Complete Resolution of a Case of TAFRO Syndrome Accompanied by Mediastinal Panniculitis, Adrenal Lesion, and Liver Damage with Hyperbilirubinemia

Ono, Shiro; Yoshimoto, Kiyomi; Nishimura, Nobushiro; Yoneima, Ryo; Kawana, Hiromasa; Kobayashi, Tadanao; Tai, Yoshiaki; Miyamoto, Makiko; Tsushima, Emiko; Yada, Noritaka; Nishio, Kenji

doi:10.2169/internalmedicine.5850-20

Cited by 6 publications

(14 citation statements)

References 25 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: 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: 99%

“…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%

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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“…In addition to these cases, we identified eight cases of TAFRO syndrome with adrenal abnormalities in the English literature (6)(7)(8)(9)(10)(11)(12). A summary of the 16 total cases, including the current case, is listed in Table 3.…”

Section: Discussionmentioning

confidence: 99%

“…Among them, 10 cases exhibited bilateral adrenal abnormalities, and no apparent laterality was observed. In particular, eight out of nine cases for which detailed clinical data were available presented with abdominal pain as the first manifestation of TAFRO syndrome (7)(8)(9)(10)(11)(12). Nishimura et al reported that the incidence of abdominal pain as the first chief complaint was 50% (15).…”

Section: Discussionmentioning

confidence: 99%

“…A significant number of cases of TAFRO syndrome present with abdominal pain as the initial manifestation; however, the diagnostic criteria and disease severity classifications include no abdominal symptoms. Furthermore, some reported cases of TAFRO syndrome have been accompanied by adrenal abnormalities on imaging, such as adrenomegaly, hemorrhaging, or infarction (6)(7)(8)(9)(10)(11)(12). More recently, the usefulness of detecting adrenal abnormalities via computed tomography (CT) for the early diagnosis of this disease has been reported (13).…”

Section: Introductionmentioning

confidence: 99%

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Acute Abdomen and Adrenal Swelling as the First Manifestations of TAFRO Syndrome

et al. 2023

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TAFRO syndrome, a rare systemic inflammatory disorder, commonly develops in an acute or subacute manner, with an aggressive clinical behavior. A substantial number of cases of TAFRO syndrome presenting with abdominal pain, and adrenal abnormalities on imaging have also been reported. A 54-year-old man developed severe acute abdominal pain. Bilateral adrenal swelling was detected on computed tomography. Although the abdominal pain resolved spontaneously, a fever and anasarca were observed. The patient was eventually diagnosed with TAFRO syndrome, and corticosteroid administration resulted in remission. TAFRO syndrome should be included in the differential diagnosis of acute abdomen and adrenal abnormalities.

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Evaluation of the correlation between multiple organ calcification on CT and disease severity in patients with TAFRO syndrome

et al. 2023

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Purpose The purpose of this study was to investigate the incidence of multiple organ calcification and the correlation between multiple organ calcification and clinical severity in patients with thrombocytopenia, anasarca, fever, reticulin fibrosis, renal dysfunction, and organomegaly (TAFRO) syndrome. Methods We retrospectively identified 13 patients with TAFRO syndrome who were treated at our hospital between February 2019 and March 2021. Computed tomography (CT) images of TAFRO patients, which were acquired at admission and one month after admission, were evaluated. Additionally, clinical and laboratory data related to organ calcification and severity classification of TAFRO syndrome were investigated. The correlation between the presence of organ calcification on CT and TAFRO syndrome-severity classification was evaluated. Results One month after admission, calcification of the myocardium, adrenal glands, gallbladder wall, pancreas, kidney, skeletal muscle, and skin were observed in 38%, 46%, 15%, 15%, 15%, 23%, and 15% of the thirteen patients, respectively. The occurrence rate of calcifications in the myocardium, adrenal glands, and skeletal muscle was significantly higher in patients with a grade 4 or higher clinical severity than in those with a level up to grade 3 (p = 0.001, p = 0.005, and p = 0.035, respectively). Conclusions Our results suggest that the higher the clinical severity in patients with TAFRO syndrome, the higher is the frequency of calcification in the myocardium, adrenal glands, and skeletal muscle; therefore, the assessment of these organ calcifications on CT images may be useful in predicting the severity of TAFRO syndrome.

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Complete Resolution of a Case of TAFRO Syndrome Accompanied by Mediastinal Panniculitis, Adrenal Lesion, and Liver Damage with Hyperbilirubinemia

Cited by 6 publications

References 25 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

Acute Abdomen and Adrenal Swelling as the First Manifestations of TAFRO Syndrome

Evaluation of the correlation between multiple organ calcification on CT and disease severity in patients with TAFRO syndrome

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