Subkutan Yağ Nekrozu Olan Süt Çocuğunda Ciddi Hiperkalsemi: Bifosfonat Tedavisi ile Başarılı Yönetim

Öz

Subkutan yağ nekrozu (SYN) nadir görülen bir durumdur ve genellikle yaşamın ilk birkaç haftasında pannikülit ile kendini gösterir. SYN’e bağlı cilt lezyonları kendiliğinden iyileşse de yaşamı tehdit eden şiddetli hiperkalsemi en önemli komplikasyonudur. Sepsis nedeniyle tedavi edilmekte olan yenidoğanda gözlenen nodüler eritemli cilt lezyonları SYN tanısı koydurdu. Olguda gelişen ciddi hiperkalsemi olağan tedavi rejimlerine yanıt vermemişti. Bu nedenle, hiperkalsemiyi düzeltmek için bifosfonat (pamidronat) tedavisi kullanıldı. Hiperkalsemi, SYN’nin ciddi bir komplikasyonudur ve uzun süreli izlem gerektirir. Bu olgularda kalsiyum seviyeleri düzenli olarak takip edilmelidir. Pamidronatın SYN’e bağlı hiperkalsemide güvenlidir ve ciddi hiperkalseminin tedavisinde ilk seçenek olarak tercih edilmelidir

Anahtar Kelimeler

• subkutan yağ nekrozu
• hiperkalsemi
• bifosfonat

Severe Hypercalcemia in an Infant with Subcutaneous Fat Necrosis: Successful Management with Bisphosphonate Treatment

Öz

Subcutaneous fat necrosis (SFN) in infancy is a rare condition and usually presents with panniculitis, especially within the first few weeks of life. These skin lesions may improve spontaneously but the most lifethreatening complication of SFN is severe hypercalcemia. Here, we report the case of an infant with severe hypercalcemia due to SFN. The infant was managed for sepsis and evaluated for nodular erythematous skin lesions, which revealed SFN. Hypercalcemia was severe and unresponsive to the usual treatment regimens. Therefore, bisphosphonate (pamidronate) was used to correct the hypercalcemia. Hypercalcemia is a serious complication in SFN and needs prolonged follow-up. Calcium levels should be monitored regularly in these patients. We report that pamidronate may be appropriate as the first line therapy to treat severe hypercalcemia due to SFN.

Anahtar Kelimeler

• Subcutaneous fat necrosis
• hypercalcemia
• bisphosphonate

Kaynakça

1. 1. Gomes MP, Porro AM, Enokihara MM, Floriano MC. Subcutaneous fat necrosis of the newborn:clinical manifestation in two cases. An Bras Dermatol. 2013;88 (6 Suppl):154-7.
2. 2. Burden AD, Krafchik BR. Subcutaneous fat necrosis of the newborn:a review of 11 cases. Pediatr Dermatol. 1999;16:384-7.
3. 3. Mahe E, Girszyn N, Hadj-Rabia A, Bodemer C, Hamel-TeillacD, De Prost Y. Subcutaneous fat necrosis of the newborn :a systematic evaluation of risk factors, clinical manisfestations, complications and outcome of 16 children. Br J Dermatol. 2007;156:709-15.
4. 4. Samedi VM, Yusuf K, Yee W, Obaid H, AI Awad EH. Neonatal hypercalcemia secondary to subcutaneous fat necrosis successfully treated with pamidronate:a case series and literature rewiev. AJP Rep. 2014; 4: 93-6.
5. 5. Alos N, Eugene D, Fillion M, Powell M, Kokta V, Chabot G. Pamidronate: Treatment for severe hypercalcemia in neonatal subcutaneous fat necrosis. Horm Res. 2006;65:289-94.
6. 6. Shumer DE, Thaker V, Taylor GA, Wassner AJ. Severe hypercalcemia due to subcutaneous fat necrosis:presentation, management and complications. Arch Dis Child Fetal Neonatal Ed. 2014; 99: 419-421.
7. 7. Sthrohm B,Hobson A, Brocklehurst P, Edwards AD, Azzopardi D;UK TOBY Cooling Register. Subcutaneous fat necrosisi after moderate therapeutic hypothermia in neonates. Pediatrics. 2011; 182:450-2.
8. 8. Tran JT, Sheth AP.Complications of subcutaneous fat necrosis of the newborn: a case report and review of the literature. Pediatr Dermatol. 2003; 20: 257-261.

9. 9. Katz DA, Huerter C, Bogard P, Braddock SW. Subcutaneous fat necrosis of the newborn. Arch Dermatol. 1984; 120:1517-18.
10. 10. Farooque A, Moss C, Zehnder D, Hewison M, Shaw N. Expression of 25-hydroxyvitamin D3-1alpha-hydroxylasein subcutaneos fat necrosis. Br J Dermatol.2009;160:423-5.
11. 11. Lewis HM, Ferryman S, Gatrad AR, Moss C. Subcutaneous fat necrosis of the newborn associated with hypercalcemia. J. R. Soc. Med. 1994; 87(8): 482-483.
12. 12. Mitra S, Dove J, Somisetty SK. Subcutaneous fat necrosis in newborn-an unusual case and review of the literature. Eur J Pediatr 2011;170: 1107-10.
13. 13. Russell RGG, Muhlbauer RC, Bisaz S, Williams DA, Fleisch H. The influence of pyrophosphate, condensed phosphates, phosphonates and other phosphate compounds on the dissolution of hydroxyapatite in vitro and on bone resorption induced by parathyroid hormone in tissue culture and in thyroparathyroidectomised rats. Calc. Tis Res. 1970; Vol.6 183-196.
14. 14. Drake MT, Clarke BL, Khosla S. Bisphosphonates: Mechanism of action and role in clinical practice. Mayo Clin. Proc. 2008; 83: 1032-45.