Mizoribine for renal sarcoidosis: effective steroid tapering and prevention of recurrence

Ito, Shuichi; Harada, Toshiro; Nakamura, Tomoko; Imagawa, Tomoyuki; Nagahama, Kiyotaka; Sasaki, Takeshi; Fujinaga, Shuichiro; Aihara, Yukoh; Yokota, Shumpei

doi:10.1007/s00467-008-0995-7

Cited by 10 publications

(4 citation statements)

References 18 publications

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“…Long term use of corticosteroids, especially in adolescents, can cause substantial side effects including diabetes, growth retardation and cataract. Alternative agents that have been attempted in treating sarcoid GIN include mycophenolate [44] and mizoribine [45,46], which are limited to case reports and have been primarily used in pediatric patients to break steroid dependence or ameliorate significant side effects. There has been great interest in the use of TNF-antagonists as another modality to treat sarcoid GIN in order to avoid use of steroids.…”

Section: Prognosis Therapy and Follow-upmentioning

confidence: 99%

Acute Renal Failure in Sarcoidosis: A Review of the Literature

Papalia¹

2016

Arch Clin Nephrol

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We report two cases of acute kidney failure (ARF) as the initial manifestation of sarcoidosis. Two patients were sent from their primary care physicians with hypercalcemia and progressive increase of serum creatinine. The renal biopsy revealed granulomatous interstitial nephritis (GIN). Both patients were started on Methylprednisolone pulse therapy, followed by administration of oral prednisolone and by a slow taper thereafter to a maintenance dose. The renal function has improved immediately in response to this therapy. Our cases demostrates thet GIN due to sarcoidosis, although a rare entity, can cause severe ARF and progressive ESRD. With early detection and appropriate therapy, the majority of patients will maintain adequate renal function. Therefore the diagnosis of renal sarcoidosis must be done quickly to prevent renal. uptake on Computed tomography (CT) scan of this chest, elevated serum ACE level, hypercalcemia (12.3 mg/dL), splenomegaly, retinal degeneration, nonischemic cardiomyophathy with hypertrophy on echocardiography, no pathognomonic skin lesions. Serologic testing showed normal complement levels and negative results for anti-nuclear antibody, double-stranded DNA, antineutrophil cytoplasmic antibody (ANCA). The presence of hypercalcemia and very low PTHi might be mistaken for Multiple Myeloma. Monoclonal Gammopathies were excluded by normal serum immunofixation, absent Bence Jones proteinuria and absent κ and λ free light chains (FLC). A skeletal survey showed no lytic or blastic osseous lesions. Active tubercolosis, fungal infection, viral hepatitis, vasculitis and malignancy were clinically excluded. The patient started with Methylprednisolone pulse therapy, followed by administration of oral prednisolone (1.0 mg/Kg/die) for 12 weeks and by a slow taper thereafter to a maintenance dose of 10 mg for an additional six months period. The renal function and hypercalcemia improved immediately in response to this therapy after one month. The follow up was marked by complete resolution of clinical and biological signs. Case No 2 We report the case of a 53-year-old Caucasian woman presenting acute renal failure (ARF), proteinuria of 1.0 gr/24H, hypercalcemia and elevated 1.25 dihydroxycholecalcipherol. She had reported the onset of fatigue, weight loss (about 12 Kg), artralgia, loss of appetite a few months before hospital admission. The serum creatinine level has been documented to be 1.0 mg/dL two months before hospitalization. Her medical history included "rheumatica peliosis" when she was 22 years old, right pleurisy and erythematous maculopapular rash on her face when she was 24 years old, onset of Diabetes when she was 46 years old. On admission on physical exam, the patient was febrile, normotensive and preserved diuresis (1800 mL/24H). Upon admission, laboratory results showed creatinine of 4.7 mg/ Case No 1 A 57-year-old Caucasian man is hospitalized in our Nephrology Department with progressive increase in serum creatinine (2.48 mg/ dl; MDRD 32.6 ml/min) and proteinuria less than 500 mg/24h. ...

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Section: Prognosis Therapy and Follow-upmentioning

confidence: 99%

Acute Renal Failure in Sarcoidosis: A Review of the Literature

Papalia¹

2016

Arch Clin Nephrol

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“…After multiple debates, the question that nephrocalcinosis and/or GIN might have a better prognosis remains uncertain. In Table S1 , we have summarized all the studies of sarcoidosis with renal involvement in children that were the basis of this review [ 53 , 54 , 55 , 56 , 57 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 65 , 66 ].…”

Section: Discussionmentioning

confidence: 99%

The Overlap of Kidney Failure in Extrapulmonary Sarcoidosis in Children—Case Report and Review of Literature

Mocanu

Bogos

Trandafir

et al. 2023

IJMS

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Sarcoidosis is a non-necrotizing granulomatous inflammatory multisystemic disorder of unknown etiology. In children, as in adults, it can involve a few or all organ systems to a varying extent and degree, entailing multisystemic manifestations. Kidney involvement in pediatric-onset adult-type sarcoidosis is rare, with a wide range of renal manifestations, most of them related to calcium metabolism. Children with renal sarcoidosis tend to be more symptomatic than adults, although male patients have a higher prevalence. We present the case of a 10-year-old boy who presented with advanced renal failure with nephrocalcinosis and important hepatosplenomegaly. The diagnosis was established by histopathological examination, with consequent cortisone therapy and hemodialysis. This review emphasizes that sarcoidosis should be considered in the differential diagnosis of pediatric patients with acute kidney insufficiency or chronic kidney disease of an unknown etiology. As far as we know, this is the first study regarding extrapulmonary sarcoidosis in children from Romania.

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“…Long term use of corticosteroids, especially in adolescents, can cause substantial side effects including diabetes, growth retardation and cataract. Alternative agents that have been attempted in treating sarcoid GIN include mycophenolate (Moudgil 2002) and mizoribine (Rajakariar et al 2006 andIto et al 2009) which are limited to case reports and have been primarily used in pediatric patients to break steroid dependence or ameliorate significant side effects. Other agents which have been tried in systemic extra-renal sarcoidosis include mycophenolate mofetil, methotrexate, azathioprine, antimalarials, and phosphodiesterase inhibitors such as pentoxifylline and thalidomide although no data on treating renal sarcoidosis exists.…”

Section: Treatmentmentioning

confidence: 99%