Inflammatory pseudotumor of the liver

Cerrato Delgado, Sergio; Castro Santiago, Maria Jesus; Atienza Cuevas, Lidia

doi:10.1016/j.medcle.2020.06.046

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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 (CNB). Complete resolution was achieved following a course of antibiotic therapy.The patient was a 62-year-old man reporting occasional smoking and a moderate drinking habit, under study for spikes of fever in the evening and a constitutional syndrome of one month’s evolution, who presented to the Emergency Department complaining of generalized abdominal pain. A complete blood count revealed a normal kidney function, electrolyte panel, and liver profile; C-reactive protein (CRP) levels of 216.8 mg/dl; a leukocyte count of 21,300/μl (79% neutrophils); a platelet count of 479,000/μl; an international normalized ratio (INR) of 1.53; and a prothrombin activity of 59%. The levels of tumor markers (alpha-fetoprotein [AFP], carcinoembryonic antigen [CEA], and cancer antigen 19.9 [CA 19.9]) were all normal and the serology tests for the Epstein-Barr virus, hepatitis A, hepatitis B, and hepatitis C were all negative. An abdominal computed tomography (CT) scan was performed, observing images compatible with acute Hinchey II diverticulitis in the sigmoid colon, without being able to rule out an underlying neoplastic process. Several low-density lesions with heterogeneous contrast enhancement and compatible with metastasis were also identified in liver segments V, VII, and VIII, one of them infiltrating the gallbladder. This study was completed with a magnetic resonance imaging (MRI) scan that revealed a nodular lesion in the gallbladder wall with intense contrast enhancement in the arterial phase, thus being suggestive of a gallbladder neoplasm, as well as two pathological lymphadenopathies in the hepatic hilum and the liver lesions compatible with metastasis described in the CT scan (hyperintense in the T2-weighted sequence and hypointense in the T1-weighted sequence). A positron emission tomography (PET)/CT scan showed radiotracer uptake in the liver lesions, the gallbladder, and the hilar adenopathies, which are findings compatible with a stage IVb gallbladder tumor. No pathological uptake was observed at the level of the sigmoid colon. An abdominal ultrasound was also performed, detecting three oval, hypoechoic lesions in the liver, without a halo and furrowed by vessels, one of them infiltrating the gallbladder wall. Injection of an echopotentiator contrast agent revealed mild, homogeneous arterial enhancement with slight washout suggestive of an infectious/inflammatory process.After presenting the case during a meeting held with a multidisciplinary committee, the conduct of a CNB of one of the hepatic lesions was agreed upon. A study of the biopsy sample revealed scarce macrovacuolar steatosis. The specimen contained areas of bile duct fibrosis and isolated hepatocytes without atypia. It also had a dense, chronic, inflammatory infiltrate with abundant plasma cells, lymphocytes, and histiocytes, as well as fibroblasts and myofibroblasts with positive immunostaining for smooth muscle actin and muscle-specific actin. An immunohistochemical study revealed negativity for CK7, CK20, CK19, CDX2, ALK, and desmin. Some plasma cells with immunohistochemical expression of IgG4 (10% of the total) were detected. All these findings are compatible with an inflammatory pseudotumor of the liver.The treatment prescribed for diverticulitis was completed with antibiotic therapy with intravenous piperacillin/tazobactam at a dosage of 4/0.5 g every 8 h administered until the time of discharge and an additional post-discharge 14day course of oral amoxicillin/clavulanic acid at a dosage of 1 g/200 mg ever 8 h. A follow-up CT scan was performed every 3 months, observing signs of radiological and clinical resolution of the condition at one year of its diagnosis. In addition, a colonoscopy was performed 6 weeks post-discharge, detecting diverticula in the sigmoid colon, but no other findings.Inflammatory pseudotumor of the liver was first described in 1953 by Pack and Baker.<a class="elsevierStyleCrossRef" href="#bib0005">1</a> Although the most common involvement of this tumor is pulmonary, some case series have described hepatic involvement in up to 8% of all extrapulmonary cases.<a class="elsevierStyleCrossRef" href="#bib0015">3</a> It usually manifests with abdominal pain associated with fever, generalized malaise, and constitutional syndrome,<a class="elsevierStyleCrossRef" href="#bib0020">4</a> although up to 20% of cases are asymptomatic and constitute incidental diagnoses.<a class="elsevierStyleCrossRef" href="#bib0010">2</a> Blood tests usually show elevated levels of acute phase reactants and, occasionally, liver enzyme alterations.<a class="elsevierStyleCrossRefs" href="#bib0010">2,5</a> Although imaging techniques such as CT, MRI, ultrasonography with an echopotentiator agent, and even PET-CT can aid in reaching the diagnosis,<a class="elsevierStyleCrossRef" href="#bib0025">5</a> because the condition lacks specific pathognomonic data, the definitive diagnosis can only be reached through a histology study.<a class="elsevierStyleCrossRefs" href="#bib0005">1,4</a> It is important to apply all available diagnostic methods before considering the administration of aggressive treatments. In our case, the results of all initial tests suggested a stage IVb gallbladder neoplasm as the main diagnosis. It was the images of the ultrasound with an echopotentiator agent that led us to consider the potential diagnosis of an inflammatory pseudotumor, which was finally confirmed by a CNB, thus completely changing both the therapeutic approach and the patient's prognosis. The evolution of this pseudotumor is unpredictable, with cases of spontaneous regression without treatment having been reported,<a class="elsevierStyleCrossRef" href="#bib0005">1</a> although most require treatment with antibiotics<a class="elsevierStyleCrossRef" href="#bib0010">2</a> and/or corticosteroids.<a class="elsevierStyleCrossRef" href="#bib0020">4</a> Surgical resection should be considered in case of persistence/recurrence of the lesion or symptoms.<a class="elsevierStyleCrossRef" href="#bib0005">1</a>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "Please cite this article as: Cerrato Delgado S, Castro Santiago MJ, Atienza Cuevas L. Seudotumor inflamatorio del hígado. Med Clin (Barc). 2021;157:303–304." ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Hepatic inflammatory pseudotumor protruding from the liver surface and directly penetrating the colon" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "N. Imazu" 1 => "M. Shibata" 2 => "Y. Koya" 3 => "K. Morino" 4 => "Y. Honma" 5 => "M. Senju" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.2169/internalmedicine.3599-19" "Revista" => array:7 [ "tituloSerie" => "Intern Med." "fecha" => "2020" "volumen" => "59" "paginaInicial" => "527" "paginaFinal" => "532" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/31611530" "web" => "Medline" ] ] "itemHostRev" => array:3 [ "pii" => "S0167527316328650" "estado" => "S300" "issn" => "01675273" ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0010" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Hepatic inflammatory pseudotumor: an important differential diagnosis in patients with a history of previous biliary procedures" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "J. Zhao" 1 => "K. Olino" 2 => "L.E. Low" 3 => "S. Qiu" 4 => "H.L. Stevenson" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.14309/crj.0000000000000015" "Revista" => array:6 [ "tituloSerie" => "ACG Case Rep J" "fecha" => "2019" "volumen" => "6" "paginaInicial" => "e00015" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/31598534" "web" => "Medline" ] ] "itemHostRev" => array:3 [ "pii" => "S0735109717419358" "estado" => "S300" "issn" => "07351097" ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0015" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Spontaneous resolution of IgG4-related hepatic inflammatory pseudotumor mimicking malignancy" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "H. Patel" 1 => "S. Nanavati" 2 => "J. Ha" 3 => "A. Shah" 4 => "W. Baddoura" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1159/000490100" "Revista" => array:6 [ "tituloSerie" => "Case Rep Gastroenterol." "fecha" => "2018" "volumen" => "12" "paginaInicial" => "311" "paginaFinal" => "316" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/30022921" "web" => "Medline" ] ] ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0020" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Recurrent hepatic inflammatory pseudotumor in asynchronism after surgery of inflammatory pseudotumor associated with granulomatous cholangitis" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "A. Imoto" 1 => "Y. Inoue" 2 => "T. Ogura" 3 => "D. Masuda" 4 => "M. Asakuma" 5 => "Y. Kurisu" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.14740/jmc3245" "Revista" => array:5 [ "tituloSerie" => "J Med Cases" "fecha" => "2019" "volumen" => "10" "paginaInicial" => "41" "paginaFinal" => "45" ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0025" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Misdiagnosing hepatic inflammatory pseudotumor as hepatocellular carcinoma: a case report" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "L. Yin" 1 => "B. Zhu" 2 => "X.Y. Lu" 3 => "W.Y. Lau" 4 => "Y.J. Zhang" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1002/jgh3.1201" "Revista" => array:6 [ "tituloSerie" => "JGH Open" "fecha" => "2017" "volumen" => "1" "paginaInicial" => "76" "paginaFinal" => "78" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/30483539" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000015700000006/v2_202201010922/S2387020621003983/v2_202201010922/en/main.assets" "Apartado" => array:4 [ "identificador" => "43309" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000015700000006/v2_202201010922/S2387020621003983/v2_202201010922/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020621003983?idApp=UINPBA00004N"]

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Vol. 157. Issue 6.

Pages 303-304 (September 2021)

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