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Filips A, Maurer MH, Montani M, Beldi G, Lachenmayer A. Inflammatory myofibroblastic tumor of the liver: A case report and review of literature. World J Hepatol 2020; 12:170-183. [PMID: 32685109 PMCID: PMC7336290 DOI: 10.4254/wjh.v12.i4.170] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/29/2019] [Revised: 03/05/2020] [Accepted: 03/12/2020] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Inflammatory myofibroblastic tumors of the liver (IMTL) are extremely rare neoplasms and very little is known about their clinical presentation, pathogenesis, and biological behavior. Due to their absolute rarity, it is almost impossible to obtain a definite diagnosis without histological examination. Because of their intermediate biological behavior with the risk for local recurrence and metastases, surgical resection is recommend whenever IMTL is suspect.
CASE SUMMARY We herein present a case of an otherwise healthy 32-year-old woman who presented with intermittent fever, unclear anemia, malaise and right flank pain 4 mo postpartum. The liver mass in segment IVa/b was highly FDG avid in the positron emission tomography-computed tomography. Hepatic resection was performed achieving a negative resection margin and an immediate resolution of all clinical symptoms. Histological analysis diagnosed the rare finding of an inflammatory myofibroblastic tumor of the liver and revealed cytoplasmic anaplastic lymphoma kinase expression by immunohistochemistry. Twelve months follow-up magnetic resonance imaging showed no recurrence and no metastases in the fully recovered patient.
CONCLUSION IMTLs are extremely rare and difficult to diagnose. Due to their intermediate biological behavior, surgical resection should be perform whenever feasible and patients should be followed-up in order to detect recurrence and metastasis as early as possible.
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Affiliation(s)
- Alexandra Filips
- Department of Visceral Surgery and Medicine, Inselspital, Bern University Hospital, University of Bern, Bern 3010, Switzerland
| | - Martin H Maurer
- Department of Radiology, Inselspital, Bern University Hospital, University of Bern, Bern 3010, Switzerland
| | - Matteo Montani
- Institute of Pathology, Inselspital, University Hospital, University Bern, Bern 3010, Switzerland
| | - Guido Beldi
- Department of Visceral Surgery and Medicine, Inselspital, Bern University Hospital, University of Bern, Bern 3010, Switzerland
| | - Anja Lachenmayer
- Department of Visceral Surgery and Medicine, Inselspital, Bern University Hospital, University of Bern, Bern 3010, Switzerland
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Yang TL, Chang HC. Inflammatory Pseudotumor of the Liver. JOURNAL OF CANCER RESEARCH AND PRACTICE 2019. [DOI: 10.4103/jcrp.jcrp_15_18] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
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Al-Hussaini H, Azouz H, Abu-Zaid A. Hepatic inflammatory pseudotumor presenting in an 8-year-old boy: A case report and review of literature. World J Gastroenterol 2015; 21:8730-8738. [PMID: 26229415 PMCID: PMC4515854 DOI: 10.3748/wjg.v21.i28.8730] [Citation(s) in RCA: 21] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/17/2014] [Revised: 02/27/2015] [Accepted: 03/31/2015] [Indexed: 02/06/2023] Open
Abstract
Hepatic inflammatory pseudotumors are uncommon benign lesions. Accurately diagnosing hepatic inflammatory pseudotumor can be very challenging because the clinical presentation and radiological appearances are nonspecific and cannot be certainly distinguished from malignant neoplastic processes. Herein, we present a case of hepatic IPT in an 8-year-old boy who presented to clinic with a 3-mo history of a tender hepatic mass, fever of unknown origin, and 9-kg weight loss. The physical examination was notable for tender hepatomegaly. Laboratory investigations were notable for a normal hepatic profile and elevated erythrocyte sedimentation rate and C-reactive protein. A T2-attenuated magnetic resonance imaging scan of the abdomen showed a 4.7 cm × 4.7 cm × 6.6 cm, contrast-enhancing, hyper-intense, well-defined lesion involving the right hepatic lobe. In view of the unremitting symptoms, tender hepatomegaly, thrombosed right hepatic vein, nonspecific radiological findings, and high suspicion of a deep-seated underlying infection or malignancy, a right hepatic lobectomy was recommended. Microscopically, the hepatic lesion exhibited a mixture of inflammatory cells (histiocytes, plasma cells, mature lymphocytes, and occasional multinucleated giant cells) in a background of dense fibrous tissue. Immunohistochemically, the cells stained negative for SMA, ALK-1, CD-21 and CD-23, diffusely positive for CD-68, and focally positive for IgG4. The final histopathological diagnosis was consistent with hepatic IPT. At the postoperative 4-mo follow-up, the patient was asymptomatic without radiological evidence of recurrence.
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Yang X, Miao R, Yang H, Chi T, Jiang C, Wan X, Xu Y, Xu H, Du S, Lu X, Mao Y, Zhong S, Zhao H, Sang X. Retrospective and comparative study of inflammatory myofibroblastic tumor of the liver. J Gastroenterol Hepatol 2015; 30:885-90. [PMID: 25387431 DOI: 10.1111/jgh.12846] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 11/03/2014] [Indexed: 12/13/2022]
Abstract
BACKGROUND AND AIM Inflammatory myofibroblastic tumor of the liver (IMTL) is a very rare benign disease with a good prognosis. The study aims to determine the clinical, radiological, and pathological characteristics of IMTL. The diagnosis and treatment strategies were discussed. METHODS A total of 11 patients with pathologically confirmed IMTL receiving treatment over a 15-year period were reviewed retrospectively. The analysis included demographics information and pertinent clinical data. Results obtained from patients with hepatocellular carcinoma (HCC), intrahepatic cholangiocarcinoma (IHCC), and metastatic liver cancer (MLC) receiving surgical resection were compared. RESULTS In comparison to HCC, IHCC, and MLC, IMTL has an earlier onset (P < 0.001). IMTL patients had significantly lower aspartate aminotransferase (P = 0.003) and higher alkaline phosphatase (P = 0.034) than HCC patients, and higher gamma-glutamyl transpeptidase (P = 0.010) than MLC patients. Increased serum α-fetoprotein level was detected in only one patient. Serum α-fetoprotein was significantly lower in patients with IMTL (P = 0.000) than in those with HCC but not IHCC (P = 0.558) or MLC (P = 0.514). In contrast to elevated serum CA19-9 in patients with HCC/IHCC/MLC, the serum CA19-9 in IMTL cases was generally normal (vs HCC P = 0.008; vs IHCC P = 0.000; vs MLC P = 0.022). In nine IMTL patients, the tumor appeared as a hypoechogenic solid mass on the ultrasonography. In contrast, most patients with HCC, IHCC, or MLC showed hybrid echo. In contrast computed tomography and magnetic resonance imaging, the lesion of IMTL and MLC appeared as peripheral enhancement. CONCLUSION Lab tests, imaging features, and patient history are helpful in the differential diagnosis of IMTL from HCC/IHCC/MLC. Surgical resection is curative for IMTL.
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Affiliation(s)
- Xiaobo Yang
- Department of Liver Surgery, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College (CAMS and PUMC), Beijing, China
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Hepatic inflammatory pseudotumor with elevated serum CA19-9 level mimicking liver metastasis from rectal cancer: report of a case. Int Surg 2015; 98:324-9. [PMID: 24229018 DOI: 10.9738/intsurg-d-13-00091.1] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/03/2023] Open
Abstract
Inflammatory pseudotumor (IPT) of the liver is rare, and differential diagnosis from malignant tumors is difficult. We herein report a case of IPT of the liver with marked elevation of carbohydrate antigen 19-9 (CA19-9). A 72-year-old woman, who underwent low anterior resection for rectal cancer, had low-grade fever and epigastric pain. The patient has well-controlled idiopathic thrombocytopenic purpura (ITP) with steroid maintenance treatment. Abnormal laboratory findings included serum carcinoembryonic antigen of 15.9 ng/mL and CA19-9 up to 14,632 U/mL. Computed tomography revealed a low-density lesion, and magnetic resonance imaging demonstrated a high intensity lesion on T1, T2-weighted, and diffusion-weighted images (55 × 22 mm in the Couinaud's segment 4 of the liver). However, 1 month after the appearance of the symptom, serum level of CA19-9 suddenly decreased to 37 U/mL. The tumor decreased spontaneously to 20 × 10 mm. Seven months later, the tumor had reduced to 18 × 11 mm. With a diagnosis of IPT, the patient remains well without regrowth of the tumor at 1 year follow-up after CA19-9 decrease to normal limits.
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Belghiti J, Cauchy F, Paradis V, Vilgrain V. Diagnosis and management of solid benign liver lesions. Nat Rev Gastroenterol Hepatol 2014; 11:737-49. [PMID: 25178878 DOI: 10.1038/nrgastro.2014.151] [Citation(s) in RCA: 72] [Impact Index Per Article: 6.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/13/2022]
Abstract
More and more asymptomatic benign liver tumours are discovered incidentally and can be divided into regenerative lesions and true neoplastic lesions. The most common regenerative lesions include hemangioma, focal nodular hyperplasia and inflammatory pseudotumours of the liver. Neoplastic lesions include hepatocellular adenomas and angiomyolipomas. Regenerative lesions rarely increase in volume, do not yield a higher risk of complications and usually do not require treatment. By contrast, hepatocellular adenomas and angiomyolipomas can increase in volume and are associated with a risk of complications. Large hepatocellular adenomas (>5 cm in diameter) are undoubtedly associated with a risk of bleeding and malignant transformation, particularly the inflammatory (also known as telangiectatic) and β-catenin mutated subtypes. Accurate diagnosis needs to be obtained to select patients eligible for surgical resection. MRI has markedly improved diagnosis and can identify the major hepatocellular adenomas subtypes. The use of biopsy results to inform the indication for resection remains questionable. However, when diagnosis remains uncertain after imaging, percutaneous biopsy could help improve diagnostic accuracy.
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Affiliation(s)
- Jacques Belghiti
- Department of Hepatobiliary Surgery and Liver Transplantation, Beaujon Hospital, University Paris-Diderot, 100 Boulevard du General Leclerc, 92110 Clichy, France
| | - François Cauchy
- Department of Hepatobiliary Surgery and Liver Transplantation, Beaujon Hospital, University Paris-Diderot, 100 Boulevard du General Leclerc, 92110 Clichy, France
| | - Valérie Paradis
- Department of Pathology, Beaujon Hospital, University Paris-Diderot, 100 Boulevard du General Leclerc, 92110 Clichy, France
| | - Valérie Vilgrain
- Department of Radiology, Beaujon Hospital, University Paris-Diderot, 100 Boulevard du General Leclerc, 92110 Clichy, France
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Al-Jabri T, Sanjay P, Shaikh I, Woodward A. Inflammatory myofibroblastic pseudotumour of the liver in association with gall stones - a rare case report and brief review. Diagn Pathol 2010. [PMID: 20718986 DOI: 10.1186/1746-1596-5-53al-jabri,] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023] Open
Abstract
Inflammatory myofibroblastic pseudotumours of the liver are rare tumour-like lesions that can mimic malignant liver neoplasms. The symptoms and radiological findings of this rare tumour can pose diagnostic difficulties. We describe a 69-year-old gentleman who was admitted to our department with symptoms suggestive of acute cholecystitis. Ultrasonography and computed tomography of the liver raised the possibility of metastatic liver disease. A core biopsy of the liver was performed to confirm the diagnosis of liver metastasis. Unexpectedly it showed no evidence of malignancy but instead revealed an inflammatory myofibroblastic pseudotumour of the liver. This case report highlights the diagnostic dilemma that arose due to the similarity of appearances between the two pathological entities on imaging and this stresses the need for accurate histological diagnosis so as to avoid unnecessary surgical intervention. To the best of our knowledge, only a minority of cases are reported in the literature associating a hepatic inflammatory myofibroblastic pseudotumour with gall stones.
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Affiliation(s)
- Talal Al-Jabri
- Department of Surgery, East and North Hertforshire NHS Trust, Hertfordshire, AL7 4HQ, UK.
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Al-Jabri T, Sanjay P, Shaikh I, Woodward A. Inflammatory myofibroblastic pseudotumour of the liver in association with gall stones - a rare case report and brief review. Diagn Pathol 2010; 5:53. [PMID: 20718986 PMCID: PMC2933681 DOI: 10.1186/1746-1596-5-53] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/09/2010] [Accepted: 08/18/2010] [Indexed: 01/17/2023] Open
Abstract
Inflammatory myofibroblastic pseudotumours of the liver are rare tumour-like lesions that can mimic malignant liver neoplasms. The symptoms and radiological findings of this rare tumour can pose diagnostic difficulties. We describe a 69-year-old gentleman who was admitted to our department with symptoms suggestive of acute cholecystitis. Ultrasonography and computed tomography of the liver raised the possibility of metastatic liver disease. A core biopsy of the liver was performed to confirm the diagnosis of liver metastasis. Unexpectedly it showed no evidence of malignancy but instead revealed an inflammatory myofibroblastic pseudotumour of the liver. This case report highlights the diagnostic dilemma that arose due to the similarity of appearances between the two pathological entities on imaging and this stresses the need for accurate histological diagnosis so as to avoid unnecessary surgical intervention. To the best of our knowledge, only a minority of cases are reported in the literature associating a hepatic inflammatory myofibroblastic pseudotumour with gall stones.
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Affiliation(s)
- Talal Al-Jabri
- Department of Surgery, East and North Hertforshire NHS Trust, Hertfordshire, AL7 4HQ, UK.
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Tsou YK, Lin CJ, Liu NJ, Lin CC, Lin CH, Lin SM. Inflammatory pseudotumor of the liver: report of eight cases, including three unusual cases, and a literature review. J Gastroenterol Hepatol 2007; 22:2143-2147. [PMID: 18031372 DOI: 10.1111/j.1440-1746.2006.04514.x] [Citation(s) in RCA: 30] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/05/2023]
Abstract
BACKGROUND Inflammatory pseudotumor (IPT) of the liver is a rare but increasingly recognized tumor-like lesion. This condition is becoming an important differential diagnosis in patients with hepatic space-occupying lesions. This study reports the clinical features of eight cases of IPT of the liver, including the first report of three cases with an unusual associated disease or clinical course. METHODS The study sample included cases of IPT diagnosed based on the histopathology in our institution from 1995 to 2004. Clinical events were reported and compared with reports in the literature. RESULTS The age of the patients ranged from 28 to 78 years. The most common symptoms were abdominal pain, fever and bodyweight loss. Three patients were diagnosed incidentally. The initial clinical diagnoses were hepatocellular carcinoma in three cases, liver abscess in two cases, metastasis in one case and uncertain nature in two cases. Two cases were associated with preceding endophthalmitis and cholangiocarcinoma, respectively. Two patients had spontaneous lesion regression, but one of these had possible recurrence 10 months later. The other two patients experienced resolution of their lesions following antibiotic treatment. Four patients underwent surgical resection with subsequent recovery. CONCLUSIONS The clinical and imaging features of IPT in this small series were found to sometimes mimic those of malignant liver tumors or abscess. Surgical resection was performed in half of the cases. However, IPT of the liver could resolve spontaneously or following antibiotics treatment.
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Affiliation(s)
- Yung-Kuan Tsou
- Liver Research Unit, Department of Hepato-Gastroenterology, Chang Gung Memorial Hospital, Chang Gung University, Taipei, Taiwan
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Boutarbouch M, Arkha Y, Rifi L, Derraz S, El Ouahabi A, El Khamlichi A. Intradural cervical inflammatory pseudotumor mimicking epidural hematoma in a pregnant woman: case report and review of the literature. ACTA ACUST UNITED AC 2007; 69:302-5. [PMID: 17765955 DOI: 10.1016/j.surneu.2006.12.056] [Citation(s) in RCA: 17] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/07/2006] [Accepted: 12/20/2006] [Indexed: 11/19/2022]
Abstract
BACKGROUND Inflammatory pseudotumors usually affect the lung and the orbit. They occur extremely rarely in the spine. We encountered a case of intradural extramedullary IPT of the cervical spine in a pregnant woman that initially presented as an epidural hematoma in MRI. CASE DESCRIPTION This is a case of a 30-year-old, HIV-negative, full-term pregnant lady presenting with 2-month history of progressive quadriparesis with rapid worsening of power in all 4 limbs for 3 days. Magnetic resonance imaging scanning revealed cervical extramedullary compressive lesion likely to be epidural bleed from its imaging characteristics. Intraoperatively, intradural extramedullary granulomatous lesion was found with dural thickening. Gross total excision was accomplished; histopathologic study assessed an IPT. The patient improved after surgery excision and is doing well at 6-month follow-up with remarkable neurological recovery. CONCLUSION Cervical intradural IPT is a very rare issue in the spine. Diagnosis can be confused with a neoplastic lesion like an "en plaque meningioma" or as epidural bleed like in the present case.
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Affiliation(s)
- Mahjouba Boutarbouch
- Department of Neurosurgery, Mohamed Vth University School of Medicine, Hôpital des Spécialités ONO, Rabat 10100, Morocco.
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Bernard M, Marie I, Houdent C, Goria O, François A, Lévesque H. [A liver in the Perigord style...and green and gray on the...]. Rev Med Interne 2005; 26 Suppl 2:S242-3. [PMID: 16129158 DOI: 10.1016/s0248-8663(05)80038-4] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Affiliation(s)
- M Bernard
- Département de médecine interne, CHU Rouen-Boisguillaume, France
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Koea JB, Broadhurst GW, Rodgers MS, McCall JL. Inflammatory pseudotumor of the liver: demographics, diagnosis, and the case for nonoperative management. J Am Coll Surg 2003; 196:226-35. [PMID: 12595051 DOI: 10.1016/s1072-7515(02)01495-3] [Citation(s) in RCA: 83] [Impact Index Per Article: 3.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
BACKGROUND Inflammatory pseudotumor of the liver (IPL) is an unusual tumor-like condition that is becoming recognized as an important differential diagnosis in the patient presenting with liver masses. This report describes six cases of IPL. STUDY DESIGN Clinical, diagnostic, pathologic, and followup data were collected prospectively on six patients presenting to a specialist hepatobiliary unit. RESULTS Six patients with IPL presented over a 2-year period. Median age was 35 years (range 2 to 79 years) and five patients were men. Three patients were Polynesian (Tongan and Samoan) and one was New Zealand Maori. Five patients presented with nonspecific symptoms (fever, arthralgia, myalgia) and IPL was an incidental finding in one patient. At presentation, four patients had elevated white cell counts, and five patients had abnormal liver function tests with elevations in alkaline phosphotase and gamma-glutamyl transferase the most commonly seen. Carcinogenic embryonic antigen and alpha fetoprotein were normal in all patients, although one was a known hepatitis B carrier. In all cases of IPL, diagnosis was made on core biopsy of the liver lesions and all patients were managed nonoperatively with complete resolution of the tumors. Two patients had marked reduction in systemic symptoms (fever and pain) from a short course of oral steroids. CONCLUSIONS IPL appears to be a relatively common problem in Maori and Polynesians. Recognition and differentiation of this condition from malignant liver lesions are important to avoid unnecessary surgery.
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Affiliation(s)
- Jonathan B Koea
- Upper Gastrointestinal/Hepatobiliary Unit, Department of Surgery, Auckland Hospital, Auckland, New Zealand
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Colović R, Grubor N, Micev M, Stojković M. [Inflammatory pseudotumors of the liver]. SRP ARK CELOK LEK 2002; 130:270-3. [PMID: 12585005 DOI: 10.2298/sarh0208270c] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/27/2022] Open
Abstract
Inflammatory pseudotumours are very rare with hardly over hundred cases reported in literature. The disease is rare in children. The tumour is usually solitary, rarely multifocal. We report on two women old 48 and 49 years. One had a solitary tumour, and the other had four multifocal inflammatory pseudotumours which were surgically removed. Preoperatively both patients had pain, loss in weight, the one had attacks of low grade temperature and the other nausea and general malaise. One patient had leucocytosis. After surgery both patients stayed symptom free with normal laboratory and ultrasonographic findings 6 and 50 months respectively.
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Affiliation(s)
- Radoje Colović
- Institute of Gastrointestinal Diseases, Clinical Centre of Serbia, Belgrade
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