Bilateral ureteral stenosis and duodenal perforation in a patient with dermatomyositis

Morita, Yoshitaka; Sakuta, Takeo; Nagasu, Hajime; Kuwabara, Atsunori; Tokuoka, Yuko; Teshigawara, Sanae; Horike, Hideyuki; Kashihara, Naoki

doi:10.1007/s10165-006-0529-8

Cited by 12 publications

(6 citation statements)

References 11 publications

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“…An emergency operation showed an ulcer and perforation at the junction of the third and fourth portions of the duodenum because of the mesenteric vasculitis [ 16 ]. Rheumatologists should be alerted about this serious complication in patients with childhood or young adult dermatomyositis presenting with abdominal complaints [ 17 ].…”

Section: Acute Intestinal Ischemia In Childrenmentioning

confidence: 99%

Intestinal Ischemia in Neonates and Children

Jeican

Ichim

Gheban

2016

Medicine and Pharmacy Reports

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The article reviews the intestinal ischemia theme on newborn and children. The intestinal ischemia may be either acute - intestinal infarction (by vascular obstruction or by reduced mesenteric blood flow besides the occlusive mechanism), either chronic.In neonates, acute intestinal ischemia may be caused by aortic thrombosis, volvulus or hypoplastic left heart syndrome.In children, acute intestinal ischemia may be caused by fibromuscular dysplasia, volvulus, abdominal compartment syndrome, Burkitt lymphoma, dermatomyositis (by vascular obstruction) or familial dysautonomia, Addison’s disease, situs inversus abdominus (intraoperative), burns, chemotherapy administration (by nonocclusive mesenteric ischemia). Chronic intestinal ischemia is a rare condition in pediatrics and can be seen in abdominal aortic coarctation or hypoplasia, idiopathic infantile arterial calcinosis.

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Section: Acute Intestinal Ischemia In Childrenmentioning

confidence: 99%

Intestinal Ischemia in Neonates and Children

Jeican

Ichim

Gheban

2016

Medicine and Pharmacy Reports

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“…This leads us to speculate that ureteral stenosis may be related to vasculopathy involving the urinary system. Morita et al [23] reported a case of dermatomyositis who developed duodenal perforation as well as ureteral stenosis, with biopsy of stenotic ureter showing calci cation with granulomatous tissue without any vasculitic lesion. Ruby Haviv et al [24] reported a case of juvenile polymyositis (JPM ) with right ureteral obstruction secondary to necrosis.…”

Section: Discussionmentioning

confidence: 99%

Gastrointestinal perforation in anti-NXP2 antibody-associated juvenile dermatomyositis: Case reports and a review of the literature

Zhou³

et al. 2020

Preprint

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Background: To summarize the characteristics of gastrointestinal (GI) perforation in anti-nuclear matrix protein 2 (NXP2) antibody-associated juvenile dermatomyositis (JDM). Methods: Five patients with GI perforation from a JDM cohort of 120 cases are described. Relevant literature was reviewed.Results: Five patients, including four females and one male, were included in the study. The age of onset of these patients ranged from 3.3 to 9.5 years with the median age of 5.0 years. When these patients were complicated by GI perforation, childhood myositis assessment score (CMAS) ranged from 1 to 5 with the median score of 2. Myositis-specific antibody (MSA) spectrum analysis indicated that the five patients were anti-NXP2 antibody positive. The initial symptoms of GI perforation were progressive abdominal pain and intermittent fever. Two patients also presented with ureteral calculus with hydronephrosis and ureteral stricture. Surgery was performed in four patients. One patient failed to undergo a repair as the perforation was high in position. For the other three patients, perforation repair was successful, of which two patients failed due to recurrent perforation. At 24 months postoperative follow-up, one patient was in complete remission on prednisone (Pred) and methotrexate(MTX) treatment, and her ureteral stricture had disappeared. The other four patients died. Adding these cases with 16 other patients described in the literature, the symptom at onset was progressive abdominal pain, which often occurred within 10 months after JDM was diagnosed. Perforation most commonly occurred in the duodenum, although it also occurred at multiple sites or was recurrent. The mortality rate of GI perforation in JDM was 38% (8/21). Conclusions: All the five perforation cases in our study subjected to MSA analysis were anti-NXP2 antibody positive. The symptom at onset was abdominal pain. The most common site of perforation was the duodenum in the retroperitoneum, and the lack of acute abdominal manifestations prevented early diagnosis. GI perforation may be a fatal complication in JDM, and early diagnosis is very important. More research is needed to determine the pathogenesis and predictive factors of GI perforation in JDM.

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“…While the incidence of ulceration and bleeding perforation of GI tracts in childhood DM is as low as 2-3%, the incidence in adult DM is even lower (~1%) [ 6 ]. The reported sites for GI involvement in adult DM include the esophagus [ 7 – 9 ], duodenum [ 10 , 11 ], and intestine [ 8 ]. Another example of perforation of the hollow organs is mediastinal emphysema that has originated from a tracheal perforation, which is considered to be related to vasculitis [ 12 , 13 ].…”

Section: Discussionmentioning

confidence: 99%

Adult Dermatomyositis with Bleeding Ulcer in the Pharynx

Kusano

Takahashi

Misaki

et al. 2014

Case Reports in Otolaryngology

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Dermatomyositis (DM) is one of the idiopathic inflammatory myopathies caused by complement-mediated vasculopathy or vasculitis in the muscle. Although the gastrointestinal (GI) mucosa has been reported to be involved as a result of vasculitis or vasculopathy, ulceration in the pharynx is a rare manifestation of DM. A 54-year-old woman complaining of muscle weakness in the extremities, low-grade fever, and dysphagia was diagnosed as having DM. Despite medical treatment with corticosteroids and immunosuppressive agents, her DM progressed rapidly, leading to exacerbation of the dysphagia. About 3 weeks after undergoing tracheostomy as a preventive measure against aspiration, the patient developed intractable respiratory tract hemorrhage. Repeated laryngoendoscopy revealed a bleeding ulceration in the pharynx that required hemostasis with electric cautery under general anesthesia. No bleeding recurred thereafter. Histopathologically, the pharynx exhibited nonspecific inflammatory cell infiltration in the muscle tissue. This rare manifestation may be considered in cases of DM with unexplainable airway bleeding.

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Bilateral ureteral stenosis and duodenal perforation in a patient with dermatomyositis

Cited by 12 publications

References 11 publications

Intestinal Ischemia in Neonates and Children

Intestinal Ischemia in Neonates and Children

Gastrointestinal perforation in anti-NXP2 antibody-associated juvenile dermatomyositis: Case reports and a review of the literature

Adult Dermatomyositis with Bleeding Ulcer in the Pharynx

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