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fever and recent-onset purpuric rash&#46; On admission she was restless&#44; tachycardic&#44; hypertensive and had abdominal distension and hyperesthesia&#46; A purpuric rash was also evident and limited to the buttocks&#44; lower limbs and feet&#44; and she also presented with tender bilateral edema &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#41;&#46; Initial blood tests showed leukocytosis&#44; elevated acute-phase reactants&#44; mild anemia&#44; abnormal renal function&#44; thrombocytopenia&#44; and abnormal thromboplastin and prothrombin time&#46; Urinalysis revealed the presence of hematuria and nephrotic proteinuria&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Blood cultures were performed and ceftriaxone was started empirically&#46; Abdominal ultrasound showed splenomegaly as well as small-to-medium sized hypoechogenic lesions in the spleen&#44; liver and both kidneys&#44; compatible with thromboembolic infarcts&#46; Due to possible infective embolic disease she was referred for cardiac assessment&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">On examination&#44; a low-grade &#40;II&#47;VI&#41; systolic murmur was detected in the lower left sternal border&#44; which radiated to the back&#46; Echocardiogram revealed coarctation of the aorta at the isthmus with a systolic gradient of 60 mmHg with diastolic run-off&#46; There appeared to be a movable filament-like structure adhering to the coarcted isthmus&#46; No valvular or intracardiac vegetations were found&#46; In order to clarify these findings&#44; a transesophageal echocardiogram &#40;TOE&#41; was carried out&#44; which showed the presence of a hyperechogenic movable &#8220;spur&#8221; at the aortic isthmus&#46; A presumptive diagnosis of infective aortic endarteritis complicated by systemic embolization&#44; hypertension and acute renal insufficiency was made&#46; Vancomycin&#44; titrated to the glomerular filtration rate&#44; was added to the antibiotic regimen&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Staphylococcus schleiferi</span> was later identified as the causative agent of the infective endarteritis&#46; This organism is easily mistaken for <span class="elsevierStyleItalic">Staphylococcus aureus</span>&#46; Antibacterial sensitivity testing showed that this agent was more sensitive to floxacillin and&#44; as such&#44; the latter was added to the therapeutic regimen and vancomycin was stopped&#46; The patient became apyretic on day 5 post-admission and subsequent blood cultures were negative&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Blood pressure control was achieved with bisoprolol and nifedipine&#44; after which the transcoarctation systolic gradient was reduced to 40 mmHg&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">At week 2 post-admission&#44; the clinical course was complicated by sudden onset of gastrointestinal &#40;GI&#41; bleeding &#40;Hb 7&#46;4 g&#47;dl&#41; leading to disseminated intravascular coagulopathy&#44; requiring plasma and red blood cell transfusions&#46; Normalization of hematological parameters was eventually achieved&#44; and total parenteral nutrition was required for 10 days&#46; Despite adequate diuresis under furosemide and spironolactone&#44; worsening renal function ensued&#44; which was characterized by high creatinine levels&#44; slightly raised urea levels&#44; nephrotic proteinuria and hematuria&#44; and hypoalbuminemia&#46; This was managed with prednisolone and&#44; by week 3&#44; renal function improved and normalized&#46; The patient completed a total of six weeks of intravenous floxacillin and was discharged home on prednisolone&#44; bisoprolol and nifedipine&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Additional imaging studies such as abdominal ultrasound&#44; Computerized Tomography and Magnetic Resonance scans were performed &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0050" class="elsevierStylePara elsevierViewall">In children&#44; aortic endarteritis is an exceedingly rare condition even in the context of congenital heart disease&#44; particularly as a source of systemic embolization&#44; as was the case with our patient&#46; Due to its rarity&#44; no precise incidence is known&#44; with very few cases found in literature&#46; As is the case with endocarditis&#44; endarteritis is a severe complication in a child with a structural heart defect where the mortality rate is &#60;10&#37; if the diagnosis is made early or approximately 25&#37; if made at a later stage&#46; Mortality is mostly due to congestive heart failure or systemic embolization complications&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">The authors wish to highlight this rare form of endarteritis and&#44; in particular&#44; in the context of systemic embolization resulting in multiple organ failure&#46; In this case&#44; endarteritis at the coarctation site explained why systemic embolization was limited to the lower part of the body&#44; 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Case report
Abdominal pain of cardiovascular origin
Dor abdominal de causa cardiovascular
Pedro Epifânio
Autor para correspondência
p.pires.epifanio@gmail.com

Corresponding author.
, Maria Emanuel Amaral, Natália Noronha, Paula Martins, Dina Rodrigues, António Pires, Eduardo Castela
Hospital Pediátrico de Coimbra, Serviço de Cardiologia Pediátrica, Coimbra, Portugal
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Gram-positive bacteria&#44; specifically alpha-hemolytic streptococci&#44; <span class="elsevierStyleItalic">Staphylococcus aureus</span> and coagulase-negative staphylococci&#44; are the most commonly involved bacteria&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">2&#44;3</span></a> Diagnosis is based on the modified Duke criteria&#44; which rely mostly on clinical assessment&#44; echocardiography and blood cultures&#46; Antibacterial treatment should ideally be targeted&#46; However&#44; if no specific bacteria have been identified&#44; patients should promptly be treated empirically with multiple drug regimens based on local resistance and the most common etiologies&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">The authors describe a case of a seven-year-old girl with classic clinical signs of endocarditis&#44; with a clinical twist&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0015" class="elsevierStylePara elsevierViewall">The authors present the case of a seven-year-old girl admitted to the emergency room with a three-day history of acute onset abdominal pain&#44; fever and recent-onset purpuric rash&#46; On admission she was restless&#44; tachycardic&#44; hypertensive and had abdominal distension and hyperesthesia&#46; A purpuric rash was also evident and limited to the buttocks&#44; lower limbs and feet&#44; and she also presented with tender bilateral edema &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Figure 1</a>&#41;&#46; Initial blood tests showed leukocytosis&#44; elevated acute-phase reactants&#44; mild anemia&#44; abnormal renal function&#44; thrombocytopenia&#44; and abnormal thromboplastin and prothrombin time&#46; Urinalysis revealed the presence of hematuria and nephrotic proteinuria&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Blood cultures were performed and ceftriaxone was started empirically&#46; Abdominal ultrasound showed splenomegaly as well as small-to-medium sized hypoechogenic lesions in the spleen&#44; liver and both kidneys&#44; compatible with thromboembolic infarcts&#46; Due to possible infective embolic disease she was referred for cardiac assessment&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">On examination&#44; a low-grade &#40;II&#47;VI&#41; systolic murmur was detected in the lower left sternal border&#44; which radiated to the back&#46; Echocardiogram revealed coarctation of the aorta at the isthmus with a systolic gradient of 60 mmHg with diastolic run-off&#46; There appeared to be a movable filament-like structure adhering to the coarcted isthmus&#46; No valvular or intracardiac vegetations were found&#46; In order to clarify these findings&#44; a transesophageal echocardiogram &#40;TOE&#41; was carried out&#44; which showed the presence of a hyperechogenic movable &#8220;spur&#8221; at the aortic isthmus&#46; A presumptive diagnosis of infective aortic endarteritis complicated by systemic embolization&#44; hypertension and acute renal insufficiency was made&#46; Vancomycin&#44; titrated to the glomerular filtration rate&#44; was added to the antibiotic regimen&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Staphylococcus schleiferi</span> was later identified as the causative agent of the infective endarteritis&#46; This organism is easily mistaken for <span class="elsevierStyleItalic">Staphylococcus aureus</span>&#46; Antibacterial sensitivity testing showed that this agent was more sensitive to floxacillin and&#44; as such&#44; the latter was added to the therapeutic regimen and vancomycin was stopped&#46; The patient became apyretic on day 5 post-admission and subsequent blood cultures were negative&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Blood pressure control was achieved with bisoprolol and nifedipine&#44; after which the transcoarctation systolic gradient was reduced to 40 mmHg&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">At week 2 post-admission&#44; the clinical course was complicated by sudden onset of gastrointestinal &#40;GI&#41; bleeding &#40;Hb 7&#46;4 g&#47;dl&#41; leading to disseminated intravascular coagulopathy&#44; requiring plasma and red blood cell transfusions&#46; Normalization of hematological parameters was eventually achieved&#44; and total parenteral nutrition was required for 10 days&#46; Despite adequate diuresis under furosemide and spironolactone&#44; worsening renal function ensued&#44; which was characterized by high creatinine levels&#44; slightly raised urea levels&#44; nephrotic proteinuria and hematuria&#44; and hypoalbuminemia&#46; This was managed with prednisolone and&#44; by week 3&#44; renal function improved and normalized&#46; The patient completed a total of six weeks of intravenous floxacillin and was discharged home on prednisolone&#44; bisoprolol and nifedipine&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Additional imaging studies such as abdominal ultrasound&#44; Computerized Tomography and Magnetic Resonance scans were performed &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Figure 2</a>&#41;&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0050" class="elsevierStylePara elsevierViewall">In children&#44; aortic endarteritis is an exceedingly rare condition even in the context of congenital heart disease&#44; particularly as a source of systemic embolization&#44; as was the case with our patient&#46; Due to its rarity&#44; no precise incidence is known&#44; with very few cases found in literature&#46; As is the case with endocarditis&#44; endarteritis is a severe complication in a child with a structural heart defect where the mortality rate is &#60;10&#37; if the diagnosis is made early or approximately 25&#37; if made at a later stage&#46; Mortality is mostly due to congestive heart failure or systemic embolization complications&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">The authors wish to highlight this rare form of endarteritis and&#44; in particular&#44; in the context of systemic embolization resulting in multiple organ failure&#46; In this case&#44; endarteritis at the coarctation site explained why systemic embolization was limited to the lower part of the body&#44; causing a lower limb purpuric rash&#44; renal insufficiency and GI bleeding&#46; As congenital heart disease is a risk factor for endocarditis&#44; it is imperative to rule out cardiovascular etiology for the embolic phenomenon&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">To our knowledge&#44; this is the first reported case of <span class="elsevierStyleItalic">Staphylococcus schleiferi</span> endarteritis originating at the site of a native aortic coarctation&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">4</span></a></p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors have no conflicts of interest to declare&#46;</p></span></span>"
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