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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Secondary osteoporosis has been associated with different diseases and drugs&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> These include endocrine diseases&#44; cancer&#44; kidney damage&#44; gastrointestinal and rheumatic diseases&#46; Steroids are also a well-recognized cause of bone mass loss&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> According to some studies&#44; 20&#37;&#8211;30&#37; of postmenopausal women and over 50&#37; of men have a second cause of osteoporosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> Endogenous Cushing&#39;s syndrome is characterized by excessive production of steroids&#44; the most common cause being Cushing&#39;s disease due to a pituitary adenoma secreting ACTH&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Case Report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A female&#44; 41&#44; attended the emergency department with generalized musculoskeletal pain&#44; especially in the chest&#44; spine and groin&#46; Initial radiographs showed multiple fractures of the upper third of the sternal body&#44; two severe stage 3 dorsal vertebral fractures and a third stages 1&#8211;2 dorsal fracture&#44; with another on the ischiopubic ramus &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A and B&#41;&#46; On examination hypertension and cushingoid features&#44; such as round face and ce obesity were later found&#46; Laboratory studies showed that cortisol and baseline ACTH determination were elevated&#46; The baseline cortisol was 319<span class="elsevierStyleHsp" style=""></span>ng&#47;ml &#40;normal between 50 and 250<span class="elsevierStyleHsp" style=""></span>ng&#47;ml&#41; and ACTH of 57&#46;4<span class="elsevierStyleHsp" style=""></span>pg&#47;ml when it is normally undetectable or less than 10<span class="elsevierStyleHsp" style=""></span>pg&#47;ml&#44; with a decreased gonadotropin level&#46; The levels of urinary free cortisol and cortisol were also elevated at night&#58; 847<span class="elsevierStyleHsp" style=""></span>nmol&#47;l and 290&#46;7<span class="elsevierStyleHsp" style=""></span>ng&#47;ml&#44; respectively&#46; The nocturnal cortisol under normal conditions is negligible&#46; A cranial MRI showed a questionable image in the pituitary&#44; so catheterization of inferior petrosal sinus was performed to study the source of ACTH overproduction&#46; There was a central-peripheral gradient of ACTH and increased focus on the left half of the hypophysis&#46; A second MRI detected a nodule of 6&#46;3<span class="elsevierStyleHsp" style=""></span>mm in the posterior left area of the pituitary&#46; Following these results the patient was diagnosed as having Cushing&#39;s disease due to a pituitary microadenoma&#46; The patient was treated from the surgical standpoint with a hemihypophysectomy of the microadenoma&#44; and infused intravenously &#40;iv&#41; with zoledronic acid 5<span class="elsevierStyleHsp" style=""></span>mg<span class="elsevierStyleHsp" style=""></span>iv<span class="elsevierStyleMonospace">&#47;</span>year as a treatment for severe osteoporosis&#44; with great improvement of bone pain&#46; After surgery&#44; cortisol levels were normalized with baseline cortisol levels of 152<span class="elsevierStyleHsp" style=""></span>ng&#47;ml and urinary cortisol of 107<span class="elsevierStyleHsp" style=""></span>nmol&#47;l&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">Although osteoporosis is a cardinal manifestation of Cushing&#39;s<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> syndrome&#44; there are few publications in this respecto&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The incidence of Cushing&#39;s syndrome of endogenous origin is 2&#8211;4 cases per million inhabitants&#47;year&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Its diagnosis requires a high clinical suspicion&#44; as most of the symptoms of this syndrome are highly prevalent in the general population &#40;hypertension&#44; glucose intolerance&#44; central obesity&#44; osteoporosis&#41; and none of these is specific&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Fractures occur in 19&#37;&#8211;50&#37; in patients with Cushing&#39;s disease&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> In the last 5 years&#44; we have only found two case reports of fractures secondary to osteoporosis due to Cushing&#39;s<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> disease&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Our patient presented with spontaneous fractures as a sentinel manifestation of Cushing&#39;s disease&#44; and in this case the causal diagnosis of osteoporosis had highly relevant prognostic and therapeutic implications&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conclusions</span><p id="par0020" class="elsevierStylePara elsevierViewall">We must always consider the possibility of secondary osteoporosis after the appearance of a spontaneous fracture&#44; especially in men and premenopausal and perimenopausal women&#44; where the frequency of secondary osteoporosis is near&#47;greater than 50&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Ethical disclosures</span><p id="par0025" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">Protection of human and animal subjects</span>&#46; The authors declare that no experiments were performed on humans or animals for this investigation&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">Confidentiality of Data</span>&#46; 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Case Report
Cushings Disease as a Cause of Severe Osteoporosis: A Clinical Challenge
Enfermedad de Cushing como causa de osteoporosis grave. Un reto clínico
Nadia Abdel-Kadera,
Corresponding author
greendoggy@hotmail.com

Corresponding author.
, Mario H. Cardielb, Victoria Navarro Compana, Juan Piedra Priegoa, Ana Gonzáleza
a Servicio de Reumatología, Hospital Universitario Virgen Macarena, Sevilla, Spain
b Servicio de Reumatología, Hospital Dr. Miguel Silva Hospital, Morelia, Mexico
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41; Severe stage 3 dorsal vertebral fractures &#40;arrows&#41;&#44; moderate stage 1&#8211;2 dorsal fracture&#46; &#40;B&#41; Fracture of the upper sternal body&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Secondary osteoporosis has been associated with different diseases and drugs&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> These include endocrine diseases&#44; cancer&#44; kidney damage&#44; gastrointestinal and rheumatic diseases&#46; Steroids are also a well-recognized cause of bone mass loss&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> According to some studies&#44; 20&#37;&#8211;30&#37; of postmenopausal women and over 50&#37; of men have a second cause of osteoporosis&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> Endogenous Cushing&#39;s syndrome is characterized by excessive production of steroids&#44; the most common cause being Cushing&#39;s disease due to a pituitary adenoma secreting ACTH&#46;<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Case Report</span><p id="par0010" class="elsevierStylePara elsevierViewall">A female&#44; 41&#44; attended the emergency department with generalized musculoskeletal pain&#44; especially in the chest&#44; spine and groin&#46; Initial radiographs showed multiple fractures of the upper third of the sternal body&#44; two severe stage 3 dorsal vertebral fractures and a third stages 1&#8211;2 dorsal fracture&#44; with another on the ischiopubic ramus &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A and B&#41;&#46; On examination hypertension and cushingoid features&#44; such as round face and ce obesity were later found&#46; Laboratory studies showed that cortisol and baseline ACTH determination were elevated&#46; The baseline cortisol was 319<span class="elsevierStyleHsp" style=""></span>ng&#47;ml &#40;normal between 50 and 250<span class="elsevierStyleHsp" style=""></span>ng&#47;ml&#41; and ACTH of 57&#46;4<span class="elsevierStyleHsp" style=""></span>pg&#47;ml when it is normally undetectable or less than 10<span class="elsevierStyleHsp" style=""></span>pg&#47;ml&#44; with a decreased gonadotropin level&#46; The levels of urinary free cortisol and cortisol were also elevated at night&#58; 847<span class="elsevierStyleHsp" style=""></span>nmol&#47;l and 290&#46;7<span class="elsevierStyleHsp" style=""></span>ng&#47;ml&#44; respectively&#46; The nocturnal cortisol under normal conditions is negligible&#46; A cranial MRI showed a questionable image in the pituitary&#44; so catheterization of inferior petrosal sinus was performed to study the source of ACTH overproduction&#46; There was a central-peripheral gradient of ACTH and increased focus on the left half of the hypophysis&#46; A second MRI detected a nodule of 6&#46;3<span class="elsevierStyleHsp" style=""></span>mm in the posterior left area of the pituitary&#46; Following these results the patient was diagnosed as having Cushing&#39;s disease due to a pituitary microadenoma&#46; The patient was treated from the surgical standpoint with a hemihypophysectomy of the microadenoma&#44; and infused intravenously &#40;iv&#41; with zoledronic acid 5<span class="elsevierStyleHsp" style=""></span>mg<span class="elsevierStyleHsp" style=""></span>iv<span class="elsevierStyleMonospace">&#47;</span>year as a treatment for severe osteoporosis&#44; with great improvement of bone pain&#46; After surgery&#44; cortisol levels were normalized with baseline cortisol levels of 152<span class="elsevierStyleHsp" style=""></span>ng&#47;ml and urinary cortisol of 107<span class="elsevierStyleHsp" style=""></span>nmol&#47;l&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0015" class="elsevierStylePara elsevierViewall">Although osteoporosis is a cardinal manifestation of Cushing&#39;s<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> syndrome&#44; there are few publications in this respecto&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The incidence of Cushing&#39;s syndrome of endogenous origin is 2&#8211;4 cases per million inhabitants&#47;year&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Its diagnosis requires a high clinical suspicion&#44; as most of the symptoms of this syndrome are highly prevalent in the general population &#40;hypertension&#44; glucose intolerance&#44; central obesity&#44; osteoporosis&#41; and none of these is specific&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> Fractures occur in 19&#37;&#8211;50&#37; in patients with Cushing&#39;s disease&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> In the last 5 years&#44; we have only found two case reports of fractures secondary to osteoporosis due to Cushing&#39;s<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> disease&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Our patient presented with spontaneous fractures as a sentinel manifestation of Cushing&#39;s disease&#44; and in this case the causal diagnosis of osteoporosis had highly relevant prognostic and therapeutic implications&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conclusions</span><p id="par0020" class="elsevierStylePara elsevierViewall">We must always consider the possibility of secondary osteoporosis after the appearance of a spontaneous fracture&#44; especially in men and premenopausal and perimenopausal women&#44; where the frequency of secondary osteoporosis is near&#47;greater than 50&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Ethical disclosures</span><p id="par0025" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">Protection of human and animal subjects</span>&#46; The authors declare that no experiments were performed on humans or animals for this investigation&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">Confidentiality of Data</span>&#46; The authors declare that they have followed the protocols of their work centre on the publication of patient data and that all the patients included in the study have received sufficient information and have given their informed consent in writing to participate in that study&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">Right to privacy and informed consent</span>&#46; The authors have obtained the informed consent of the patients and&#47;or subjects mentioned in the article&#46; The author for correspondence is in possession of this document&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Disclosures</span><p id="par0030" class="elsevierStylePara elsevierViewall">The authors have no disclosures to make&#46;</p></span></span>"
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        "resumen" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La osteoporosis secundaria est&#225; frecuentemente infraestimada en el estudio de la osteoporosis&#46; Existe una causa segunda de osteoporosis en cerca o m&#225;s de la mitad de hombres y mujeres premenop&#225;usicas&#44; y en torno a un quinto de las mujeres posmenop&#225;usicas&#46; Presentamos a una mujer de 41 a&#241;os&#44; con dolor &#243;seo por fracturas m&#250;ltiples&#44; cuyo diagn&#243;stico final fue enfermedad de Cushing&#46;</p>"
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ISSN: 21735743
Original language: English
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Idiomas
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