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Elhalaby I, Koura O, Elhalaby R, Zeina W, Shareef M, Elhalaby E. Inflammatory myofibroblastic tumors of the colon in pediatrics: clinical presentation, management, and outcomes-A case report and systematic review of literature. Int J Colorectal Dis 2025; 40:94. [PMID: 40234278 PMCID: PMC12000112 DOI: 10.1007/s00384-025-04869-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 03/20/2025] [Indexed: 04/17/2025]
Abstract
PURPOSE Inflammatory myofibroblastic tumors (IMTs) of the colon represent an exceptionally rare entity in the pediatric population. This systematic review aims to comprehensively analyze the clinical presentation, diagnostic workup, management strategies, and outcomes of colorectal IMTs in children. METHODS A systematic literature review was conducted across multiple electronic databases (inception to January 2025), including MEDLINE (via PubMed), Embase, Cochrane, Web of Science, and Google Scholar. Two independent reviewers screened abstracts, reviewed studies, and extracted data on all reported cases of colorectal IMTs in the pediatric population, including one previously unreported case from our institution. RESULTS Including our case, 53 pediatric patients with colorectal IMTs were identified from 39 studies. The mean age at diagnosis was 7 years (range: 5 months-17 years) with a slight female preponderance. The IMTs comprised a wide range of anatomic locations with rectum (27%) and ascending colon (24%) being the most common. Abdominal pain (54%), gastrointestinal bleeding (29%), and fever (21%) were the predominant symptoms. Anemia was the most common laboratory abnormality (62%). Surgical resection was the primary treatment modality in 98% of cases. After a mean follow-up of 38 months ( range: 3-181 months), the local recurrence rate was 11%, with no distant metastases reported. CONCLUSION Colorectal IMTs in children present diagnostic and therapeutic challenges. While complete surgical resection remains the gold standard treatment, emerging therapies such as ALK inhibitors and NSAIDs warrant further investigation. The potential for late recurrence mandates long term follow-up.
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Affiliation(s)
- Ismael Elhalaby
- Department of Surgery, Faculty of Medicine, Tanta University, Tanta, Egypt.
- Kenanah Children's Medical Center, Tanta, Egypt.
| | - Omar Koura
- Department of Surgery, Faculty of Medicine, Tanta University, Tanta, Egypt
- Kenanah Children's Medical Center, Tanta, Egypt
| | - Rofyda Elhalaby
- Department of Pathology, Faculty of Medicine, Tanta University, Tanta, Egypt
| | - Wael Zeina
- Kenanah Children's Medical Center, Tanta, Egypt
- Benha Children Hospital, Benha, Egypt
| | - Mohamed Shareef
- Department of Pathology, Faculty of Medicine, Tanta University, Tanta, Egypt
| | - Essam Elhalaby
- Department of Surgery, Faculty of Medicine, Tanta University, Tanta, Egypt
- Kenanah Children's Medical Center, Tanta, Egypt
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Billingsley BC, Chaudhary R, Morris MW, Cox JA, Camacho-Gomez SM, Varshney N. Inflammatory Myofibroblastic Tumor of the Esophagus and Stomach Successfully Treated With ALK Inhibitor in a Pediatric Patient: A Case Report and Concise Review of Literature. Int J Surg Pathol 2025; 33:145-152. [PMID: 38656257 DOI: 10.1177/10668969241246470] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 04/26/2024]
Abstract
An inflammatory myofibroblastic tumor (IMT) is a rare mesenchymal neoplasm of borderline malignant potential. Nearly half of all IMTs have rearrangement of anaplastic lymphoma kinase (ALK) locus on chromosome 2p23 which can be treated with targeted therapy. Herein, we describe an unusual presentation of IMT involving an anatomical region rarely implicated in this disease process. A 15-year-old male patient came to the ER with dysphagia and coffee ground emesis. On esophagogastroscopy, a nodular luminal obstructing 30 × 50 mm mass in the lower esophagus was found, which was continuous with a large, partially circumferential gastric mass extending from the mid-body to the proximal antrum. Biopsies from esophageal and gastric masses revealed submucosal lesions composed of cytologically bland spindle and epithelioid cells, intermingled with inflammatory infiltrate, for which several immunohistochemical (IHC) stains were performed. The molecular study demonstrated ATIC::ALK fusion. Based on morphological, IHC, and molecular study findings, the diagnosis of ALK-positive IMT was rendered. Because surgical excision was deemed infeasible, the patient was started on ALK-inhibiting therapy with crizotinib. The patient responded well with no evidence of residual or recurrent disease on follow-up imaging or surveillance esophagogastroduodenoscopy. Crizotinib was ultimately discontinued after 10 months of therapy, and the patient continues to undergo surveillance imaging for monitoring of disease burden.
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Affiliation(s)
- Benjamin C Billingsley
- Department of Internal Medicine, University of Mississippi Medical Center, Jackson, MS, USA
| | - Ritica Chaudhary
- Department of Pathology, University of Mississippi Medical Center, Jackson, MS, USA
| | - Michael W Morris
- Division of Pediatric, Department of Surgery, University of Mississippi Medical Center, Jackson, MS, USA
| | - Jennifer A Cox
- Division of Hematology and Oncology, Department of Pediatrics, University of Mississippi Medical Center, Jackson, MS, USA
| | - Sandra M Camacho-Gomez
- Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, University of Texas at Austin, Austin, TX, USA
| | - Neha Varshney
- Department of Pathology, University of Mississippi Medical Center, Jackson, MS, USA
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Li X, Li J, Liang C, Zou Q. Case report: Intra-abdominal inflammatory myofibroblastic tumor with mucinous features: a case of rapid recurrence and dissemination post-surgery. Front Oncol 2025; 14:1517710. [PMID: 39871941 PMCID: PMC11770369 DOI: 10.3389/fonc.2024.1517710] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/26/2024] [Accepted: 12/09/2024] [Indexed: 01/29/2025] Open
Abstract
Inflammatory myofibroblastic tumors (IMTs) are rare mesenchymal neoplasms with intermediate biological potential and are characterized by spindle-shaped myofibroblastic cells and significant inflammatory infiltrates. This case report describes a 24-year-old male with diabetes who was admitted to the hospital for over three days of vomiting and abdominal pain and was initially diagnosed with diabetic ketoacidosis. Upon admission, an abdominal CT scan revealed a large cystic-solid mass in the abdominal cavity and multiple nodules in the mesentery, omentum, and peritoneum, suggesting a preliminary diagnosis of an intra-abdominal mesenchymal tumor with peritoneal metastasis. The patient underwent tumor resection, and postoperative pathology confirmed it to be an IMT rich in mucin, with a Ki-67 proliferation index of 50%. Despite the initial symptom improvement after surgery, the patient experienced rapid recurrence with more extensive abdominal lesions. The patient refused further treatment, and died shortly thereafter. The case underscores the aggressive nature of inflammatory myofibroblastic tumors (IMTs) characterized by significant mucinous features, which are prone to recurrence and may suggest a poor prognosis. Radiological examinations and preoperative fine-needle aspiration biopsy may play a crucial role in managing such cases. Furthermore, alternative non-surgical treatment options or adjunct postoperative treatments could have a positive impact on the prognosis of this patient group. Further research is vital for enhancing our understanding of this rare tumor type and optimizing treatment strategies.
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Affiliation(s)
| | | | | | - Qing Zou
- Department of Radiology, People’s Hospital of Deyang City,
Deyang, Sichuan, China
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Kavirayani V, Pai NG, Nayal B, Prabhu S. Infantile inflammatory myofibroblastic tumour of the sigmoid colon: a diagnostic dilemma. BMJ Case Rep 2023; 16:e256505. [PMID: 37832973 PMCID: PMC10583037 DOI: 10.1136/bcr-2023-256505] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/15/2023] Open
Abstract
An inflammatory myofibroblastic tumour (IMT) is an uncommon neoplasm composed of inflammatory cells and myofibroblasts in a fibrous stroma. They are mostly seen in the lungs and rarely involve the gastrointestinal tract. An 8-month-old infant presented with a history of lower abdominal lump for 2 months. Her CT scan confirmed a large, lobulated mass in the retroperitoneum arising from the pelvis. The mass was found to be arising from the sigmoid colon on laparotomy which was excised. Histopathology showed a cellular tumour composed of spindle cells and inflammatory lymphocytic infiltrate. Immunohistochemistry revealed positive staining for anaplastic lymphoma kinase and smooth muscle actin, confirming the diagnosis of IMT. The patient is doing well at her 6-month follow-up. Ours is the youngest case of sigmoid IMT among the only other series of eight cases reported in the literature indicating its rarity.
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Affiliation(s)
- Vaishnavi Kavirayani
- Pediatric Surgery, Kasturba Medical College Manipal, Manipal academy of Higher education, Manipal, Karnataka, India
| | - Nitin G Pai
- Pediatric Surgery, Kasturba Medical College Manipal, Manipal academy of Higher education, Manipal, Karnataka, India
| | - Bhavna Nayal
- Pathology, Kasturba Medical College Manipal, Manipal academy of Higher education, Manipal, Karnataka, India
| | - Santosh Prabhu
- Pediatric Surgery, Kasturba Medical College Manipal, Manipal academy of Higher education, Manipal, Karnataka, India
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Li YY, Zang JF, Zhang C. Laparoscopic treatment of inflammatory myofibroblastic tumor in liver: A case report. World J Clin Cases 2022; 10:11853-11860. [PMID: 36405255 PMCID: PMC9669864 DOI: 10.12998/wjcc.v10.i32.11853] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/11/2022] [Revised: 07/12/2022] [Accepted: 08/30/2022] [Indexed: 11/07/2022] Open
Abstract
BACKGROUND Inflammatory myofibroblastic tumor in the liver (IMTL) is a rare borderline mesenchymal tumor. Neither clinical symptoms nor laboratory tests have absolute specificity for the diagnosis of IMTL, and imaging also lacks obvious specificity. Although there are sporadic reports of recurrence after surgical treatment, surgical resection is the mainstay of treatment.
CASE SUMMARY A 29-year-old man complained of general weakness, slight discomfort in the upper abdomen, with a history of upper respiratory tract infection for 1 wk before admission. Plain and enhanced upper abdominal magnetic resonance imaging showed a mass in liver segments II and III (48 mm × 53 mm). He was treated by laparoscopic left lateral segmentectomy. Postoperative pathological examination with hematoxylin and eosin staining suggested that the mass in liver segments II and III was IMTL. During 21 mo postoperative follow-up, no obvious residual or recurrent lesions were observed.
CONCLUSION There is a risk of malignant degeneration in IMTL. The principal choice of treatment is laparoscopic left lateral segmentectomy.
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Affiliation(s)
- Yang-Yang Li
- Department of Hepatobiliary Surgery, Taizhou People’s Hospital, The Fifth Affiliated Hospital of Medical School of Nantong University, Taizhou 225300, Jiangsu Province, China
| | - Jin-Feng Zang
- Department of Hepatobiliary Surgery, Taizhou People’s Hospital, The Fifth Affiliated Hospital of Medical School of Nantong University, Taizhou 225300, Jiangsu Province, China
| | - Chi Zhang
- Department of Hepatobiliary Surgery, Taizhou People’s Hospital, The Fifth Affiliated Hospital of Medical School of Nantong University, Taizhou 225300, Jiangsu Province, China
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Al Shenawi H, Al-Shaibani SA, Al Saad SK, Al-Sindi F, Al-Sindi K, Al Shenawi N, Naguib Y, Yaghan R. An extremely rare case of malignant jejunal mesenteric inflammatory myofibroblastic tumor in a 61-year-old male patient: A case report and literature review. Front Med (Lausanne) 2022; 9:1042262. [PMID: 36425100 PMCID: PMC9679529 DOI: 10.3389/fmed.2022.1042262] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/12/2022] [Accepted: 10/18/2022] [Indexed: 11/03/2023] Open
Abstract
Introduction A mesenteric inflammatory myofibroblastic tumor (IMT) is a rare solid tumor of intermediate malignant potential that affects children, adolescents, and young adults predominantly. IMT is mostly encountered in the lung. We report a case of malignant jejunal mesenteric IMT in a 61-year-old male patient who presented with vague abdominal pain and generalized weakness. CT scan revealed a mesenteric mass displacing the attached jejunum. Surgical resection was curative. Discussion An extensive literature review was performed to update and further analyze the already available data. A total of 35 cases with mesenteric IMT were reported previously. Only five cases of jejunal mesenteric IMT were reported. Mesenteric IMT demands vast effort to reveal the diagnosis due to its vagueness in the clinical presentation. Mesenteric IMT resembles each other in plenty of pathological and immunohistochemical characteristics. Conclusion To the best of our knowledge, this is the first case of malignant jejunal mesenteric IMT in the elderly. Surgical resection was curative.
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Affiliation(s)
- Hamdi Al Shenawi
- Department of Surgery, College of Medicine and Medical Sciences, Arabian Gulf University, Manama, Bahrain
| | | | - Suhair K. Al Saad
- Department of Surgery, College of Medicine and Medical Sciences, Arabian Gulf University, Manama, Bahrain
| | - Fedaa Al-Sindi
- Department of Pathology, King Hamad University Hospital, Busaiteen, Bahrain
| | - Khalid Al-Sindi
- Department of Pathology, King Hamad University Hospital, Busaiteen, Bahrain
| | - Noor Al Shenawi
- College of Medicine and Medical Sciences, Arabian Gulf University, Manama, Bahrain
| | - Yahya Naguib
- Department of Physiology, College of Medicine and Medical Sciences, Arabian Gulf University, Manama, Bahrain
- Department of Clinical Physiology, Faculty of Medicine, Menoufia University, Menoufia, Egypt
| | - Rami Yaghan
- Department of Surgery, College of Medicine and Medical Sciences, Arabian Gulf University, Manama, Bahrain
- Department of Surgery, Jordan University of Science and Technology, Irbid, Jordan
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Karaisli S, Kamer E, Ekinci N, Cengiz F, Er A, Peskersoy M. Inflammatory myofibroblastic tumour of the colon: 2 case reports and a comprehensive review of the literature. Int J Colorectal Dis 2020; 35:947-958. [PMID: 32100112 DOI: 10.1007/s00384-020-03522-0] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 01/26/2020] [Indexed: 02/04/2023]
Abstract
PURPOSE Inflammatory myofibroblastic tumour (IMT), which is also named as plasma cell granuloma (PCG) or inflammatory pseudotumour (IPT), is a rare tumour which rarely develops in the colorectal region. We aimed to review all reported cases to draw attention about this rare tumour. METHODS We present two new cases of colonic IMT with no recurrence during the follow-up period. We also reviewed previously reported colorectal IMT/IPT/PCG patients to investigate demographics, diagnosis and treatment modalities. RESULTS A total of 60 patients which including our 2 patients and 58 patients from 42 published articles were analysed. Male/female ratio was 34/26. Mean age was found to be 31.84 ± 22.26 years (9 months-82 years). Abdominal pain (56.7%) and fever (23.3%) were the most common complaints in the first admission. Fifty-nine (98.3%) out of 60 patients underwent surgery. During follow-up, 7 (14.3%) patients developed a local recurrence. CONCLUSION IMT may occur at any age. IMT is considered to be a borderline tumour with the potential for recurrence or distant metastasis. Complete resection of the tumour is recommended for treatment. Long-time follow-up is necessary due to recurrence potential of the tumour even many years after complete surgical resection. TRIAL REGISTRATION The study follows the regulation of the Institutional Review Board for human research at Izmir Katip Celebi University Ataturk Training and Research Hospital. Written informed consents were obtained from the patients who participated in this study.
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Affiliation(s)
- Serkan Karaisli
- Department of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey.
| | - Erdinc Kamer
- Department of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Nese Ekinci
- Department of Pathology, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Fevzi Cengiz
- Department of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Ahmet Er
- Department of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
| | - Mustafa Peskersoy
- Department of General Surgery, Izmir Katip Celebi University Ataturk Training and Research Hospital, Izmir, Turkey
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Alan O, Kuzhan O, Koca S, Telli TA, Basoglu T, Ercelep O, Filinte D, Sengul Y, Arikan H, Kaya S, Babacan NA, Dane F, Yumuk PF. How long should we continue crizotinib in ALK translocation-positive inflammatory myofibroblastic tumors? Long-term complete response with crizotinib and review of the literature. J Oncol Pharm Pract 2019; 26:1011-1018. [PMID: 31615346 DOI: 10.1177/1078155219879757] [Citation(s) in RCA: 12] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/12/2022]
Abstract
INTRODUCTION Inflammatory myofibroblastic tumor is a rare disease which is typically seen in children and young adults. Approximately half of the inflammatory myofibroblastic tumors contain translocations that result in over-expression of anaplastic lymphoma kinase gene. Herein, we present two anaplastic lymphoma kinase-positive cases with long-term remission with crizotinib. We do not know how long these therapies need to be continued. CASE REPORTS We present two cases of inflammatory myofibroblastic tumor treated with anaplastic lymphoma kinase inhibitor therapies: an 8-year-old Turkish boy and a 21-year-old Caucasian man. MANAGEMENT AND OUTCOME Two cases, both with good tumor control under crizotinib, but one who progressed on drug holiday, responded again to the same drug, and had a very short period of response after restarting crizotinib. CONCLUSION A molecular-targeted drug (anaplastic lymphoma kinase inhibitor) was found to be extremely effective as selective therapy for inflammatory myofibroblastic tumor with anaplastic lymphoma kinase translocation. Here, we want to emphasize the continuation of this treatment after achieving a good response until progression or a major side effect.
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Affiliation(s)
- Ozkan Alan
- Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Okan Kuzhan
- Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Yeditep University, Istanbul, Turkey
| | - Sinan Koca
- Division of Medical Oncology, Umraniye Research and Training Hospital, Istanbul, Turkey
| | - Tugba Akin Telli
- Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Tugba Basoglu
- Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Ozlem Ercelep
- Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Deniz Filinte
- Department of Pathology, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Yildiz Sengul
- Department of Radiology, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Huseyin Arikan
- Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Serap Kaya
- Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Nalan Akgul Babacan
- Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Faysal Dane
- Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey
| | - Perran Fulden Yumuk
- Division of Medical Oncology, Department of Internal Medicine, Faculty of Medicine, Marmara University, Istanbul, Turkey
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Gallegos-Sierra C, Belmonte-Chico Goerne MA, Ramírez-Jaimez J, de la Cruz-Temores S, Leonher-Ruezga KL. [Intestinal intussusception secondary to myofibroblastic tumor in an elderly patient. Case report]. CIR CIR 2016; 85:444-448. [PMID: 27568400 DOI: 10.1016/j.circir.2016.06.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/09/2015] [Revised: 04/09/2016] [Accepted: 06/02/2016] [Indexed: 11/29/2022]
Abstract
BACKGROUND Intussusception is defined as a segment of the gastrointestinal tract and mesentery within the lumen of an adjacent segment. It is a rare condition in adults that can occur anywhere in the gastrointestinal tract from the stomach to the rectum. Only 5% of all intussusceptions are presented in adults, and in 1-5% of all cases of intestinal obstruction. Inflammatory myofibroblastic tumour is rare, and is usually found in the lung, and rarely detected in some intestinal portions. It causes a variety of non-specific symptoms, with those that present as an intussusception being uncommon. CLINICAL CASE A female of 69 years with partial bowel obstruction secondary to intestinal intussusception due to an inflammatory myofibroblastic tumour, a rarely diagnosed condition and never published before. DISCUSSION Inflammatory myofibroblastic tumours are rare, and in this case with an atypical presentation that was surgically resolved satisfactorily. These entities are difficult to diagnose, with histopathology giving the definitive diagnosis. A literature review was performed to gather recent information about their diagnosis and treatment. CONCLUSIONS Inflammatory myofibroblastic tumours require a high level of suspicion, as diagnosis prior to surgery is difficult. Surgery is considered the treatment of choice, requiring leaving free surgical edges to prevent recurrences.
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Affiliation(s)
| | | | - Juan Ramírez-Jaimez
- Servicio de Cirugía General, Hospital Dr. Valentín Gómez Farías, Zapopan, Jalisco, México
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Dalton BGA, Thomas PG, Sharp NE, Manalang MA, Fisher JE, Moir CR, St Peter SD, Iqbal CW. Inflammatory myofibroblastic tumors in children. J Pediatr Surg 2016; 51:541-4. [PMID: 26732283 DOI: 10.1016/j.jpedsurg.2015.11.015] [Citation(s) in RCA: 42] [Impact Index Per Article: 4.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/27/2015] [Revised: 11/16/2015] [Accepted: 11/17/2015] [Indexed: 01/22/2023]
Abstract
BACKGROUND Inflammatory myofibroblastic tumor (IMFT) is an uncommon neoplasm in children. METHODS Retrospective review from 1993 to 2014 of patients ≤18years of age with a histopathologic diagnosis of IMFT treated at two tertiary centers. RESULTS Thirty-two patients were diagnosed with IMFT. Mean (±SD) age was 9.3±5.7years at diagnosis. Tumor location was variable: abdomen/pelvis (28%), head/neck region (22%), intrathoracic (22%), genitourinary (9%), bowel (6%) liver (6%), and musculoskeletal (6%). Median follow-up was 2.6±4.6years, with 3 recurrences and 2 deaths, which occurred only after recurrence. Positive microscopic margin after resection was associated with recurrence, compared to those that had a negative margin (40% vs. 0%, p=0.04). Recurrence was associated with increased mortality (67% vs 0%, p=0.01). Time from first symptoms to resection was shorter in those with recurrence (25.8±22 vs. 179±275days, p=0.01) and in nonsurvivors (44.0±8.0 vs. 194.3±53.4days, p=0.02). Adjuvant chemotherapy, not including steroid monotherapy, either given before or after resection, was administered more often to nonsurvivors (100% vs 4%, p=0.009), and use of corticosteroids was also higher in the nonsurvivors (100% vs. 15%, p=0.04). CONCLUSIONS IMFT is a rare pediatric neoplasm with variable locations. Complete excision is critical for cure. Proposed guidelines for diagnosis, treatment and surveillance of theses tumors in children are reported.
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Affiliation(s)
| | | | - Nicole E Sharp
- Children's Mercy Hospital, Kansas City, MO, United States
| | | | - James E Fisher
- Mayo Clinic Division of Pediatric Surgery, Rochester, MN, United States
| | | | | | - Corey W Iqbal
- Children's Mercy Hospital, Kansas City, MO, United States.
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11
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Kiziltan R, Yilmaz O, Almali N, Peksen C. Inflammatory myofibroblastic tumor: A rare cause of invagination in adults. Pak J Med Sci 2016; 32:260-2. [PMID: 27022387 PMCID: PMC4795880 DOI: 10.12669/pjms.321.9326] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/15/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) is a distinct pseudosarcomatous lesion arising in the soft tissues and interior organs of children and young adults. It is rarely seen in adults. It was first described in lungs. IMT can occur in any location in the body. However, it is seen most commonly in lungs, intestinal mesentery and liver. Non-mesenteric alimentary tract IMT's are quite rare. The presented case is an ileal IMT that caused small bowel invagination. A 38 year-old male patient presented to the emergency department with the complaint of diffuse abdominal pain, distension and no passage of gas or stools for two days. An abdominal examination revealed distension and tenderness in the abdomen with no guarding or rebound tenderness. Computerized tomography (CT) of the abdomen was ordered. CT revealed an image compatible with invagination of the right lower quadrant of the abdomen and a mass inside the lumen measuring 4x3x3cm. The mass causing invagination was detected during the surgical operation. A segmentary small bowel resection and ileoileal anastomosis was performed. The patient was discharged uneventfully on the postoperative sixth day. The diagnosis of IMT was confirmed histologically and immunochemically.
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Affiliation(s)
- Remzi Kiziltan
- Remzi Kiziltanepartment of General Surgery, DursunOdabas Medical Center, School of Medicine, University of Yuzuncuyil, Van, Turkey
| | - Ozkan Yilmaz
- Ozkan Yilmazepartment of General Surgery, DursunOdabas Medical Center, School of Medicine, University of Yuzuncuyil, Van, Turkey
| | - Necat Almali
- Necat Almali, Department of General Surgery, Training and Research Hospital, Van, Turkey
| | - Caghan Peksen
- Caghan Peksenepartment of General Surgery, DursunOdabas Medical Center, School of Medicine, University of Yuzuncuyil, Van, Turkey
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12
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Voluminous omental inflammatory myofibroblastic tumor in an elderly man: a case report and literature review. Case Rep Surg 2015; 2015:873758. [PMID: 25688324 PMCID: PMC4320915 DOI: 10.1155/2015/873758] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/10/2014] [Revised: 12/29/2014] [Accepted: 01/01/2015] [Indexed: 12/22/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) is a rare neoplasm of intermediate biologic potential, with uncertain etiology. This tumor occurs primarily in the lung, but the tumor may affect any organ system. A 75-year-old male was evaluated for voluminous palpable high abdominal mass with continuous and moderately abdominal pain, associated with abdominal distension for the last two months. Abdominal computed tomography showed a large (32 × 29 × 15 cm) heterogeneously enhanced mass with well-defined margins. At surgery, the mass originated from the greater omentum was completely excised. Histologically the tumor was a mesenchymal neoplasm in smooth muscle differentiation and was characterized by spindle-cell proliferation with lymphocytes, plasma cells, and rare eosinophils. Immunohistochemically, the tumor cells were positive for vimentin and smooth muscle actin and negative for anaplastic lymphoma kinase. Complete surgical resection of IMTs remains the mainstay of treatment associated with a low rate of recurrence. Final diagnosis should be based on histopathological and immunohistochemical findings. Appropriate awareness should be exercised by surgeons to abdominal IMTs in combination with constitutional symptoms, abnormal hematologic findings, and radiological definition, to avoid misdiagnosed.
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13
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Zhao JJ, Ling JQ, Fang Y, Gao XD, Shu P, Shen KT, Qin J, Sun YH, Qin XY. Intra-abdominal inflammatory myofibroblastic tumor: Spontaneous regression. World J Gastroenterol 2014; 20:13625-13631. [PMID: 25309095 PMCID: PMC4188916 DOI: 10.3748/wjg.v20.i37.13625] [Citation(s) in RCA: 25] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/16/2014] [Revised: 04/01/2014] [Accepted: 05/29/2014] [Indexed: 02/06/2023] Open
Abstract
Inflammatory myofibroblastic tumors are usually treated by surgical resection. We herein report two cases of intra-abdominal inflammatory myofibroblastic tumors that were unresectable and underwent spontaneous regression without any treatment. Our case report and literature review show that regression is more common in the middle-aged and older male populations. Abdominal discomfort and fever were the most common symptoms, but the majority of patients had no obvious physical signs. There was no specific indicator for diagnosis. The majority of the lesions regressed within 3 mo and nearly all of the masses completely resolved within 1 year. We conclude that the clinical characteristics of inflammatory myofibroblastic tumors are variable and, accordingly, the disease needs to be subdivided and treated on an individual basis. Surgery is always the first-line treatment; however, for those masses assessed as unresectable, conservative therapy with intense follow-up should be considered.
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14
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Oh E, Ro JY, Gardner JM, Kim JW, Jung WH, Yoon SO. Inflammatory myofibroblastic tumor of the appendix arising after treatment of gastric cancer: a case report and review of the literature. APMIS 2014; 122:657-9. [PMID: 24989630 DOI: 10.1111/apm.12205] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/10/2013] [Accepted: 08/30/2013] [Indexed: 12/28/2022]
Abstract
Inflammatory myofibroblastic tumor (IMT) is a rare soft tissue neoplasm of uncertain malignant potential and unclear etiology. IMT involving the appendix is very rare. Herein, we report a case of IMT of the appendix in a gastric cancer patient who was treated with radical gastrectomy and adjuvant systemic chemotherapy. Rare cases of IMT associated with preceding events have been described in other organs/sites, but not in the appendix. A previous intra-abdominal operation for gastric cancer may contribute to the development of IMT in the appendix as seen in the present patient. To our knowledge, this is the first case of appendiceal IMT arising after a previous operation.
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Affiliation(s)
- Eunji Oh
- Department of Pathology, Gangnam Severance Hospital, Yonsei University College of Medicine, Seoul, Korea
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15
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Appak YÇ, Sahin GE, Ayhan S, Taneli C, Kasırga E. Inflammatory myofibroblastic tumor of the colon with an unusual presentation of intestinal intussusception. European J Pediatr Surg Rep 2014; 2:54-7. [PMID: 25755972 PMCID: PMC4336076 DOI: 10.1055/s-0034-1370774] [Citation(s) in RCA: 24] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/21/2013] [Accepted: 12/16/2013] [Indexed: 12/29/2022] Open
Abstract
Inflammatory myfibroblastic tumor (IMT), also known as inflammatory pseudotumor is unusual, benign solid tumor. This tumor is commonly reported in the lungs but can be present in extrapulmonary sites as well. We present the case of a 7-year-old girl with IMT in an unusual location. The patient was admitted with abdominal pain, and ultrasound showed a solid mass in the abdomen. She was operated and colocolic intussusception secondary to a mass was found. Histologic evaluation of mass revealed IMT.
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Affiliation(s)
- Yeliz Çağan Appak
- Department of Pediatric Gastroenterology, Faculty of Medicine, Celal Bayar University, Manisa, Turkey
| | - Gülseren Evirgen Sahin
- Department of Pediatric Gastroenterology, Faculty of Medicine, Celal Bayar University, Manisa, Turkey
| | - Semin Ayhan
- Department of Pathology, Faculty of Medicine, Celal Bayar University, Manisa, Turkey
| | - Can Taneli
- Department of Pediatric Surgery, Faculty of Medicine, Celal Bayar University, Manisa, Turkey
| | - Erhun Kasırga
- Department of Pediatric Gastroenterology, Faculty of Medicine, Celal Bayar University, Manisa, Turkey
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16
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He CY, Dong GH, Liu HG. Recurrent laryngeal inflammatory myofibroblastic tumor with positive anaplastic lymphoma kinase mimicking recurrent respiratory papillomatosis: a case report. World J Surg Oncol 2014; 12:54. [PMID: 24602144 PMCID: PMC3996035 DOI: 10.1186/1477-7819-12-54] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/30/2013] [Accepted: 02/19/2014] [Indexed: 12/24/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) of the larynx is an unusual lesion, particularly in the pediatric age group. Laryngeal IMTs in children follow a benign clinical course with reports of only rare recurrences and no metastases. Although anaplastic lymphoma kinase (ALK) has been associated with IMTs, there is only one pediatric laryngeal IMT reported to be ALK-positive with immunohistochemical staining. Here, we present a case of a 10-year-old boy with a laryngeal IMT that recurred four times and was misdiagnosed as recurrent respiratory papillomatosis after the initial three operations. ALK positivity was demonstrated by both immunohistochemical staining and fluorescence in situ hybridization. To the best of our knowledge, this case report is the first to describe a laryngeal IMT that recurred multiple times and was confirmed to be ALK-positive at the molecular level.
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Affiliation(s)
| | | | - Hong-gang Liu
- Department of Pathology, Beijing Tongren Hospital, Capital Medical University, No,1 Dongjiaominxiang Street, Dongcheng District, Beijing 100730, China.
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17
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Fu J, Wu Q, Zhang Y, Zhang X, Wang Z. Primary inflammatory myofibroblastic tumor of the diaphragm: Report of a case. Thorac Cancer 2013; 4:449-452. [PMID: 28920230 DOI: 10.1111/j.1759-7714.2012.00155.x] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/28/2022] Open
Abstract
We report a case of primary inflammatory myofibroblastic tumor of the diaphragm in a 64-year-old man. The patient was hospitalized for a computed tomography (CT)-detected large tissue mass at the left lower lung field. Complete tumor excision followed by pathological investigation was performed. Microscopically, the tumor showed staggered arrangements of spindle myoepithelial cells, lymphoblastic and eosinophil cells. Immunohistochemically, the proliferating spindle cells showed positive staining for smooth muscle actin, vimentin, CD68 and Desmin, but negative for cytokeratin, leukocyte common antigen, epithelial membrane antigen, and S-100. This is the first reported inflammatory myofibroblastic tumor of the diaphragm found in an adult. The postoperative course was uneventful and the patient had no recurrence 18 months after surgery.
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Affiliation(s)
- Junke Fu
- Department of Thoracic Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China Department of Hepatobiliary Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China
| | - Qifei Wu
- Department of Thoracic Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China Department of Hepatobiliary Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China
| | - Yong Zhang
- Department of Thoracic Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China Department of Hepatobiliary Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China
| | - Xufeng Zhang
- Department of Thoracic Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China Department of Hepatobiliary Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China
| | - Zhe Wang
- Department of Thoracic Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China Department of Hepatobiliary Surgery, the First Affiliated Hospital of Medical Collage, Xi'an Jiaotong University, Xi'an, Shaanxi Province, China
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18
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Yi XL, Lu HY, Wu YX, Li WH, Meng QG, Cheng JW, Tang Y, Liu Y, Bai XZ. Inflammatory myofibroblastic tumor with extensive involvement of the bladder in an adolescent: a case report. World J Surg Oncol 2013; 11:206. [PMID: 23957966 PMCID: PMC3765871 DOI: 10.1186/1477-7819-11-206] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/15/2013] [Accepted: 08/07/2013] [Indexed: 11/10/2022] Open
Abstract
Inflammatory myofibroblastic tumor (IMT) is a rare lesion of unclear pathogenesis that shows a wide, highly variable spectrum of clinical behavior. We describe the case of a 17-year-old boy with a large IMT that infiltrated the bladder, ileocecal junction, peritoneum and pelvic retroperitoneal space. The tumor was associated with extensive toughening and thickening of the bladder, and, although it showed a tendency for invasive growth, it affected mainly the bladder and adjacent tissue. To the best of our knowledge, this case report is the first to describe an IMT involving the entire bladder and several adjacent pelviabdominal organs. The bladder wall was tough and could hardly be cut by scalpel. Levels of inflammatory response markers such as C-reactive protein fell after surgery.
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Affiliation(s)
- Xin Lin Yi
- Department of Urology, Cancer Hospital of Guangxi Medical University & Guangxi Cancer Research Institute, Nanning 530021, People's Republic of China.
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19
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Gupta RK, Samalavicius NE, Sapkota S, Sah PL, Kafle SU. Colonic inflammatory myofibroblastic tumours: an institutional review. Colorectal Dis 2013; 15:e239-43. [PMID: 23350604 DOI: 10.1111/codi.12149] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/25/2012] [Accepted: 09/13/2012] [Indexed: 02/08/2023]
Abstract
AIM The aim of the study was to present the largest series of colonic inflammatory myofibroblastic tumour (C-IMFT) in the literature so far and to provide a review of this condition. METHOD A retrospective review was carried out of a consecutive series of patients diagnosed with a C-IMFT at a community-based hospital with a specialized gastrointestinal unit between 2002 and 2011. The main outcome measures were success rate and postoperative complications. Using a set of terms we searched the PubMed database for papers published on C-IMFT. We reviewed the data from these studies and case reports. RESULTS There were seven patients with a histopathologically proven C-IMFT. The patients' mean age was 39 ± 11.3 years. Four presented with clinical features of intestinal obstruction of varying severity and three with symptoms of anaemia. Complete surgical resection with end-to-end anastomosis was performed. The gross morphology included polypoidal myxoid tumours that served as a lead point for intussusception in two cases, a whorled mass in two and a circumferential infiltrative tumour in three. Microscopically, all tumours had typical features of IMFT with a variable expression of anaplastic lymphoma kinase (ALK-1) and tumour-free resection margins. All patients were well without local recurrence or metastasis at a mean follow-up of 46.8 ± 11.9 months. CONCLUSION Surgical resection is effective for this rare tumour which mostly behaves in a benign manner. Our review supports the need for patients to be followed up for long periods because of the possibility of metastasis or late recurrence.
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Affiliation(s)
- R K Gupta
- Department of Surgery, B. P. Koirala Institute of Health Sciences, Dharan, Nepal.
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20
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Affiliation(s)
- Simona Gurzu
- Department of Surgery, University of Medicine and Pharmacy of Targu-Mures, Targu Mures, Romania.
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21
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Satahoo SS, Brathwaite C, Davis JS, Burnweit C. Obstructing apple core lesion of the rectum: a case report of inflammatory pseudotumor masquerading as colorectal carcinoma. J Pediatr Surg 2013; 48:677-80. [PMID: 23480933 DOI: 10.1016/j.jpedsurg.2013.01.034] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/05/2012] [Revised: 01/02/2013] [Accepted: 01/09/2013] [Indexed: 12/17/2022]
Abstract
Inflammatory pseudotumor is a rare lesion which can occur, typically in children and young adults, in many different organ systems. The tumor often clinically behaves like a cancer but without histological evidence of malignancy. This case study of a 14 year-old boy is the first report in the literature of an inflammatory pseudotumor presenting as an obstructing apple core lesion, mimicking a rectal carcinoma. A six-week course of non-steroidal, anti-inflammatory drugs (NSAIDs) led to complete resolution of the mass, and following resection of a residual stricture, the patient has been recurrence-free for seven years.
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Affiliation(s)
- Shevonne S Satahoo
- Department of Surgery, University of Miami Miller School of Medicine, Miami, FL 33136, USA.
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22
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Kye BH, Kim HJ, Kang SG, Yoo C, Cho HM. A case of inflammatory myofibroblastic tumor originated from the greater omentum in young adult. JOURNAL OF THE KOREAN SURGICAL SOCIETY 2012; 82:380-4. [PMID: 22708101 PMCID: PMC3373989 DOI: 10.4174/jkss.2012.82.6.380] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 11/09/2011] [Revised: 12/05/2011] [Accepted: 12/12/2011] [Indexed: 01/21/2023]
Abstract
Inflammatory myofibroblastic (IMF) tumor is a rare solid tumor that often affects children. IMF tumors occur primarily in the lung, but the tumor may affect any organ system with protean manifestations. A 22-year-old woman was evaluated for palpable low abdominal mass that had been increasing in size since two months prior. Abdominal computed tomography showed a lobulated, heterogeneous contrast enhancing soft tissue mass, 6.5 × 5.7 cm in size in the ileal mesentery. At surgery, the mass originated from the greater omentum laying in the pelvic cavity and was completely excised without tumor spillage. Histologically, the mass was a spindle cell lesion with severe atypism and some mitosis. Immunohistochemistry for anaplastic lymphoma kinase-1 revealed that the lesion was an IMF tumor. Because of its local invasiveness and its tendency to recur, this tumor can be confused with a soft tissue sarcoma. Increasing physician awareness of this entity should facilitate recognition of its clinical characteristics and laboratory findings.
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Affiliation(s)
- Bong Hyeon Kye
- Department of Surgery, St. Vincent's Hospital, The Catholic University of Korea School of Medicine, Suwon, Korea
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23
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Kim EY, Lee IK, Lee YS, Yang N, Chung DJ, Yim KI, Kim JI, Oh ST. Inflammatory myofibroblastic tumor in colon. JOURNAL OF THE KOREAN SURGICAL SOCIETY 2011; 82:45-9. [PMID: 22324046 PMCID: PMC3268143 DOI: 10.4174/jkss.2012.82.1.45] [Citation(s) in RCA: 29] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 06/13/2011] [Revised: 06/21/2011] [Accepted: 06/24/2011] [Indexed: 01/22/2023]
Abstract
Inflammatory myofibroblastic tumor (IMT) is an uncommon mesenchymal solid tumor commonly documented in children and young adults. Here, we report a case of IMT in colon confirmed pathologically after laparoscopic anterior resection. A 35-year-old man presented with anal bleeding after defecation for 2 weeks. Colonoscopy demonstrated a mass with shallow ulceration in the central area and irregular margin accompanied by intact mucosa in the descending colon. Computer tomography showed a well-demarcated and homogenous solitary mass in the descending colon. We performed laparoscopic anterior resection. This case was diagnosed as IMT after microscopic examination. The tumor was composed of a proliferation of spindle-shaped cells arranged in the hyaline material with chronic inflammatory cells, composed mainly of plasma cells and lymphocytes. Immunohistochemically, tumor cells were positive for smooth muscle actin, and vimentin, and negative for desmin, CD117 (c-kit), anaplastic lymphoma kinase-1.
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Affiliation(s)
- Eun Young Kim
- Department of Surgery, The Catholic University of Korea School of Medicine, Seoul, Korea
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24
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Fragoso AC, Eloy C, Estevão-Costa J, Campos M, Farinha N, Lopes JM. Abdominal inflammatory myofibroblastic tumor a clinicopathologic study with reappraisal of biologic behavior. J Pediatr Surg 2011; 46:2076-82. [PMID: 22075336 DOI: 10.1016/j.jpedsurg.2011.07.009] [Citation(s) in RCA: 26] [Impact Index Per Article: 1.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/09/2011] [Revised: 07/04/2011] [Accepted: 07/04/2011] [Indexed: 12/17/2022]
Abstract
BACKGROUND AND PURPOSE Inflammatory myofibroblastic tumor (IMT) is a proliferative lesion of controversial nosology and uncertain prognosis. In an attempt to acquire further understanding of pathogenesis and biologic behavior, we surveyed abdominal IMTs managed over the last 12 years at a single institution. METHODS Intra-abdominal IMTs treated between 1995 and 2007 were reviewed concerning demographic, clinical, and pathologic features as well as therapeutic management and outcome. All specimens were reevaluated by histologic examination and immunohistochemistry. RESULTS There were 7 patients (4 males; age range, 28 days to 14 years). Five lesions were located in alimentary tract: 1 gastric presenting with bleeding, 1 hepatic presenting with a thoracic wall mass, 1 pancreatic and 2 colonic presenting with obstructive symptoms. One splenic IMT was found incidentally. The remaining case arose from the adrenal gland and presented with a palpable mass. The gastric and adrenal IMTs had evidence of a previous or concomitant infectious setting. Five lesions were excised. The pancreatic IMT underwent a drainage procedure followed by steroid administration, and the hepatic lesion received antibiotics. Histopathology revealed characteristic findings of IMT. Expression of anaplastic lymphoma kinase was negative in all cases. At a median follow-up of 6 years (range, 3-15), all children were asymptomatic with no recurrences. The hepatic and pancreatic IMT displayed complete and near total regression, respectively. CONCLUSION A benign behavior of abdominal IMTs was observed even in patients not undergoing surgical excision. Although IMT remains a surgical disease, a conservative approach may be reasonable in select cases.
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Affiliation(s)
- Ana Catarina Fragoso
- Division of Pediatric Surgery, Faculty of Medicine, University of Porto, Hospital S. João, 4200-319 Porto, Portugal
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25
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Diagnostic puzzle of inflammatory pseudotumor of the urinary bladder: a case report with brief literature review. South Med J 2010; 103:563-6. [PMID: 20710142 DOI: 10.1097/smj.0b013e3181de0ecb] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
Abstract
Inflammatory pseudotumor of the urinary bladder is a rare reactive proliferation with a clinical presentation similar to malignant neoplasms. We present the case of a 35-year-old woman who presented with left lower quadrant pain and gross hematuria. A diagnosis of cystitis glandularis was initially considered; however, the symptoms did not resolve following transurethral resection. Subsequently, a partial cystectomy was performed after malignancy was excluded based on intraoperative frozen sections. Further histopathological evaluation confirmed the diagnosis of inflammatory pseudotumor.
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26
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Böhme B, Ngendahayo P, Hamaide A, Heimann M. Inflammatory pseudotumours of the urinary bladder in dogs resembling human myofibroblastic tumours: a report of eight cases and comparative pathology. Vet J 2010; 183:89-94. [PMID: 18926743 DOI: 10.1016/j.tvjl.2008.08.025] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/24/2008] [Revised: 08/19/2008] [Accepted: 08/30/2008] [Indexed: 01/15/2023]
Abstract
Eight cases of inflammatory myofibroblastic tumours (IMT) of the urinary bladder in dogs are described, including their clinical, imaging, morphological, and histopathological aspects. Old small breed female dogs were more commonly affected. Haematuria was the main clinical sign, often associated with dysuria and crystalluria. The IMT appeared as single or multiple, polypoid, firm masses, measuring 1-7 cm. Histologically, the masses were well delineated, covered by a benign hyperplastic urothelium and diffusely infiltrated by a mixed inflammatory component. The cells were spindle-shaped, adopting a fascicular, pallisading or whirling organisation and were vimentin and variably desmin and actin positive. The nuclei were globular, containing open chromatin and prominent nucleoli. Mild to moderate pleomorphism and little mitotic activity were present. These morphological features are compatible with human myofibroblastic inflammatory tumours.
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Affiliation(s)
- Beatrice Böhme
- Department of Companion Animal Sciences, Small Animal Surgery Section, School of Veterinary Medicine, University of Liège, Belgium
| | - Placide Ngendahayo
- Human Pathology Department of the Institute of Pathology and Genetic, Gosselies, Belgium
| | - Annick Hamaide
- Department of Companion Animal Sciences, Small Animal Surgery Section, School of Veterinary Medicine, University of Liège, Belgium
| | - Marianne Heimann
- Veterinary Pathology Department of Bio.be, 25 rue Georges Lemaitre, 6041 Gosselies, Hainaut, Belgium.
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