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CASE REPORT
Year : 2018  |  Volume : 19  |  Issue : 2  |  Page : 146-147

Neonatal hypercalcemia secondary to subcutaneous fat necrosis presenting as severe dehydration


Department of Dermatology, Venerology and Leprology, Rajendra Institute of Medical Sciences, Ranchi, Jharkhand, India

Date of Web Publication26-Mar-2018

Correspondence Address:
Ravish Singhal
Flat No. 101, Triambak Apartments, 7-Baxi Compound, Bariatu, Ranchi - 834 009, Jharkhand
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.IJPD_5_17

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  Abstract 


Subcutaneous fat necrosis (SCFN) is a rare fat tissue inflammation of the newborn that generally occurs in term or postterm newborns exposed to perinatal stressing factors such as cord prolapse, perinatal asphyxia, therapeutic hypothermia, meconium aspiration, and sepsis. Although most cases of SCFN resolve spontaneously, SCFN may be associated with symptomatic hypercalcemia presenting as lethargy, hypotonia, irritability, vomiting, polyuria, polydipsia, constipation, and dehydration. We describe a case of newborn affected with the disease, its presentation, clinical course, and treatment.

Keywords: Hypercalcemia, newborn, perinatal asphyxia, subcutaneous fat necrosis


How to cite this article:
Garg A, Singhal R, Chaudhary SS. Neonatal hypercalcemia secondary to subcutaneous fat necrosis presenting as severe dehydration. Indian J Paediatr Dermatol 2018;19:146-7

How to cite this URL:
Garg A, Singhal R, Chaudhary SS. Neonatal hypercalcemia secondary to subcutaneous fat necrosis presenting as severe dehydration. Indian J Paediatr Dermatol [serial online] 2018 [cited 2020 Nov 28];19:146-7. Available from: https://www.ijpd.in/text.asp?2018/19/2/146/211819




  Introduction Top


Subcutaneous fat necrosis (SCFN) is a noninfectious panniculitis that occurs in term or postterm infants who experience significant distress in the first week of life,[1] including cord prolapse, perinatal asphyxia, therapeutic hypothermia, meconium aspiration, and sepsis. It is characterized by hard and painful nodules, purple and erythematous, typically located on the shoulders, buttocks, back, faces, and distal extensors of arms and legs. It can occur from birth to the first 6 weeks of life.[2] The importance of this condition lies in the associated complications, mainly abscess formation and hypercalcemia. Significant hypercalcemia is rare, but it can have severe consequences, and it is potentially lethal.


  Case Report Top


A full term 3.4 kg female infant, born by emergency cesarean section due to fetal distress, prolonged rupture of membrane and nonprogress of labor to a 28-year-old primigravida mother whose pregnancy was complicated with gestational diabetes controlled by diet. Resuscitation in the delivery room included oro-nasal suctioning followed by positive pressure ventilation for 2 min. Her Apgar scores were 2, 6, and 8 at 1, 5, and 10 min, respectively. The baby was referred to the Department of Pediatrics and Neonatology for postresuscitation care. On admission, venous blood gas showed pH 7.15 and base excess (BE) was −9.8. The baby had grunting and chest retractions hence started on continuous positive airway pressure. Sepsis screen was sent and intravenous antibiotics were started in v/o probable sepsis. By day 7 of life, while the baby was recovering, a large patch of indurated, erythematous-purplish skin lesion appeared on his back and buttock [Figure 1]. Dermatology consultation suggested that SCFN and biopsy of the lesion showed organized fat necrosis. Blood investigations showed ionic calcium to be 1.694 mmol/L (normal: 1.120–1.320 mmol/L) which progressed to 2.240 mmol/L on day 10 of life. Treated with iv fluids and injection furosemide. Normalization of ionic calcium levels was achieved in 3 days. The baby was discharged after the completion of antibiotics and called for follow-up after 7 days. Baby reported on the 24th day of life with the complaint of new erythematous hard nodular lesions around the left knee joint [Figure 2]. There was a history of frequent urination and signs of severe dehydration and weight loss (current weight 2.9 kg). Sepsis screen was negative, ionic calcium was 3.350 mmol/dl. The baby was treated with iv fluids, injection furosemide and oral prednisolone were started in dose of 1 mg/kg/day. Serum triglyceride, renal ultrasound found to be normal, and serum parathormone level was decreased. Ionic calcium normalized in 4 days and baby regained weight and was discharged. The oral steroid was continued on the same dose for 3 months and serial monitoring on follow-up revealed normal calcium levels.
Figure 1: Erythematous-purplish nodular lesion on buttocks and thigh

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Figure 2: Erythematous indurated plaque on the left knee

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  Discussion Top


SCFN is a noninfectious inflammatory disorder of adipocytes presenting as erythematous, indurated, painful, subcutaneous nodules usually located over the cheeks, back, shoulders, and buttocks. Although the cause is unknown, generally, there is a history of intrauterine or perinatal distress in almost all cases. Predisposing conditions include ischemic injury, perinatal hypoxia, hypothermia, meconium aspiration, Rh incompatibility, obstetric trauma, sepsis, gestational diabetes, smoking or exposure to passive smoking during pregnancy, maternal medications (calcium channel blockers, and cocaine), and preeclampsia.[3],[4] The subcutaneous fat in infants has a high concentration of saturated fatty acids (stearic and palmitic acids) with a high melting point. In addition, enzyme systems involved in fatty acid metabolism are not fully developed.[5] Different stressors may exacerbate this enzyme defect and result in increased levels of saturated fatty acids in fat deposits. At the same time, high melting point of the saturated fatty acids may cause crystallization of fat, result in adipocyte necrosis triggering a granulomatous reaction.[6]

SCFN is mostly a self-limited process that resolves within weeks or months. However, it is known to be associated with frequent metabolic complications as hypoglycemia, hypercalcemia, hypertriglyceridemia, or with hematological complications as anemia and thrombocytopenia. The differential diagnoses include sclerema neonatorum, erythema nodosum, and bacterial cellulitis. Hypercalcemia usually develops when skin lesions begin to resolve. However, skin lesions usually resolve over a period of weeks to several months, whereas hypercalcemia can persist longer and require regular monitoring. The exact cause of hypercalcemia in SCFN is unknown. Possible explanations include high levels of 1,25-dihydroxyvitamin D3 secreted by macrophages resulting in increased calcium absorption, increased prostaglandin activity leading to osteoclast activation, and direct calcium release from necrotic fat cells.[7] Neonates with hypercalcemia tend to present with lethargy, hypotonia, irritability, vomiting, polyuria, polydipsia, constipation, and dehydration. If left untreated, moderate to severe hypercalcemia may lead to complications as nephrocalcinosis, nephrolithiasis, and renal failure; calcification of falx cerebri, skin, myocardium, and gastric mucosa may also occur. The first-line of treatment for hypercalcemia in infants with SCFN is hyperhydration, calcium wasting diuretics, and dietary Vitamin D and calcium restriction. In the case of dangerously high hypercalcemia, alternative choices are corticosteroid therapy and pamidronate. Regular monitoring of serum calcium is recommended until the age of 6 months, in infants with SCFN.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Calisici E, Oncel MY, Degirmencioglu H, Sandal G, Canpolat FE, Erdeve O, et al. Aneonate with subcutaneous fat necrosis after passive cooling: Does polycythemia have an effect? Case Rep Pediatr 2013;2013:254089.  Back to cited text no. 1
[PUBMED]    
2.
Paige DG, Gennery AR, Cant AJ. The neonate. In: Burns T, Breathnach S, Cox N, Griffiths C, editors. Rook's Textbook of Dermatology. 8th ed. Massachusetts: Blackwell Publishing; 2010. p. 590-2.  Back to cited text no. 2
    
3.
Hicks MJ, Levy ML, Alexander J, Flaitz CM. Subcutaneous fat necrosis of the newborn and hypercalcemia: Case report and review of the literature. Pediatr Dermatol 1993;10:271-6.  Back to cited text no. 3
[PUBMED]    
4.
Tran JT, Sheth AP. Complications of subcutaneous fat necrosis of the newborn: A case report and review of the literature. Pediatr Dermatol 2003;20:257-61.  Back to cited text no. 4
[PUBMED]    
5.
Caple JI, Reyes S. Subcutaneous fat necrosis of the newborn: A case presentation. J Perinatol 1996;16(2 Pt 1):140-1.  Back to cited text no. 5
    
6.
Sivanandan S, Rabi Y, Kamaluddeen M, Akierman A, Lodha A. Subcutaneous fat necrosis as a complication of therapeutic hypothermia in a term neonate. Indian J Pediatr 2012;79:664-6.  Back to cited text no. 6
[PUBMED]    
7.
Bonnemains L, Rouleau S, Sing G, Bouderlique C, Coutant R. Severe neonatal hypercalcemia caused by subcutaneous fat necrosis without any apparent cutaneous lesion. Eur J Pediatr 2008;167:1459-61.  Back to cited text no. 7
[PUBMED]    


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