Case 42-1989

Cabot, Richard C.; Scully, Robert E.; Mark, Eugene J.; McNeely, William F.; McNeely, Betty U.; Yood, Robert A.; Fienberg, Robert

doi:10.1056/nejm198910193211608

Cited by 14 publications

(6 citation statements)

References 79 publications

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“…Mean duration of GB-related (right-upper quadrant or abdominal) symptoms was 9.5 weeks (median, 1 wk; range, 0–60 wk) prior to diagnosis. Lithiasic cholecystitis and/or chronic cholelithiasis was the clinical presentation in 44% of patients, 3 , 4 , 11 , 14 , 17 , 19 , 31 , 34 , 38 , 40 , 46 , 48 , 50 , 51 , 61 45.7% presented with acalculous cholecystitis, 1 , 3 , 5 , 12 , 16 , 18 , 20 , 22 , 23 , 28 , 32 , 36 – 38 , 42 – 48 , 51 , 58 – 60 3.4% with bile duct obstruction, 10 , 15 and 6.7% did not exhibit abdominal symptoms; GV was discovered because of abnormal GB findings in imaging studies, 9 , 38 exploratory laparotomy (current study Patient 5), or necropsy. 56 …”

Section: Resultsmentioning

confidence: 99%

“…4 , 51 Twenty-seven patients underwent biopsies in other regions. 1 , 3 , 9 – 11 , 14 , 15 , 19 , 23 , 32 , 36 , 38 , 40 , 42 , 43 , 45 , 46 , 48 , 50 , 51 , 54 , 60 Abdominal angiography was performed in 7 patients and vascular abnormalities were detected in 3 who had GB-SV (Table 4 ). 9 , 10 , 12 , 37 , 46 , 54 …”

Section: Resultsmentioning

confidence: 99%

“…GB-SOV was found in 20 (33%) patients and GB-SV in 41 (67%). Forms of SV that affected the GB were PAN (n = 10), 4 , 12 , 22 , 32 , 38 , 45 , 46 , 51 , 54 HBV-associated vasculitis (n = 8), 10 , 11 , 17 , 38 , 46 , 56 , 60 cryoglobulinemic (essential or HCV-associated) vasculitis (n = 6), 9 , 14 , 19 , 36 , 50 MPA (n = 4), 5 , 31 , 38 , 40 EGPA (Churg-Strauss) (n = 4), 20 , 43 , 48 , 61 IgA vasculitis (Henoch-Schönlein) (n = 2), 23 , 28 giant cell arteritis (n = 1), 47 and vasculitis associated with autoimmune diseases (n = 6) 11 , 16 , 18 , 42 , 58 (see Table 4 ). Systemic diseases with associated vasculitis included RA (n = 3), 11 , 16 , 18 SLE (n = 2), 42 , 58 and systemic sclerosis (n = 1).…”

Section: Resultsmentioning

confidence: 99%

“…Angiographic studies were performed in 2 GB-SOV patients 37 , 54 and in 5 GB-SV patients, 9 , 10 , 12 , 46 and were suggestive of SV in 3 of them. 10 , 12 , 46 Among GB-SV patients, 23 (59%) had a biopsy performed in regions other than the GB 9 – 11 , 14 , 19 , 23 , 32 , 36 , 38 , 40 , 42 , 43 , 45 , 46 , 48 , 50 , 51 , 54 , 60 , 61 and 82% of them confirmed vasculitis. SV (including GV) was diagnosed at necropsy in 1 patient.…”

Section: Resultsmentioning

confidence: 99%

“…Among GB-SOV patients, only 3 received glucocorticoids for 6 to 16 weeks, 15 , 37 whereas all GB-SV patients were treated with glucocorticoids (p = 0.001),; 4 , 5 , 9 , 10 , 12 , 14 , 16 – 18 , 20 , 22 , 23 , 28 , 31 , 32 , 36 , 38 , 40 , 42 , 43 , 46 – 48 , 54 , 58 , 60 , 61 54% of patients with SV also received cytotoxic agents 5 , 12 , 14 , 16 , 17 , 22 , 23 , 31 , 36 , 40 , 42 , 46 , 48 , 58 , 60 (see Table 2 ). Among GB-SV patients with adequate information, 17/20 patients were receiving glucocorticoid therapy at the end of follow-up.…”

Section: Resultsmentioning

confidence: 99%

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Single-Organ Gallbladder Vasculitis

et al. 2014

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Systemic vasculitis (SV) involving abdominal structures usually has a poor prognosis. Gallbladder vasculitis (GV) has been reported as part of SV (GB-SV) and focal single-organ vasculitis (GB-SOV). We analyzed clinical and histologic characteristics of patients with GV to identify features that differentiate GB-SOV from the systemic forms of GV. To identify affected patients with GV we used pathology databases from our institution and an English-language PubMed search. Clinical manifestations, laboratory and histologic features, treatment administered, and outcomes were recorded. Patients were divided in 2 groups, GB-SOV and GB-SV. As in previous studies of single-organ vasculitis, GB-SOV was only considered to be a sustainable diagnosis if disease beyond the gallbladder was not apparent after a follow-up period of at least 6 months. Sixty-one well-characterized patients with GV were included (6 from our institution). There was no significant sex bias (32 female patients, 29 male). Median age was 52 years (range, 18–94 yr). GB-SOV was found in 20 (33%) and GB-SV in 41 (67%) patients. No differences were observed in age, sex frequency, or duration of gallbladder symptoms between groups. Past episodes of recurrent right-upper quadrant or abdominal pain and lithiasic cholecystitis were more frequent in GB-SOV patients, whereas acalculous cholecystitis occurred more often in GB-SV. In GB-SV, gallbladder-related symptoms occurred more often concomitantly with or after the systemic features, but they sometimes appeared before SV was fully developed (13.5%). Constitutional and musculoskeletal symptoms were reported only in GB-SV patients. Compared to GB-SOV, GB-SV patients presented more often with fever (62.5% vs 20%; p = 0.003) and exhibited higher erythrocyte sedimentation rate levels (80 ± 28 vs 37 ± 25 mm/h, respectively; p = 0.006). All GB-SV patients required glucocorticoids and 50% of them also received cytotoxic agents. Mortality in GB-SV was higher than in GB-SOV (35.5% vs 10%; p = 0.05). Nongranulomatous inflammation with fibrinoid necrosis of medium-sized vessels occurred equally in both groups (>90%). Forms of SV affecting the gallbladder included polyarteritis nodosa (n = 10), hepatitis B virus-associated vasculitis (n = 8), cryoglobulinemic (essential or hepatitis C virus-associated) vasculitis (n = 6), vasculitis associated with autoimmune diseases (n = 6), microscopic polyangiitis (n = 4), eosinophilic granulomatosis with polyangiitis (Churg-Strauss) (n = 4), IgA vasculitis (Henoch-Schönlein) (n = 2), and giant cell arteritis (n = 1).GV is uncommon. Its histology most often consists of a nongranulomatous necrotizing vasculitis affecting medium-sized vessels. GB-SOV is usually discovered after routine cholecystectomy performed because of the presence of local symptoms, gallstone-associated cholecystitis, and contrary to GB-SV, GB-SOV is usually not associated with systemic symptoms. Acute phase reactants and surrogate markers of autoimmunity are usually normal or negative in GB-SOV. GB-SOV does n...

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

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Single-Organ Gallbladder Vasculitis

et al. 2014

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Acalculous ischemic gallbladder necrosis in the catastrophic antiphospholipid syndrome

Dessailloud

Papo

Vaneecloo

et al. 1998

Arthritis & Rheumatism

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A 29-year-old woman was referred for abdominal pain. Results of tests for lupus anticoagulant and antibodies to phosphatidylserine and to P,-glycoprotein I were positive, but the patient had no features of systemic lupus erythematosus (SLE). Abdominal ultrasonography showed a thickening of the gallbladder wall without cholelithiasis. A surgical procedure revealed necrotic areas of the gallbladder wall, and a cholecystectomy was performed. Histologic examination of the gallbladder showed multiple thrombi and no vasculitis. Despite full-dose heparin, the patient developed a catastrophic antiphospholipid syndrome (APS) and subsequently died. Among connective tissue disorders, acute acalculous cholecystitis has been reported in patients with polyarteritis nodosa and/or SLE. APS should be considered as a possible cause of acalculous cholecystitis.The antiphospholipid syndrome (APS) may be responsible for diverse manifestations affecting the liver and/or gut (1,2), but to date, gallbladder involvement has not been described. We describe here a patient who developed acalculous ischemic gallbladder necrosis as a feature of catastrophic APS. CASE REPORTA 29-year-old nulliparous woman was referred for abdominal pain of recent onset. Her medical history was unremarkable. She had been taking an estrogencontaining contraceptive pill for years, and used to smoke 10 cigarettes per day. On cxaniination, she was

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Neuromuscular and Central Nervous System Manifestations of Clostridium perfringens Infections

Finsterer

Hess

2007

Infection

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Infections with Clostridium perfringens usually manifest locally or spread to sepsis with multiorgan involvement, hemolysis or septic shock. Central nervous system (CNS) manifestations are rare and most frequently comprise meningitis with or without pneumencephalon, encephalitis, plexitis, cerebral abscess, or subdural empyema. The course of CNS affections is usually foudroyant and the outcome fatal. Neuromuscular manifestations of C. perfringens infections are much more frequent than CNS manifestations and comprise myonecrosis (gas gangrene), rhabdomyolysis, myositis, fasciitis, affection of the neuromuscular transmission, or affection of the peripheral nerves. C. perfringens infections usually start from the site of a recent surgical wound or trauma, a gastrointestinal or urogenital problem, or occur in association with malignancy. In quite a number of cases the source of origin remains speculative. Treatment of choice is surgical debridement of the infectious focus with radical removal of all necrotic tissue, resection of the corresponding lymphatics in addition to antibiotic therapy with penicillin G, aminoglycosides, or clindamycin or hyperbaric oxygenation. Despite these therapeutic options, the prognosis of CNS and neuromuscular involvement in an infection with C. perfringens is still poor. Only focal infections or clostridial brain abscesses may eventually have a more favorable outcome, if surgery and antibiotics are instantly provided. Generally, early recognition of the infectious agent is of paramount importance to prevent from spreading and the development of severe hemolysis, septic shock, or death.

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Case 42-1989

Cited by 14 publications

References 79 publications

Single-Organ Gallbladder Vasculitis

Single-Organ Gallbladder Vasculitis

Acalculous ischemic gallbladder necrosis in the catastrophic antiphospholipid syndrome

Neuromuscular and Central Nervous System Manifestations of Clostridium perfringens Infections

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