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• W3135283280 abstract "HomeCirculation: Heart FailureVol. 14, No. 3A Case of Rapidly Progressing Granulomatous Myocarditis Free AccessCase ReportPDF/EPUBAboutView PDFView EPUBSections ToolsAdd to favoritesDownload citationsTrack citationsPermissions ShareShare onFacebookTwitterLinked InMendeleyRedditDiggEmail Jump toSupplementary MaterialsFree AccessCase ReportPDF/EPUBA Case of Rapidly Progressing Granulomatous MyocarditisWhat Is the Diagnosis? Jessica Cao, MD Yosef Y. Schwartz, MD Jon W. Lomasney, MD Akhil Narang, MD Ike S. Okwuosa, MD Ryan Avery, MD Rod S. Passman, MD Leslie T. Cooper Jr, MD Esther VorovichMD Jessica CaoJessica Cao Correspondence to: Jessica Cao, MD, Department of Internal Medicine, Northwestern University Feinberg School of Medicine, 676 North St. Clair St, Arkes Suite 2330, Chicago IL, 60611. Email E-mail Address: [email protected] https://orcid.org/0000-0001-9385-5179 Department of Internal Medicine (J.C., Y.Y.S.), Northwestern University Feinberg School of Medicine, Chicago, IL. Search for more papers by this author , Yosef Y. SchwartzYosef Y. Schwartz Department of Internal Medicine (J.C., Y.Y.S.), Northwestern University Feinberg School of Medicine, Chicago, IL. Search for more papers by this author , Jon W. LomasneyJon W. Lomasney https://orcid.org/0000-0002-0438-2427 Department of Pathology (J.W.L.), Northwestern University Feinberg School of Medicine, Chicago, IL. Search for more papers by this author , Akhil NarangAkhil Narang Division of Cardiology (A.N., I.S.O., R.A., R.S.P., E.V.), Northwestern University Feinberg School of Medicine, Chicago, IL. Search for more papers by this author , Ike S. OkwuosaIke S. Okwuosa Division of Cardiology (A.N., I.S.O., R.A., R.S.P., E.V.), Northwestern University Feinberg School of Medicine, Chicago, IL. Search for more papers by this author , Ryan AveryRyan Avery Division of Cardiology (A.N., I.S.O., R.A., R.S.P., E.V.), Northwestern University Feinberg School of Medicine, Chicago, IL. Search for more papers by this author , Rod S. PassmanRod S. Passman https://orcid.org/0000-0001-8718-1534 Division of Cardiology (A.N., I.S.O., R.A., R.S.P., E.V.), Northwestern University Feinberg School of Medicine, Chicago, IL. Search for more papers by this author , Leslie T. Cooper JrLeslie T. Cooper Jr https://orcid.org/0000-0003-1002-3313 Department of Cardiovascular Medicine, Mayo Clinic, Jacksonville, FL (L.T.C.). Search for more papers by this author and Esther VorovichEsther Vorovich Division of Cardiology (A.N., I.S.O., R.A., R.S.P., E.V.), Northwestern University Feinberg School of Medicine, Chicago, IL. Search for more papers by this author Originally published8 Mar 2021https://doi.org/10.1161/CIRCHEARTFAILURE.120.007800Circulation: Heart Failure. 2021;14:e007800Giant cell myocarditis (GCM) and cardiac sarcoidosis (CS) are causes of inflammatory cardiomyopathy that can present with heart failure and arrhythmias. We present a case with an initial subacute presentation with extracardiac lymph node biopsy demonstrating noncaseating granulomas (NCG) consistent with CS. Several weeks thereafter, the patient rapidly decompensated with endomyocardial biopsy (EMBx) confirming GCM. This case demonstrates an atypical presentation of GCM and highlights the utility of a multimodality diagnostic approach.Case ReportA 59-year-old male with history of hypertension presented with 2 weeks of lightheadedness and fatigue. ECG showed new high-degree atrioventricular block with intermittent complete heart block (Figure 1). On admission, troponin I was 3.11 ng/mL (reference: 0.00–0.04 ng/mL), B-type natriuretic peptide was 52 pg/mL, and transthoracic echocardiography demonstrated an ejection fraction of 62%. Coronary angiogram and Lyme serologies were unrevealing. Cardiac magnetic resonance imaging (Movies I and II in the Data Supplement; Figure 2) revealed delayed gadolinium enhancement diffusely involving the right ventricle; right ventricle ejection fraction was 39%. High capture thresholds were noted during pacemaker implantation. He was discharged with improvement albeit not resolution of symptoms.Download figureDownload PowerPointFigure 1. Complete heart block with junctional escape, left axis deviation, and right bundle branch block.Download figureDownload PowerPointFigure 2. The irregular thickening has a striking pattern of diffuse late gadolinium enhancement throughout the right ventricle with relative sparing of the left ventricle.One week later, outpatient FDG-PET demonstrated diffuse right ventricle inflammation, patchy left ventricle involvement, and hypermetabolic mediastinal lymphadenopathy (Figure 3). Our institutional CS imaging protocol utilizes FDG-PET/computed tomography with a 48-hour low-carbohydrate diet prior. PET imaging assessing perfusion was not performed as our institution does not have current access to PET myocardial radiotracers. Subsequent lymph node biopsy revealed NCG consistent with CS (Figure 4A). He was referred to a sarcoidosis specialist but represented 2 days later with profound fatigue and dyspnea.Download figureDownload PowerPointFigure 3. FDG-PET: both ventricles demonstrate diffuse patchy hypermetabolic activity with somewhat more diffuse involvement of the right ventricle. These findings are compatible with active myocardial inflammation. The exam was also notable for multiple hypermetabolic, nonenlarged lymph nodes predominately seen in the right paratracheal region.Download figureDownload PowerPointFigure 4. Endomyocardial biopsy ultimately provided the diagnosis of giant cell myocarditis.A, Lymph node. Noncaseating granulomatous inflammation consistent with sarcoidosis. Special stains for microorganisms were negative (Grocott's methenamine silver, acid-fast bacteria). B, Native biopsy. Extensive cardiomyocyte damage, early granulation tissue, heavy aggressive inflammatory infiltrate with numerous eosinophils, and multinucleate giant cells. Granulomas and fibrosis are absent. C, Cardiac explant. Extensive cardiomyocyte damage and replacement, granulation tissue, heavy aggressive inflammatory infiltrate with numerous eosinophils and multinucleate giant cells. Granulomas are absent. Transmural in some areas (posterior left ventricle). Bundle of His, right and left bundle branches involved. D, Cardiac allograft biopsy showing recurrent giant cell myocarditis (GCM). Endomyocardium with a small focus of cardiomyocyte damage, multinucleate giant cells, and abundant eosinophils consistent with recurrent GCM.Repeat ejection fraction was 20% with mild left ventricle dilatation (left ventricular end-diastolic diameter 5.8 cm). Troponin I was 65.99 ng/mL. B-type natriuretic peptide was 1746 pg/mL. Right heart catheterization demonstrated normal filling pressures and severely depressed cardiac index (1.72 L/minute per m2). Lactate was elevated (2.4 mmol/L) and a femoral balloon pump implanted. Emergent EMBx revealed extensive GCM (Figure 4B). Cyclosporine and steroids were initiated, and he was listed for heart transplant (Figure 4C), which he underwent on hospital day 7. Thymoglobulin induction (1 mg/kg) was given with thrombocytopenia precluding further doses. Therapeutic tacrolimus levels were attained on posttransplant day 7. He was maintained on tacrolimus (goal level 10–15 ng/mL), mycophenolate mofetil (2 g/day), and prednisone taper as per protocol.The second routine EMBx (posttransplant day 16) revealed GCM recurrence in 2/5 samples (Figure 4D); graft function and hemodynamics were normal. He was treated with 3 mg/kg of thymoglobulin, and mycophenolate mofetil was increased to 3 g/day with resolution of GCM on EMBx. His tacrolimus level decreased transiently to 8.6 ng/mL concomitant with a decrease in prednisone (20 mg to 17.5 mg), and EMBx again demonstrated GCM recurrence. He was treated with a steroid pulse and taper again with resolution. Graft function and hemodynamics consistently remained normal.DiscussionHistorically, there has been debate whether CS and GCM are separate disorders or if GCM represents a severe phenotype of the CS/granulomatous myocarditis spectrum.1 Both diseases can present with heart failure and arrhythmias, although conduction abnormalities occur more frequently with CS.2 However, GCM evolves more rapidly, frequently presenting with fulminant myocarditis, while CS typically has a more indolent course. EMBx performed for CS demonstrates poor diagnostic performance (sensitivity 20%–30%), so extracardiac tissue biopsy is typically pursued.3Our patient’s initial presentation of isolated atrioventricular block, extracardiac NCG and imaging findings initially strongly favored the diagnosis of CS, although magnetic resonance imaging findings of right ventricle fibrosis with relative left ventricle sparing were relatively atypical for CS.4 Our current PET/computed tomography protocol does not evaluate myocardial perfusion which if detected could have potentially raised suspicion for GCM earlier.The patient’s rapid clinical deterioration quickly shifted our focus to GCM. EMBx, which carries a class I recommendation for suspected GCM, ultimately provided the diagnosis.3 Sarcoidosis is characterized by NCG compromised of epithelioid cells that can fuse to form occasional Langhans giant cells and well-formed granulomas. Eosinophils are rare and while fibrosis can be extensive, necrosis is typically absent. In contrast, well-formed granulomas are rare in GCM and the sine qua non is the presence of giant cells and prominent eosinophils with extensive necrosis disproportionate to degree of inflammation. In our patient, lymph node histology was consistent with sarcoidosis. However, 5% to 10% of GCM cases have extracardiac granulomatous inflammation.2 Therefore, extracardiac granulomas should not rule out a diagnosis of GCM.Interestingly, the patient’s GCM recurrence at 16 days is the earliest published report. Histologically, GCM recurrence is distinguished from allograft rejection by the presence of giant cells, necrosis, and prominent eosinophils in the absence of aggressive lymphocytic infiltrates typically seen in allograft rejection.In conclusion, we present a case of GCM whose initial clinical presentation of atrioventricular block with extracardiac NCG erroneously suggested CS. Thus, in patients with atypical clinical or imaging findings of CS, regardless of extracardiac presence of NCG, EMBx should be considered for timely diagnosis to hopefully prevent decompensation.Sources of FundingNone.Disclosures None.FootnotesThis manuscript was sent to Anita Deswal, MD, MPH, Guest Editor, for review by expert referees, editorial decision, and final disposition.The Data Supplement is available at https://www.ahajournals.org/doi/suppl/10.1161/CIRCHEARTFAILURE.120.007800.Correspondence to: Jessica Cao, MD, Department of Internal Medicine, Northwestern University Feinberg School of Medicine, 676 North St. Clair St, Arkes Suite 2330, Chicago IL, 60611. Email jessica.[email protected]eduReferences1. Okura Y, Dec GW, Hare JM, Kodama M, Berry GJ, Tazelaar HD, Bailey KR, Cooper LT. A clinical and histopathologic comparison of cardiac sarcoidosis and idiopathic giant cell myocarditis.J Am Coll Cardiol. 2003; 41:322–329. doi: 10.1016/s0735-1097(02)02715-8CrossrefMedlineGoogle Scholar2. Blauwet LA, Cooper LT. Idiopathic giant cell myocarditis and cardiac sarcoidosis.Heart Fail Rev. 2013; 18:733–746. doi: 10.1007/s10741-012-9358-3CrossrefMedlineGoogle Scholar3. Cooper LT, Baughman KL, Feldman AM, Frustaci A, Jessup M, Kuhl U, Levine GN, Narula J, Starling RC, Towbin J, et al.; American Heart Association; American College of Cardiology; European Society of Cardiology; Heart Failure Society of America; Heart Failure Association of the European Society of Cardiology. The role of endomyocardial biopsy in the management of cardiovascular disease: a scientific statement from the American Heart Association, the American College of Cardiology, and the European Society of Cardiology. Endorsed by the Heart Failure Society of America and the Heart Failure Association of the European Society of Cardiology.J Am Coll Cardiol. 2007; 50:1914–1931. doi: 10.1016/j.jacc.2007.09.008CrossrefMedlineGoogle Scholar4. Lamacie MM, Almufleh A, Nair V, Stadnick E, Birnie D, Beanlands RSB, Chih S. Serial 18F-fluorodeoxyglucose positron emission tomography imaging in a patient with giant cell myocarditis.Circ Cardiovasc Imaging. 2020; 13:e009940. doi: 10.1161/CIRCIMAGING.119.009940LinkGoogle Scholar Previous Back to top Next FiguresReferencesRelatedDetails March 2021Vol 14, Issue 3Article InformationMetrics Download: 170 © 2021 American Heart Association, Inc.https://doi.org/10.1161/CIRCHEARTFAILURE.120.007800PMID: 33677978 Originally publishedMarch 8, 2021 Keywordsbiopsymyocarditisinflammationgiant cellssarcoidosishypertensionheart failurePDF download SubjectsHeart Failure" @default.
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