Computed tomography findings of early-stage TAFRO syndrome and associated adrenal abnormalities

Kurokawa, Ryo; Gonoi, Wataru; Yokota, Hajime; Isshiki, Saiko; Ohira, Kenji; Mizuno, Hideaki; Kiguchi, Takao; Inui, Shigeru; Kurokawa, Mariko; Kato, Shimpei; Matsuki, Mitsuru; Takeda, Taro; Yokoyama, Kota; Ota, Yoshiaki; Nakai, Yudai; Maeda, Eriko; Mori, Hiromu; Abe, Osamu

doi:10.1007/s00330-020-06919-1

Cited by 15 publications

(40 citation statements)

References 32 publications

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“…These presentations and the results of the lymph node biopsy led to the diagnosis of TA-FRO syndrome. The bilateral adrenal infarctions were not accompanied by adrenal insufficiency and spontaneously improved before the initiation of the treatments for TAFRO (2) 24 Female Adrenal infarction Bilateral 2 (2) 50 Male Adrenal infarction Unilateral 3 (2) 71 Male Adrenomegaly Unilateral 4 (2) 33 Male Adrenal infarction Bilateral 5 (2) 55 Male Adrenal infarction Bilateral 6 (2) 53 Male Adrenal infarction Bilateral 7 (2) 35 Male Adrenomegaly Bilateral 8 (4) 46 Male Adrenal infarction Unilateral 9 (6) 48 Male Adrenal hemorrhage Unilateral 10 (7) 43 Male Adrenomegaly Unilateral 11 (8) 19 Male Adrenal hemorrhage Bilateral 12 (8) 31 Female Adrenal hemorrhage Bilateral 13 (9) 48 Male Adrenal hemorrhage Bilateral 14(Our case) 53 Female Adrenal infarction Bilateral syndrome.…”

Section: Discussionmentioning

confidence: 95%

“…It has also been reported in patients with TAFRO syndrome. In our literature search, we found 14 cases of TAFRO syndrome with adrenal abnormalities (2,4,(6)(7)(8)(9). We have summarized these cases in Table 3.…”

Section: Discussionmentioning

confidence: 99%

“…Adrenal lesions have also been reported in Castleman disease, but they included adrenal tumors, and there have been no reports of adrenal hemorrhaging or infarction (11,12). Furthermore, adrenal biopsies of patients with Castleman disease have shown tissue comparable to that found in the lymph nodes, and the pathogenesis is different from that of adrenal lesions in TAFRO syndrome (2,4,11). Although most reports of adrenal tumors have been in patients with unicentric Castleman disease, the type of adrenal lesion may be useful for differentiating this disease from TAFRO syndrome.…”

Section: Discussionmentioning

confidence: 99%

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Bilateral Adrenal Infarctions as an Initial Manifestation of TAFRO Syndrome: A Case Report and Review of the Literature

et al. 2022

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TAFRO syndrome is a systemic inflammatory disorder resembling multicentric Castleman disease; it is characterized by thrombocytopenia, anasarca, a fever, reticulin fibrosis, and organomegaly. Involvement of the adrenal glands, including adrenal infarction, hemorrhaging, and adrenomegaly, has recently been reported in several cases and been considered a characteristic early-stage symptom. We herein report a case of TAFRO syndrome initially presenting with bilateral adrenal infarctions and review the literature on TAFRO syndrome related to adrenal involvement. This case suggests that adrenal abnormalities as an early clinical feature of TAFRO syndrome may be useful for the early diagnosis.

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Section: Discussionmentioning

confidence: 95%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Bilateral Adrenal Infarctions as an Initial Manifestation of TAFRO Syndrome: A Case Report and Review of the Literature

et al. 2022

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“…Kurokawa et al . 20 noted adrenomegaly and adrenal ischemia in 46.2% and 30.8% of patients, respectively, in the early stage of TAFRO syndrome. This suggests that adrenal hemorrhage occurs before the exacerbation of thrombocytopenia and/or coagulopathy due to inflammatory changes in the early stage of the disease.…”

Section: Discussionmentioning

confidence: 87%

“…This suggests that adrenal hemorrhage in TAFRO syndrome is caused by destruction of the adrenal gland vasculature induced by post-peritoneal inflammation. Kurokawa et al 20 noted adrenomegaly and adrenal ischemia in 46.2% and 30.8% of patients, respectively, in the early stage of TAFRO syndrome. This suggests that adrenal hemorrhage occurs before the exacerbation of thrombocytopenia and/or coagulopathy due to inflammatory changes in the early stage of the disease.…”

Section: Fig 3 Clinical Coursementioning

confidence: 95%

Fatal case of TAFRO syndrome with unilateral adrenal hemorrhage in early-stage disease

Okamoto

Ochi

Motokawa³

et al. 2021

J Clin Exp Hematopathol

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Thrombocytopenia, anasarca, fever, reticulin fibrosis/renal failure, and organomegaly comprise TAFRO syndrome, which was proposed as a distinct clinical entity from iMCD without TAFRO syndrome (iMCD-NOS) due to its aggressive clinical course, refractoriness to corticosteroids, presence of thrombocytopenia, increased level of alkaline phosphatase, and normal level of gammaglobulin. However, diagnosing TAFRO syndrome in its early stages is challenging because it is rare and its diagnostic criteria are complicated. We describe a patient with TAFRO syndrome and adrenal hemorrhage who demonstrated a rapid decline in her clinical condition and did not respond to steroid pulse therapy, resulting in a fatal outcome. In the early stage of her clinical course, she developed unilateral adrenal hemorrhage with mild thrombocytopenia and normal clotting times, suggesting adrenal hemorrhage as a unique manifestation of TAFRO syndrome. In general, patients with TAFRO syndrome exhibit a more aggressive clinical course and poorer outcome than those with iMCD-NOS. To ameliorate this poor prognosis, it is important to diagnose the disease early and immediately start powerful immunosuppressive agents such as tocilizumab. Based on this case, adrenal hemorrhage may suggest TAFRO syndrome, and facilitate the rapid diagnosis of this complicated and rare disease.

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Validated international definition of the thrombocytopenia, anasarca, fever, reticulin fibrosis, renal insufficiency, and organomegaly clinical subtype (TAFRO) of idiopathic multicentric Castleman disease

et al. 2021

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Thrombocytopenia, anasarca, fever, reticulin fibrosis, renal insufficiency, and organomegaly (TAFRO) syndrome is a heterogeneous entity manifesting with a constellation of symptoms described above that can occur in the context of idiopathic multicentric Castleman disease (iMCD) as well as infectious diseases, malignancies, and rheumatologic disorders. So, iMCD-TAFRO is an aggressive subtype of iMCD with TAFRO syndrome and often hyper-vascularized lymph nodes. Since we proposed diagnostic Yoshito Nishimura and Noriko Iwaki contributed equally to this study.

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Computed tomography findings of early-stage TAFRO syndrome and associated adrenal abnormalities

Cited by 15 publications

References 32 publications

Bilateral Adrenal Infarctions as an Initial Manifestation of TAFRO Syndrome: A Case Report and Review of the Literature

Bilateral Adrenal Infarctions as an Initial Manifestation of TAFRO Syndrome: A Case Report and Review of the Literature

Fatal case of TAFRO syndrome with unilateral adrenal hemorrhage in early-stage disease

Validated international definition of the thrombocytopenia, anasarca, fever, reticulin fibrosis, renal insufficiency, and organomegaly clinical subtype (TAFRO) of idiopathic multicentric Castleman disease

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