Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s disease
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...
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| Format: | Article |
| Language: | English |
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SMC MEDIA SRL
2023-05-01
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| Series: | European Journal of Case Reports in Internal Medicine |
| Subjects: | |
| Online Access: | https://www.ejcrim.com/index.php/EJCRIM/article/view/3887 |
| _version_ | 1797809392726310912 |
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| author | Pierre Rossignon Alain Soupart |
| author_facet | Pierre Rossignon Alain Soupart |
| author_sort | Pierre Rossignon |
| collection | DOAJ |
| description | 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. |
| first_indexed | 2024-03-13T06:51:59Z |
| format | Article |
| id | doaj.art-b6573f5d8ace4a0bab96bbeb389197df |
| institution | Directory Open Access Journal |
| issn | 2284-2594 |
| language | English |
| last_indexed | 2024-03-13T06:51:59Z |
| publishDate | 2023-05-01 |
| publisher | SMC MEDIA SRL |
| record_format | Article |
| series | European Journal of Case Reports in Internal Medicine |
| spelling | doaj.art-b6573f5d8ace4a0bab96bbeb389197df2023-06-07T14:14:15ZengSMC MEDIA SRLEuropean Journal of Case Reports in Internal Medicine2284-25942023-05-0110.12890/2023_0038873422Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s diseasePierre Rossignon0Alain Soupart1Department of Internal Medicine, Moliere-Longchamp Hospital, Brussels, BelgiumResearch Unit for the Study of Hydromineral Metabolism, Department of Internal Medicine, Erasmus University Hospital, ULB, Brussels, BelgiumAdult-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.https://www.ejcrim.com/index.php/EJCRIM/article/view/3887adult-onset still’s diseasehypokalemiaintestinal pseudo-obstruction |
| spellingShingle | Pierre Rossignon Alain Soupart Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s disease European Journal of Case Reports in Internal Medicine adult-onset still’s disease hypokalemia intestinal pseudo-obstruction |
| title | Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s disease |
| title_full | Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s disease |
| title_fullStr | Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s disease |
| title_full_unstemmed | Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s disease |
| title_short | Intestinal pseudo-obstruction with life-threatening hypokalaemia in a patient with adult-onset Still’s disease |
| title_sort | intestinal pseudo obstruction with life threatening hypokalaemia in a patient with adult onset still s disease |
| topic | adult-onset still’s disease hypokalemia intestinal pseudo-obstruction |
| url | https://www.ejcrim.com/index.php/EJCRIM/article/view/3887 |
| work_keys_str_mv | AT pierrerossignon intestinalpseudoobstructionwithlifethreateninghypokalaemiainapatientwithadultonsetstillsdisease AT alainsoupart intestinalpseudoobstructionwithlifethreateninghypokalaemiainapatientwithadultonsetstillsdisease |