Subcutaneous Fat Necrosis of the Newborn Secondary to Sepsis: A Case Report

Apoorva Dhananjay Chopkar; Bhagyashree Babanrao Supekar; Jayesh Ishwardas Mukhi; Rajesh Pratap Singh; Dharitri M Bhat

doi:10.4103/ijpd.IJPD_52_20

CASE REPORT

Year : 2021 | Volume : 22 | Issue : 1 | Page : 73-76

Subcutaneous Fat Necrosis of the Newborn Secondary to Sepsis: A Case Report

Apoorva Dhananjay Chopkar¹, Bhagyashree Babanrao Supekar¹, Jayesh Ishwardas Mukhi¹, Rajesh Pratap Singh¹, Dharitri M Bhat²
¹ Department of Dermatology, Venereology and Leprology, Government Medical College and Hospital, Nagpur, Maharashtra, India
² Department of Pathology, Government Medical College and Hospital, Nagpur, Maharashtra, India

Date of Submission	05-Apr-2020
Date of Decision	07-May-2020
Date of Acceptance	11-Jun-2020
Date of Web Publication	31-Dec-2020

Correspondence Address:
Bhagyashree Babanrao Supekar
Department of Dermatology, Venereology and Leprology, Government Medical College and Hospital, Nagpur, Maharashtra
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/ijpd.IJPD_52_20

Abstract

Subcutaneous fat necrosis of the newborn (SCFN) is a rare noninfectious type of panniculitis, which primarily affects term and postterm infants. It is usually a benign, self-limiting condition, but in rare cases can be complicated by hypercalcemia, hypoglycemia, thrombocytopenia, etc. We report a case of SCFN in a 4-week-old male infant, treated successfully without complications.

Keywords: Sepsis, subcutaneous fat necrosis of the newborn, term neonate

How to cite this article:
Chopkar AD, Supekar BB, Mukhi JI, Singh RP, Bhat DM. Subcutaneous Fat Necrosis of the Newborn Secondary to Sepsis: A Case Report. Indian J Paediatr Dermatol 2021;22:73-6

How to cite this URL:
Chopkar AD, Supekar BB, Mukhi JI, Singh RP, Bhat DM. Subcutaneous Fat Necrosis of the Newborn Secondary to Sepsis: A Case Report. Indian J Paediatr Dermatol [serial online] 2021 [cited 2021 Apr 22];22:73-6. Available from: https://www.ijpd.in/text.asp?2021/22/1/73/305810

Introduction

Subcutaneous fat necrosis of the newborn (SCFN or SFN) is a rare form of lobular panniculitis that is seen in term or postterm infants during the first few weeks of life. SCFN is usually self-limiting with a favorable prognosis unless complicated by hypercalcemia.^[1],[2] This condition is also known as adiponecrosis subcutanea.^[3] There are very few reports of SCFN secondary to sepsis described in literature from India.

Case Report

One-month-old full-term male neonate, born out of nonconsanguineous marriage delivered through cesarean section due to oligohydramnios to a 26-year-old primigravida mother, was referred to us with multiple reddish to purple swelling over scalp, trunk, and extremities for 15 days. The neonate was lethargic and had feeding difficulties for 10–12 days. Prenatal history was unremarkable. The baby had cried immediately after birth. The weight was 1.9 kg at birth. The initial few days of life were uneventful. Mother gave a history of two episodes of seizures on 20^th and 25^th day of life. On cutaneous examination, there were multiple erythematous to violaceous plaques with overlying ulceration and yellowish discharge present over axilla, occipital region of scalp, lower back, and buttocks [Figure 1]a,[Figure 1]b,[Figure 1]c. There were multiple erythematous, tender, indurated subcutaneous nodules and plaques present over bilateral upper and lower limbs [Figure 1]d and [Figure 1]e. The neonate was diagnosed as a case of severe malnutrition (weight <3SD) evident by loose skin folds over arms and thighs. The length of the baby was 48 cm (between median and 1SD). Hematological investigations revealed anemia (9.2 g%), leukocytosis (18,000/mm³), and neutrophilia (79%). Peripheral smear examination revealed toxic granules and cytoplasmic vacuoles within neutrophils with reactive lymphocytosis. Serum calcium levels were normal. Blood culture and sensitivity demonstrated methicillin-resistant Staphylococcus aureus sensitive to linezolid. Renal function tests were within normal limits. An ultrasound scan of soft tissue on the lateral side of the left thigh showed hypoechoic lesion noted in subcutaneous plane. Ultrasonography of abdomen and pelvis was normal. Computed tomography of head revealed hypodensity in the right parieto-temporal region of average attenuation of 14 HU with hydrocephalus suggestive of infective etiology [Figure 2]. Histopathological examination from the nodule over back revealed clusters of fibrinous necrotic material containing few residual viable adipocytes and infiltration of acute inflammatory cells suggestive of subcutaneous fat necrosis [Figure 3]a,[Figure 3]b,[Figure 3]c. Special stain for fat was not done due to the lack of resources. On the basis of history, clinical examination, histopathology, and investigations, a final diagnosis of SCFN secondary to sepsis was reached. The infant was managed in pediatric unit with intravenous fluid hydration therapy and parenteral broad-spectrum antibiotics. The infant was treated with intravenous meropenem 60 mg thrice daily, intravenous vancomycin 20 mg once daily, and syrup linezolid 0.75 ml thrice daily for 14 days. The skin lesions began to regress and infant started feeding well. There was complete resolution of subcutaneous lesions at the end of 2 weeks of treatment with hyperpigmented scars [Figure 4]a,[Figure 4]b,[Figure 4]c,[Figure 4]d. The infant was discharged and followed up for 6 months with serial monitoring of calcium levels.

Figure 1: Multiple erythematous to violaceous plaques with overlying ulceration with yellowish discharge over left axilla (a), lower back (b) occipital region of scalp (c), and buttocks. Multiple erythematous, tender, indurated subcutaneous nodules and plaques present over bilateral upper and lower limbs (d and e)

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Figure 2: Computed tomography of head revealed hypodensity in the right parieto-temporal region of average attenuation of 14 HU with hydrocephalus, suggestive of infective etiology

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Figure 3: Histopathological examination from the nodule over back revealed clusters of fibrinous necrotic material containing few residual viable adipocytes and infiltration of acute inflammatory cells (H and E, a: 10x and b,c: 40x), suggestive of subcutaneous fat necrosis

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Figure 4: Complete resolution of subcutaneous lesions at the end of 2 weeks of treatment with hyperpigmented atrophic scars over scalp (a), back (b), and legs (c and d)

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Discussion

SCFN is a rare inflammatory pathology affecting subcutaneous and adipose tissue of postterm or term infants, during the first few weeks of life.^[4] The neonatal fat has higher melting point than adult fat due to higher content of saturated fatty acids such as stearic and palmitic acid.^[5] Although etiology is not known, various maternal and fetal predisposing factors are thought to be implicated. These predisposing risk factors include hypoxic ischemic injury, perinatal asphyxia, therapeutic hypothermia, meconium aspiration, Rh incompatibility, obstetric trauma, cord prolapse, sepsis, gestational diabetes, preeclampsia, smoking, or exposure to passive smoking during pregnancy and maternal mediations (calcium channel blockers and cocaine).^[2],[6] Most of the case reports that have been described until now are associated with perinatal hypothermia and obstetric risk factors. In our case, sepsis was a predisposing factor for causation of SCFN. Pathophysiology of SCFN may involve a precocious phase of impaired tissue perfusion with subsequent tissue hypoxia which leads to crystallization of neonatal subcutaneous fat followed by necrosis and formation of granulomatous infiltrates.^[7] It is a very rare disorder with no gender predilection. SCFN usually appears a few days or weeks after birth. In acute phase, the lesions are tender later on which becomes asymptomatic. It presents as erythematous to violaceous, firm to indurated painful subcutaneous nodules usually located over cheeks, trunk, shoulders, and buttocks. The lesions may be flesh colored or have overlying erythema. Rarely, these lesions may become fluctuant and ulcerate with spontaneous discharge of fat.^[8] Our case also had tender, erythematous, subcutaneous indurated nodules and plaques distributed over scalp, trunk, buttocks, and legs with ulceration evident on scalp, back, and legs. Histopathological findings include inflammatory histiocytic reaction with fat tissue necrosis. The presence of extensive granulocytic infiltrate includes macrophages, giant cells, eosinophils, and lymphocytes. Furthermore, fat cells and giant cells containing radial crystals which stained with oil red O represent triglyceride deposition. Areas of calcification may be present occasionally. It is a self-limiting condition with favorable prognosis unless complicated by hypercalcemia, hypoglycemia, anemia, and thrombocytopenia, among which hypercalcemia is a common life-threatening complication.^[9],[10] It has been postulated that production of 1,25(OH)₂ Vitamin D3 by granulomatous reaction and bone resorption due to elevated parathyroid hormone and prostaglandin E2 contributed to hypercalcemia. Furthermore, increased expression of Vitamin D3-activating enzyme 1-alpha-hydroxylase in skin biopsy sample was seen.^[11],[12] Increased prostaglandins level also initiates an inflammatory cascade resulting in fever, thrombocytopenia, and increase in C-reactive protein levels.^[13] The resultant hypercalcemia may lead to lethargy, irritability, hypotonia, polyuria, polydipsia, vomiting, dehydration, and constipation.^[14] Our case had no complication at the time of admission and at 6 months of follow-up. In most of the cases, treatment should be conservative except for the lesions that may benefit from aspiration to prevent rupture, infections, and scarring. Periodic monitoring of calcium levels until the age of 6 months is required as there can be delayed elevations. If hypercalcemia is present, management includes hydration, use of calcium wasting furosemide, and low calcium formula. Furthermore, systemic glucocorticoids and bisphosphonates can be used to treat hypercalcemia.^[15] SCFN usually has a favorable prognosis with spontaneous resolution within months. Delayed diagnosis can lead to severe complications such as hypercalcemia, renal failure, sudden cardiac arrest, hypoglycemia, and metastatic calcifications (myocardial calcifications, calcification of the falx cerebri, liver, gastric mucosa, etc.). Hence, prompt treatment of the triggering factors and meticulous monitoring with particular reference to serum calcium is needed until 6 months of age. To the best our knowledge, there is a paucity of literature describing SCFN secondary to sepsis. Thus, we report this case to create awareness among the pediatricians and dermatologists to rule out other rare causes.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Alsofyani KA. Neonatal subcutaneous fat necrosis with hypercalcemia treatment using calcitonin. Saudi Med J 2018;39:622-6.
2.	Tran JT, Sheth AP. Complications of subcutaneous fat necrosis of the new born: A case report and review of the literature. Pediatr Dermatol 2003;20:257-61.
3.	Chikaodinaka AA, Jude AC. Subcutaneous fat necrosis of the newborn: A case report of a term infant presenting with malaise and fever at age of 9 weeks. Case Rep Pediatr 2015;2015:638962.
4.	Paige DG, Gennery AR, Cant AJ. The neonate. In: Burns T, Breathnach S, Cox N, Griffiths C, editors. Rook's Textbook of Dermatology. 8^th ed. ???: Wiley-Blackwell; 2010. p. 17.36 7.
5.	Caple JI, Reyes S. Subcutaneous fat necrosis of the new born: A case presentation. J Perinatol 1996;16 (2 Pt 1):140-1.
6.	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.
7.	Coondoo A, Lahiry R, Choudhury A, Sengupta S. Tender skin nodules in a newborn. Indian J Dermatol 2013;58:328. [PUBMED] [Full text]
8.	Mahé E, Girszyn N, Hadj-Rabia S, Bodemer C, Hamel-Teillac D, De Prost Y. Subcutaneous fat necrosis of the newborn: A systematic evaluation of risk factors, clinical manifestations, complications and outcome of 16 children. Br J Dermatol 2007;156:709-15.
9.	Gomes MP, Porro AM, Enokihara MM, Floriano MC. Subcutaneous fat necrosis of the newborn: Clinical manifestations in two cases. An Bras Dermatol 2013;88:154-7.
10.	Woods AG, Cederholm CK. Subcutaneous fat necrosis and whole-body cooling therapy for neonatal encephalopathy. Adv Neonatal Care 2012;12:345-8.
11.	Farooque A, Moss C, Zehnder D, Hewison M, Shaw NJ. Expression of 25hydroxyvitamin D31alphahydroxylase in subcutaneous fat necrosis. Br J Dermatol 2009;160:423-5.
12.	Norwood-Galloway A, Lebwohl M, Phelps RG, Raucher H. Subcutaneous fat necrosis of the newborn with hypercalcemia. J Am Acad Dermatol 1987;16:435-9.
13.	Sharata H, Postellon DC, Hashimoto K. Subcutaneous fat necrosis, hypercalcemia, and prostaglandin E. Pediatr Dermatol 1995;12:43-7.
14.	Choudhary R, Sachdeva G, Katoch G. Neonatal subcutaneous fat necrosis as a close differential of neonatal sepsis: Case report and review of literature. Indian J Paediatr Dermatol 2019;21:11-4.
15.	Lake EP, Worobec SM, Aronson IK. Subcutaneous tissue disorders. In: Kang S, Amagai M, Bruckner AL, Enk AH, Margolis DJ, Mcmichael AJ, et al., editors. Fitzpatrick's Dermatology. 9^th ed. New York: McGraw-Hill Education; 2019. p. 1279-80.