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Emergency splenectomy during gastroenteritis and primary EBV infection
Abstracts / Digestive and Liver Disease 47S (2015) e237–e276
In two patients the disease was refractory to medical and nutritional treatment, hence surgical intervention was required (gastrointestinal bypass with “Roux-en-Y” and gastroenteroanastomosis after temporary jejunostomy in one case and duodenal strictureplasty in the second case). The third case was responsive to medical treatment (steroids, azathioprine, and subsequently infliximab). Despite gastroduodenal localisation of CD is rare, it should be suspected in patients with upper GIT symptoms. An accurate history is important. Medical treatment should be attempted in first instance but surgery may be required in cases of severe obstructive disease unresponsive to the medical approach. http://dx.doi.org/10.1016/j.dld.2015.07.107 P060 MULTICENTRIC STUDY ON AZATHIOPRINE DOSE AND PHARMACOKINETICS IN EARLY-ONSET PEDIATRIC INFLAMMATORY BOWEL DISEASE G. Stocco 1,∗ , S. Martelossi 2 , S. Arrigo 3 , A. Barabino 3 , M. Aloi 4 , M. Martinelli 5 , E. Miele 5 , S. De Iudicibus 2 , D. Favretto 2 , E. Cuzzoni 6 , G. Decorti 1 , A. Ventura 6 1 Department of Life Sciences, University of Trieste, Trieste, Italy 2 Institute for Maternal and Child Health Burlo Garofolo, Trieste, Italy 3 Gastroenterology and Endoscopy Unit, G. Gaslini Institute for Children, Genoa, Italy 4 Pediatric Gastroenterology and Liver Unit, Sapienza University of Rome, Rome, Italy 5 Department of Translational Medical Science, Section of Pediatrics, University of Naples Federico II, Italy 6 Department of Medical and Surgical Sciences, University of Trieste, Trieste, Italy
Introduction: Despite the introduction of highly effective biological drugs, treatment with glucocorticoids and azathioprine continues to be first-choice approach even for early-onset inflammatory bowel disease (IBD); however, azathioprine response is reduced in early-onset IBD. Aim: To evaluate azathioprine doses, metabolites’ concentrations and their associations with patients’ age in children with IBD treated at four tertiary referral centers for pediatric IBD. Methods: Azathioprine doses, metabolites and clinical effects were assessed after at least 3 months of therapy in 18 early-onset (age <8 years) and a control group of 88 late-onset (age >12 and <18 years) pediatric IBD patients. Azathioprine dose was titrated on therapeutic efficacy (response and adverse effects). Clinical efficacy was defined as disease activity score below 10. Results: Frequency of clinical efficacy was not different among early-onset and control group (82.3% and 81.0% respectively; pvalue = 0.90). However, early-onset patients required higher doses of azathioprine (median 2.7 vs 2.0 mg/kg/day, p-value = 0.0002), which resulted in comparable azathioprine active thioguaninenucleotide (TGN) metabolites concentrations (median 257 vs 361 U, p-value = 0.37) and higher methyl-mercaptopurine-nucleotides (MMPN) metabolites in early-onset patients (median 1978 vs 1363 U, p-value = 0.049). Interestingly, early-onset patients presented also higher MMPN/TGN ratio (median 6.2 vs 4.1, p-value = 0.0039). Conclusion: This study confirms previous observations that early-onset IBD patients present an increased inactivating
e259
azathioprine metabolism, putatively through elevated activity of the enzyme thiopurine-S-methyl transferase. http://dx.doi.org/10.1016/j.dld.2015.07.108 P061 WHEN ULCERATIVE COLITIS BECOMES A REAL CHALLENGE TO PHYSICIAN M. Citrano 1,∗ , G.M. Sciarrabone 2 , R. Ganci 1 , M. Violante 1 , N. Cassata 1 1 U.O.C Pediatria, Ospedali Riuniti Villa Sofia-Cervello, Palermo, Italy 2 Dipartimento di Scienze per la Promozione della Salute e Materno-Infantile “G. D’Alessandro”, Università degli studi di Palermo, Italy
Introduction: The acute attack of ulcerative colitis (UC) is a medical emergency. According to scientific evidence steroid therapy is the first line, but sometimes for the establishment of cortico-resistance, it is necessary to undertake second-line treatments with infliximab (IFX). These treatments are able, in most cases, to control the disease. Case report: We present the case of a 7-year-old child suffering from chronic-active pancolitis early-onset (3 years), characterized by cortico-resistance and refractory to treatment with biologics (IFX and adalimumab), undergoing therapy with Thalidomide, with good clinical response. The child, from diagnosis, had a good clinical control of the disease for 4 years. At the onset, he has performed double immunosuppression (cyclosporine and azathioprine) and subsequently continued with azathioprine and mesalazine. However, during the last year, he presented three episodes of severe exacerbation (PUCAI 70), with no response to several lines of treatment (high-dose intravenous steroid, then IFX and ADA). Therefore, the treatment failure should nominate the patient to surgery. Our decision was to use thalidomide (dose 2.5 mg/kg/day) as a last treatment option. The patient is being treated for 8 weeks. The clinical follow-up and laboratory parameters monitoring show an improvement (PUCAI 20). Conclusions: Our case report highlights the aggressive clinical course of ulcerative colitis, especially if early onset, and strengthens the published evidence about the positive impact of thalidomide therapy as a possible therapeutic option to increase the time of need for surgery in patients with cortico-resistance and refractory to biological therapy. http://dx.doi.org/10.1016/j.dld.2015.07.109 P062 EMERGENCY SPLENECTOMY DURING GASTROENTERITIS AND PRIMARY EBV INFECTION L. Balanzoni ∗ , E. Battisti, A.M. Colombari, G. Muscolino, C. Voltolina, Y. Suzuki, A. Piccoli, S. Bonetti, S. Vecchini, M. Salani, M. Cinquetti U.O.C. Pediatria, Osp. G. Fracastoro, S.Bonifacio, Verona, Italy We describe the case of a 11 year-old girl with splenic rupture in the course of an acute EBV infection. The patient is brought to the Emergency Department with dehydration secondary to vomit and diarrhoea; when she arrives she has already undergone a 4 days
e260
Abstracts / Digestive and Liver Disease 47S (2015) e237–e276
treatment with Amoxicillin/Clavulanic acid for pharyngotonsillitis with fever. Treated with parenteral rehydration and intravenous Ceftriaxone, in the hospital the patient at first shows a clinical improvement, but afterwards complaints with abdominal pain especially in the right iliac fossa, with reappearance of vomit and diarrhoea. Serial ultrasounds show hyperechoic effusion in the right iliac fossa. Blood tests reveal mild anemia and low platelet count. Due to abdominal tension she undergoes laparotomy which shows massive splenic infarction with capsular laceration and hemoperitoneum. Splenectomy is performed. Serology is consistent with EBV primary infection. We described this case with the aim of underlining the possibility (as already referred by other authors in anecdotal cases) of a splenic rupture in patients with infectious mononucleosis with gastroenteritis. This event needs to be treated in a setting of emergency surgery. http://dx.doi.org/10.1016/j.dld.2015.07.110 P063 A RECURRENT ILEITIS B. Parma ∗ , M.T. Ortisi, R. Picchi, V. Chierici, L. Abbagnato U.O. Pediatria. Azienda Ospedaliera Sant’Anna, Como, Italy Objective: To raise awareness about a rare infectious cause of ileitis. Methods: Description of a clinical case. Case report: A 16 years old boy was operated for appendicitis with cecal abscess. 2 months later he presented low-grade fever and abdominal pain. Regular bowel function. Examinations: High values of inflammatory index. An abdominal ultrasound (US) showed an ileitis and a cecal abscess. EnteroMRI confirmed the US picture. Calprotectin was slightly elevated without blood. Colonscopy evinced only a lymphoid nodular hyperplasia. He was subjected to 21 days (d.) of cephalosporin with biochemical and MRI negativization. After one month there was a resumption of symptoms with increase of inflammatory index. US showed again an ileocecal inflammatory activity with cecal abscess. The coproculture documented an Aereomonas spp (hydrophila/ caviae). It was set therapy with gentamicin for 10 d. and ciprofloxacin for 28 d. with negativization of the US and EnteroMRI picture. Discussion: Aereomonas spp. are ubiquitous inhabitants of fresh, brackish water, and also of tap water. 3 species (hydrophila, caviae, veronii) are major associated with human diseases. They are, Gram-negative, principally associated with gastroenteritis, soft tissue infection and, even if uncommon, with sepsis, intrabdominal abscess and peritonitis. Aeromonas spp. are resistant to penicillin and ampicillin, usually but not invariably susceptible to third generation cephalosporins and carbapenems. Aminoglicosides and fluoroquinolones are usually active.
Conclusions: It is important to consider Aeromonas spp. as a potential etiology of recurrent ileitis, appendicitis and cecal abscess, particularly in people who may assume unchecked water. http://dx.doi.org/10.1016/j.dld.2015.07.111 PO64 ENVIRONMENTAL RISK FACTORS IN PEDIATRIC IBD ACCORDING TO THE HYGIENE HYPOTHESIS: A CASE CONTROL STUDY C. Strisciuglio 1,∗ , F.P. Giugliano 2 , M. Martinelli 2 , L. Greco 2 , S. Cenni 2 , A. Staiano 2 , E. Miele 2 1 Department of Women and Children and General and Specialized Surgery, Seconda Università degli Studi di Napoli, 80138 Naples, Italy 2 Department of Translational and Medical Science, Section of Pediatrics, University Federico II, Naples, Italy
Objectives and study: The rapid spread in Inflammatory Bowel Disease (IBD) incidence over the past century cannot be accounted by genetic predisposition alone. We investigated the impact of the emerging “Hygiene Hypothesis” in an IBD pediatric cohort to find a correlation between the exposure to environmental factors and the risk of IBD. Methods: 663 pediatric subjects were enrolled, among these 262 were IBD patients, 230 were IBD healthy siblings and 171 were age- and sex-matched healthy controls. All patients underwent a questionnaire including 5 groups of potential environmental IBD risk factors: family history of IBD, perinatal period, home amenities, childhood diseases, diet. Results: Family history of IBD and autoimmune diseases were strong risk factors for developing Crohn’s disease (CD) (OR: 9.494 and 2.442) and Ulcerative Colitis (UC) (OR: 20.667 and 2.875). In both IBD groups, an higher number of family stressing events (OR: 1.624 and 2.290) and an increased number of toilets (OR: 1.733 and 2.167) were found to be more frequent compared to controls. Breastfeeding >3 months was a significant risk factor for CD and UC (OR: 2.701 and 1.523), as well as gluten introduction before the 6th month of age (OR: 2.834 and 2.992) and low adherence to Mediterranean diet (OR: 1.998 and 2.895). In IBD groups, a lower gestational age, a higher number of siblings, pet owing and family history for Intestinal Parasitosis and Helicobacter Pylori were protective factors. Conclusion: Our work confirms that environmental factors are closely linked to IBD. Prospective studies are necessary to validate these results in order to offer possible interventions for IBD prevention in genetically predisposed individuals. http://dx.doi.org/10.1016/j.dld.2015.07.112
In two patients the disease was refractory to medical and nutritional treatment, hence surgical intervention was required (gastrointestinal bypass with “Roux-en-Y” and gastroenteroanastomosis after temporary jejunostomy in one case and duodenal strictureplasty in the second case). The third case was responsive to medical treatment (steroids, azathioprine, and subsequently infliximab). Despite gastroduodenal localisation of CD is rare, it should be suspected in patients with upper GIT symptoms. An accurate history is important. Medical treatment should be attempted in first instance but surgery may be required in cases of severe obstructive disease unresponsive to the medical approach. http://dx.doi.org/10.1016/j.dld.2015.07.107 P060 MULTICENTRIC STUDY ON AZATHIOPRINE DOSE AND PHARMACOKINETICS IN EARLY-ONSET PEDIATRIC INFLAMMATORY BOWEL DISEASE G. Stocco 1,∗ , S. Martelossi 2 , S. Arrigo 3 , A. Barabino 3 , M. Aloi 4 , M. Martinelli 5 , E. Miele 5 , S. De Iudicibus 2 , D. Favretto 2 , E. Cuzzoni 6 , G. Decorti 1 , A. Ventura 6 1 Department of Life Sciences, University of Trieste, Trieste, Italy 2 Institute for Maternal and Child Health Burlo Garofolo, Trieste, Italy 3 Gastroenterology and Endoscopy Unit, G. Gaslini Institute for Children, Genoa, Italy 4 Pediatric Gastroenterology and Liver Unit, Sapienza University of Rome, Rome, Italy 5 Department of Translational Medical Science, Section of Pediatrics, University of Naples Federico II, Italy 6 Department of Medical and Surgical Sciences, University of Trieste, Trieste, Italy
Introduction: Despite the introduction of highly effective biological drugs, treatment with glucocorticoids and azathioprine continues to be first-choice approach even for early-onset inflammatory bowel disease (IBD); however, azathioprine response is reduced in early-onset IBD. Aim: To evaluate azathioprine doses, metabolites’ concentrations and their associations with patients’ age in children with IBD treated at four tertiary referral centers for pediatric IBD. Methods: Azathioprine doses, metabolites and clinical effects were assessed after at least 3 months of therapy in 18 early-onset (age <8 years) and a control group of 88 late-onset (age >12 and <18 years) pediatric IBD patients. Azathioprine dose was titrated on therapeutic efficacy (response and adverse effects). Clinical efficacy was defined as disease activity score below 10. Results: Frequency of clinical efficacy was not different among early-onset and control group (82.3% and 81.0% respectively; pvalue = 0.90). However, early-onset patients required higher doses of azathioprine (median 2.7 vs 2.0 mg/kg/day, p-value = 0.0002), which resulted in comparable azathioprine active thioguaninenucleotide (TGN) metabolites concentrations (median 257 vs 361 U, p-value = 0.37) and higher methyl-mercaptopurine-nucleotides (MMPN) metabolites in early-onset patients (median 1978 vs 1363 U, p-value = 0.049). Interestingly, early-onset patients presented also higher MMPN/TGN ratio (median 6.2 vs 4.1, p-value = 0.0039). Conclusion: This study confirms previous observations that early-onset IBD patients present an increased inactivating
e259
azathioprine metabolism, putatively through elevated activity of the enzyme thiopurine-S-methyl transferase. http://dx.doi.org/10.1016/j.dld.2015.07.108 P061 WHEN ULCERATIVE COLITIS BECOMES A REAL CHALLENGE TO PHYSICIAN M. Citrano 1,∗ , G.M. Sciarrabone 2 , R. Ganci 1 , M. Violante 1 , N. Cassata 1 1 U.O.C Pediatria, Ospedali Riuniti Villa Sofia-Cervello, Palermo, Italy 2 Dipartimento di Scienze per la Promozione della Salute e Materno-Infantile “G. D’Alessandro”, Università degli studi di Palermo, Italy
Introduction: The acute attack of ulcerative colitis (UC) is a medical emergency. According to scientific evidence steroid therapy is the first line, but sometimes for the establishment of cortico-resistance, it is necessary to undertake second-line treatments with infliximab (IFX). These treatments are able, in most cases, to control the disease. Case report: We present the case of a 7-year-old child suffering from chronic-active pancolitis early-onset (3 years), characterized by cortico-resistance and refractory to treatment with biologics (IFX and adalimumab), undergoing therapy with Thalidomide, with good clinical response. The child, from diagnosis, had a good clinical control of the disease for 4 years. At the onset, he has performed double immunosuppression (cyclosporine and azathioprine) and subsequently continued with azathioprine and mesalazine. However, during the last year, he presented three episodes of severe exacerbation (PUCAI 70), with no response to several lines of treatment (high-dose intravenous steroid, then IFX and ADA). Therefore, the treatment failure should nominate the patient to surgery. Our decision was to use thalidomide (dose 2.5 mg/kg/day) as a last treatment option. The patient is being treated for 8 weeks. The clinical follow-up and laboratory parameters monitoring show an improvement (PUCAI 20). Conclusions: Our case report highlights the aggressive clinical course of ulcerative colitis, especially if early onset, and strengthens the published evidence about the positive impact of thalidomide therapy as a possible therapeutic option to increase the time of need for surgery in patients with cortico-resistance and refractory to biological therapy. http://dx.doi.org/10.1016/j.dld.2015.07.109 P062 EMERGENCY SPLENECTOMY DURING GASTROENTERITIS AND PRIMARY EBV INFECTION L. Balanzoni ∗ , E. Battisti, A.M. Colombari, G. Muscolino, C. Voltolina, Y. Suzuki, A. Piccoli, S. Bonetti, S. Vecchini, M. Salani, M. Cinquetti U.O.C. Pediatria, Osp. G. Fracastoro, S.Bonifacio, Verona, Italy We describe the case of a 11 year-old girl with splenic rupture in the course of an acute EBV infection. The patient is brought to the Emergency Department with dehydration secondary to vomit and diarrhoea; when she arrives she has already undergone a 4 days
e260
Abstracts / Digestive and Liver Disease 47S (2015) e237–e276
treatment with Amoxicillin/Clavulanic acid for pharyngotonsillitis with fever. Treated with parenteral rehydration and intravenous Ceftriaxone, in the hospital the patient at first shows a clinical improvement, but afterwards complaints with abdominal pain especially in the right iliac fossa, with reappearance of vomit and diarrhoea. Serial ultrasounds show hyperechoic effusion in the right iliac fossa. Blood tests reveal mild anemia and low platelet count. Due to abdominal tension she undergoes laparotomy which shows massive splenic infarction with capsular laceration and hemoperitoneum. Splenectomy is performed. Serology is consistent with EBV primary infection. We described this case with the aim of underlining the possibility (as already referred by other authors in anecdotal cases) of a splenic rupture in patients with infectious mononucleosis with gastroenteritis. This event needs to be treated in a setting of emergency surgery. http://dx.doi.org/10.1016/j.dld.2015.07.110 P063 A RECURRENT ILEITIS B. Parma ∗ , M.T. Ortisi, R. Picchi, V. Chierici, L. Abbagnato U.O. Pediatria. Azienda Ospedaliera Sant’Anna, Como, Italy Objective: To raise awareness about a rare infectious cause of ileitis. Methods: Description of a clinical case. Case report: A 16 years old boy was operated for appendicitis with cecal abscess. 2 months later he presented low-grade fever and abdominal pain. Regular bowel function. Examinations: High values of inflammatory index. An abdominal ultrasound (US) showed an ileitis and a cecal abscess. EnteroMRI confirmed the US picture. Calprotectin was slightly elevated without blood. Colonscopy evinced only a lymphoid nodular hyperplasia. He was subjected to 21 days (d.) of cephalosporin with biochemical and MRI negativization. After one month there was a resumption of symptoms with increase of inflammatory index. US showed again an ileocecal inflammatory activity with cecal abscess. The coproculture documented an Aereomonas spp (hydrophila/ caviae). It was set therapy with gentamicin for 10 d. and ciprofloxacin for 28 d. with negativization of the US and EnteroMRI picture. Discussion: Aereomonas spp. are ubiquitous inhabitants of fresh, brackish water, and also of tap water. 3 species (hydrophila, caviae, veronii) are major associated with human diseases. They are, Gram-negative, principally associated with gastroenteritis, soft tissue infection and, even if uncommon, with sepsis, intrabdominal abscess and peritonitis. Aeromonas spp. are resistant to penicillin and ampicillin, usually but not invariably susceptible to third generation cephalosporins and carbapenems. Aminoglicosides and fluoroquinolones are usually active.
Conclusions: It is important to consider Aeromonas spp. as a potential etiology of recurrent ileitis, appendicitis and cecal abscess, particularly in people who may assume unchecked water. http://dx.doi.org/10.1016/j.dld.2015.07.111 PO64 ENVIRONMENTAL RISK FACTORS IN PEDIATRIC IBD ACCORDING TO THE HYGIENE HYPOTHESIS: A CASE CONTROL STUDY C. Strisciuglio 1,∗ , F.P. Giugliano 2 , M. Martinelli 2 , L. Greco 2 , S. Cenni 2 , A. Staiano 2 , E. Miele 2 1 Department of Women and Children and General and Specialized Surgery, Seconda Università degli Studi di Napoli, 80138 Naples, Italy 2 Department of Translational and Medical Science, Section of Pediatrics, University Federico II, Naples, Italy
Objectives and study: The rapid spread in Inflammatory Bowel Disease (IBD) incidence over the past century cannot be accounted by genetic predisposition alone. We investigated the impact of the emerging “Hygiene Hypothesis” in an IBD pediatric cohort to find a correlation between the exposure to environmental factors and the risk of IBD. Methods: 663 pediatric subjects were enrolled, among these 262 were IBD patients, 230 were IBD healthy siblings and 171 were age- and sex-matched healthy controls. All patients underwent a questionnaire including 5 groups of potential environmental IBD risk factors: family history of IBD, perinatal period, home amenities, childhood diseases, diet. Results: Family history of IBD and autoimmune diseases were strong risk factors for developing Crohn’s disease (CD) (OR: 9.494 and 2.442) and Ulcerative Colitis (UC) (OR: 20.667 and 2.875). In both IBD groups, an higher number of family stressing events (OR: 1.624 and 2.290) and an increased number of toilets (OR: 1.733 and 2.167) were found to be more frequent compared to controls. Breastfeeding >3 months was a significant risk factor for CD and UC (OR: 2.701 and 1.523), as well as gluten introduction before the 6th month of age (OR: 2.834 and 2.992) and low adherence to Mediterranean diet (OR: 1.998 and 2.895). In IBD groups, a lower gestational age, a higher number of siblings, pet owing and family history for Intestinal Parasitosis and Helicobacter Pylori were protective factors. Conclusion: Our work confirms that environmental factors are closely linked to IBD. Prospective studies are necessary to validate these results in order to offer possible interventions for IBD prevention in genetically predisposed individuals. http://dx.doi.org/10.1016/j.dld.2015.07.112