Sarcoidosis and calcium homeostasis disturbances—Do we know where we stand?

Gwadera, Łukasz; Białas, Adam J.; Iwański, Mikołaj; Górski, Paweł; Piotrowski, Wojciech J.

doi:10.1177/1479973119878713

Cited by 24 publications

(18 citation statements)

References 94 publications

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“…A more frequent sign of dysregulated calcium homeostasis in sarcoidosis is hypercalciuria which affects 20–40% of patients. As a consequence, nephrolithiasis is more frequent in sarcoidosis than in the general population as it occurs in 10–14% of patients during the course of the disease [ 82 ]. It is important to remember that sarcoidosis does not exclude other causes of hypercalcemia, starting with the most important: hyperparathyroidism and neoplasia, particularly lymphoma which can be associated with increased calcitriol production in granulomas [ 83 ].…”

Section: Clinical Manifestationsmentioning

confidence: 99%

Sarcoidosis: A Clinical Overview from Symptoms to Diagnosis

Sève

Pachéco

Durupt

et al. 2021

Cells

225

144

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Sarcoidosis is a multi-system disease of unknown etiology characterized by the formation of granulomas in various organs. It affects people of all ethnic backgrounds and occurs at any time of life but is more frequent in African Americans and Scandinavians and in adults between 30 and 50 years of age. Sarcoidosis can affect any organ with a frequency varying according to ethnicity, sex and age. Intrathoracic involvement occurs in 90% of patients with symmetrical bilateral hilar adenopathy and/or diffuse lung micronodules, mainly along the lymphatic structures which are the most affected system. Among extrapulmonary manifestations, skin lesions, uveitis, liver or splenic involvement, peripheral and abdominal lymphadenopathy and peripheral arthritis are the most frequent with a prevalence of 25–50%. Finally, cardiac and neurological manifestations which can be the initial manifestation of sarcoidosis, as can be bilateral parotitis, nasosinusal or laryngeal signs, hypercalcemia and renal dysfunction, affect less than 10% of patients. The diagnosis is not standardized but is based on three major criteria: a compatible clinical and/or radiological presentation, the histological evidence of non-necrotizing granulomatous inflammation in one or more tissues and the exclusion of alternative causes of granulomatous disease. Certain clinical features are considered to be highly specific of the disease (e.g., Löfgren’s syndrome, lupus pernio, Heerfordt’s syndrome) and do not require histological confirmation. New diagnostic guidelines were recently published. Specific clinical criteria have been developed for the diagnosis of cardiac, neurological and ocular sarcoidosis. This article focuses on the clinical presentation and the common differentials that need to be considered when appropriate.

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Section: Clinical Manifestationsmentioning

confidence: 99%

Sarcoidosis: A Clinical Overview from Symptoms to Diagnosis

Sève

Pachéco

Durupt

et al. 2021

Cells

225

144

View full text Add to dashboard Cite

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“…Rates of hypercalcemia have varied among studies (2-63%), although patients are rarely symptomatic [1,2]. Typically, patients exhibit increased production of 1,25(OH)2D with low levels of 25-hydroxy-vitamin D and no increases in parathyroid hormone levels [2,7,8,9]. In our patient, 1,25(OH)2D levels were elevated at the time of diagnosis and were more correlated with disease reactivation than serum ACE levels.…”

Section: Discussionmentioning

confidence: 57%

“…As in the present case, abnormalities in liver and kidney function may also be observed, in which case ndings from liver or kidney biopsies can aid in histological diagnosis. Rates of hypercalcemia have varied among studies (2-63%), although patients are rarely symptomatic [1,2]. Typically, patients exhibit increased production of 1,25(OH)2D with low levels of 25-hydroxy-vitamin D and no increases in parathyroid hormone levels [2,7,8,9].…”

Section: Discussionmentioning

confidence: 99%

“…Although serum angiotensin converting enzyme (ACE) is among the most commonly used diagnostic biomarkers for sarcoidosis, the test is non-speci c and lacks sensitivity [1]. Hypercalciuria, hypercalcemia, and increased levels of 1,25-dihydroxy vitamin D (1,25(OH)2D) may also occur in patients with sarcoidosis due to over-production of 25-hydroxy vitamin D-1 α-hydroxylase [1,2].…”

Section: Introductionmentioning

confidence: 99%

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Biomarkers in Juvenile Systemic Sarcoidosis: A Case Report and Literature Review

Alsarhan

Habeeb

Popatia

et al. 2020

Preprint

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Background: Juvenile sarcoidosis represents a spectrum of rare granulomatous inflammatory conditions characterized by non-caseating granulomas that can affect multiple systems. Nonetheless, no laboratory markers can conclusively indicate a diagnosis of sarcoidosis. In this report, we discuss the role of serum soluble interleukin-2 receptor (sIL-2r) levels in supporting the diagnosis of sarcoidosis in a child with ocular, renal, and pulmonary involvement. We further discuss trends in this marker, as well as serum angiotensin converting enzyme (ACE) and 1,25(OH)2D levels, in relation to disease activity and response to treatment. Case Presentation: A previously healthy 14-year-old Arabic girl presented with fever, anorexia, and dyspnea upon exertion. She exhibited elevated levels of acute-phase reactants, anemia, elevated serum creatinine, and proteinuria. An eye exam revealed bilateral uveitis. High-resolution computed tomography (CT) of the chest revealed subpleural interlobar septal thickening with reticular changes and subtle ground glass opacities. The results of an extensive work-up to rule out infectious etiology, immune deficiency, malignancy, and other rheumatic diseases were unremarkable. Serum levels of sIL-2r were extraordinarily high at 18,589 U/L (normal range: 223–710 U/L), and the trend of subsequent measurements correlated with disease response and disease reactivation. Although ferritin levels were elevated at 1,136 ng/mL (normal: 13–68 ng/mL), bone marrow biopsy was negative for hemophagocytosis. Kidney biopsy revealed granulomatous interstitial nephritis with tubular atrophy. Serum ACE and 1,25(OH)2D levels were also elevated at diagnosis. The patient was treated with glucocorticosteroids, mycophenolate mofetil, and anti-tumor necrosis factor (anti-TNF). Clinical responses to such treatment were correlated with trends in serum sIL-2r and 1,25 (OH)2D levels, but not with trends in serum levels of ACE. Conclusions: Our findings highlight the promise of sIL-2r as a marker of diagnosis and disease activity during the treatment period in patients with juvenile sarcoidosis. Further prospective studies involving large cohorts of patients with childhood sarcoidosis are required to elucidate the diagnostic and prognostic roles of serum sIL-2r levels.

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“…Sarcoidosisban hypercalcaemiát először 1981-ben írtak le [12]. A hypercalcaemia hátterében a sarcoidosisos betegekben az aktív D-vitamin (kalcitriol) endogén túlprodukciója áll, melyet az aktivált makrofágok fokozott 1-alfa-hidroxiláz-aktivitása okoz [13].…”

Section: Esetismertetésunclassified

Hypercalcaemia és veseelégtelenség – merre induljunk?

Garam

Tislér

Pethő

et al. 2021

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Összefoglaló. Közleményünkben egy 63 éves férfi esetét ismertetjük, aki fáradékonyság, fogyás miatt végzett laboratóriumi vizsgálatokon igazolódó veseelégtelenség és hypercalcaemia miatt került felvételre Klinikánkra. A területen végzett röntgenvizsgálaton a koponyán frontalisan és a sacrumon csonteltérések (temporofrontalisan 13 mm-es, körülírtabb, mérsékelten intenzív árnyék és az S1-es rés sclerosisa) ábrázolódtak, ultrahangvizsgálat során lépmegnagyobbodás volt látható. Tünetei hátterében endokrin vagy malignus betegség nem igazolódott. A háttérben elsősorban myeloma multiplex merült fel, ugyanakkor azt célzott vizsgálatokkal sem megerősíteni, sem kizárni nem lehetett, így csontvelő-biopszia történt. A vesefunkció-romlás okának tisztázása végett vesebiopsziát végeztünk, melynek előzetes eredménye interstitialis nephritist véleményezett óriássejtekkel. Az angiotenzinkonvertáló enzim szérumszintjének ez okból történő vizsgálata emelkedett szintet mutatott, így esetünket Boeck-sarcoidosis extrapulmonalis manifesztációjának tartottuk. Per os szteroidkezelésre a beteg tünetei egyértelmű regressziót mutattak. A csontvelő- és vesebiopszia eredménye megerősítette a Boeck-sarcoidosis diagnózisát. A sarcoidosis ezen extrapulmonalis formája hypercalcaemiával és veseérintettséggel – de tüdőérintettség nélkül – rendkívül ritka, különös tekintettel a vesét érintő formára. Hypercalcaemia nagyjából 7,9%-ban, veseelégtelenség 1,4%-ban fordul elő. Ezen eset alapján fontos hangsúlyozni, hogy a hypercalcaemia és a veseelégtelenség hátterében a gyakoribb endokrin, malignus, hematológiai okok mellett a Boeck-sarcoidosisnak is fel kell merülnie a differenciáldiagnosztika során. Orv Hetil. 2021; 162(13): 514–518. Summary. We present the case of a 63-year-old male patient who was admitted to our Clinic with fatigue, weight loss, hypercalcemia, renal insufficiency and anemia. X-ray showed lesions on the frontal skull and sacral region. On abdominal ultrasound, splenomegaly was detected. Based on these, myeloma multiplex was the most likely initial diagnosis; this, however, could not be confirmed with targeted serum tests, therefore bone marrow biopsy was performed. To clarify the underlying cause of decreased kidney function, renal biopsy was performed, the preliminary results of which revealed interstitial nephritis accompanied by giant cells. Serum angiotensin converting enzyme level was elevated, which led to the diagnosis of Boeck sarcoidosis with extrapulmonary manifestations. Oral corticosteroid therapy was commenced that was followed by regression of the patient’s symptoms and laboratory abnormalities. Both the bone marrow and the kidney biopsies supported the diagnosis of Boeck sarcoidosis. Presentation of sarcoidosis with hypercalcemia and renal insufficiency but without the involvement of the lungs is extremely rare. Hypercalcemia occurs in about 7.9% and renal insufficiency in 1.4% of the cases. Based on this case, it is important to highlight that in the background of hypercalcemia and renal failure – beside the more frequent causes such as endocrine and hematological diseases, malignancy – one is to consider the possibility of Boeck sarcoidosis as well. Orv Hetil. 2021; 162(13): 514–518.

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Sarcoidosis and calcium homeostasis disturbances—Do we know where we stand?

Cited by 24 publications

References 94 publications

Sarcoidosis: A Clinical Overview from Symptoms to Diagnosis

Sarcoidosis: A Clinical Overview from Symptoms to Diagnosis

Biomarkers in Juvenile Systemic Sarcoidosis: A Case Report and Literature Review

Hypercalcaemia és veseelégtelenség – merre induljunk?

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