IgG4-Associated Adrenalitis—a Case Report

Saeger, Wolfgang; Lohe, B.; Engels, Christina; Werner, Ulrike

doi:10.1007/s12022-018-9531-x

Cited by 4 publications

(3 citation statements)

References 23 publications

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“…Kuo et al described 5 out of 10 SANTs with abdominal disseminated calcifying fibrous tumors sharing features overlapping with IgG4 RD [21]. Scattered cases also demonstrate higher IgG4: IgG ratio in tissues and elevated serous IgG, IgG4 ratio, suggesting their possible association with IgG4 RD [22–24]. However, other reports did not show any increased positivity IgG4 or IgG: IgG4 and failed to provide straightforward evidence of the relationship between CFT and IgG4 RD [25, 26].…”

Section: Discussionmentioning

confidence: 99%

Clinicopathologic Study of Calcifying Fibrous Tumor Emphasizing Different Anatomical Distribution and Favorable Prognosis

Zhou

et al. 2019

BioMed Research International

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Aims. Calcifying fibrous tumor (CFT) is a very rare begin fibroblastic tumor featuring a widely anatomical distribution and may mimic various spindle cell tumors. Misdiagnosis and hence mistreatment are likely caused due to unfamiliarity to clinicians or junior pathologists. We collected a relatively large series of CFTs in our institution aiming at further summarizing their clinicopathologic features in Chinese patients and discussing the diagnosis and differential diagnosis in clinical practice. Methods. Clinicopathologic data of 22 CFTs were retrospectively reviewed. Histologic features were reevaluated and summarized. Immunostaining markers include CD34, SMA, Desmin, keratin, S100, ALK1, CD117, IgG, IgG4, and Ki-67. Follow-up of all cases was performed. Results. 22 CFTs include gastric (n=8), pulmonary (n=2), hepatic (n=2), cervical (n=1), appendiceal (n=1), esophageal (n=1), retroperitoneal (n=1), intra-abdominal (n=1), diaphragmatic (n=1), spermatic cord and scrotum (n=1), anconeal (n=1), mesenteric (n=1), and omental (n=1) lesions. Coexisting hepatocellular carcinoma, pancreatic carcinoma, pheochromocytoma, Castleman disease, and leiomyoma of the uterus and other metabolic or functional disorders were also appreciated. CFT histologically features spindle cells embedded dense hyalinized stroma with scattered psammomatous calcifications and lymphoplasmacytic infiltration and immunohistochemically for CD34. None of any individuals die of CFT per se. Conclusion. Our study discloses that CFT is a bona fide benign fibroblastic lesion, regardless of its developing location. Involvement of digestive tract seems much more common in the Chinese population. Awareness of the clinicopathologic characteristics of this rare entity and its mimickers contribute to avoiding misdiagnosis and mistreatment in clinical practice.

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Section: Discussionmentioning

confidence: 99%

Clinicopathologic Study of Calcifying Fibrous Tumor Emphasizing Different Anatomical Distribution and Favorable Prognosis

Zhou

et al. 2019

BioMed Research International

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“…IgG4-related disease of the adrenal glands is very uncommon, and only anecdotal cases have been reported, presenting as pseudotumor masses that, at histological analysis, showed the characteristic features of IgG4-RD (storiform fibrosis, dense plasma cell infiltrate, high IgG4/IgG ratio, and high numbers of IgG4 + plasma cells) [ 232 , 233 ].…”

Section: Organ-specific Inflammatory Diseasesmentioning

confidence: 99%

Inflammatory and Infectious Disorders in Endocrine Pathology

et al. 2023

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A variety of inflammatory conditions may directly involve the endocrine glands, leading to endocrine dysfunction that can cause severe consequences on patients’ health, if left untreated. Inflammation of the endocrine system may be caused by either infectious agents or other mechanisms, including autoimmune and other immune-mediated processes. Not infrequently, inflammatory and infectious diseases may appear as tumor-like lesions of endocrine organs and simulate neoplastic processes. These diseases may be clinically under-recognized and not infrequently the diagnosis is suggested on pathological samples. Thus, the pathologist should be aware of the basic principles of their pathogenesis, as well as of their morphological features, clinicopathological correlates, and differential diagnosis. Interestingly, several systemic inflammatory conditions show a peculiar tropism to the endocrine system as a whole. In turn, organ-specific inflammatory disorders are observed in endocrine glands. This review will focus on the morphological aspects and clinicopathological features of infectious diseases, autoimmune disorders, drug-induced inflammatory reactions, IgG4-related disease, and other inflammatory disorders involving the endocrine system. A mixed entity-based and organ-based approach will be used, with the aim to provide the practicing pathologist with a comprehensive and practical guide to the diagnosis of infectious and inflammatory disorders of the endocrine system.

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“…High concentrations of serum IgG4 occur in other diseases [ 8 - 10 ], and wide variability in the diagnostic performance of serum IgG4 for IgG4-RD has been reported owing to different sample sizes, test methods, and ethnic dissimilarities among studies [ 11 - 14 ]. Given the lack of in-depth understanding of this disease, there are limited studies on the diagnostic efficacy of serum IgG4 in patients with IgG4-RD, especially in China, with the majority of published studies being case reports [ 15 , 16 ]. The majority of clinical descriptions of IgG4-RD have been concentrated on the Japanese and Caucasian populations and are confined to studies of a single organ [ 17 - 20 ].…”

Section: Introductionmentioning

confidence: 99%

Clinical Significance of Serum IgG4 in the Diagnosis and Treatment Response of IgG4-Related Disease in Adults of Southwest China: A Retrospective Study

Wei

Guo

et al. 2023

Ann Lab Med

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Background: There is no standard cut-off value of serum IgG4 concentration and serum IgG4/total IgG ratio for the diagnosis of IgG4-related disease (IgG4-RD) or as a marker of treatment responses. We aimed to explore this issue through a retrospective cohort analysis of adults in southwest China. Methods:The diagnostic performance of serum IgG4 concentration and IgG4/IgG ratio for IgG4-RD was evaluated in a retrospective analysis of 177 adults newly diagnosed as having IgG4-RD and 877 adults without IgG4-RD. Dynamic analysis was performed to evaluate the significance of serum IgG4 concentration on IgG4-RD treatment responses.Results: The serum IgG4 concentration differed according to sex. The optimal cut-off values of serum IgG4 concentration and IgG4/IgG ratio for IgG4-RD diagnosis were 1.92 g/L and 0.12 in males and 1.83 g/L and 0.11 in females, respectively. For patients with serum IgG4 concentration > 2.01 g/L, the cut-off values in the total population were > 3.00 g/L and 0.19, respectively. The median serum IgG4 concentration decreased over time, and the decrease rate increased over time. The serum IgG4 concentration significantly decreased at > 1 week post-treatment (P = 0.004), and the median decrease rate was close to 50% at > 4 weeks post-treatment.Conclusions: Serum IgG4 can be a good indicator for IgG4-RD diagnosis; however, different diagnostic cut-off values should be determined according to sex. The decreasing rate is more conducive than the serum IgG4 concentration to monitor treatment efficacy. The IgG4/IgG ratio did not improve the diagnostic efficacy for IgG4-RD.

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IgG4-Associated Adrenalitis—a Case Report

Cited by 4 publications

References 23 publications

Clinicopathologic Study of Calcifying Fibrous Tumor Emphasizing Different Anatomical Distribution and Favorable Prognosis

Clinicopathologic Study of Calcifying Fibrous Tumor Emphasizing Different Anatomical Distribution and Favorable Prognosis

Inflammatory and Infectious Disorders in Endocrine Pathology

Clinical Significance of Serum IgG4 in the Diagnosis and Treatment Response of IgG4-Related Disease in Adults of Southwest China: A Retrospective Study

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