Severe neonatal hypercalcemia caused by subcutaneous fat necrosis without any apparent cutaneous lesion

Bonnemains, Laurent; Rouleau, Stéphanie; Sing, Gaelle; Bouderlique, C; Coutant, Régis

doi:10.1007/s00431-008-0682-5

Cited by 14 publications

(20 citation statements)

References 13 publications

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“…Individual lesions vary from a few millimeters to several centimeters and may coalesce. SCFN can present without any obvious visible skin changes and might need careful examination [2]. The disorder has been described in the literature as an entity mostly confined to term or post-term infants.…”

Section: Discussionmentioning

confidence: 97%

Subcutaneous fat necrosis in newborn—an unusual case and review of literature

2011

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Subcutaneous fat necrosis (SCFN) of the newborn is an uncommon, self-limiting panniculitis mostly occurring within the first few weeks after birth. SCFN has been described mostly in term or post-term newborn infants in literature. We report a preterm infant developing extensive subcutaneous fat necrosis within the first week of life after significant perinatal hypoxic injury. The infant was conservatively managed for subcutaneous fat necrosis but developed hypercalcaemia and required prolonged medical treatment. Hypercalcaemia is a rare but serious complication of subcutaneous fat necrosis and needs prolonged follow-up. The etiopathogenesis of both subcutaneous fat necrosis in newborn and the resultant hypercalcaemia are poorly understood. Conclusion: Significant subcutaneous fat necrosis can develop in both preterm and term infants, and preterm infants also develop significant complications including hypercalcaemia.Keywords Subcutaneous fat necrosis of newborn . Hypercalcaemia . Perinatal hypoxia . Hypoglycaemia Case reportA male preterm infant weighing 2,300 g was born after a crash Caesarean section at a gestational age of 33+6 weeks. His mother presented with a foetal heart of 30/min and a history of reduced foetal movements for 24 h. The infant was born in poor condition (blue, floppy with no respiratory effort) and no heart rate was noted at birth. The infant needed full cardiopulmonary resuscitation till 4 min of age. First heart rate was detected at 2 min of age, and first gasping effort was noted at 9 min of age, The infant needed continued ventilatory support in view of poor respiratory effort. Apgar scores were 1 at 1 min and 5 at 5 min. Cord blood gases reflected severe combined respiratory and metabolic acidosis. He was ventilated for the first 24 h of life.He suffered from severe hypoxic ischaemic encephalopathy, and abnormal movements of both the upper extremities were noted from 11 h of age. In view of his prematurity, therapeutic hypothermia was not considered appropriate. He was treated with phenobarbitone for the first 6 days of life. He developed abnormal liver and renal functions and also developed severe hypoglycaemia requiring high concentration of dextrose infusion (25% dextrose) to maintain normoglycaemia (total fluid intake was restricted initially in view of severe hypoxic ischaemic encephalopathy (HIE)). Generalised hypertonia was noted very early in life. He developed large bilateral intraventricular haemorrhages which subsequently resulted in bilateral posthaemorrhagic hydrocephalus.He developed an erythematous rash over the dorsum of the chest, abdomen, and right arm which felt warm in comparison to the rest of the body on the third day of life. This developed into a sclerematous appearance by day 5. A punch biopsy of skin confirmed subcutaneous fat necrosis

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Section: Discussionmentioning

confidence: 97%

Subcutaneous fat necrosis in newborn—an unusual case and review of literature

2011

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“…SCFN is considered a rare cause of hypercalcemia in newborns and infants. However, when an unexplained hypercalcemia is detected in a neonate, especially following an abnormal perinatal period, one should perform a meticulous search for these nodules 10 . Hyperparathyroidism was excluded in our patient due to the adequately low PTH level.…”

Section: Discussionmentioning

confidence: 99%

Subcutaneous Fat Necrosis and Hypercalcemia Following Therapeutic Hypothermia – a Patient Report and Review of the Literature

Zifman

Mouler

Eliakim

et al. 2010

Journal of Pediatric Endocrinology and Metabolism

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Subcutaneous fat necrosis (SCFN) of the newborn is an uncommon dermatologic disorder characterized by firm, palpable subcutaneous nodules or plaques with or without erythema. Despite its benign course, SCFN may become complicated by extracutaneous manifestations. Hypercalcemia is considered a rare complication, but it is potentially fatal if unrecognized. During the last several years therapeutic hypothermia (TH) became an essential new therapeutic modality for severe neonatal asphyxia. We report a neonate who presented with SCFN and hypercalcemia following hypothermia therapy for hypoxic ischemic encephalopathy (HIE) and provide a review of the contemporary literature on the topic. We believe that this is important since the use of TH is rapidly increasing, and therefore, the possible side effects including SCFN and hypercalcemia may also become more prevalent. This prompts the need for awareness by treating physicians for this complication.

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“…da başlangıçta gözle görülen tipik deri bulguları olmayan detaylı bir muayene sonrası deri altı nodüllerin fark edilebildiği şiddetli hiperkalsemisi olan YSYN'li bir olgu tanımlamış-lardır. 10 Bu nedenle de hiperkalsemisi olan yenidoğanlarda dikkatli bir dermatolojik incelemenin etiyolojiye ışık tutabileceği vurgulanmıştır.…”

Section: Resi̇m 4: Subkütan Yağ Nekrozu (He X300)unclassified

“…[1][2][3] Genel olarak kendiliğinden gerilemekle birlikte hiperkalsemi, trombositopeni, hipertrigliseridemi gibi ciddi sistemik bulguların eşlik edebildiği vakalarda bildirilmektedir. [4][5][6][7][8][9][10][11][12] Tanı klinik bulgu ve histopatolojik inceleme ile birlikte konulmaktadır. Bu çalışmada; miadında sezaryen ile Yenidoğanın Subkütan Yağ Nekrozu Ö ÖZ ZE ET T Yenidoğanın subkütan yağ nekrozu (YSYN), çoğunlukla doğum travması, mekonyum aspirasyonu hikâyesi olan ya da yaşamın ilk haftalarında hipotermi tedavisi alan, miadında yenidoğan-larda gözlenen nadir bir pannikülit formudur.…”

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“…11 Mekonyum aspirasyonu, plasenta previa, umbilikal kord prolapsusu, preeklampsi gibi perinatal hipoksemi nedenleri, gestasyonel diyabet gibi hipoglisemi nedenleri, hipotermi ve zorlu doğum gibi nedenler hastalığı presipite eden faktörler olarak suçlan-maktadır. [1][2][3][4][5][6][7][8][9][10][11] Hastalığın doğum sırasında meydana gelen iskemik hasarlanma ile başladığı düşünül-mektedir. Doğum sonrasında hipotermi tedavisi alan ya da hipotermik kalp cerrahisi sonrası gelişen birkaç olgu bildirimi de mevcuttur.…”

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Neonatal Subcutaneous Fat Necrosis: Case Report

Kundak¹,

Girgin²,

Gülez³

et al. 2016

Turkiye Klinikleri J Dermatol

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168enidoğanın subkütan yağ nekrozu (YSYN), tipik olarak zamanında doğmuş yenidoğanlarda yaşamın ilk 2 ve 3. haftalarında ortaya çıkan, subkütan dokuya lokalize, iyi sınırlı, mobil, kırmızı-mor renkli, bazen simetrik yerleşimli olabilen, genellikle yanaklar, omuz, sırt, kalça ve uyluklarda ortaya çıkan, endürasyon gösteren nodül veya plaklar şek-linde görülen bir pannikülit tablosudur. Sklerema neonatorumun aksine YSYN iyi prognozu olan lokalize bir hastalıktır. Hastalığın etiyolojisi tam bilinmemektedir. Hipoksi, lokal travma, hipotermi gibi fetüse ait nedenler ile birlikte annenin doğum öncesi dönemde kalsiyum antagonistleri ve kokain kullanması gibi anneye ait faktörlerin de rol alabileceği bildirilmektedir.1-3 Genel olarak kendiliğinden gerilemekle birlikte hiperkalsemi, trombositopeni, hipertrigliseridemi gibi ciddi sistemik bulguların eşlik edebildiği vakalarda bildirilmektedir. [4][5][6][7][8][9][10][11][12] Tanı klinik bulgu ve histopatolojik inceleme ile birlikte konulmaktadır. Bu çalışmada; miadında sezaryen ile Yenidoğanın Subkütan Yağ Nekrozu Ö ÖZ ZE ET T Yenidoğanın subkütan yağ nekrozu (YSYN), çoğunlukla doğum travması, mekonyum aspirasyonu hikâyesi olan ya da yaşamın ilk haftalarında hipotermi tedavisi alan, miadında yenidoğan-larda gözlenen nadir bir pannikülit formudur. YSYN'li çoğu hasta kendiliğinden gerilemekle birlikte, hiperkalsemi, hipoglisemi, hipertrigliseridemi gibi metabolik komplikasyonların veya anemi ve trombositopeni gibi hematolojik komplikasyonların eşlik edebildiği hastalar olabilmektedir. Herhangi bir metabolik ve hematolojik komplikasyonun eşlik etmediği miadında bir yenidoğan olgusu, hastalığın nadir görülmesi sebebiyle bildirilmeye uygun bulunmuştur.A An na ah ht ta ar r K Ke el li im me el le er r: : Pannikülit; yağ nekrozu A AB BS ST TR RA AC CT T Neonatal subcutaneous fat necrosis (NSFN) is a rare form of panniculitis that occurs in term infants who experience birth trauma, meconium aspiration or therapeutic cooling in the first week of life. Although most cases of NSFN resolve spontaneously, it may be associated with metabolic complications as hypercalsemia, hypoglycaemia, hypertriglyceridemia or with heamatological complications as anemia and thrombocytopenia. Because the disease is rarely seen, we want to report the case of term newborn who did not have any metabolic and heamatologic complications.

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Severe neonatal hypercalcemia caused by subcutaneous fat necrosis without any apparent cutaneous lesion

Abstract: Subcutaneous fat necrosis may induce severe hypercalcemia without any visible cutaneous lesion.

Cited by 14 publications

References 13 publications

Subcutaneous fat necrosis in newborn—an unusual case and review of literature

Subcutaneous fat necrosis in newborn—an unusual case and review of literature

Subcutaneous Fat Necrosis and Hypercalcemia Following Therapeutic Hypothermia – a Patient Report and Review of the Literature

Neonatal Subcutaneous Fat Necrosis: Case Report

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