Postoperative expression of Cushing disease in a young male: metamorphosis of silent corticotroph adenoma?

Jahan, Sharmin; Hasanat, Muhammad Abul; Mahmood, Tahseen; Morshed, Md Shahed; Haq, Raziul; Fariduddin, .

doi:10.1530/edm-19-0046

Cited by 4 publications

(6 citation statements)

References 26 publications

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“…All the tumors were macroadenomas. The invasion was detected as follows: cavernous sinus invasion in seven cases (43.8%) ( 17 , 18 ), sphenoid sinus invasion in two cases (12.5%) ( 13 ), simultaneous sphenoid sinus and cavernous sinus invasion in two cases (12.5%) ( 14 ), and no sphenoid sinus or cavernous sinus invasion in five cases (31.3%) ( 15 , 16 ). The MRI images from six cases and CT images from one case in our center are shown in Figure 1 .…”

Section: Resultsmentioning

confidence: 99%

“…Most SCA cases remained "silent" for a long time. Very rare cases of SCAs have the potential to transform into clinically functional corticotroph adenomas (13)(14)(15)(16)(17)(18). To investigate the clinical features of SCAs with transformation, a total of 16 SCAs with detailed data were collected from our medical center and the literature.…”

Section: Discussionmentioning

confidence: 99%

“…It is worth noting that the first case of NFPA that transformed into a functional corticotroph adenoma, after 7 years of follow-up, was reported by Cooper et al (12), causing some concern among clinicians. Subsequently, a few cases of SCAs that transformed from silent to clinically functional corticotroph adenomas have been reported (13)(14)(15)(16)(17)(18). However, the underlying mechanism of the transformation of function has not yet been elucidated.…”

Section: Introductionmentioning

confidence: 99%

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Clinical, Laboratory, and Treatment Profiles of Silent Corticotroph Adenomas That Have Transformed to the Functional Type: A Case Series With a Literature Review

et al. 2020

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Purpose: Silent corticotroph adenoma (SCA) is clinically non-functional pituitary adenoma with expression of corticotropin or Tpit. To further understand the characteristics of this rare type of SCA transforming to a functional SCA, we retrospectively reviewed SCAs that converted to typical Cushing's syndrome at a tertiary medical center and the relevant literature. Methods: Patients were identified based on the diagnosis of pituitary adenoma without symptoms of hypercortisolism at the initial visit with positive Immunohistochemical (IHC) staining for corticotropin or Tpit after surgery and subsequent transformation to functional SCAs during the follow-up period from March 1990 to January 2020 at Peking Union Medical College Hospital and in the literature. The characteristics of the clinical manifestations, biochemical results, imaging findings, pathology findings and outcome were analyzed. Results: Altogether, 16 patients were included in the study with an average age of 42.0 ± 12.48 (18-65) years at the first visit. Females were slightly predominant (F:M = 1.3:1). The median time of conversion from the nonfunctional to the functional type was 30 (13.0, 68.3) months. Once a functional SCA developed, the adrenocorticotropic hormone (ACTH) level and 24-h urine free cortisol were increased 3.8-(2.6, 12.9) and 5.3-(2.6, 19.3) fold, respectively, above the normal range. Approximately 50% of the patients had macrocystic changes on pituitary MRI. All 16 patients experienced 1-5 surgeries with a median of 2.5 (2.0, 4.0) surgeries. The proportion of patients with Ki-67 ≥ 3% increased from 22.2% (2/9) at the beginning to 50% (7/14) at the time of functional SCA diagnosis. Thirteen patients received radiotherapy, and 4 patients (30.8%) achieved remission. Four patients with refractory functional SCAs received temozolomide treatment with the normalization of cortisol in 4 cases and reduced tumor volume in 3 cases. Zheng et al. Clinical Features of SCA Transforming to Functional Conclusion: In this study, all cases that transformed to functional SCAs were macroadenomas. Hypercortisolism was more severe in functional SCA patients. The tumors tended to have frequent recurrence and were highly invasive. Temozolomide could be a promising treatment for refractory functional SCA cases. Long-term follow-up is needed for nonfunctional SCAs since some cases have the potential to transform to clinical Cushing's syndrome.

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Section: Resultsmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Clinical, Laboratory, and Treatment Profiles of Silent Corticotroph Adenomas That Have Transformed to the Functional Type: A Case Series With a Literature Review

et al. 2020

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“…AN has been reported in Cushing’s syndrome, a complex conditions which also brings together IR and hyperandrogenemia in women [ 158 , 159 ]. A 12-year-old girl with newly diagnosed celiac disease and Cushing’s disease was admitted with AN [ 160 ].…”

Section: Discussionmentioning

confidence: 99%

Acanthosis Nigricans: Pointer of Endocrine Entities

et al. 2022

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Acanthosis nigricans (AN) has been reported in relation to insulin resistance (IR). We aim to review AN through an endocrine and metabolic perspective focusing on IR in association with metabolic complications such as obesity, diabetes mellitus (DM), and metabolic syndrome (MS) with/without polycystic ovary syndrome (PCOS). We revised English papers on PubMed covering publications from the last 5 years. The current prevalence of AN varies from 4.5 to 74% (or even 100%, depending on the studied population), with equal distribution among females and males. Despite higher incidence with an age-dependent pattern, an alarming escalation of cases has been noted for obesity and MS in younger populations . Most frequent IR-associated sites are the neck, axilla, and knuckles, but unusual locations such as the face have also been reported. Quantitative scales such as Burke have been used to describe the severity of the dermatosis, particularly in correlation with IR elements. Dermoscopic examination are required, for instance, in cases with sulcus cutis, hyperpigmented spots, crista cutis, and papillary projections. A skin biopsy may be necessary, but it is not the rule. Both IR that clinically manifests with or without obesity/MS correlates with AN; most studies are cross-sectional, with only a few longitudinal. The approach varied from screening during school periodic checkups/protocols/programs to subgroups of individuals who were already known to be at high cardio-metabolic risk. AN was associated with type2DM, as well as type 1DM. Females with PCOS may already display metabolic complications in 60–80% of cases, with AN belonging to the associated skin spectrum. AN management depends on underlying conditions, and specific dermatological therapy is not generally required, unless the patient achieves metabolic control, has severe skin lesions, or desires cosmetic improvement. In IR cases, lifestyle interventions can help, including weight control up to bariatric surgery. In addition, metformin is a key player in the field of oral medication against DM type 2, a drug whose indication is extended to PCOS and even to AN itself, outside the specific panel of glucose anomalies. In terms of cosmetic intervention, limited data have been published on melatonin, urea cream, topical retinoids, vitamin D analogs, or alexandrite laser. In conclusion, awareness of IR and its associated clinical features is essential to provide prompt recognition of underlying conditions. AN represents a useful non-invasive surrogate marker of this spectrum in both children and adults. The pivotal role of this dermatosis could massively improve endocrine and metabolic assessments.

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“…Adrenocorticotropic hormone (ACTH)-secreting pituitary tumors that cause Cushing's disease account for 5% to 10% of pituitary adenomas. Silent corticotroph adenomas are an uncommon subgroup of nonfunctioning pituitary tumors (NFPAs) (1) characterized by no biochemical or clinical evidence of Cushing's disease (2,3). They are generally diagnosed after surgery by positive immunostaining for ACTH (4).…”

Section: Introductionmentioning

confidence: 99%

Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression

Demarchi

Perrone²,

Romero³

et al. 2022

Front. Endocrinol.

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Clinically silent corticotroph tumors are usually macroadenomas that comprise 20% of ACTH tumors. They frequently progress to aggressive tumors with high recurrence, invasiveness, and on rare occasions, they may become hormonally active causing Cushing’s disease. Trustable biomarkers that can predict their aggressive course, as well as their response to traditional or new therapies, are paramount. Aberrant β-Catenin expression and localization have been proposed as responsible for several malignancies including pituitary tumors. Nevertheless, the role of β-Catenin in the aggressive transformation of silent corticotropinomas and their response to Temozolomide salvage treatment have not been explored yet. In this work, we present a case of a silent corticotroph tumor that invaded cavernous sinus and compressed optic chiasm and, after a first total resection and tumor remission it recurred six years later as an aggressive ACTH-secreting tumor. This lesion grew with carotid compromise and caused Cushing’s signs. It required multiple medical treatments including Cabergoline, Ketoconazole, TMZ, and radiotherapy. Besides, other two surgeries were needed until it could be controlled. Interestingly, we found α-SMA vascular area reduction and differential β-Catenin cell localization in the more aggressive tumor stages characterized by high Ki-67 indexes and p53 expression. Our results may indicate a role of angiogenesis and β-Catenin trigged events in the pituitary tumor progression, which could in turn affect the response to TMZ and/or conventional treatments. These molecular findings in this unusual case could be useful for future management of aggressive pituitary tumors.

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Postoperative expression of Cushing disease in a young male: metamorphosis of silent corticotroph adenoma?

Cited by 4 publications

References 26 publications

Clinical, Laboratory, and Treatment Profiles of Silent Corticotroph Adenomas That Have Transformed to the Functional Type: A Case Series With a Literature Review

Clinical, Laboratory, and Treatment Profiles of Silent Corticotroph Adenomas That Have Transformed to the Functional Type: A Case Series With a Literature Review

Acanthosis Nigricans: Pointer of Endocrine Entities

Case Report: Progression of a Silent Corticotroph Tumor to an Aggressive Secreting Corticotroph Tumor, Treated by Temozolomide. Changes in the Clinic, the Pathology, and the β-Catenin and α-SMA Expression

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