IVIG resistance in KD. 20 Bagrul et al presented a 3-yearold boy who was diagnosed with atypical KD, with coronary arteries dilatation, sterile pyuria and progressive bowel oedema, severe abdominal pain, hepatosplenomegaly and hydrops of the gallbladder.…”

Section: Discussionmentioning

confidence: 99%

<p>Unusual Presentation Of Kawasaki Disease With Gastrointestinal And Renal Manifestations</p>

Lazea¹,

Man

Sur³

et al. 2019

TCRM

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Diagnosis of Kawasaki disease (KD) is based on well-established clinical criteria. In incomplete or atypical KD, the diagnosis is challenging, because of the paucity of clinical signs or because of the presence of clinical manifestations that generally are not seen in KD. We describe the case of a 3-year-old female patient with persistent high fever, vomiting, watery diarrhea, metabolic acidosis and severe hypopotassemia. On the fourth day of fever, bilateral conjunctivitis, mucous and extremity changes were registered. Urine changes as glycosuria and proteinuria were also noticed. Echocardiography revealed ectasia of the left anterior descending coronary artery, and diagnosis of KD was established. The treatment consisted of intravenous immunoglobulin (IVIG) and oral aspirin. Recurrence of disease was recorded on the 23rd day of the disease, with favorable evolution after the second dose of IVIG was infused.

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Incidence of chronic immune-mediated inflammatory diseases after diagnosis with Kawasaki disease: a population-based cohort study

et al. 2021

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Objectives Kawasaki disease (kDa) is an immune-mediated vasculitis of childhood with multi-organ inflammation. We determined the risk of subsequent immune-mediated inflammatory disease (IMID), including arthritis, type 1 diabetes, inflammatory bowel disease (IBD), autoimmune liver disease (ALD), primary sclerosing cholangitis (PSC) and multiple sclerosis (MS). Methods We conducted a matched population-based cohort study using health administrative data from Ontario, Canada. Children aged <18 years born between 1991–2016 diagnosed with kDa (n = 3,753) were matched to 5 non-kDa controls from the general population (n = 18 749). We determined the incidence of IMIDs after resolution of kDa. Three- and 12-month washout periods were used to exclude kDa-related symptoms. Results There was an elevated risk of arthritis in kDa patients compared with non-kDa controls starting three months after index date (103.0 vs 12.7 per 100 000 person-years (PYs); incidence rate ratio (IRR) 8.07 (95% CI 4.95–13.2); hazard ratio (HR) 8.08 (95% CI 4.95–13.2), resulting in the overall incidence of IMIDs being elevated in kDa patients (175.1 vs 68.0 per 100 000 PYs; IRR 2.58 (95% CI 1.93–3.43); HR 2.58, 95% CI 1.94–3.43). However, there was no increased risk for diabetes, IBD, ALD, PSC or MS in kDa patients. Similar results were observed using a 12-month washout period. Conclusions Children diagnosed with kDa were at increased risk of arthritis following the acute kDa event, but not other IMIDs. Health care providers should monitor for arthritis in children following a diagnosis of kDa.

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Autoimmune sclerosing cholangitis: An atypical association with kawasaki disease

Cited by 6 publications

References 6 publications

Atypically Protracted Course of Liver Involvement in Kawasaki Disease. Case Report and Literature Review

Atypically Protracted Course of Liver Involvement in Kawasaki Disease. Case Report and Literature Review

<p>Unusual Presentation Of Kawasaki Disease With Gastrointestinal And Renal Manifestations</p>

Incidence of chronic immune-mediated inflammatory diseases after diagnosis with Kawasaki disease: a population-based cohort study

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