Successful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the lung

Abstract Background Inflammatory myofibroblastic tumor (IMT) of the lung is rare. This disease often shows neoplastic features with anaplastic lymphoma kinase positiveness as well as inflammatory features, such as steroid-responsive immunoglobulin G4 (IgG4)-related sclerosing disease. Since many cas...

Full description

Saved in:
Bibliographic Details
Main Authors: Hironosuke Watanabe, Naoya Yamasaki, Takuro Miyazaki, Keitaro Matsumoto, Tomoshi Tsuchiya, Kuniko Abe, Takeshi Nagayasu
Format: Article
Language:English
Published: Japan Surgical Society 2019-07-01
Series:Surgical Case Reports
Subjects:
Online Access:http://link.springer.com/article/10.1186/s40792-019-0674-x
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1849414194408980480
author Hironosuke Watanabe
Naoya Yamasaki
Takuro Miyazaki
Keitaro Matsumoto
Tomoshi Tsuchiya
Kuniko Abe
Takeshi Nagayasu
author_facet Hironosuke Watanabe
Naoya Yamasaki
Takuro Miyazaki
Keitaro Matsumoto
Tomoshi Tsuchiya
Kuniko Abe
Takeshi Nagayasu
author_sort Hironosuke Watanabe
collection DOAJ
description Abstract Background Inflammatory myofibroblastic tumor (IMT) of the lung is rare. This disease often shows neoplastic features with anaplastic lymphoma kinase positiveness as well as inflammatory features, such as steroid-responsive immunoglobulin G4 (IgG4)-related sclerosing disease. Since many cases have been reported as advanced, various treatment strategies should be considered based on clinical and biological features of each case. Case presentation We report a 49-year-old male with IMT, which seemed invading the left atrium from preoperative imaging modalities. Serological and pathological examinations from the biopsy specimen revealed high expression of anaplastic lymphoma kinase expression in the tumor. On the other hand, IgG4/IgG ration in the tumor was small, where a therapeutic effect of steroid was not expected, leading to surgical treatment rather than a steroid administration. The tumor was completely resected en bloc with the right lower lobe of the lung and a part of the left atrium. The postoperative course of the patient was uneventful. The patient has remained recurrence free over 5 years from the surgery. Conclusion In this case, preoperative biological assessment prior to the treatment led to a good clinical course. We believe that molecular biological examination is important in the determination of treatment strategy for this rare disease as well as imaging modalities.
format Article
id doaj-art-68146e134e624ac28a862bbb625f8caa
institution Kabale University
issn 2198-7793
language English
publishDate 2019-07-01
publisher Japan Surgical Society
record_format Article
series Surgical Case Reports
spelling doaj-art-68146e134e624ac28a862bbb625f8caa2025-08-20T03:33:54ZengJapan Surgical SocietySurgical Case Reports2198-77932019-07-01511410.1186/s40792-019-0674-xSuccessful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the lungHironosuke Watanabe0Naoya Yamasaki1Takuro Miyazaki2Keitaro Matsumoto3Tomoshi Tsuchiya4Kuniko Abe5Takeshi Nagayasu6Division of Surgical Oncology, Department of Surgery, Nagasaki University Graduate School of Biomedical SciencesDivision of Surgical Oncology, Department of Surgery, Nagasaki University Graduate School of Biomedical SciencesDivision of Surgical Oncology, Department of Surgery, Nagasaki University Graduate School of Biomedical SciencesDivision of Surgical Oncology, Department of Surgery, Nagasaki University Graduate School of Biomedical SciencesDivision of Surgical Oncology, Department of Surgery, Nagasaki University Graduate School of Biomedical SciencesDepartment of Pathology, Nagasaki University Graduate School of Biomedical SciencesDivision of Surgical Oncology, Department of Surgery, Nagasaki University Graduate School of Biomedical SciencesAbstract Background Inflammatory myofibroblastic tumor (IMT) of the lung is rare. This disease often shows neoplastic features with anaplastic lymphoma kinase positiveness as well as inflammatory features, such as steroid-responsive immunoglobulin G4 (IgG4)-related sclerosing disease. Since many cases have been reported as advanced, various treatment strategies should be considered based on clinical and biological features of each case. Case presentation We report a 49-year-old male with IMT, which seemed invading the left atrium from preoperative imaging modalities. Serological and pathological examinations from the biopsy specimen revealed high expression of anaplastic lymphoma kinase expression in the tumor. On the other hand, IgG4/IgG ration in the tumor was small, where a therapeutic effect of steroid was not expected, leading to surgical treatment rather than a steroid administration. The tumor was completely resected en bloc with the right lower lobe of the lung and a part of the left atrium. The postoperative course of the patient was uneventful. The patient has remained recurrence free over 5 years from the surgery. Conclusion In this case, preoperative biological assessment prior to the treatment led to a good clinical course. We believe that molecular biological examination is important in the determination of treatment strategy for this rare disease as well as imaging modalities.http://link.springer.com/article/10.1186/s40792-019-0674-xInflammatory myofibroblastic tumorAnaplastic lymphoma kinaseImmune globulin G 4-related sclerosing disease
spellingShingle Hironosuke Watanabe
Naoya Yamasaki
Takuro Miyazaki
Keitaro Matsumoto
Tomoshi Tsuchiya
Kuniko Abe
Takeshi Nagayasu
Successful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the lung
Surgical Case Reports
Inflammatory myofibroblastic tumor
Anaplastic lymphoma kinase
Immune globulin G 4-related sclerosing disease
title Successful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the lung
title_full Successful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the lung
title_fullStr Successful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the lung
title_full_unstemmed Successful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the lung
title_short Successful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the lung
title_sort successful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase positive inflammatory myofibroblastic tumor of the lung
topic Inflammatory myofibroblastic tumor
Anaplastic lymphoma kinase
Immune globulin G 4-related sclerosing disease
url http://link.springer.com/article/10.1186/s40792-019-0674-x
work_keys_str_mv AT hironosukewatanabe successfultreatmentbasedonmolecularbiologicalassessmentofinvasiveanaplasticlymphomakinasepositiveinflammatorymyofibroblastictumorofthelung
AT naoyayamasaki successfultreatmentbasedonmolecularbiologicalassessmentofinvasiveanaplasticlymphomakinasepositiveinflammatorymyofibroblastictumorofthelung
AT takuromiyazaki successfultreatmentbasedonmolecularbiologicalassessmentofinvasiveanaplasticlymphomakinasepositiveinflammatorymyofibroblastictumorofthelung
AT keitaromatsumoto successfultreatmentbasedonmolecularbiologicalassessmentofinvasiveanaplasticlymphomakinasepositiveinflammatorymyofibroblastictumorofthelung
AT tomoshitsuchiya successfultreatmentbasedonmolecularbiologicalassessmentofinvasiveanaplasticlymphomakinasepositiveinflammatorymyofibroblastictumorofthelung
AT kunikoabe successfultreatmentbasedonmolecularbiologicalassessmentofinvasiveanaplasticlymphomakinasepositiveinflammatorymyofibroblastictumorofthelung
AT takeshinagayasu successfultreatmentbasedonmolecularbiologicalassessmentofinvasiveanaplasticlymphomakinasepositiveinflammatorymyofibroblastictumorofthelung