Gallbladder Vasculitis: An Unexpected and Puzzling Finding

Carrascosa, Miguel F; Daroca-Pérez, Rafael; Silva-Carmona, Mary

doi:10.1016/j.cgh.2016.11.019

Cited by 2 publications

(2 citation statements)

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“…Single-organ vasculitis (SOV) is one of the categories of vasculitis and a rare disease, which occurs in arteries or veins of any size in a single organ of an adult, and does not exhibit any feature that it is a limited expression of a systemic vasculitis [21]. In the digestive organs, approximately 80 cases of SOV in the GI tract, 24 cases of SOV in the gallbladder [22–26], and one case of SOV in the pancreas have been reported in English articles [27]. Similar to the present case, Carrascosa et al reported a case of SOV in the gallbladder of an adult with necrotizing vasculitis of small- and medium-sized arteries and eosinophil-rich inflammation, but their case did not fulfill the criteria of ACR for EGPA [26].…”

Section: Discussionmentioning

confidence: 99%

“…In the digestive organs, approximately 80 cases of SOV in the GI tract, 24 cases of SOV in the gallbladder [22–26], and one case of SOV in the pancreas have been reported in English articles [27]. Similar to the present case, Carrascosa et al reported a case of SOV in the gallbladder of an adult with necrotizing vasculitis of small- and medium-sized arteries and eosinophil-rich inflammation, but their case did not fulfill the criteria of ACR for EGPA [26]. In addition, Gonzalez-Gay et al reported a case of SOV in the pancreas of an adult with polyarteritis nodosa-like arteritis in medium-sized peripancreatic arteries, but pancreatitis was not clinically or histologically observed [27].…”

Section: Discussionmentioning

confidence: 99%

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An Autopsy Case of a 5-Year-Old Child with Acute Pancreatitis Caused by Eosinophilic Granulomatosis with Polyangiitis-like Necrotizing Vasculitis

Yagi

Takahashi

Kibe

et al. 2019

Case Reports in Rheumatology

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In children, acute pancreatitis has been reported in IgA vasculitis, Kawasaki disease, systemic lupus erythematosus-associated vasculitis, and juvenile dermatomyositis-associated vasculitis. However, its frequency in these vasculitides has been shown to be low. In other childhood-onset vasculitides, acute pancreatitis is seldom reported. The patient was a 5-year-old Japanese boy who suddenly presented with gastrointestinal (GI) bleeding. Therapy with antiulcer drugs successfully stopped bleeding, but subsequently, high fever, leukocytosis, and hypoxia appeared. He died 12 days after he presented with GI bleeding. An autopsy unexpectedly revealed that necrotizing vasculitis with marked eosinophilic and histiocytic infiltration of the pancreas led to acute pancreatitis, and gastric ulcer with eosinophilic infiltration was shown to be the origin of GI bleeding. In addition, eosinophilic infiltration was found in the small intestine, lungs, and bone marrow. Necrotizing vasculitis with eosinophilic and histiocytic infiltration of the pancreas, eosinophilic infiltration of the airway wall, and eosinophilic gastroenteritis with gastric ulcer were histologically confirmed, suggesting that the present case may be an early stage of eosinophilic granulomatosis with polyangiitis- (EGPA-) like vasculitis. To our knowledge, this might be the first reported case of EGPA-like vasculitis presenting with acute pancreatitis in a child.

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