Inflammatory pseudotumor of the liver successfully treated with nonsteroidal anti-inflammatory drugs: A challenge diagnosis for one not so rare entity

2nd Propaedeutic Department of Internal Medicine, Ippokration Hospital, Aristotle University of Thessaloniki, Greece.
European Journal of Gastroenterology & Hepatology (Impact Factor: 2.25). 12/2007; 19(11):1016-20. DOI: 10.1097/MEG.0b013e32821acdd2
Source: PubMed


Inflammatory pseudotumor of the liver is a rare, benign lesion characterized by a well-circumscribed mass of chronic inflammatory cell infiltration and proliferating fibrous tissue. Its etiology remains unclear, although inflammatory processes have been proposed. It is often misdiagnosed as a malignant tumor, and the management has been traditionally surgical. We report the case of a 16-year-old boy who was referred from another hospital with a fever of >38 degrees C with rigor and right upper quadrant pain which he had suffered from for 5 days. The ultrasonographic computed tomography and MRI findings were not diagnostic, and we performed a needle biopsy from the lesion that was consistent with inflammatory pseudotumor (of liver, mixed fibrous tissue and chronic inflammatory cell infiltration). The patient was treated with nonsteroidal anti-inflammatory drugs and had an uneventful clinical course. During follow-up, the lesion subsequently shrank to completely vanish 1 year later.

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Available from: Themistoklis Vassiliadis, Mar 25, 2015
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    • "Due to its diagnostic ambiguity, some cases with IPT of the liver were diagnosed and treated through surgical resection. Other cases were reported to be successfully managed with antibiotics, nonsteroidal anti-inflammatory drugs,16 or no medication.2,12,17,18 "
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    ABSTRACT: Inflammatory pseudotumor (IPT) of the liver is a rare disease characterized by chronic infiltration of inflammatory cells. However, the clinical characteristics and outcomes of IPT remain uncertain. Clinical features, image findings, and outcomes of 55 patients with histologically proven IPT were evaluated. They consisted of 26 men and 19 women with median age of 65 years. Serum carcinoembryonal antigen and carbohydrate antigen 19-9 levels were normal in 42 patients (93.3%). Enhanced CT scans indicated poorly defined peripheral enhancement (82.5%) at the arterial phase and poorly defined hyperattenuating lesions with internal hypoattenuating areas at the equilibrium phase (77.0%). Gadolinium-enhancement MRI revealed poorly defined peripheral rim-like enhancement (77.8%). Ten patients underwent surgical resection and 35 were treated conservatively with or without antibiotics. No recurrence was noted after surgical resection during follow-up (1 to 48 months). In all patients who received conservative treatment, complete resolution or size reduction was noted during follow-up (1 to 192 months). CT and MRI provide clues to the diagnosis of IPT in patients with liver masses and normal tumor markers. However, due to the lack of pathognomonic findings, the clinician's suspicion and histological diagnosis are necessary to make an accurate diagnosis of IPT.
    Full-text · Article · Jan 2014 · Gut and Liver
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    • "NSAID therapy induced CR by 40 days. Vassiliadis et al(16) also reported on a patient with a solitary liver mass. In this case, the patient had fever, leucocytosis thrombocytosis and an acute phase reaction serology, i.e., the same clinical presentation as the case of Chan et al(13). "
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    ABSTRACT: We report a case in which a 52-year-old female developed a multifocal inflammatory myofibroblastic tumor (IMT) of the lung. The tumor did not overexpress the anaplastic lymphoma kinase (ALK) protein, indicating a lack of ALK rearrangement. The patient required two wedge resections in 15 months due to recurrent disease. Recurrence after the second surgery was treated with corticosteroids, which only led to a transient response (6 months). Introduction of celecoxib, a cyclooxygenase-2 inhibitor, induced a complete remission in the patient. Maintenance on celecoxib further led to a progression-free survival of 34 months. A literature review retrieved a total of eight case reports, comprising ten patients, of IMT of various anatomical sites successfully treated with non-steroidal anti-inflammatory agent (NSAID) therapy. Nine of the ten patients achieved durable complete remission. Remission occurred rapidly and persisted even after termination of NSAID therapy. Although such a successful outcome may only be achieved rarely, a trial of an NSAID should be considered in any patient in whom complete resection is not an option. Our case also demonstrates that NSAID therapy may be successful in a non-ALK rearranged tumor in which ALK inhibition is not an option.
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    ABSTRACT: A 50-year-old man presented with an asymptomatic, 1.5 × 1.5 cm, dark-brown noduloplaque with a rubbery consistency (Fig. 1) on the lateral aspect of the left lower leg of uncertain duration. His general condition was healthy, and he did not recall any trauma or insect bite at this site. No similar skin lesions were found elsewhere and no lymphadenopathy was observed. The lesion revealed a nonencapsulated, but well-circumscribed, deep dermal nodule with several lymphoid aggregates and germinal center-like structures within the tumor and also at the periphery, when examined microscopically at scanning power (Fig. 2a). The epidermis showed no remarkable changes, except for basal hyperpigmentation. At higher power, a mixed inflammatory infiltrate composed of histiocytes, foamy histiocytes (Fig. 2b), lymphocytes, and abundant plasma cells (Fig. 2c) with Russell bodies was revealed. The stroma contained mainly hyalinized and sclerotic collagen fibers (Fig. 2d). Prominent venules were noted, especially in the sclerotic areas, and some were surrounded by dense collagen fibers. No vasculitis or emperipolesis was found. No foreign materials were observed by polarization microscopy, and no organisms could be identified by periodic acid–Schiff (PAS), Grocott methenamine silver (GMS), Giemsa, Gram, acid-fast, or fite stains. The results of testing for infection by Epstein–Barr virus (EBV) (latent membrane protein 1, LMP-1) were negative. No spindle cells were found in the lesion. Figure 1. A brownish noduloplaque, about 1.5 × 1.5 cm in size, located over the lateral aspect of the left lower leg Download figure to PowerPoint Figure 2. (a) A well-circumscribed deep dermal nodule with lymphoid aggregates and germinal center-like structures within the tumor and at its periphery. (b) Foamy histiocytes in serial sections. (c) Plasma cells in the infiltrate. (d) Hyalinized collagenous stroma with perivascular patterned fibrosis (arrow). Hematoxylin and eosin. Original magnification: (a) ×10; (b, c) ×400; (d) ×200 Download figure to PowerPoint Immunohistochemical studies demonstrated mature plasma cells stained with CD138, and polyclonality was confirmed by the expression of both kappa and lambda light chains. The germinal center-like lymphoid aggregates were found to be B cells, which reacted positively with CD20. Scarce S100-positive cells and even rarer CD1a-positive cells were detected. Test results for smooth muscle actin (SMA) and anaplastic lymphoma kinase (ALK) were negative. Abundant CD68+ macrophages were observed within the lesion (Fig. 3a), and about 50–75% of the inflammatory cells were found to express cyclooxygenase-2 (COX-2) (Fig. 3b). The patient's condition was diagnosed as cutaneous plasma cell granuloma (CPCG). One year after excision, no evidence of recurrence was observed. Figure 3. (a) Abundant CD68+ macrophages in the lesion (immunohistochemical staining, ×200). (b) Positive reaction with anti-cyclooxygenase-2 (anti-COX-2) in 50–75% of inflammatory cells (immunohistochemical staining, ×400) Download figure to PowerPoint
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