Hypercalcemia, hyperkalemia and supraventricular tachycardia in a patient with subcutaneous fat necrosis

Bahadur, Kandy; Johnson, Scott H.; Lentzner, Benjamin; Gangat, Mariam; Carlson, Joann; Balachandar, Sadana

doi:10.1515/jpem-2017-0365

Cited by 5 publications

(5 citation statements)

References 8 publications

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“…One hypothesis is, that this prolonged disease course is related to the tendency of larger skin lesions in newborns with SCFN and hypercalcaemia. It is assumed, that the size of skin lesion correlates with the extent of inflammation and the resulting immune response [ 10 , 32 , 33 ]. Since we observed in a subgroup of patients with reported levels of serum 1,25(OH)2D3 and parathyroid hormone (PTH) that the majority of these patients showed a suppressed PTH level, independent of serum 1,25(OH)2D3 levels, a PTH-independent but immune-stimulated hypercalcaemia is assumed.…”

Section: Discussionmentioning

confidence: 99%

“…A PTH-independent hypercalcaemia is assumed. Within literature an extrarenal 1,25(OH)2D3 formation as a trigger for the elevation of serum calcium levels in newborns with SCFN is discussed [ 10 , 32 , 52 ]. The formation of the active 1,25(OH)2D3 (calcitriol) from its biologically inactive form 25OHD3 (=calcidiol) occurs by hydroxylation by the enzyme 25-hydroxyvitamin D3-1α-hydroxylase (1α-hydroxylase).…”

Section: Discussionmentioning

confidence: 99%

“…This reaction step is stimulated by the increase in 1,25(OH)2D3, ensuring a constant 1,25(OH)2D3 level in plasma [ 56 ]. Bahadur et al [ 32 ] observed elevated 1,25(OH)2D3 levels in their patient with SCFN concomitant with low levels of the inactive degradation product 24,25(OH)2D. They concluded an impaired degradation by the enzyme 24α-hydroxylase, which is encoded by the gene CYP2A41 [ 62 ].…”

Section: Discussionmentioning

confidence: 99%

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Subcutaneous fat necrosis in newborns: a systematic literature review of case reports and model of pathophysiology

Frank

Brandt²,

Wabitsch³

2022

Mol Cell Pediatr

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Background Subcutaneous fat necrosis of the newborn (SCFN) is a rare disease occurring in the first days of life. Characteristically, the infants show hard nodules in subcutaneous tissue, purple or erythematous in color and appear on the upper back, cheeks, buttocks and limbs. In most cases, SCFN is a self-limiting disease, as the nodules disappear in up to 6 months. A severe complication associated with SCFN is hypercalcaemia. Pathophysiological mechanisms causing SCFN or associated hypercalcaemia are not fully understood yet. Methods A systematic literature research including the six biggest databases for medical research has been used to identify all published case reports of SCFN. N = 206 publications has been identified containing n = 320 case reports. All cases have been classified into four subgroups (depending on reported serum-calcium-level): hypercalcaemia, normocalcaemia, hypocalcaemia or no information given. Reported maternal factors, birth characteristics, details about SCFN, diagnostics, therapy and long-term observations have been extracted from publications. Results This is the first systematic literature research that summed up all published cases of SCFN from 1948 up to 2018. Information about serum calcium level was given in 64.3% of the cases. From those, the majority showed hypercalcaemia (70.5%) (normocalcaemia 25.1%, hypocalcemia 4.3%). 89.3% of newborns with hypercalcaemia showed suppressed levels of the parathormone. Maternal gestational diabetes, maternal hypertensive diseases during pregnancy, macrosomia (> 4000g), asphyxia and therapeutic hypothermia are risk factors for SCFN. Histological findings showed a granulomatous inflammation in 98% of cases. Conclusion We identified that maternal, birth characteristics and therapeutic measures are probably risk factors for SCFN. These risk factors should be taken into account within the care of neonates.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Subcutaneous fat necrosis in newborns: a systematic literature review of case reports and model of pathophysiology

Frank

Brandt²,

Wabitsch³

2022

Mol Cell Pediatr

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“…11 Hypertriglyceridemia may be related to the neonate's innate increased level of saturated fats that can crystalize in adipocytes at lower temperatures. 12 In 2018, Bahadur et al 13 also described a possible new sequela with SCFN: hyperkalemia. Their case report described an infant with a subcutaneous nodule who was also treated for SVT with ice therapy.…”

Section: Discussionmentioning

confidence: 99%

Cold Panniculitis and Subcutaneous Fat Necrosis of the Newborn: The Continual Sequelae in the Non-pharmacological Conversion of Supraventricular Tachycardia

Freeman

Moreaux

Beavers

et al. 2022

Clin Pediatr (Phila)

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“…Metabolic consequences like moderate to severe hypercalcemia [1], metastatic calcifications (disseminated sub cutaneous calcifications, nephrocalcinosis and myocardial calcifications) [6], hypocalcaemia [12], hypoglycaemia [13], hyperkalaemia [14] hypertriglyceridemia [1,10] have been reported to be associated with SCFN. Although the pathophysiology of hypercalcemia in SCFN remains unclear, a widely accepted theory proposes that the elevated 1, 25-Hydroxyvitamin D3 secreted from the granulomas of the skin lesions can stimulate intestinal calcium uptake [6] and decreased clearance of calcium by the kidneys [3] results in hypercalcemia.…”

Section: Discussionmentioning

confidence: 99%

Subcutaneous fat necrosis (SCFN) secondary to therapeutic hypothermia with persistent leucocytosis in a neonate

Govindarajan¹

2021

IJCCR

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We report a term male neonate, born to consanguineous parents through a difficult labour who needed resuscitation at birth, followed by ventilation for respiratory distress. In view of suspected Hypoxic Ischemic Injury, he was started on whole body cooling for 72 hours. He was treated as suspected sepsis with 5 days of intravenous antibiotics. On day 6, he was noted to have hard, painless skin nodules of his back and left arm which was clinically diagnosed as subcutaneous fat necrosis. His serum calcium and phosphate levels remained normal throughout hospital stay. His full blood counts and bone profile were monitored regularly. During follow-up at 6 weeks, his white cell counts continued to be high with 69% lymphocytes. To our knowledge, this is the first case of SCFN of a neonate associated with persistent leucocytosis. Haematological and metabolic complications should be closely monitored in a neonate with subcutaneous fat necrosis.

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Hypercalcemia, hyperkalemia and supraventricular tachycardia in a patient with subcutaneous fat necrosis

Abstract: Our case highlights the importance of remaining vigilant in the workup of atypical rashes of the infant, which should include early assessment of serum electrolytes.

Cited by 5 publications

References 8 publications

Subcutaneous fat necrosis in newborns: a systematic literature review of case reports and model of pathophysiology

Subcutaneous fat necrosis in newborns: a systematic literature review of case reports and model of pathophysiology

Cold Panniculitis and Subcutaneous Fat Necrosis of the Newborn: The Continual Sequelae in the Non-pharmacological Conversion of Supraventricular Tachycardia

Subcutaneous fat necrosis (SCFN) secondary to therapeutic hypothermia with persistent leucocytosis in a neonate

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