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Successful Reversal of Furosemide-Induced Secondary Hyperparathyroidism with Cinacalcet Tarak Srivastava, Shahryar Jafri, William E

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Successful Reversal of Tarak Srivastava, MD, a Shahryar Jafri, a William E. Truog, MD, b Judith Sebestyen Furosemide-InducedVanSickle, MD, a Winston M. Manimtim, MD, b Uri S. Alon, MDa Secondary Hyperparathyroidism With Cinacalcet abstract Secondary hyperparathyroidism (SHPT) is a rare complication of furosemide therapy that can occur in patients treated with the loop diuretic for a long period of time.‍ We report a 6-month-old 28-weeks premature infant treated chronically with furosemide for his bronchopulmonary dysplasia, who developed hypocalcemia and severe SHPT, adversely affecting his bones.‍ Discontinuation of the loop diuretic and the addition of supplemental calcium and calcitriol only partially reversed the SHPT, aSections of Nephrology, Bone and Mineral Disorder Clinic, bringing serum parathyroid hormone level down from 553 to 238 pg/mL.‍ and bNeonatology, The Children’s Mercy Hospitals and Clinics, University of Missouri at Kansas City, Kansas City, After introduction of the calcimimetic Cinacalcet, we observed a sustained Missouri normalization of parathyroid hormone concentration at 27 to 63 pg/mL and, with that correction, of all biochemical abnormalities and healing of the Drs Srivastava and Alon conceptualized and designed the study and drafted the initial bone disease.‍ No adverse effects were noted.‍ We conclude that in cases of manuscript; Drs Truog, Manimtim, Sebestyen SHPT due to furosemide in which traditional treatment fails, there may be VanSickle, and Mr Jafri carried out the initial room to consider the addition of a calcimimetic agent.‍ analyses and reviewed and revised the manuscript; and all authors approved the final manuscript as submitted. DOI: https:// doi. org/ 10. 1542/ peds. 2016- 3789 Furosemide therapy in the endocrinopathy may persist for a Accepted for publication Mar 10, 2017 neonatal/infantile period is well prolonged period.‍ Address correspondence to Uri S. Alon, MD, Section known to be associated with of Nephrology, The Children’s Mercy Hospital, 2401 medullary nephrocalcinosis from In recent years, a calcimimetic agent, Gillham Rd, Kansas City, MO 64108. E-mail: ualon@ cmh.edu hypercalciuria1, 2caused by the Cinacalcet, which acts on the calcium- PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, loop diuretic.‍ A less known sensing receptor 9has become available complication of chronic treatment to suppress PTH.‍ The calcimimetic 1098-4275). with furosemide– is the development agent changes the configuration of Copyright © 2017 by the American Academy of Pediatrics of secondary3 6 hyperparathyroidism the calcium-sensing receptor in the (SHPT).‍ The latter is attributed parathyroid glands, making them more FINANCIAL DISCLOSURE: The authors have to the hypercalciuria, leading to the sensitive to circulating serum calcium, indicated they have no financial relationships development of hypocalcemia, which resulting in decreased secretion of relevant to this article to disclose. stimulates parathyroid hormone (PTH) PTH.‍ Although originally created FUNDING: Supported by the Sam and Helen Kaplan 6, 7 Research Fund in Pediatric Nephrology and Eric secretion, but recent literature for use in adults with SHPT, there McClure Research Fund in Pediatric Bone and also suggests an additional, direct have been now several reports of Mineral Disorders. stimulating effect of furosemide on the successful use of this agent in children 8 – POTENTIAL CONFLICT OF INTEREST: The authors parathyroid glands.‍ It is expected that with SHPT from various10 13 genetic and have indicated they have no potential conflicts of discontinuation of the loop diuretic, at acquired etiologies.‍ We report interest to disclose. times combined with supplementation our novel experience in a premature with calcium and active vitamin infant with severe bronchopulmonary To cite: Srivastava T, Jafri S, Truog WE, et al. D metabolites, will reverse the dysplasia (BPD) necessitating long- Suc cessful Reversal of Furosemide-Induced Sec- SHPT.‍ However, if the parathyroid term treatment with furosemide who ondary Hyperparathyroidism With Cinacalcet. Pedi­ glands achieve a significant level of developed SHPT.‍ After observing only atrics. 2017;140(6):e20163789 hyperplasia, the task of controlling partial response to traditional therapy SHPT will be difficult, and the with calcium and calcitriol, the SHPT Downloaded from www.aappublications.org/news by guest on September 28, 2021 PEDIATRICS Volume 140, number 6, December 2017:e20163789 CASE REPORT Srivastava et al 2017 ROUGH GALLEY PROOF Successful Reversal of Furosemide-Induced https://doi.‍org/10.‍1542/peds.‍2016-3789 December 2017 Secondary Hyperparathyroidism With Cinacalcet 6 140 Pediatrics VDR tapered and discontinued next, and was successfully reversed with CASRwith inheritedPHEX CYP27B1 forms ofCYP2R1 rickets – Cinacalcet.‍ DMP1and/orCLCN7 osteopetrosisSOST LRP4 (namelySNX10 , finally Cinacalcet was stopped (Table 1, , , , , weeks 11.‍5 16.‍5).‍ The child was CASE REPORT TMFSF11 TNFRSF11A OSTM1 , , , , , then able to maintain serum calcium , , and ) and PTH levels in their normal ’ were negative.‍ A microarray did not ranges until discharge 8 weeks later.‍ The child was born at 28 weeks identify any deletion or duplication.‍ At follow-up a month later, serum gestation with a birth weight of Calcitriol was added to address calcium and PTH were normal at 830 g.‍ He was transferred from his his hypocalcemia.‍ Over the next 3 10.‍2 mg/dL and 78 pg/mL, respectively.‍ weeks, his serum PTH decreased but The child tolerated therapy with birth hospital at 2 months of age − for ongoing needs of prematurity, remained elevated at 257 pg/mL Cinacalcet without any adverse severe BPD, and possible need for (Table 1, week 3.‍4).‍ At this point, effects.‍ Throughout this course, tracheostomy.‍ On admission to our our team was consulted, and the his serum creatinine and urine institution, furosemide was added diagnosis of furosemide-induced calcium/creatinine ratio remained to his ongoing thiazide therapy SHPT was made.‍ Furosemide was normal.‍ The renal ultrasound to treat his respiratory condition.‍ discontinued and the chlorothiazide on last follow-up showed no At age 5.‍5 months, he underwent dose doubled.‍ In coming weeks, a nephrocalcinosis.‍ Serum ALP, which tracheostomy and placement of further decrease in PTH levels was was originally elevated, gradually ventriculoperitoneal shunt for noted, but the dose of calcitriol and normalized.‍ Follow-up radiographs hydrocephalus.‍ MRI of the brain calcium supplementation had to be done at 1 year of age showed normal performed for evaluation of the decreased to avoid hypercalcemia.‍ mineralization with resolution of ventriculoperitoneal shunt showed Nevertheless, despite sustained metaphyseal cupping and fraying severe demineralization of the normalization of serum calcium over (Fig 1).‍ this 6-week period, his serum PTH bones (Fig 1).‍ The skeletal survey DISCUSSION conducted the next day showed remained elevated (Table 1, week 0).‍ abnormal bony demineralization After discussion with the family, a concerning for a metabolic bone decision was made to initiate oral disorder.‍ That day serum ionized Cinacalcet 3 mg/day (0.‍4 mg/kg/day) Furosemide is a potent loop diuretic – calcium (iCa) was 0.‍99 mmol/L in 2 divided doses.‍ Over the next with action in the loop of Henle that (normal 1.‍13 1.‍37), and he was 2 weeks (Table 1: Cinacalcet Course A), results in decreased sodium and started on calcium supplementation.‍ PTH normalized at 15 to 25 pg/mL.‍ water reabsorption; it is often used − 14 Further detailed evaluation in the Due to hypocalcemia, calcium in infants with BPD.‍ However, coming days (Table 1, week 7.‍0), supplementation was increased.‍ its action also results in decreased when his chronological age was 6 With serum PTH and calcium levels calcium reabsorption because months and weight 6.‍3 kg, detected within their normal range, Cinacalcet calcium and sodium15 reabsorption are decreased serum calcium (8.‍9 mg/dL); was subsequently discontinued coupled at this site.‍ The increased iCa 1.‍09 mmol/L; elevated PTH (Table 1, week 2.‍0).‍ Within the urinary calcium excretion may (553 pg/mL), alkaline phosphatase next 2 weeks, the serum PTH result in formation of medullary1, 2 (ALP; 420 U/L), and 1,25(OH) 2- rebounded to 151 to 234 pg/mL.‍ nephrocalcinosis or stones.‍ In vitamin D (689 pg/mL); and normal This suggested significant older patients, furosemide has also phosphorus (5.‍4 mg/dL), 25(OH)- hyperplasia of the parathyroid glands been reported to be associated with vitamin D (65 ng/mL), and urine requiring sustained pharmacological decreased bone– mineral density, calcium/creatinine ratio (0.‍22 mg/mg).‍ suppression of PTH.‍ An attempt osteoporosis,16 18 and high risk for At that time, his treatment of BPD to image the parathyroid glands fractures.‍ In addition, there have included furosemide (3 mg/kg/day), by using neck ultrasound failed been reports– about the association of chlorothiazide (22.‍5 mg/kg/day), because of neck size and presence the loop 3diuretic8 with5 development and prednisone (0.‍5 mg/kg every of tracheostomy.‍ At this point, of SHPT.‍ Coe et al in 1973 48 hours).‍ Renal ultrasound showed Cinacalcet was reinitiated (Table 1: demonstrated that adults treated no medullary nephrocalcinosis.‍ Cinacalcet Course B) with with furosemide had elevated PTH His nutritional formula provided concomitant calcitriol and calcium similar to adults with persistent 6 elemental calcium, phosphorus, supplementation.‍ Serum PTH
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