Treatment with bisphosphonates in severe hypercalcemia due to subcutaneous fat necrosis in an infant with hypoxic-ischemic encephalopathy

Abstract Background Subcutaneous fat necrosis (SCFN) can be complicated by severe hypercalcemia, which is frequently asymptomatic. Nephrocalcinosis is associated with hypercalcemia and, in other clinical settings, has been linked to furosemide and glucocorticoid use. First-line bisphosphonate therapy treating hypercalcemia in neonatal SCFN is not well described. Objectives To describe the biochemical changes and risk of nephrocalcinosis in infants with hypercalcemia, secondary to neonatal SCFN, treated with initial pamidronate. Methods A retrospective chart review of five infants treated with initial pamidronate and without furosemide or glucocorticoids. Data were collected on the following: timing of presentation, therapeutic response, and presence of nephrocalcinosis. Results Hypercalcemia resolved after 2.8±1.7 days; this is compared to 7.6±2.8 days from previously reported cases utilising alternative therapies (P=0.012). There were no episodes of rebound hypercalcemia or hypocalcemia. Nephrocalcinosis was present in four of five cases. When including published cases, age at diagnosis was associated with presenting serum calcium (P=0.003) and nephrocalcinosis was associated with higher serum calcium (P=0.014) and time from SCFN to hypercalcemia diagnosis (P=0.002). Conclusions This retrospective case series demonstrates that first-line pamidronate treatment was effective and safe in the resolution of hypercalcemia. Nephrocalcinosis was observed, despite the avoidance of furosemide and glucocorticoid therapy, and associated with greater disease severity and duration of hypercalcemia.

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A Case Study of Severe Hypercalcemia Secondary to Subcutaneous Fat Necrosis: A Diagnosis of Exclusion

Neonatal Network The Journal of Neonatal Nursing ◽

10.1891/0730-0832.38.4.236 ◽

2019 ◽

Vol 38 (4) ◽

pp. 236-241 ◽

Cited By ~ 1

Author(s):

Mary Whalen

Keyword(s):

Tissue Injury ◽

Rare Complication ◽

Subcutaneous Fat ◽

Ischemic Injury ◽

The Body ◽

Whole Body ◽

Fat Necrosis ◽

Subcutaneous Fat Necrosis ◽

Severe Hypercalcemia

Subcutaneous fat necrosis (SCFN) is a rare complication, usually occurring in otherwise healthy full-term infants who have experienced some level of trauma that causes ischemic injury to adipose tissue. Tissue injury usually occurs in areas of the body that are exposed to excessive pressure as during delivery. Tissue injury has also been described secondary to therapeutic cooling. This case study presents an infant who received whole body cooling for hypoxic ischemic injury and later developed severe hypercalcemia at one month of age without the skin lesions consistent with SCFN. The differential diagnosis for hypercalcemia and how it relates to SCFN is presented, as well as clinical presentation, treatment, and prognosis.

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Use of Zoledronic Acid in a Neonate with Subcutaneous Fat Necrosis Complicated with Severe, Refractory Hypercalcemia

American Journal of Perinatology ◽

10.1055/s-0039-1691777 ◽

2019 ◽

Vol 36 (S 02) ◽

pp. S134-S138

Author(s):

Maria Angela Militello ◽

Maria Paola Re ◽

Giuliana Vitaliti ◽

Francesca Finazzo ◽

Paolo Manzoni ◽

...

Keyword(s):

Zoledronic Acid ◽

Low Dose ◽

Standard Treatment ◽

Subcutaneous Fat ◽

Rare Condition ◽

Inflammatory Disorder ◽

Fat Necrosis ◽

Subcutaneous Fat Necrosis ◽

Severe Hypercalcemia ◽

Healthy Infants

Objective Subcutaneous fat necrosis (SCFN) is a rare condition that may occur in the neonatal period. SCFN is an inflammatory disorder of the adipose tissue, usually found in full-term healthy infants who have a history of intrauterine or perinatal distress. It is usually a self-limited condition; however, in some cases, it can get complicated, leading to severe hypercalcemia that may be life-threatening. Study Design We report and describe a classic presentation of SCFN that led to severe hypercalcemia refractory to standard treatment. The diagnosis of SCFN was made based on the finding of subcutaneous nodules and of hypercalcemia. The serum calcium level reached 16.6 mg/dL. Hypercalcemia was treated first with intravenous infusions of fluids and furosemide and then of methylprednisolone. This standard treatment was not effective; therefore, we administered a single low dose of zoledronic acid, which, in turn, was efficacious in ultimately managing the hypercalcemia. Conclusion Our case shows how a single low dose of zoledronic acid was safe and effective in managing severe hypercalcemia unresponsive to conventional treatment while minimizing the risk of hypocalcemic rebounds.

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Subcutaneous Fat Necrosis and Hypercalcemia with Nephrocalcinosis in Infancy: Case Report and Review of the Literature

Children ◽

10.3390/children8050374 ◽

2021 ◽

Vol 8 (5) ◽

pp. 374

Author(s):

Katerina Chrysaidou ◽

Georgios Sargiotis ◽

Vasiliki Karava ◽

Dimitrios Liasis ◽

Victor Gourvas ◽

...

Keyword(s):

Therapeutic Hypothermia ◽

Late Onset ◽

Subcutaneous Fat ◽

Birth Asphyxia ◽

Fat Necrosis ◽

Subcutaneous Fat Necrosis ◽

Severe Hypercalcemia ◽

Life Threatening ◽

Term Newborns

Subcutaneous fat necrosis is an uncommon benign panniculitis affecting more commonly full-term newborns. It has been associated with birth asphyxia and meconium aspiration, as well as therapeutic hypothermia. Although the prognosis is generally favorable, complications such as hypercalcemia, thrombocytopenia, hypoglycemia and hypertriglyceridemia may complicate its course. The most serious complication is hypercalcemia that may reach life threatening levels and can be associated with nephrocalcinosis. We thereby describe a case of subcutaneous fat necrosis after therapeutic hypothermia, which presented with late-onset refractory severe hypercalcemia and persistent nephrocalcinosis during the follow up of the patient. Due to the risk of the development of chronic kidney disease, we highlight the importance of careful monitoring of hypercalcemia and review the literature of subcutaneous fat necrosis related to nephrocalcinosis.

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Pamidronate—An Effective Treatment for Severe Hypercalcemia due to Subcutaneous Fat Necrosis

Canadian Journal of Diabetes ◽

10.1016/j.jcjd.2016.08.089 ◽

2016 ◽

Vol 40 (5) ◽

pp. S31

Author(s):

Danielle Adam ◽

Laura Swaney ◽

Marianne Mckenna ◽

Jennifer O'dea ◽

Susan Sanderson ◽

...

Keyword(s):

Effective Treatment ◽

Subcutaneous Fat ◽

Fat Necrosis ◽

Subcutaneous Fat Necrosis ◽

Severe Hypercalcemia

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Neonatale subcutane vetnecrose met opvolging van de hypercalciëmie

Tijdschrift voor Geneeskunde ◽

10.47671/tvg.77.20.199 ◽

2021 ◽

Author(s):

L. VERBEKE ◽

K. KAMOEN ◽

L. AEYELS ◽

J. VERAMME ◽

N. DE CONO

Keyword(s):

Skin Biopsy ◽

Therapeutic Hypothermia ◽

Subcutaneous Fat ◽

Fat Necrosis ◽

Skin Damage ◽

Subcutaneous Fat Necrosis ◽

Life Threatening ◽

The Right ◽

Ischemic Encephalopathy

Subcutaneous fat necrosis of the newborn with follow-up of the hypercalcemia A thirteen-day-old boy is diagnosed with subcutaneous fat necrosis of the newborn after the appearance of erythematous, subcutaneous plaques on the right shoulder. This rare, usually self-limiting cutaneous disease is mainly diagnosed in term and postterm infants. The diagnosis is confirmed by a skin biopsy. Various etiological hypotheses are suggested, although the exact pathogenesis needs to be elucidated. The disease might be associated with several maternal and foetal predisposing factors, including therapeutic hypothermia at birth as part of hypoxic ischemic encephalopathy. It is a benign inflammation of the subcutaneous adipocytes that can be accompanied by life-threatening complications, such as hypercalcemia and thrombocytopenia. The latter requires rigorous follow-up and, in some cases, also treatment. The skin damage should only be treated symptomatically with analgesia.

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Subcutaneous Fat Necrosis of the Newborn With Initial Hypocalcemia and Familial Recurrence: A Case Report

Journal of Cutaneous Medicine and Surgery ◽

10.1177/1203475417733459 ◽

2017 ◽

Vol 22 (2) ◽

pp. 223-225 ◽

Cited By ~ 1

Author(s):

Amelia Kellar ◽

Jennifer Aileen Tangtatco ◽

Miriam Weinstein ◽

Natasha Saunders

Keyword(s):

Case Report ◽

Perinatal Asphyxia ◽

Subcutaneous Fat ◽

Fat Necrosis ◽

Extensive Disease ◽

Subcutaneous Fat Necrosis ◽

Severe Hypercalcemia ◽

Familial Association ◽

History Of ◽

Posterior Trunk

Subcutaneous fat necrosis of the newborn (SFNN) is a rare disorder characterised by indurated plaques or nodules with or without erythema, typically distributed over the cheeks, extremities, posterior trunk, buttocks, and thighs, and appears during the first 2 weeks of life. It is commonly associated with perinatal asphyxia and, while usually self-limited, can lead to severe hypercalcemia. There is no known familial association. The present article describes a neonate with extensive disease, counterintuitive hypocalcemia, and a family history of SFNN and reviews the existing literature on SFNN.

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