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YOUNG FEMALE WITH ACTIVE ULCERATIVE COLITIS (UC) AND CEREBRAL SINUS THROMBOSIS: CLINICAL PRESENTATION AND LITERATURE REVIEW
S138
Abstracts / Digestive and Liver Disease 41S (2009), S1–S167
of mild to moderately active ulcerative colitis (UC) and in relapse prevention. In vivo metabolism of 5-ASA produces two hydroxylated derivatives (2,3- and 2,5-DHBA). 2,3-DHBA can be formed by direct hydroxyl radical attack. thus appearing a useful marker of in vivo OH. production. The aim was to evaluate the mucosal concentration of both 5-ASA and 2,3-DHBA in UC patients with active and non-active disease. Material and methods: The study included 130 consecutive ulcerative colitis patients (mean age 48.5, range 23-84; 62 males and 38 females) on continuous oral 5-ASA treatment (2.4g/day). After informed consent, patients undergoing colonoscopy had two biopsies taken from the sigmoid region (25 cm from the anal verge) for conventional histology and 5-ASA concentration. Endoscopic and histological disease activity were recorded as remission or active disease. 5-ASA (ng/mg) and 2,3DHBA (pg/mg) concentrations were measured in tissue homogenates by high-pressure liquid chromatography equipped with electrochemical detector. The t-test was used for statistical analysis. Results: Patients with endoscopic remission showed higher concentrations of mucosal 5-ASA than those with a mild to moderately active endoscopic disease (58.4±5.9 vs. 42.6±5.2, p=0.04). Similarly, mucosal 5-ASA concentrations in patients with normal histology were higher than in those with active histological inflammation (67.04±7.11 vs. 39.6±4.4, p=0.001). Conversely, 2,3–DHBA mucosal levels were significantly lower in patients in remission with respect to patients with active disease both endoscopically and histologically (0.6±0.1 vs. 4.9±1.9, p=0.03 and 0.6±0.1 vs. 3.6±1.3 p=0.03, respectively). Conclusions: Patients with active ulcerative colitis show lower 5-ASA concentration in the colonic mucosa while its hydroxylation product increases. The determination of 2,3-DHBA in the colonic mucosa may be suggested as a novel marker of inflammation-derived oxidative damage. # L. Inflammatory bowel diseases 3. Ulcerative colitis
P.171 YOUNG FEMALE WITH ACTIVE ULCERATIVE COLITIS (UC) AND CEREBRAL SINUS THROMBOSIS: CLINICAL PRESENTATION AND LITERATURE REVIEW A. Zelante ∗ , L. Simone, N. Fusetti, A. Carella, M. Grazia, S. Gullini Azienda Ospedaliero-Universitaria Sant’Anna, Ferrara Background and aim: Extraintestinal manifestations are common in UC with a life-time prevalence of 25-40%.The risk of thromboembolism is well-known in young patients, particularly of deep venous thrombosis and pulmonary embolism; cerebral vascular involvement is rare. Material and methods: A 23-year old female with 3-year history of UC diagnosed in 2005, initially controlled disease with corticosteroids i.v. then with azathioprine 100mg/day. She had a history of recurrent hemicrania and she was in therapy with oral contraceptives with a normal coagulation profile (including S and C proteins). In June 2008, she developed bloody diarrhoea (7 stools/day) and intense abdominal pain not relieved by beclometasone enemas. Results: She was admitted to day hospital, starting corticosteroids (1mg/kg i.v.) and ciprofloxacin (1000mg/day) for a concomitant lower urinary tract infection. After 4 days she referred a mild headache responsive to paracetamol; neurologic examination was negative. After 8 days, she suddenly developed upper limbs paraesthesias and intense headache. Within a few hours she fell into a comatose state with elevated XDP 1636 ng/ml; WBC 6.70×103 /μl; Hb 9.9 g/dl; PLTS 146×103 /μl; INR 1.21; RCP 3.00 mg/dl. Cerebral TC and angio-MRI revealed thrombosis of the right transverse and sagittal superior sinus with widespread haemorrhagic edges. She was transferred to Intensive Care Unit, submitted to a decompressing craniotomy without clinical improvement. Laboratory tests revealed platelets consumption (plts
69×103 /μl), elevated INR (1.77) and XDP (5471 ng/ml) with normal fibrinogen value (412 mg/dl). Neuroimaging described increased haemorrhagic areas resulting in irreversible cerebral compression and patient died. Conclusions: A systematic literature search regarding similar clinical presentation and neuroimaging revealed only few case reports. The sudden onset of neurologic symptoms in IBD, even mild, need immediate neurological examination with CT if necessary. In female patients with UC, the use of oral contraceptives has to be reconsidered in association with corticosteroid therapy. UC might have a pathogenetic association with thrombo-embolism although the mechanism of the procoagulant activity is unclear. Elevated risk patients (female with severe disease, contraceptive therapy, etc) should be informed about thrombotic manifestations. # L. Inflammatory bowel diseases 3. Ulcerative colitis
P.172 PULMONARY EMBOLISM IN SEVERE STEROID RESISTANT ULCERATIVE COLITIS: RESCUE THERAPY WITH INFLIXIMAB G. Forti ∗ , G. Pianese, N. Duranti, A. Sgro, R. Masala S. Maria Goretti Hospital, Latina Background and aim: A serious and potentially fatal complication of UC (ulcerative colitis) is pulmonary embolism resulting from thrombosis of the leg or pelvic veins. Case Report: a 45 ys. woman admitted to Hospital in March ’07 for DVT (deep venous thrombosis) of iliac-femoral axis and long standing UC. Material and methods: A CT scan revealed left renal vein and right pulmonary artery thrombosis without involvement of inferior vena cava. The woman also showed failure of clinical conditions for worsening of long standing UC with >10 stool movements a day, hypoalbuminemia, anoemia (Hb <7.5 g/dl) and weigt loss (UCDAI>8). There were no haemostatic values alteration nor presence of autoantibodies neither presence of cancer markers. Because of higher risk for colonic bleeding, no anticoagulation therapy was started, instead an “umbrella” was percutaneously inserted into the inferior vena cava. A following colonoscopy confirmed severe UC. No improvement of general conditions despite of TPN (total parenteral nutrition), i.v. steroid medication (prednisone 80 mg), mesalazine (4.8 g tid),and i.v. antibiotics (ciprofloxacin). Results: A first rescue therapy was performed by the use of cyclosporin i.v. (2.5 mg per kg.) for 7 day without any improvement of pt. clinical status. A further rescue therapy was started by using of anti-TNFα antibody (infliximab) (5 mg per kg.) i.v. at 0,2 and 6 weeks with progressive reduction of steroid amount. There was a fast improvement of clinical conditions with decrease of UCDAI (<6) and keeping of kidney functionality. A following CT scan showed decrease of DVT, still current after 2 months. The improved clinical status allowed starting therapy with LMWH (low molecular weigth heparin) and the removal after 63 days of inferior vena cava filter. The woman was discharged in good general conditions, decrease of UCDAI, Hb 10g/dl, and recovery of body weigth. She is still attending maintenance therapy with infliximab every 8 weeks, no thrombocitosis appearance, no steroids intake and UCDAI <3. Conclusions: Thromboembolism is a not rare extraintestinal complication of severe UC with mortality rate of 30%. For this reason a prophilactic therapy with LMWH could be reasonable in severe UC pts. Use of infliximab, just well evaluated for mucosal healing and improvement of steroid resistant UC, could be evaluated to improve and/or to prevent extraintestinal manifestations of severe UC. # L. Inflammatory bowel diseases 3. Ulcerative colitis
Abstracts / Digestive and Liver Disease 41S (2009), S1–S167
of mild to moderately active ulcerative colitis (UC) and in relapse prevention. In vivo metabolism of 5-ASA produces two hydroxylated derivatives (2,3- and 2,5-DHBA). 2,3-DHBA can be formed by direct hydroxyl radical attack. thus appearing a useful marker of in vivo OH. production. The aim was to evaluate the mucosal concentration of both 5-ASA and 2,3-DHBA in UC patients with active and non-active disease. Material and methods: The study included 130 consecutive ulcerative colitis patients (mean age 48.5, range 23-84; 62 males and 38 females) on continuous oral 5-ASA treatment (2.4g/day). After informed consent, patients undergoing colonoscopy had two biopsies taken from the sigmoid region (25 cm from the anal verge) for conventional histology and 5-ASA concentration. Endoscopic and histological disease activity were recorded as remission or active disease. 5-ASA (ng/mg) and 2,3DHBA (pg/mg) concentrations were measured in tissue homogenates by high-pressure liquid chromatography equipped with electrochemical detector. The t-test was used for statistical analysis. Results: Patients with endoscopic remission showed higher concentrations of mucosal 5-ASA than those with a mild to moderately active endoscopic disease (58.4±5.9 vs. 42.6±5.2, p=0.04). Similarly, mucosal 5-ASA concentrations in patients with normal histology were higher than in those with active histological inflammation (67.04±7.11 vs. 39.6±4.4, p=0.001). Conversely, 2,3–DHBA mucosal levels were significantly lower in patients in remission with respect to patients with active disease both endoscopically and histologically (0.6±0.1 vs. 4.9±1.9, p=0.03 and 0.6±0.1 vs. 3.6±1.3 p=0.03, respectively). Conclusions: Patients with active ulcerative colitis show lower 5-ASA concentration in the colonic mucosa while its hydroxylation product increases. The determination of 2,3-DHBA in the colonic mucosa may be suggested as a novel marker of inflammation-derived oxidative damage. # L. Inflammatory bowel diseases 3. Ulcerative colitis
P.171 YOUNG FEMALE WITH ACTIVE ULCERATIVE COLITIS (UC) AND CEREBRAL SINUS THROMBOSIS: CLINICAL PRESENTATION AND LITERATURE REVIEW A. Zelante ∗ , L. Simone, N. Fusetti, A. Carella, M. Grazia, S. Gullini Azienda Ospedaliero-Universitaria Sant’Anna, Ferrara Background and aim: Extraintestinal manifestations are common in UC with a life-time prevalence of 25-40%.The risk of thromboembolism is well-known in young patients, particularly of deep venous thrombosis and pulmonary embolism; cerebral vascular involvement is rare. Material and methods: A 23-year old female with 3-year history of UC diagnosed in 2005, initially controlled disease with corticosteroids i.v. then with azathioprine 100mg/day. She had a history of recurrent hemicrania and she was in therapy with oral contraceptives with a normal coagulation profile (including S and C proteins). In June 2008, she developed bloody diarrhoea (7 stools/day) and intense abdominal pain not relieved by beclometasone enemas. Results: She was admitted to day hospital, starting corticosteroids (1mg/kg i.v.) and ciprofloxacin (1000mg/day) for a concomitant lower urinary tract infection. After 4 days she referred a mild headache responsive to paracetamol; neurologic examination was negative. After 8 days, she suddenly developed upper limbs paraesthesias and intense headache. Within a few hours she fell into a comatose state with elevated XDP 1636 ng/ml; WBC 6.70×103 /μl; Hb 9.9 g/dl; PLTS 146×103 /μl; INR 1.21; RCP 3.00 mg/dl. Cerebral TC and angio-MRI revealed thrombosis of the right transverse and sagittal superior sinus with widespread haemorrhagic edges. She was transferred to Intensive Care Unit, submitted to a decompressing craniotomy without clinical improvement. Laboratory tests revealed platelets consumption (plts
69×103 /μl), elevated INR (1.77) and XDP (5471 ng/ml) with normal fibrinogen value (412 mg/dl). Neuroimaging described increased haemorrhagic areas resulting in irreversible cerebral compression and patient died. Conclusions: A systematic literature search regarding similar clinical presentation and neuroimaging revealed only few case reports. The sudden onset of neurologic symptoms in IBD, even mild, need immediate neurological examination with CT if necessary. In female patients with UC, the use of oral contraceptives has to be reconsidered in association with corticosteroid therapy. UC might have a pathogenetic association with thrombo-embolism although the mechanism of the procoagulant activity is unclear. Elevated risk patients (female with severe disease, contraceptive therapy, etc) should be informed about thrombotic manifestations. # L. Inflammatory bowel diseases 3. Ulcerative colitis
P.172 PULMONARY EMBOLISM IN SEVERE STEROID RESISTANT ULCERATIVE COLITIS: RESCUE THERAPY WITH INFLIXIMAB G. Forti ∗ , G. Pianese, N. Duranti, A. Sgro, R. Masala S. Maria Goretti Hospital, Latina Background and aim: A serious and potentially fatal complication of UC (ulcerative colitis) is pulmonary embolism resulting from thrombosis of the leg or pelvic veins. Case Report: a 45 ys. woman admitted to Hospital in March ’07 for DVT (deep venous thrombosis) of iliac-femoral axis and long standing UC. Material and methods: A CT scan revealed left renal vein and right pulmonary artery thrombosis without involvement of inferior vena cava. The woman also showed failure of clinical conditions for worsening of long standing UC with >10 stool movements a day, hypoalbuminemia, anoemia (Hb <7.5 g/dl) and weigt loss (UCDAI>8). There were no haemostatic values alteration nor presence of autoantibodies neither presence of cancer markers. Because of higher risk for colonic bleeding, no anticoagulation therapy was started, instead an “umbrella” was percutaneously inserted into the inferior vena cava. A following colonoscopy confirmed severe UC. No improvement of general conditions despite of TPN (total parenteral nutrition), i.v. steroid medication (prednisone 80 mg), mesalazine (4.8 g tid),and i.v. antibiotics (ciprofloxacin). Results: A first rescue therapy was performed by the use of cyclosporin i.v. (2.5 mg per kg.) for 7 day without any improvement of pt. clinical status. A further rescue therapy was started by using of anti-TNFα antibody (infliximab) (5 mg per kg.) i.v. at 0,2 and 6 weeks with progressive reduction of steroid amount. There was a fast improvement of clinical conditions with decrease of UCDAI (<6) and keeping of kidney functionality. A following CT scan showed decrease of DVT, still current after 2 months. The improved clinical status allowed starting therapy with LMWH (low molecular weigth heparin) and the removal after 63 days of inferior vena cava filter. The woman was discharged in good general conditions, decrease of UCDAI, Hb 10g/dl, and recovery of body weigth. She is still attending maintenance therapy with infliximab every 8 weeks, no thrombocitosis appearance, no steroids intake and UCDAI <3. Conclusions: Thromboembolism is a not rare extraintestinal complication of severe UC with mortality rate of 30%. For this reason a prophilactic therapy with LMWH could be reasonable in severe UC pts. Use of infliximab, just well evaluated for mucosal healing and improvement of steroid resistant UC, could be evaluated to improve and/or to prevent extraintestinal manifestations of severe UC. # L. Inflammatory bowel diseases 3. Ulcerative colitis