A woman with adult‐onset Still's disease and acute intestinal pseudo‐obstruction

Hori, Hiroshi; Yabe, Hiroki; Fukuchi, Takeo; Sugawara, Hitoshi

doi:10.1002/ccr3.3488

Cited by 3 publications

(5 citation statements)

References 20 publications

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“…The presence of small bowel inflammation in patients affected by AOSD has previously been reported in a few cases in association with Crohn's disease, coeliac disease and chronic intestinal pseudo-obstruction. [2][3][4][5][6][7] The association between AOSD and raised intraepithelial lymphocytes 11 or between juvenile idiopathic arthritis (the juvenile counterpart of AOSD) and the presence of activated natural killer lymphocytes in the bowel wall 12 has also been described. Moreover, a pathogenetic role of the gut immune system in spondyloarthropathies has been hypothesised.…”

Section: Discussionmentioning

confidence: 99%

“…1 Small bowel involvement has been reported only in a few cases in association with Crohn's disease, coeliac disease and chronic intestinal pseudo-obstruction. [2][3][4][5][6][7]…”

Section: Introductionmentioning

confidence: 99%

“…Laboratory investigations usually reveal leucocytosis with neutrophilia, raised ferritin, elevated C reactive protein (CRP) and erythrocyte sedimentation rate 1. Small bowel involvement has been reported only in a few cases in association with Crohn’s disease, coeliac disease and chronic intestinal pseudo-obstruction 2–7…”

Section: Introductionmentioning

confidence: 99%

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Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease

et al. 2023

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Adult-onset Still’s disease (AOSD) is a rare systemic autoinflammatory disease with a broad spectrum of clinical manifestations including fever, skin rash, arthralgia and neutrophilic leucocytosis. Small bowel inflammation in AOSD has been reported in association with Crohn’s disease, coeliac disease and chronic intestinal pseudo-obstruction. We have here reported the first-time case of AOSD with small bowel involvement, presenting with chronic vomiting. Fluorodeoxyglucose total-body positron emission tomography/CT showed high uptake of the duodenum and first jejunal loop. Faecal calprotectin increased. Duodenal histology revealed neutrophilic infiltrate among duodenal enterocytes and severe chronic lymphoplasmacellular infiltrate of the lamina propria. Vomiting disappeared when duodenojejunal inflammation was controlled by immunomodulatory treatments for AOSD, suggesting a possible causal relation between duodenojejunal inflammation and the symptom.

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Section: Discussionmentioning

confidence: 99%

“…1 Small bowel involvement has been reported only in a few cases in association with Crohn's disease, coeliac disease and chronic intestinal pseudo-obstruction. [2][3][4][5][6][7]…”

Section: Introductionmentioning

confidence: 99%

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Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease

et al. 2023

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“…In this case report, we highlight acute intestinal pseudo-obstruction as a rare complication of AOSD. Only four other cases with pseudoobstruction have been previously reported [5][6][7][8] . Additionally, a recent case report described duodenojejunal inflammation causing chronic vomiting [9] , suggesting that further investigation into potential gastrointestinal involvement in AOSD may be warranted.…”

Section: Discussionmentioning

confidence: 99%

“…Additionally, a recent case report described duodenojejunal inflammation causing chronic vomiting [9] , suggesting that further investigation into potential gastrointestinal involvement in AOSD may be warranted. Previous reports have implicated an affectation of the sympathetic tone of the bowel and an imbalance in the autonomic enteric nervous system, leading to colonic distension [8] . However, recent literature suggests that other mechanisms, such as a high level of IL-18 associated with AOSD, may also play a role.…”

Section: Discussionmentioning

confidence: 99%

Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s disease

Rossignon,

Soupart

2023

European Journal of Case Reports in Internal Medicine

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Adult-onset Still’s disease (AOSD) is a rare autoinflammatory disorder that can lead to a cytokine storm, causing a range of symptoms. Acute intestinal pseudo-obstruction is another rare condition that results in intestinal obstruction without anatomical cause. Although the two conditions are rarely reported together, we present the case of a 62-year-old male who developed acute intestinal pseudo-obstruction in the context of an AOSD flare. This led to severe hypokalaemia and a critical condition. Other symptoms included a high-spiking fever lasting for weeks, polyarthralgias and a typical salmon-coloured rash. After ruling out other potential causes, the patient was diagnosed with AOSD. Our findings suggest that the cytokine storm associated with this disease triggered the acute intestinal pseudo-obstruction and life-threatening hypokalaemia, establishing a causal relationship. Only four other cases of AOSD complicated by intestinal pseudo-obstruction have been reported, and this is the first to present with life-threatening hypokalaemia. This case serves as a crucial reminder that, despite being a diagnosis of exclusion, Still’s disease should be considered as a potential cause of intestinal pseudo-obstruction, as prompt recognition and treatment of the underlying cause is crucial in managing this potentially life-threatening condition.

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A Usual Presentation of an Unusual Case

Zawadka,

Mosaad,

Wong

2023

Lessons From the ICU

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A woman with adult‐onset Still's disease and acute intestinal pseudo‐obstruction

Abstract: This is an open access article under the terms of the Creative Commons Attribution-NonCommercial License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited and is not used for commercial purposes.

Cited by 3 publications

References 20 publications

Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease

Duodenojejunal inflammation causing chronic vomiting in adult-onset Still’s disease

Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s disease

A Usual Presentation of an Unusual Case

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