Ectopic corticotropin-releasing hormone (CRH) syndrome from metastatic small cell carcinoma a case report and review of the literature.docVIP

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Ectopic corticotropin-releasing hormone (CRH) syndrome from metastatic small cell carcinoma a case report and review of the literature.doc

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Ectopic corticotropin-releasing hormone (CRH) syndrome from metastatic small cell carcinoma a case report and review of the literature

Shahanietal.DiagnosticPathology2010,5:56 /content/5/1/56 CASE REPORT OpenAccess Ectopiccorticotropin-releasinghormone(CRH) syndromefrommetastaticsmallcellcarcinoma: acasereportandreviewoftheliterature SadekaShahani1,RodolfoJNudelman2,RamaswamiNalini1,3,Han-SeobKim2,3,SusanLSamson1,3* Abstract Background:Cushing’sSyndrome(CS)whichiscausedbyisolatedCorticotropin-releasinghormone(CRH) production,ratherthanadrenocorticotropin(ACTH)production,isextremelyrare. Methods:Wedescribetheclinicalpresentation,course,laboratoryvaluesandpathologicfindingsofapatientwith isolatedectopicCRHcausingCS.Wereviewtheliteratureofthetypesoftumorsassociatedwiththisunusual syndromeandthebehaviorofthesetumorsbyendocrinetesting. Results:A56yearoldwomanpresentedwithclinicalandlaboratoryfeaturesconsistentwithACTH-dependentCS. Pituitaryimagingwasnormalandcortisoldidnotsuppresswithahighdosedexamethasonetest,consistentwith adiagnosisofectopicACTH.CTimagingdidnotrevealanydiscretelunglesionsbutthereweremediastinaland abdominallymphadenopathyandmultipleliverlesionssuspiciousformetastaticdisease.Laboratorytestingwas positiveforelevatedserumcarcinoembryonicantigenandtheneuroendocrinemarkerchromograninA.Serum markersofcarcinoid,medullarythyroidcarcinoma,andpheochromocytomawereinthenormalrange.Becausethe primarytumorcouldnotbeidentifiedbyimaging,biopsyofthepresumedmetastaticliverlesionswasperformed. Immunohistochemistrywasconsistentwithaneuroendocrinetumor,specificallysmallcellcarcinoma. ImmunostainingforACTHwasnegativebutwasstronglypositiveforCRHandlaboratorytestingrevealedaplasma CRHof10pg/ml(normal0to10pg/ml)whichshouldhavebeensuppressedinthepresenceofhighcortisol. Conclusions:ThiscaseillustratestheimportanceofconsideringtheectopicproductionofCRHinthedifferential diagnosisforpresentationsofACTH-dependentCushing’ sSyndrome. Introduction Theremaining ACTH-dependent cases mostly involve Cushing’ssyndrome(CS)comprisestheclinicalmanifes- non-pituitarymalignancies(EctopicACTH)whichoften tationsofexposuret