A Novel Diagnostic Model for Primary Adrenal Lymphoma

Yu, Kai; Xue, Qingping; Zhou, Fangli; Tian, Haoming; Qiao, Xin; Chen, Tao; Ren, Yan

doi:10.3389/fendo.2021.636658

Cited by 4 publications

(1 citation statement)

References 47 publications

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“…PAL is suspect on the basis of clinical and imaging assessment, however should be definitively confirmed by histopathology. Recently it has been proposed [ 11 ] a quick and efficient diagnostic method to guide PAL detection.…”

Section: Introductionmentioning

confidence: 99%

An unusual cause of adrenal insufficiency with elevation of 17-hydroxyprogesterone: case report

Teti¹,

Bezante

Gatto

et al. 2023

BMC Endocr Disord

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Background We present an intriguing case of primary adrenal lymphoma, with associated primary adrenal insufficiency (PAI), in a patient presenting a transitory partial 21-hydroxylase deficiency during the active phase of the adrenal disease. Case presentation An 85-years old woman was referred because of worsening asthenia, lumbar pain, generalized myalgia and arthralgia. During investigations a computed tomography (CT) scan evidenced two large bilateral adrenal masses, highly suspicious for primary adrenal tumor. The hormonal assessment revealed very low levels of morning plasma cortisol and 24-h urinary cortisol, elevated ACTH levels with low plasma concentration of aldosterone, pointing to the diagnosis of PAI. After diagnosis of PAI our patient started glucocorticoid and mineralcorticoid replacement therapy with clinical benefit. In order to further characterize the adrenal lesions, adrenal biopsy, was performed. The histology revealed a high grade non-Hodgkin lymphoma with an immunophenotype consistent with intermediate aspects between diffuse large B-cell and Burkitt lymphoma, with a high proliferation index (KI-67 > 90%). The patient received chemotherapy with epirubicin, vincristine, cyclophosphamide, and rituximab, associated with methylprednisolone that resulted in a complete clinical and radiological remission within one year. After 2 years from the diagnosis and a total of 6 cycles of rituximab, the patient was in good clinical condition and was taking only the replacement therapy for PAI. The patient initially presented also a slight increase of 17-hydroxyprogesterone (17-OHP) for age that normalize after resolution of lymphoproliferative disease. Conclusions In the presence of bilateral adrenal disease and/or in the presence of signs and symptoms of PAI clinicians must exclude the presence of PAL. The evidence of elevated ACTH-stimulated 17-OHP levels also in patients with other adrenal masses, together with the detection of elevated basal 17-OHP levels in our patient make it more plausible, in our view, an effect of the lesion on the “healthy” adrenal tissue residue than a direct secretory activity by the adrenal tumor.

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Section: Introductionmentioning

confidence: 99%

An unusual cause of adrenal insufficiency with elevation of 17-hydroxyprogesterone: case report

Teti¹,

Bezante

Gatto

et al. 2023

BMC Endocr Disord

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The combination of early identification, chemotherapy, and autologous stem cell transplantation obtained favorable outcomes in unilateral primary adrenal lymphoma: A case report

Huang

Xiao²,

Zhang³

et al. 2022

Endocrine

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sIL-2R: A Novel Diagnostic Biomarker for Primary Adrenal Lymphoma

Xie

Zhao

et al. 2022

The Journal of Clinical Endocrinology &Amp; Metabolism

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Context Primary adrenal lymphoma (PAL) is difficult to distinguish from other adrenal masses. Soluble interleukin-2 receptor (sIL-2R) is a diagnostic biomarker for nodal non-Hodgkin lymphoma, whose association with PAL is unknown. Objective The aim of this study was to determine the diagnostic utility of serum sIL-2R for Patients with PAL. Design Prospective cohort study. Settings and Participants A total of 118 patients with adrenal masses who were willing to be tested for levels of serum sIL-2R from a tertiary hospital between 2019 and 2021 were included. Main Outcomes and Measures Serum sIL-2R and lactate dehydrogenase (LDH) levels. Results Patients with PAL had significantly higher sIL-2R levels than those of patients with other adrenal masses with indetermined and benign CT features (both p<0.001). The LDH levels of patients with PAL were also significantly higher than those of patients with other adrenal masses with indeterminate and benign CT features (both p<0.001). Good discrimination of patients with PAL from other patients (PAL vs. other adrenal masses with indeterminate CT features/non-PAL) was achieved with an area under the receiver operating characteristic (ROC) curve (AUC) of 0.984 (95% CI, 0.95-1)/0.992 (95% CI, 0.975-1) using the serum levels of sIL-2R and further improved (AUC=0.998, 95% CI, 0.994-1; AUC=0.999, 95% CI, 0.996-1) after adjusting by LDH category. Conclusions For the first time, we have identified that serum sIL-2R and LDH category-adjusted sIL-2R levels have good diagnostic performances for PAL.

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A Novel Diagnostic Model for Primary Adrenal Lymphoma

Cited by 4 publications

References 47 publications

An unusual cause of adrenal insufficiency with elevation of 17-hydroxyprogesterone: case report

An unusual cause of adrenal insufficiency with elevation of 17-hydroxyprogesterone: case report

The combination of early identification, chemotherapy, and autologous stem cell transplantation obtained favorable outcomes in unilateral primary adrenal lymphoma: A case report

sIL-2R: A Novel Diagnostic Biomarker for Primary Adrenal Lymphoma

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