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Gorham's Disease With Chest Wall Involvement: A Case Report and a Review of the Literature

INTRODUCTION: Gorham's disease is a rare disorder characterized by osteolysis and abnormal vascular growth within bones. Diagnosis of Gorham's disease is often delayed and for accurate and early diagnosis high clinical suspicion is crucial. No specific treatment is available. Management op...

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Autores principales: Daneshvar Kakhaki, Abolghasem, Khodadad, Kian, Pejhan, Saviz, Karimi, Shirin, Arab, Mehrdad, Saghebi, Reza, Behgam Shadmehr, Mohammad, Farzanegan, Roya
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Kowsar 2014
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4329932/
https://www.ncbi.nlm.nih.gov/pubmed/25763205
http://dx.doi.org/10.5812/ircmj.12180
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author Daneshvar Kakhaki, Abolghasem
Khodadad, Kian
Pejhan, Saviz
Karimi, Shirin
Arab, Mehrdad
Saghebi, Reza
Behgam Shadmehr, Mohammad
Farzanegan, Roya
author_facet Daneshvar Kakhaki, Abolghasem
Khodadad, Kian
Pejhan, Saviz
Karimi, Shirin
Arab, Mehrdad
Saghebi, Reza
Behgam Shadmehr, Mohammad
Farzanegan, Roya
author_sort Daneshvar Kakhaki, Abolghasem
collection PubMed
description INTRODUCTION: Gorham's disease is a rare disorder characterized by osteolysis and abnormal vascular growth within bones. Diagnosis of Gorham's disease is often delayed and for accurate and early diagnosis high clinical suspicion is crucial. No specific treatment is available. Management options include surgery, radiation therapy and medical therapy. We aimed to present the first case of Gorham’s disease with chest wall involvement in Iran. By review of the literature we discussed important issues of this rare disease including clinical findings, diagnosis and treatment options. CASE PRESENTATION: We present a 48-year-old man with a history of dyspnea following a blunt chest trauma who was admitted to our clinic several times due to reaccumulation of pleural fluid and chylothorax. Gorham's disease was finally established according to clinical manifestations and radiological findings including massive osteolysis in his left ribs and also histological examination. DISCUSSION: According to review of the literature and considering all treatment modalities the patients was successfully treated with a combination of radiotherapy, pamidronate and thalidomide. We suggest that this disease should be considered among differential diagnoses of patients with chest pain, pleural effusion and/or chylothorax with an unknown reason and more importantly history of chest trauma. In suspected cases, it is essential to examine biopsy specimens of the bone adjacent to the inflammated tissues in order to confirm diagnosis.
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spelling pubmed-43299322015-03-11 Gorham's Disease With Chest Wall Involvement: A Case Report and a Review of the Literature Daneshvar Kakhaki, Abolghasem Khodadad, Kian Pejhan, Saviz Karimi, Shirin Arab, Mehrdad Saghebi, Reza Behgam Shadmehr, Mohammad Farzanegan, Roya Iran Red Crescent Med J Case Report INTRODUCTION: Gorham's disease is a rare disorder characterized by osteolysis and abnormal vascular growth within bones. Diagnosis of Gorham's disease is often delayed and for accurate and early diagnosis high clinical suspicion is crucial. No specific treatment is available. Management options include surgery, radiation therapy and medical therapy. We aimed to present the first case of Gorham’s disease with chest wall involvement in Iran. By review of the literature we discussed important issues of this rare disease including clinical findings, diagnosis and treatment options. CASE PRESENTATION: We present a 48-year-old man with a history of dyspnea following a blunt chest trauma who was admitted to our clinic several times due to reaccumulation of pleural fluid and chylothorax. Gorham's disease was finally established according to clinical manifestations and radiological findings including massive osteolysis in his left ribs and also histological examination. DISCUSSION: According to review of the literature and considering all treatment modalities the patients was successfully treated with a combination of radiotherapy, pamidronate and thalidomide. We suggest that this disease should be considered among differential diagnoses of patients with chest pain, pleural effusion and/or chylothorax with an unknown reason and more importantly history of chest trauma. In suspected cases, it is essential to examine biopsy specimens of the bone adjacent to the inflammated tissues in order to confirm diagnosis. Kowsar 2014-11-17 /pmc/articles/PMC4329932/ /pubmed/25763205 http://dx.doi.org/10.5812/ircmj.12180 Text en Copyright © 2014, Iranian Red Crescent Medical Journal. http://creativecommons.org/licenses/by-nc/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 International License (http://creativecommons.org/licenses/by-nc/4.0/) which permits copy and redistribute the material just in noncommercial usages, provided the original work is properly cited.
spellingShingle Case Report
Daneshvar Kakhaki, Abolghasem
Khodadad, Kian
Pejhan, Saviz
Karimi, Shirin
Arab, Mehrdad
Saghebi, Reza
Behgam Shadmehr, Mohammad
Farzanegan, Roya
Gorham's Disease With Chest Wall Involvement: A Case Report and a Review of the Literature
title Gorham's Disease With Chest Wall Involvement: A Case Report and a Review of the Literature
title_full Gorham's Disease With Chest Wall Involvement: A Case Report and a Review of the Literature
title_fullStr Gorham's Disease With Chest Wall Involvement: A Case Report and a Review of the Literature
title_full_unstemmed Gorham's Disease With Chest Wall Involvement: A Case Report and a Review of the Literature
title_short Gorham's Disease With Chest Wall Involvement: A Case Report and a Review of the Literature
title_sort gorham's disease with chest wall involvement: a case report and a review of the literature
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4329932/
https://www.ncbi.nlm.nih.gov/pubmed/25763205
http://dx.doi.org/10.5812/ircmj.12180
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