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LetterLetters to the Editor

Regarding “Waxing and Waning Presentation of Isolated Cardiac Sarcoidosis on Sequential 18F-FDG PET Exams”

David H. Birnie
Journal of Nuclear Medicine Technology September 2021, 49 (3) 292; DOI: https://doi.org/10.2967/jnmt.120.254276
David H. Birnie
University of Ottawa Heart Institute 40 Ruskin St. Ottawa, ON, K1Y 4W7
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TO THE EDITOR: I read with interest the article by Ostwani et al. (1) and, for 3 reasons, am concerned that the patient does not have cardiac sarcoidosis (CS). First, because of the lack of a positive biopsy result, the patient does not meet guideline criteria for the diagnosis of CS (2). Second, corticosteroid-refractory sarcoidosis is considered very rare (3). Third, accumulating data suggest that there may not be a pathophysiologic entity of truly isolated CS. It is clear that there are many patients with manifest CS who have no clinically apparent disease in other organs—that is, who have what can be termed clinically isolated CS. However, sarcoidosis is, by definition and biology, a systemic disease. Hence, a key starting point to understand isolated CS is to agree on a standardized definition. The 2017 version of the Japanese CS guidelines tackled, for the first time, the definition of and criteria for the diagnosis for isolated CS (4). They included the following 3 criteria: no clinical findings characteristic of sarcoidosis are observed in any organs other than the heart; 67Ga scintigraphy or 18F-FDG PET reveals no abnormal tracer accumulation in any organs other than the heart; and a chest CT scan reveals no shadowing along the lymphatic tracts in the lungs or no hilar and mediastinal lymphadenopathy (minor axis > 10 mm).

Using a similar definition, my group found imaging-isolated CS in only 1 in 31 patients (5). However, other data suggest that even these apparent isolated cases are unlikely to be truly isolated. Petek et al. investigated 10 patients with presentations and cardiac imaging consistent with the Japanese definition of isolated CS. Four of these 10 had granulomas on bronchial biopsy (6). Hence, these data suggest that there is a small subset of patients who at the moment of 18F-FDG PET imaging have PET-detectable inflammation only in their heart. However, it also follows that additional or interval investigation will likely reveal extracardiac disease.

This debate is more than just semantics, as the overdiagnosis of “imaging-isolated CS” can, as in this case (1), lead to unnecessary immunosuppression or a missed alternative diagnosis.

Footnotes

  • Published online September 4, 2021.

REFERENCES

  1. 1.↵
    1. Ostwani W,
    2. Hanna C,
    3. Brice AE,
    4. Wymer DC
    . Waxing and waning presentation of isolated cardiac sarcoidosis on sequential 18F-FDG-PET exams. J Nucl Med Technol June 9, 2020 [Epub ahead of print].
  2. 2.↵
    1. Birnie DH,
    2. Sauer WH,
    3. Bogun F,
    4. et al
    . HRS expert consensus statement on the diagnosis and management of arrhythmias associated with cardiac sarcoidosis. Heart Rhythm 2014;11: 1305–1323.
    OpenUrlCrossRefPubMed
  3. 3.↵
    1. Goldman C,
    2. Judson MA
    . Corticosteroid refractory sarcoidosis. Respir Med 2020;171:106081.
    OpenUrl
  4. 4.↵
    1. Terasaki FYK
    . New guidelines for diagnosis of cardiac sarcoidosis in Japan. Ann Nucl Cardiol 2017;3:42–45.
    OpenUrl
  5. 5.↵
    1. Juneau D,
    2. Nery P,
    3. Russo J,
    4. et al
    . How common is isolated cardiac sarcoidosis? Extra-cardiac and cardiac findings on clinical examination and whole-body 18F-fluorodeoxyglucose positron emission tomography. Int J Cardiol 2018;253:189–193.
    OpenUrlPubMed
  6. 6.↵
    1. Petek BJ,
    2. Rosenthal DG,
    3. Patton KK,
    4. et al
    . Cardiac sarcoidosis: diagnosis confirmation by bronchoalveolar lavage and lung biopsy. Respir Med 2018;144(suppl):S13–S19.
    OpenUrl
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Journal of Nuclear Medicine Technology: 49 (3)
Journal of Nuclear Medicine Technology
Vol. 49, Issue 3
September 1, 2021
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Regarding “Waxing and Waning Presentation of Isolated Cardiac Sarcoidosis on Sequential 18F-FDG PET Exams”
David H. Birnie
Journal of Nuclear Medicine Technology Sep 2021, 49 (3) 292; DOI: 10.2967/jnmt.120.254276

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Regarding “Waxing and Waning Presentation of Isolated Cardiac Sarcoidosis on Sequential 18F-FDG PET Exams”
David H. Birnie
Journal of Nuclear Medicine Technology Sep 2021, 49 (3) 292; DOI: 10.2967/jnmt.120.254276
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