scholarly journals Double trouble: two cases of dual adrenal pathologies in one adrenal mass

Author(s):  
Vasileios Chortis ◽  
Christine J H May ◽  
Kassiani Skordilis ◽  
John Ayuk ◽  
Wiebke Arlt ◽  
...  

Summary Context Adrenal incidentalomas (AI) represent an increasingly common problem in modern endocrine practice. The diagnostic approach to AIs can be challenging and occasionally reveals surprising features. Here we describe two rare cases of complex adrenal lesions consisting of phaeochromocytomas with synchronous metastases from extra-adrenal primaries. Case descriptions Patient 1 – a 65-year-old gentleman with a newly diagnosed malignant melanoma was found to harbour an adrenal lesion with suspicious radiographic characteristics. Percutaneous adrenal biopsy was consistent with adrenocortical adenoma. After excision of the skin melanoma and regional lymphatic metastases, he was followed up without imaging. Three years later, he presented with abdominal discomfort and enlargement of his adrenal lesion, associated with high plasma metanephrines. Adrenalectomy revealed a mixed tumour consisting of a large phaeochromocytoma with an embedded melanoma metastasis in its core. Patient 2 – a 63-year-old lady with a history of NF-1-related phaeochromocytoma 20 years ago and previous breast cancer presented with a new adrenal lesion on the contralateral side. Plasma normetanephrine was markedly elevated. Elective adrenalectomy revealed an adrenal tumour consisting of chromaffin cells intermixed with breast carcinoma cells. Conclusions Adrenal incidentalomas require careful evaluation to exclude metastatic disease, especially in the context of a history of previous malignancy. Adrenal biopsy provides limited and potentially misleading information. Phaeochromocytomas are highly vascularised tumours that may function as a sieve, extracting and retaining irregularly shaped cancer cells, thereby yielding adrenal masses with intriguing dual pathology. Learning points: Adrenal incidentalomas require careful evaluation focused on exclusion of underlying hormone excess and malignant pathology. Adrenal biopsy can be misleading and should only be considered in select cases. Phaeochromocytomas harbouring intratumoural metastases from other, extra-adrenal primary malignancies represent rare pathological entities that highlight the complexities that can be presented by adrenal tumours.

Author(s):  
Angelo Paci ◽  
Ségolène Hescot ◽  
Atmane Seck ◽  
Christel Jublanc ◽  
Lionel Mercier ◽  
...  

Summary Mitotane (o,p′-DDD) is the standard treatment for advanced adrenocortical carcinoma (ACC). Monitoring of plasma mitotane levels is recommended to look for a therapeutic window between 14 and 20mg/L, but its positive predictive value requires optimization. We report the case of an ACC patient with a history of dyslipidemia treated with mitotane in whom several plasma mitotane levels >30mg/L were found together with an excellent neurological tolerance. This observation led us to compare theoretical or measured o,p′-DDD and o,p′-DDE levels in a series of normolipidemic and dyslipidemic plasma samples to explore potential analytical issues responsible for an overestimation of plasma mitotane levels. We demonstrate an overestimation of mitotane measurements in dyslipidemic patients. Mitotane and o,p′-DDE measurements showed a mean 20% overestimation in hypercholesterolemic and hypertriglyceridemic plasma, compared with normolipidemic plasma. The internal standard p,p′-DDE measurements showed a parallel decrease in hypercholesterolemic and hypertriglyceridemic plasma, suggesting a matrix effect. Finally, diluting plasma samples and/or using phospholipid removal cartridges allowed correcting such interference. Learning points Hypercholesterolemia (HCH) and hypertriglyceridemia (HTG) induce an overestimation of plasma mitotane measurements. We propose a routine monitoring of lipidemic status. We propose optimized methodology of measurement before interpreting high plasma mitotane levels.


Author(s):  
Ya-Wun Guo ◽  
Chii-Min Hwu ◽  
Justin Ging-Shing Won ◽  
Chia-Huei Chu ◽  
Liang-Yu Lin

Summary A functional lesion in corticotrophin (ACTH)-independent Cushing’s syndrome is difficult to distinguish from lesions of bilateral adrenal masses. Methods for distinguishing these lesions include adrenal venous sampling and 131I-6β-iodomethyl-19-norcholesterol (131I-NP-59) scintigraphy. We present a case of a 29-year-old Han Chinese female patient with a history of hypercholesterolaemia and polycystic ovary syndrome. She presented with a 6month history of an 8kg body weight gain and gradual rounding of the face. Serial examinations revealed loss of circadian rhythm of cortisol, elevated urinary free-cortisol level and undetectable ACTH level (<5pg/mL). No suppression was observed in both the low- and high-dose dexamethasone suppression tests. Adrenal computed tomography revealed bilateral adrenal masses. Adrenal venous sampling was performed, and the right-to-left lateralisation ratio was 14.29. The finding from adrenal scintigraphy with NP-59 was consistent with right adrenal adenoma. The patient underwent laparoscopic right adrenalectomy, and the pathology report showed adrenocortical adenoma. Her postoperative cortisol level was 3.2μg/dL, and her Cushingoid appearance improved. In sum, both adrenal venous sampling and 131I-NP-59 scintigraphy are good diagnostic methods for Cushing’s syndrome presenting with bilateral adrenal masses. Learning points The clinical presentation of Cushing’ syndrome includes symptoms and signs of fat redistribution and protein-wasting features. The diagnosis of patients with ACTH-independent Cushing’s syndrome with bilateral adrenal masses is challenging for localisation of the lesion. Both adrenal venous sampling and 131I-NP-59 scintigraphy are good methods to use in these patients with Cushing’s syndrome presenting with bilateral adrenal masses.


2018 ◽  
Author(s):  
Yasir Elhassan ◽  
Fares Alahdab ◽  
Alessandro Prete ◽  
Danae Delivanis ◽  
Aakanksha Khanna ◽  
...  

2018 ◽  
Vol 35 (8) ◽  
pp. 522.1-522
Author(s):  
Charlotte Delcourt ◽  
Jean Cyr Yombi ◽  
Halil Yildiz

Clinical introductionA 37-year-old man with history of lymph node tuberculosis presented with bilateral inguinal swelling with night sweats but no fever for 2 weeks. He had a cat but he had no history of scratches. He had an extraconjugal sexual intercourse a few weeks before. Physical examination revealed 5 cm tender, erythematous and painful bilateral inguinal adenopathy (figure 1A) and a small ulceration at the base of the penis (figure 1B). Vital signs were normal.Figure 1(A) Inguinal lymphadenopathy. (B) Ulceration at the base of the penis.QuestionWhat is the most likely diagnosis?ToxoplasmosisTuberculosisCat-scratch diseaseLymphogranuloma venereumSyphilis


2015 ◽  
Vol 2015 ◽  
pp. 1-4
Author(s):  
Keishi Maruo ◽  
Toshiya Tachibana ◽  
Shinichi Inoue ◽  
Fumihiro Arizumi ◽  
Shinichi Yoshiya

Most unilateral pedicle stress fractures occur on the contralateral side of patients with unilateral spondylolysis. However, there are few reports of unilateral pedicle stress fractures in patients with bilateral spondylolysis and spondylolisthesis. We report a unique case of unilateral pedicle stress fracture in a long-term hemodialysis patient with isthmic spondylolisthesis. A 65-year-old man who had undergone hemodialysis presented with lower back pain that had persisted for several years. The patient experienced severe right lower extremity pain with no history of trauma. Computed tomography revealed unilateral pedicle fracture with bilateral L5 spondylolysis and spondylolisthesis with progression of scoliosis. The patient underwent Gill laminectomy of L5 with pedicle screw fixation at L4-S1 and interbody fusion at L5-S1. The patient’s leg pain ceased immediately, and he began walking without leg pain. In our present patient, development of scoliosis caused by destructive spondyloarthropathy may have contributed to a unilateral pedicle fracture.


2016 ◽  
Vol 16 (1) ◽  
pp. 40-59 ◽  
Author(s):  
Claudio Maccone

AbstractIn two recent papers (Maccone 2013, 2014) as well as in the book (Maccone 2012), this author described the Evolution of life on Earth over the last 3.5 billion years as a lognormal stochastic process in the increasing number of living Species. In (Maccone 2012, 2013), the process used was ‘Geometric Brownian Motion’ (GBM), largely used in Financial Mathematics (Black-Sholes models). The GBM mean value, also called ‘the trend’, always is an exponential in time and this fact corresponds to the so-called ‘Malthusian growth’ typical of population genetics. In (Maccone 2014), the author made an important generalization of his theory by extending it to lognormal stochastic processes having an arbitrary trend mL(t), rather than just a simple exponential trend as the GBM have.The author named ‘Evo-SETI’ (Evolution and SETI) his theory inasmuch as it may be used not only to describe the full evolution of life on Earth from RNA to modern human societies, but also the possible evolution of life on exoplanets, thus leading to SETI, the current Search for ExtraTerrestrial Intelligence. In the Evo-SETI Theory, the life of a living being (let it be a cell or an animal or a human or a Civilization of humans or even an ET Civilization) is represented by a b-lognormal, i.e. a lognormal probability density function starting at a precise instant b (‘birth’) then increasing up to a peak-time p, then decreasing to a senility-time s (the descending inflexion point) and then continuing as a straight line down to the death-time d (‘finite b-lognormal’).(1)Having so said, the present paper describes the further mathematical advances made by this author in 2014–2015, and is divided in two halves: Part One, devoted to new mathematical results about the History of Civilizations as b-lognormals, and(2)Part Two, about the applications of the Evo-SETI Theory to the Molecular Clock, well known to evolutionary geneticists since 50 years: the idea is that our EvoEntropy grows linearly in time just as the molecular clock. (a)Summarizing the new results contained in this paper: In Part One, we start from the History Formulae already given in (Maccone 2012, 2013) and improve them by showing that it is possible to determine the b-lognormal not only by assigning its birth, senility and death, but rather by assigning birth, peak and death (BPD Theorem: no assigned senility). This is precisely what usually happens in History, when the life of a VIP is summarized by giving birth time, death time, and the date of the peak of activity in between them, from which the senility may then be calculated (approximately only, not exactly). One might even conceive a b-scalene (triangle) probability density just centred on these three points (b, p, d) and we derive the relevant equations. As for the uniform distribution between birth and death only, that is clearly the minimal description of someone's life, we compare it with both the b-lognormal and the b-scalene by comparing the Shannon Entropy of each, which is the measure of how much information each of them conveys. Finally we prove that the Central Limit Theorem (CLT) of Statistics becomes a new ‘E-Pluribus-Unum’ Theorem of the Evo-SETI Theory, giving formulae by which it is possible to find the b-lognormal of the History of a Civilization C if the lives of its Citizens Ci are known, even if only in the form of birth and death for the vast majority of the Citizens.(b)In Part Two, we firstly prove the crucial Peak-Locus Theorem for any given trend mL(t) and not just for the GBM exponential. Then we show that the resulting Evo-Entropy grows exactly linearly in time if the trend is the exponential GMB trend.(c)In addition, three Appendixes (online) with all the relevant mathematical proofs are attached to this paper. They are written in the Maxima language, and Maxima is a symbolic manipulator that may be downloaded for free from the web.In conclusion, this paper further increases the huge mathematical spectrum of applications of the Evo-SETI Theory to prepare Humans for the first Contact with an Extra-Terrestrial Civilization.


Author(s):  
Elda Kara ◽  
Elisa Della Valle ◽  
Sara De Vincentis ◽  
Vincenzo Rochira ◽  
Bruno Madeo

Summary Spontaneous or fine-needle aspiration (FNAB)-induced remission of primary hyperparathyroidism (PHPT) may occur, especially for cystic lesions. However, the disease generally relapses over a short time period. We present a case of PHPT due to an enlarged hyperfunctioning parathyroid that underwent long-term (almost 9 years) clinical and ultrasonographic remission after the disappearance of the lesion following ultrasound (US)-assisted FNAB. A 67-year-old woman with PHPT underwent biochemical and US examinations that confirmed the diagnosis and showed a lesion suggestive for parathyroid adenoma or hyperplasia. US-FNAB of the lesion confirmed its parathyroid nature by means of elevated levels of parathyroid hormone within the needle washing fluid. At the second visit, the patient referred slight neck swelling that resolved spontaneously in the days after the US-FNAB. At subsequent follow-up, the enlarged parathyroid was not found; it was visible neither with US nor with magnetic resonance imaging. Biochemical remission persists after 9 years. This is the first reported case of cure of PHPT after US-FNAB performed on a hyperfunctioning parathyroid resulting in its complete disappearance over a period of 9 years of negative biochemical and ultrasonographic follow-up. Learning points: Spontaneous or fine-needle aspiration-induced remission of primary hyperparathyroidism can occur. Both circumstances may present disease relapse over a variable time period, but definite remission is also possible even though long-term periodic follow-up should be performed. Parathyroid damage should be ruled out in case of neck symptomatology after parathyroid fine-needle aspiration or spontaneous symptomatology in patients with history of primary hyperparathyroidism.


Author(s):  
Hiroaki Iwasaki

Summary A 45-year-old female was referred for endocrine evaluation of an incidental mass (31×24 mm in diameter) on the right adrenal gland. The patient was normotensive and nondiabetic, and had no history of generalised obesity (body weight, 46 kg at 20 years of age and 51.2 kg on admission); however, her waist-to-hip ratio was 0.97. Elevated urinary free cortisol levels (112–118 μg/day) and other findings indicated adrenocorticotrophic hormone-independent Cushing's syndrome due to right adrenocortical adenoma. Echocardiography before adrenalectomy revealed concentric left ventricular (LV) hypertrophy with a particular increase in interventricular septum thickness leading to impaired systolic and diastolic functions. Upon surgical remission of hypercortisolism, the asymmetric hypertrophy disappeared and the cardiac dysfunctions were considerably ameliorated. Although the mechanism(s) by which excessive cortisol contributes to LV wall thickness remain(s) unclear, serial echocardiography and cardiac multidetector-row computed tomography may support the notion that abnormal fat deposition in the myocardium owing to hypercortisolism appears to be an important factor for the reversible change in the cardiac morphology. Learning points Patients with Cushing's syndrome occasionally exhibit severe LV hypertrophy related to systolic and diastolic dysfunctions although they have neither hypertension nor diabetes mellitus. Biological remission of hypercortisolism can normalise structural and functional cardiac parameters and help in differentiating the cardiac alterations induced by excessive cortisol from those induced by other diseases. Excessive lipid accumulation within the heart before myocardial fibrosis may be implicated in reversible alterations in the cardiac morphology by Cushing's syndrome. Early diagnosis and treatment of Cushing's syndrome appear to be pivotal in preventing irreversible cardiac dysfunctions subsequent to cardiovascular events and heart failure.


Author(s):  
Yasuhiro Oda ◽  
Masayuki Yamanouchi ◽  
Hiroki Mizuno ◽  
Rikako Hiramatsu ◽  
Tatsuya Suwabe ◽  
...  

Summary We report the renal histology of a 66-year-old man with hypertension, cardiovascular disease, and a 30-year history of type 2 diabetes mellitus with proliferative diabetic retinopathy, diabetic neuropathy, and diabetic foot status post toe amputation. Urinary protein excretion was 1.4 g/gCr, serum creatinine level 0.86 mg/dL, estimated glomerular filtration rate 69 mL/min/1.73 m2, and HbA1c 13–15%, despite using insulin. Light microscopy showed global glomerulosclerosis in 37% of the glomeruli, but the remaining glomeruli were intact. Significant polar vasculosis was present, while arteriolar sclerosis was mild. Electron microscopy revealed a thickened glomerular basement membrane, which is compatible with the early stage of diabetic glomerulopathy. The presented case was unique because glomerular changes seen typically in diabetes were not seen in the patient, despite the long-standing history of diabetes and diabetic comorbidities, while prominent polar vasculosis was found. Polar vascular formation helps preserve the glomeruli by allowing hyperosmotic blood bypass the glomeruli; this decreases intraglomerular pressure and minimizes glomerular endothelial damage. Learning points: A 66-year-old man with a 30-year history of type 2 diabetes mellitus with poor glycemic control underwent renal biopsy, which showed scarce glomerular changes typically seen in diabetic kidney disease and instead revealed significant polar vasculosis. Past studies demonstrated that the increased small vessels around the vascular hilus in diabetic patients originated from the afferent arterioles and drained into the peritubular capillaries. Polar vascular formation may preserve glomerular function by allowing the blood flow to bypass the glomeruli and decreasing the intraglomerular pressure, which minimizes endothelial damage of the glomerular tufts.


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