Clinical and pathological characteristics of patients with pulmonary inflammatory pseudotumors: An 18-year retrospective study of 31 cases.

ABSTRACT: To investigate the clinical and pathological characteristics in patients with pulmonary inflammatory pseudotumors (PIP).This retrospective study included 31 patients with PIP from 2001 to 2019. Preoperative computed tomography scan was performed in all patients. Clinical and pathological characteristics were collected and analyzed.Thirty-one patients (16 female and 15 male) were recruited, with a median age of 57 years (range, 11-72 years). Eight (25.8%) patients were asymptomatic, and the others had symptoms characterized by cough with sputum, chest and back pain, dry cough, fever and blood in sputum, or hemoptysis. All cases were single lesions, including 23 cases in the right lung, and 8 cases in the left lung. Computed tomography scan demonstrated irregular lobulated nodules or masses in 14 patients, and regular round or oval nodules or masses in 11 cases. The blurred edge of tumors and spiculation was found in 12 cases. Microscopic results were characterized by the collection of inflammatory mesenchymal cells. Immunohistochemical examination showed vimentin, smooth muscle actin, and anaplastic lymphoma kinase positive. Complete tumor resection was obtained in all cases. No recurrence or metastasis was observed during the follow-up period.PIP has a variety of manifestations. Preoperative diagnosis is difficult to reach. The final diagnosis still depends on the pathological and immunohistochemical examination. Complete surgical resection is the main treatment at present, and the overall prognosis is good.

Hepatic Inflammatory Pseudotumor Protruding from the Liver Surface and Directly Penetrating the Colon.

A 69-year-old man was diagnosed with a liver abscess and received antibiotics at a local hospital. He was referred to our hospital due to a persistent fever. He had hepatic masses protruding from the liver surface toward the transverse colon. We reached a diagnosis of inflammatory pseudotumor (IPT) by a percutaneous liver biopsy. Colonoscopy showed direct invasion of IPT to the colon. His condition improved by the intravenous administration of antibiotics. Hepatic IPT is often misdiagnosed as a malignant tumor. We should consider IPT when we encounter hepatic tumors, and a percutaneous liver biopsy is useful for avoiding unnecessary excessive treatments.

Unusual olecranon mass with ulnar nerve compressive neuropathy caused by a haemophilic pseudotumour.

A 55-year-old man with a history of haemophilia A and bilateral haemophilic arthropathy of the elbows presented with an enlarging left elbow mass and worsening paresthesias in the ulnar distribution of the left hand. The mass, originally thought to be olecranon bursitis and treated as such, was found to be due to a haemophilic pseudotumour. The patient underwent successful excision of the haemophilic pseudotumour with concomitant ulnar nerve decompression and had sustained resolution of the pseudotumour and symptoms at 10 years of follow-up. This case demonstrates the need for consideration of haemophilic pseudotumour in the differential diagnosis for olecranon masses and cubital tunnel compressive neuropathy in patients with haemophilia, and highlights the viability of surgical excision as a therapeutic option for the treatment of haemophilic pseudotumours in the elbow.

Inflammatory myofibroblastic tumor of the epiglottis excised with a carbon dioxide laser: Case report and literature review.

Inflammatory myofibroblastic tumor (IMT) is a benign neoplasm of intermediate biologic potential. It rarely occurs in the larynx, and it has not been previously reported in the epiglottis. We treated a 66-year-old woman who presented with progressive dysphonia and a mass on her suprahyoid epiglottis. The tumor was completely excised with a CO2 laser; no adjuvant therapy was administered. Histopathology revealed that the mass was an IMT. No evidence of recurrence was noted after 6 months of follow-up. We present what we believe is the first case of an epiglottic IMT to be reported in the literature, and we propose CO2 laser excision without adjuvant therapy as an acceptable treatment.

Spinal Epidural Inflammatory Pseudotumor: A Case Report and Review of Literature.

BACKGROUND: Inflammatory pseudotumor is a histologically proven benign tumor-like lesion of unknown etiopathogenesis. It is less commonly found in central nervous system, and least commonly in the spinal canal. Spinal epidural inflammatory pseudotumor is a rarely reported entity. CASE DESCRIPTION: We report a patient who presented with paraplegia, in whom a spinal epidural mass was diagnosed as an inflammatory pseudotumor after surgical excision followed by histopathology. CONCLUSIONS: It was a radiologic dilemma; thus, complete excision was required for diagnosis and a better outcome.

Thoracic involvement in IgG4-related disease in a UK-based patient cohort.

IgG4-related disease (IgG4-RD) is a multi-system fibro-inflammatory disorder with classical histopathological findings, often in the context of elevated serum IgG4 levels. The thoracic manifestations of IgG4-RD are numerous and can mimic several common and better known conditions. The objective of this study was to outline the frequency and nature of thoracic involvement in a prospective cohort of IgG4-RD patients who met defined diagnostic criteria. Over 40% of IgG4-RD patients had clinicoradiological and/or histological evidence of thoracic involvement, predominantly mediastinal lymphadenopathy, the majority associated with multi-system disease outside the chest. Thoracic involvement was associated with a higher serum IgG4 level, potentially representing greater disease activity or spread. Our data highlight the diverse nature of thoracic IgG4-RD, and the importance of knowledge and recognition of the condition among respiratory physicians who are likely to encounter this disease entity on an increasing basis.

IgG4-related disease presenting with an epidural inflammatory pseudotumor: a case report.

BACKGROUND: Inflammatory pseudotumor is a rare clinical condition that can be related to immunoglobulin G4 disease. Only a few cases of spinal inflammatory pseudotumors have been reported in the literature and an association with immunoglobulin G4 disease was not conclusive in any of them. We describe what we believe to be the first biopsy-proven case of an epidural inflammatory pseudotumor related to immunoglobulin G4 disease. CASE PRESENTATION: A 57-year-old Caucasian woman presented to our hospital with severe paraparesis, gait disturbance, and sensory loss secondary to a relapsing epidural mass. Examination of a biopsy specimen revealed a lymphoplasmacytic infiltration with fibrosis and an immunoglobulin G4-positive plasma cell ratio of over 50 %, which are compatible with a diagnosis of immunoglobulin G4-related inflammatory pseudotumor. Our patient was successfully treated with systemic and epidural administration of glucocorticoids. CONCLUSION: Immunoglobulin G4-related disease is an emerging clinical condition in which central nervous system involvement is still uncommon. We describe the case of a patient with an epidural mass with medullar compression, which was proved to be an immunoglobulin G4-related epidural inflammatory pseudotumor. Our findings suggest a new manifestation of immunoglobulin G4-related disease. This disorder should be considered in the differential diagnosis of spinal tumors as a potentially treatable condition with glucocorticoids.

Pseudotumours and IgG4-related disease: a case report.

We report a case of a patient presenting with abdominal pain, weight loss and CT imaging showing mass lesions in the chest and abdomen associated with lymphadenopathy. He was diagnosed as having IgG4-related disease and responded well to steroid treatment.

Inflammatory myofibroblastic tumor of the larynx-a case report.

OBJECTIVES: Inflammatory myofibroblastic tumor (IMT) is a borderline neoplasm with uncertain malignant potential. It is a rare disease also referred to as an inflammatory pseudotumor, a plasma cell granuloma, and an inflammatory fibrosarcoma. IMT rarely also involves the head and neck region with only 50 cases of laryngeal IMT reported in the literature, and this is the first case with reported magnetic resonance imaging (MRI) findings. METHODS: A 37-year-old man with a 1-year history of hoarseness, dysphagia, and fatigue presented with a right vocal fold submucosal mass and was treated conservatively. RESULTS: The MRI of the neck revealed a mildly spontaneously hyperintense right true vocal fold on GRE images and relative hyperintensity on fat-saturation T2-weighted images. A biopsy of the right-sided submucosal laryngeal mass was performed and the pathologic examination revealed a lesion consistent with an IMT. CONCLUSION: IMT is a borderline neoplasm with uncertain malignant potential. There are many variants of IMT and its etiology is not truly understood. In general, IMT of the larynx has a benign clinical course with low rates of recurrence.

[Inflammatory myofibroblastic tumor of the lung: problems in differential diagnosis].

Inflammatory myofibroblastic tumor is a neoplasm of intermediate biological potential, a marked inflammatory component, and characteristic genetic changes. Once it was identified as an independent nosological entity from a rather heterogeneous group of inflammatory pseudotumors. This paper describes a case of inflammatory myofibroblastic tumor of the chest in a child, by discussing the criteria for differential diagnosis in the use of up-to-date radiology techniques. When thoracic neoplasms are detected, it is necessary to determine their site and the most likely origin organ as exactly as possible and to assess the X-ray pattern of the pathological process. By applying the state-of-the-art radiodiagnostic techniques, the clarification of the pattern of the disease makes it possible to choose an optimal treatment policy and a surgical procedure and scope, to avoid anesthesia overload, and to decrease the probability of resurgery. Our observation is to demonstrate possible problems in the diagnosis of inflammatory myofibroblastic tumor of the lung in childhood and to recall again the algorithm of radiological approaches to detecting mediastinal and lung masses.

Metal-on-metal bearings, inflammatory pseudotumours and their neurological manifestations.

Metal-on-metal bearings have become more popular for hip arthroplasty in younger patients in the last 15 years. We present a review of the neurological consequences relating to such bearing surfaces illustrated by an appropriate case report. The mechanisms of increased metal ion production and pseudotumour formation are discussed. If unexplained neurology, hip/groin pain or a localised mass around the hip are present with a metal bearing hip then urgent referral to a specialist orthopaedic surgeon is warranted for consideration of exchange arthroplasty.

Transformation of a cranial fusiform aneurysm into a pseudotumoral-like mass prior to spontaneous occlusion and regression.

This report documents the evolution of a non-ruptured cranial fusiform aneurysm that underwent both spontaneous occlusion and regression. During this process, unique images of the aneurysm as a pseudotumoral-like mass were obtained. The pseudotumoral-like mass most likely reflected inflammation and secondary neovascularization within the aneurysm, supporting the theory that spontaneous aneurysmal healing involves an inflammatory process.

The relationship between head-neck ratio and pseudotumour formation in metal-on-metal resurfacing arthroplasty of the hip.

Pseudotumour is a rare but important complication of metal-on-metal hip resurfacing that occurs much more commonly in women than in men. We examined the relationship between head-neck ratio (HNR) and pseudotumour formation in 18 resurfaced hips (18 patients) revised for pseudotumour and 42 asymptomatic control resurfaced hips (42 patients). Patients in whom pseudotumour formation had occurred had higher pre-operative HNR than the control patients (mean 1.37 (sd 0.10) vs mean 1.30 (sd 0.08) p = 0.001). At operation the patients with pseudotumours had a greater reduction in the size of their femoral heads (p = 0.035) and subsequently had greater neck narrowing (mean 10.1% (sd 7.2) vs mean 3.8% (sd 3.2) p < 0.001). No female patient with a pre-operative HNR ≤ 1.3 developed a pseudotumour. We suggest that reducing the size of the femoral head, made possible by a high pre-operative HNR, increases the risk of impingement and edge loading, and may contribute to high wear and pseudotumour formation. As the incidence of pseudotumour is low in men, it appears safe to perform resurfacing in men. However, this study suggests that it is also reasonable to resurface in women with a pre-operative HNR ≤ 1.3.

Extramedullary hematopoiesis: a rare occurrence in the sinonasal tract.

Extramedullary hematopoiesis (EMH) is a systemic reaction to inadequate hematopoiesis. We report two exceedingly rare cases of EMH involving the paranasal sinuses. The first patient, a 30-year-old man, presented with a maxillary sinus mass. The lesion was excised by endoscopic surgery: definitive histology identified foci of EMH within an inflammatory fibromyxoid pseudotumor. The second case occurred in a 29-year-old man affected by intermediate beta-thalassemia. He was hospitalized with a diagnosis of sphenoid sinus mucocele secondary to an ethmoid lesion. The patient underwent endoscopic excision of the mass and drainage of the sphenoid mucocele. At definitive histology, a diagnosis of EMH was established. Herein, the presenting modalities, imaging profile, and treatment options of this rare EMH localization are reviewed.

Hip resurfacings revised for inflammatory pseudotumour have a poor outcome.

Inflammatory pseudotumours occasionally occur after metal-on-metal hip resurfacing and often lead to revision. Our aim was to determine the severity of this complication by assessing the outcome of revision in these circumstances and by comparing this with the outcome of other metal-on-metal hip resurfacing revisions as well as that of matched primary total hip replacements. We identified 53 hips which had undergone metal-on-metal hip resurfacing and required revision at a mean of 1.59 years (0.01 to 6.69) after operation. Of these, 16 were revised for pseudotumours, 21 for fracture and 16 for other reasons. These were matched by age, gender and diagnosis with 103 patients undergoing primary total hip replacement with the Exeter implant. At a mean follow-up of three years (0.8 to 7.2) the outcome of metal-on-metal hip resurfacing revision for pseudotumour was poor with a mean Oxford hip score of 20.9 (sd 9.3) and was significantly worse (p < 0.001) than the outcome for fracture with a mean Oxford hip score of 40.2 (sd 9.2) or that for other causes with a mean Oxford hip score of 37.8 (sd 9.4). The clinical outcome of revision for pseudotumour was also significantly worse (p < 0.001) than the outcome of matched primary total hip replacements. By contrast, the outcome for fracture and other causes was not significantly different from that of matched primary total hip replacements (p = 0.065). After revision for pseudotumour there were three cases of recurrent dislocation, three of palsy of the femoral nerve, one of stenosis of the femoral artery and two of loosening of the component. Five hips required further revision. In three of these there was evidence of recurrent pseudotumour, and one is currently awaiting further revision. The incidence of major complications after revision for pseudotumour (50%) was significantly higher (p = 0.018) than that after revision for other causes (14%). The outcome of revision for pseudotumour is poor and consideration should be given to early revision to limit the extent of the soft-tissue destruction. The outcome of resurfacing revision for other causes is good.

Radiographic analysis of the cervical spine in patients with retro-odontoid pseudotumors.

STUDY DESIGN: A retrospective review of 10 consecutive patients with a noninflammatory retro-odontoid pseudotumor. OBJECTIVE: To examine the radiographic characteristics in patients with a retro-odontoid pseudotumor and to evaluate the efficacy of posterior fusion. SUMMARY OF BACKGROUND DATA: A retro-odontoid pseudotumor, a reactive fibrocartilaginous mass, is known to develop after chronic atlantoaxial instability; however, one-third of the reported cases showed no overt atlantoaxial instability. The pathomechanism for such "atypical" cases remains unclear, although altered cervical motion secondary to ossification of the anterior longitudinal ligament (OALL) or severe spondylosis has been implicated. METHODS: We reviewed the charts and radiographs of 10 patients with a retro-odontoid pseudotumor who underwent surgery. Preoperative radiographs were evaluated for atlas-dens interval (ADI), presence of OALL, range of motion, and segmental motion adjacent to the atlantoaxial joint. Computed tomography was evaluated for degenerative changes of zygapophysial joints. RESULTS: There were 6 men and 4 women. Atlantoaxial instability (ADI >4 mm) was observed in 2 patients. ADI was less than 3 mm in 5 patients. Frequent association of OALL (6 patients) and marked decrease in C2 to C7 range of motion (mean, 17.6 degrees ; range, 3 degrees-36 degrees ) were noted. Ankylosis of O-C1 was observed in 4 patients and C2 to C3 in 6. Severe degenerative change of C2 to C3 zygapophysial joint was observed in 4 patients. The patients underwent occipito-cervical fusion (9 patients) or direct removal of the pseudotumor (1 patient). Postoperative magnetic resonance imaging invariably demonstrated the mass regression. CONCLUSION: Retro-odontoid pseudotumors were not always associated with radiographic atlantoaxial instability. Our data indicate that extensive OALL and ankylosis of the adjacent segments are risk factors for the formation of the pseudotumor. Retro-odontoid pseudotumors may develop as an "adjacent segment disease" after altered biomechanics of the cervical spine, especially those in the adjacent segments. Posterior fusion was effective even in cases without radiographic atlantoaxial instability.

Inflammatory myofibroblastic tumor ofabdomen: computerized tomographic (CT) and pathological findings.

Inflammatory Myofibroblastic Tumor (IMT) is the recent name of Inflammatory pseudotumor which was in intermediate group of fibrous-origin tumor. The authors retrospectively reviewed such cases in Ramathibodi Hospital from January 2001 to December 2005. There were 115 cases of fibrous-origin tumor which was IMT in 17 cases. Nine cases occurred in the abdomen and only 3 of these had complete computerized tomographic (CT) imaging. One was hypodense liver mass with thick rim enhancement. Another one in the liver presented as a liver abscess which appeared as multiloculated hypodense mass with enhanced septum. The third case was a large malignant-looking retroperitoneal mass and having a small accompanying hepatic lesion which rapidly grew in the follow up study at nine months. IMT in the abdomen was scanty. The diagnosis was done with difficulty because of different signs and symptoms such as fever and palpable abdominal mass. The laboratory findings were nonspecific or within normal limits. Tissue biopsy was the way of definite diagnosis. We reported 3 cases of abdominal IMT with variable imaging findings that may lead to inappropriate treatment. Recognization of such findings will help achieve correct diagnosis.