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Letter to the Editor
Inflammatory pseudotumor of the liver
Seudotumor inflamatorio del hígado
Sergio Cerrato Delgado
Corresponding author
sergio_0892@hotmail.com

Corresponding author.
, Maria Jesus Castro Santiago, Lidia Atienza Cuevas
Hospital Universitario Puerta del Mar, Cadiz, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Inflammatory pseudotumor is a rare lesion of unknown etiology that presents as circumscribed lesions with clear radiological signs of malignancy despite being benign in nature&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> It constitutes a significant diagnostic challenge that occasionally leads to the application of unnecessary aggressive surgical treatments&#46; In this paper we present the case of an inflammatory pseudotumor of the liver that radiologically mimicked a stage IVb gallbladder tumor and whose 0definitive diagnosis was reached through a core needle biopsy &#40;CNB&#41;&#46; Complete resolution was achieved following a course of antibiotic therapy&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was a 62-year-old man reporting occasional smoking and a moderate drinking habit&#44; under study for spikes of fever in the evening and a constitutional syndrome of one month&#8217;s evolution&#44; who presented to the Emergency Department complaining of generalized abdominal pain&#46; A complete blood count revealed a normal kidney function&#44; electrolyte panel&#44; and liver profile&#59; C-reactive protein &#40;CRP&#41; levels of 216&#46;8&#8239;mg&#47;dl&#59; a leukocyte count of 21&#44;300&#47;&#956;l &#40;79&#37; neutrophils&#41;&#59; a platelet count of 479&#44;000&#47;&#956;l&#59; an international normalized ratio &#40;INR&#41; of 1&#46;53&#59; and a prothrombin activity of 59&#37;&#46; The levels of tumor markers &#40;alpha-fetoprotein &#91;AFP&#93;&#44; carcinoembryonic antigen &#91;CEA&#93;&#44; and cancer antigen 19&#46;9 &#91;CA 19&#46;9&#93;&#41; were all normal and the serology tests for the Epstein-Barr virus&#44; hepatitis A&#44; hepatitis B&#44; and hepatitis C were all negative&#46; An abdominal computed tomography &#40;CT&#41; scan was performed&#44; observing images compatible with acute Hinchey II diverticulitis in the sigmoid colon&#44; without being able to rule out an underlying neoplastic process&#46; Several low-density lesions with heterogeneous contrast enhancement and compatible with metastasis were also identified in liver segments V&#44; VII&#44; and VIII&#44; one of them infiltrating the gallbladder&#46; This study was completed with a magnetic resonance imaging &#40;MRI&#41; scan that revealed a nodular lesion in the gallbladder wall with intense contrast enhancement in the arterial phase&#44; thus being suggestive of a gallbladder neoplasm&#44; as well as two pathological lymphadenopathies in the hepatic hilum and the liver lesions compatible with metastasis described in the CT scan &#40;hyperintense in the T2-weighted sequence and hypointense in the T1-weighted sequence&#41;&#46; A positron emission tomography &#40;PET&#41;&#47;CT scan showed radiotracer uptake in the liver lesions&#44; the gallbladder&#44; and the hilar adenopathies&#44; which are findings compatible with a stage IVb gallbladder tumor&#46; No pathological uptake was observed at the level of the sigmoid colon&#46; An abdominal ultrasound was also performed&#44; detecting three oval&#44; hypoechoic lesions in the liver&#44; without a halo and furrowed by vessels&#44; one of them infiltrating the gallbladder wall&#46; Injection of an echopotentiator contrast agent revealed mild&#44; homogeneous arterial enhancement with slight washout suggestive of an infectious&#47;inflammatory process&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">After presenting the case during a meeting held with a multidisciplinary committee&#44; the conduct of a CNB of one of the hepatic lesions was agreed upon&#46; A study of the biopsy sample revealed scarce macrovacuolar steatosis&#46; The specimen contained areas of bile duct fibrosis and isolated hepatocytes without atypia&#46; It also had a dense&#44; chronic&#44; inflammatory infiltrate with abundant plasma cells&#44; lymphocytes&#44; and histiocytes&#44; as well as fibroblasts and myofibroblasts with positive immunostaining for smooth muscle actin and muscle-specific actin&#46; An immunohistochemical study revealed negativity for CK7&#44; CK20&#44; CK19&#44; CDX2&#44; ALK&#44; and desmin&#46; Some plasma cells with immunohistochemical expression of IgG4 &#40;10&#37; of the total&#41; were detected&#46; All these findings are compatible with an inflammatory pseudotumor of the liver&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The treatment prescribed for diverticulitis was completed with antibiotic therapy with intravenous piperacillin&#47;tazobactam at a dosage of 4&#47;0&#46;5&#8239;g every 8&#8239;h administered until the time of discharge and an additional post-discharge 14day course of oral amoxicillin&#47;clavulanic acid at a dosage of 1&#8239;g&#47;200&#8239;mg ever 8&#8239;h&#46; A follow-up CT scan was performed every 3 months&#44; observing signs of radiological and clinical resolution of the condition at one year of its diagnosis&#46; In addition&#44; a colonoscopy was performed 6 weeks post-discharge&#44; detecting diverticula in the sigmoid colon&#44; but no other findings&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Inflammatory pseudotumor of the liver was first described in 1953 by Pack and Baker&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Although the most common involvement of this tumor is pulmonary&#44; some case series have described hepatic involvement in up to 8&#37; of all extrapulmonary cases&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> It usually manifests with abdominal pain associated with fever&#44; generalized malaise&#44; and constitutional syndrome&#44;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> although up to 20&#37; of cases are asymptomatic and constitute incidental diagnoses&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Blood tests usually show elevated levels of acute phase reactants and&#44; occasionally&#44; liver enzyme alterations&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;5</span></a> Although imaging techniques such as CT&#44; MRI&#44; ultrasonography with an echopotentiator agent&#44; and even PET-CT can aid in reaching the diagnosis&#44;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> because the condition lacks specific pathognomonic data&#44; the definitive diagnosis can only be reached through a histology study&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a> It is important to apply all available diagnostic methods before considering the administration of aggressive treatments&#46; In our case&#44; the results of all initial tests suggested a stage IVb gallbladder neoplasm as the main diagnosis&#46; It was the images of the ultrasound with an echopotentiator agent that led us to consider the potential diagnosis of an inflammatory pseudotumor&#44; which was finally confirmed by a CNB&#44; thus completely changing both the therapeutic approach and the patient&#39;s prognosis&#46; The evolution of this pseudotumor is unpredictable&#44; with cases of spontaneous regression without treatment having been reported&#44;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> although most require treatment with antibiotics<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> and&#47;or corticosteroids&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Surgical resection should be considered in case of persistence&#47;recurrence of the lesion or symptoms&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p></span>"
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ISSN: 23870206
Original language: English
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