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Case Reports
. 2022 Feb;9(1):761-765.
doi: 10.1002/ehf2.13771. Epub 2021 Dec 29.

Inflammatory cardiomyopathy of possibly overlapping aetiology: a case posing treatment dilemma and potential association

Shun Nakagama  1 , Katherine Candray  2   3 , Tasuku Yamamoto  1 , Yuta Tsugeno  4 , Yu Nakagama  2 , Yasutoshi Kido  2 , Yuko Nitahara  2 , Yasuhiro Maejima  1 , Tetsuo Sasano  1
Affiliations
Case Reports

Inflammatory cardiomyopathy of possibly overlapping aetiology: a case posing treatment dilemma and potential association

Shun Nakagama et al. ESC Heart Fail. 2022 Feb.

Abstract

We report on a 52-year-old Brazilian immigrant woman with past histories of chronic kidney disease and uveitis, presenting with symptomatic atrioventricular block. Her country of origin being endemic for Trypanosoma cruzi infection, we suspected Chagas disease as the aetiology, diagnosis of which was confirmed by serological tests. Further systemic workup identified an emerging nodular lesion in the lung, which turned out to be a sarcoid epithelioid granuloma on biopsy. Involvement of the kidneys and eyes was suggestive of systemic extension of the lung sarcoidosis. Although imaging modalities did not detect inflammatory foci in the myocardium, the rare coexistence of histologically proven sarcoidosis raised the intriguing concept of cardiac manifestation having arisen from two possibly overlapping aetiologies: Chagas disease and cardiac sarcoidosis. The case highlights a treatment dilemma increasingly likely to be encountered in this globalized world, and also raises the potential, but intriguing, association of these two diseases.

Keywords: Atrioventricular block; Chagas disease; Inflammatory cardiomyopathy; Latin America; Sarcoidosis; Trypanosoma cruzi.

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Conflict of interest statement

None declared.

Figures

Figure 1
Figure 1
Clinical findings and the results of Trypanosoma cruzi serological testing. (A) The baseline electrocardiogram was remarkable only for left anterior hemiblock. (B) The telemetry electrocardiogram revealed advanced 2:1 atrioventricular block with a ventricular response rate of 33 beats per minute. (C) A lateral flow immunoassay (Trypanosoma Detect Rapid Test; InBios, Seattle, USA) detected a positive band (arrowhead). (D) In the enzyme‐linked immunosorbent assay ( T. cruzi IgG CELISA II; CELLABS, Sydney, Australia), absorbance at 450 nm was measured. Each row indicates negative control (NC), positive control (PC) and the patient's sample, all measured in duplicates. Samples suggested a positive result.
Figure 2
Figure 2
Histopathological findings. (A) A transbronchial lung biopsy revealed noncaseating epithelioid granulomas (arrows). Scale bars represent 50 μm. (B) Immunohistochemical staining with anti‐ Propionibacterium acnes ( P. acnes ) antibody revealed the localization of P. acnes in granulomas (arrowheads). Scale bar represents 50 μm.
See this image and copyright information in PMC

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