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1</a>&#41;&#46; Magnetic resonance confirmed the described osteolysis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#44; diffuse osteolysis in vertebral bodies and posterior elements of the cervical&#44; dorsal and lumbar spine&#44; multiple vertebral and dorsal flattening and secondary hyperkyphosis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Computerised tomography demonstrated destruction of the cortical bone in vertebral bodies &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Imaging tests showed a left laterocervical lymphangioma &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>&#41; and multicystic splenomegalia compatible with diagnosis of diffuse abdominal lymphangioma &#40;<a class="elsevierStyleCrossRef" href="#fig0030">Fig&#46; 6</a>&#41;&#46; The bone biopsy revealed discreet medullar lymphoid infiltration&#44; with no signs of malignancy&#46;</p><elsevierMultimedia ident="fig0025"></elsevierMultimedia><elsevierMultimedia ident="fig0030"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Based on clinical&#44; radiological and histopathological findings a diagnosis of Gorham-Stout syndrome was made&#46; The patient rejected radiotherapy and began oral treatment with alendronic acid &#40;70<span class="elsevierStyleHsp" style=""></span>mg&#47;week&#41; and sirolimus &#40;2<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#41; until the next control visit&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion&#47;comment</span><p id="par0025" class="elsevierStylePara elsevierViewall">Gorham-Stout syndrome is a sporadic bone disease&#44; 300 cases of which have been described&#44;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> approximately 50 in the vertebral spine&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> It is characterized by a progressive osteolysis with no bone regeneration&#44; with destruction of the cortical bone&#44; triggered by non neoplasic lymphangiogenesis and hemangiogenesis at medullar level&#44; which may infiltrate soft tissues and lead to fibrosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;5</span></a> Involvement is usually regional or multifocal&#44;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> mostly in the axial skeleton&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The evolutional course of the disease is unpredictable&#44; with up to 16&#37; mortality&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Diagnosis is made by exclusion&#44; ruling out lymphangiomatosis&#44; which does not injure the cortical bone&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;7</span></a> Magnetic resonance with gadolium contrast agent is used to monitor patient evolution&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> showing lymphangiogenesis in the active lesions&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Multifocal location and extensive involvement of the spine makes this case exceptional&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interests</span><p id="par0030" class="elsevierStylePara elsevierViewall">The authors have no conflict of interest to declare&#46;</p></span></span>"
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Images in Clinical Rheumatology
Massive osteolysis in a patient with Gorham-Stout syndrome
Osteolisis generalizada en un paciente con síndrome de Gorham-Stout
Irene María López García
Corresponding author
irene.lopgar1@gmail.com

Corresponding author.
, María Teresa Lorenzo Domínguez, Ana María Fernández Martínez, María Teresa Cuesta Marcos
Servicio de Radiodiagnóstico, Complejo Asistencial Universitario de León, León, Spain
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1</a>&#41;&#46; Magnetic resonance confirmed the described osteolysis &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#44; diffuse osteolysis in vertebral bodies and posterior elements of the cervical&#44; dorsal and lumbar spine&#44; multiple vertebral and dorsal flattening and secondary hyperkyphosis &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Computerised tomography demonstrated destruction of the cortical bone in vertebral bodies &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Imaging tests showed a left laterocervical lymphangioma &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>&#41; and multicystic splenomegalia compatible with diagnosis of diffuse abdominal lymphangioma &#40;<a class="elsevierStyleCrossRef" href="#fig0030">Fig&#46; 6</a>&#41;&#46; The bone biopsy revealed discreet medullar lymphoid infiltration&#44; with no signs of malignancy&#46;</p><elsevierMultimedia ident="fig0025"></elsevierMultimedia><elsevierMultimedia ident="fig0030"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Based on clinical&#44; radiological and histopathological findings a diagnosis of Gorham-Stout syndrome was made&#46; The patient rejected radiotherapy and began oral treatment with alendronic acid &#40;70<span class="elsevierStyleHsp" style=""></span>mg&#47;week&#41; and sirolimus &#40;2<span class="elsevierStyleHsp" style=""></span>mg&#47;day&#41; until the next control visit&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion&#47;comment</span><p id="par0025" class="elsevierStylePara elsevierViewall">Gorham-Stout syndrome is a sporadic bone disease&#44; 300 cases of which have been described&#44;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> approximately 50 in the vertebral spine&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> It is characterized by a progressive osteolysis with no bone regeneration&#44; with destruction of the cortical bone&#44; triggered by non neoplasic lymphangiogenesis and hemangiogenesis at medullar level&#44; which may infiltrate soft tissues and lead to fibrosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;5</span></a> Involvement is usually regional or multifocal&#44;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> mostly in the axial skeleton&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> The evolutional course of the disease is unpredictable&#44; with up to 16&#37; mortality&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Diagnosis is made by exclusion&#44; ruling out lymphangiomatosis&#44; which does not injure the cortical bone&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;7</span></a> Magnetic resonance with gadolium contrast agent is used to monitor patient evolution&#44;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> showing lymphangiogenesis in the active lesions&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Multifocal location and extensive involvement of the spine makes this case exceptional&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflict of interests</span><p id="par0030" class="elsevierStylePara elsevierViewall">The authors have no conflict of interest to declare&#46;</p></span></span>"
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ISSN: 21735743
Original language: English
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