AJOPS | CASE REPORT PUBLISHED: 30-09-2019 Aes

Large-volume liposuction in acquired partial lipodystrophy Yasiru Gehan Karunaratne MD,1 Marc Langbart MBBS FRACS (Plast),1 Jerry R Greenfield MBBS (Hons 1) FRACP PhD,2,3,4 James Southwell-Keely MBBS (Hons) FRACS (Plas)1

1 Department of Plastic Introduction St Vincent’s Hospital Lipodystrophy syndromes, a clinically diverse group Darlinghurst, New South Wales AUSTRALIA of conditions with selective deficiency of as a central feature, are classified as inherited 2 Department of and Endocrinology or acquired, with further sub-classification based St Vincent’s Hospital on aetiology and clinical features.1 The acquired Darlinghurst, New South Wales lipodystrophy subtypes include localised, partial, AUSTRALIA generalised and HIV-associated.1 Acquired partial 3 St Vincent’s Clinical School lipodystrophy, also known as Barraquer-Simons Faculty of Medicine syndrome, is a rare condition first described in University of New South Wales 1885 by Mitchell.2 It is characterised by progressive Kensington, New South Wales accumulation of surplus subcutaneous fat in the hips AUSTRALIA and lower limbs, contrasted with a deficiency of fat in 4 Diabetes and Metabolism Group the upper body, as well as metabolic complications of Garvan Institute of Medical Research hypertriglyceridaemia and .3 Precise Darlinghurst, New South Wales pathogenesis is unclear; however, it is commonly AUSTRALIA preceded by , and reports have implicated OPEN ACCESS tetanus antitoxin, surgery, psychological stress and sunburn.4 Management options are limited1 and Correspondence scarce literature regarding liposuction in acquired James Southwell-Keely Name: partial lipodystrophy exists, though it has been Address: St Vincent’s Clinic used effectively in other lipodystrophy syndromes, Suite 709, 438 Victoria Street albeit rarely.5,6 Medical management and large- Darlinghurst, New South Wales, 2010 volume liposuction are effective in improving the AUSTRALIA metabolic profile of patients without lipodystrophy, Email: [email protected] as well as cosmesis and quality of life.5,7,8 The Phone: +61 (0)2 9369 2800 removal of large volumes of fat via liposuction is not without potential complications, and postoperative Citation: Karunaratne YG, Langbart M, Greenfield JR, 9,10 Southwell-Keely J. Large-volume liposuction in acquired medical management is complex. We report a partial lipodystrophy. Aust J Plast Surg. 2019;2(2):67–69. case of acquired partial lipodystrophy treated with https://doi.org/ 10.34239/ajops.v2n2.14 large-volume liposuction of the lower limbs with

Accepted for publication: 2 December 2018 subsequent metabolic improvements.

Copyright © 2019. Authors retain their copyright in the Keywords: lipodystrophy, lipectomy, metabolome article. This is an open access article distributed under the Creative Commons Attribution Licence which permits unrestricted use, distribution and reproduction in any Case presentation medium, provided the original work is properly cited. A 29-year-old female with acquired partial Section: Aesthetic lipodystrophy was referred to the team by her endocrinologist for consideration of liposuction. She developed lipodystrophy at age four years following varicella infection.

Australasian Journal of Plastic Surgery 67 Volume 2 Number 2 2019 Karunaratne, Langbart, Greenfield, Southwell-Keely: Large-volume liposuction in acquired partial lipodystrophy AJOPS | CASE REPORT

Her medical history included hypothyroidism, tonsillectomy, adenoidectomy and excision of cervical intraepithelial neoplasia II. Her medications were daily metformin XR 500 mg, thyroxine 50 mcg and the cyproterone acetate/ ethinylestradiol oral contraceptive. On examination, she weighed 67.5 kg, with significant deficiency of subcutaneous fat in her face, upper limbs and torso. There was excess subcutaneous fat in her lower limbs, with predominance in the upper thighs and sparing of the buttocks (Figure 1). Circumferences were 74.0 Fig 1. Preoperative cm at the waist, 60.0 cm bilaterally at a point 40.0 cm below the iliac crest and 33.0 cm bilaterally at a point 30.0 cm above the lateral malleolus. Biochemical markers showed total cholesterol 6.9 mmol/L, low-density lipoprotein (LDL) cholesterol 4.1 mmol/L, high-density lipoprotein (HDL) cholesterol 1.5 mmol/L and triglycerides 2.8 mmol/L. She exhibited hyperinsulinaemia (24.5 mU/L), while her leptin level was 25.6 ng/mL (reference range 3.7–11.1 ng/mL). Liver ultrasound was suggestive of mild fatty infiltration. Following consultation between the plastic surgery and Fig 2. Three years postoperative endocrinology teams, consensus for surgery was reached. Table 1. Pre- and postoperative comparison Preoperative Three months The operation involved tumescent infiltration of 10 postoperative L normal saline, with a total of 200 mg ropivacaine Weight (kg) 67.5 58.0 and 10 mg of 1:1,000,000 (epinephrine). Waist circumference 74.0 – Large-volume liposuction of the calves and thighs (cm) was conducted via multiple stab incisions with total Leg circumference (cm) 60.0 47.0 (left), 49.5 40 cm below iliac crest bilaterally (right) aspirate of 18,950 mL. The postoperative course Leg circumference (cm) 33.0 30.0 bilaterally had minimal complications, though was prolonged 30 cm above lateral bilaterally when compared with routine liposuction. She malleolus had tender calves but was able to mobilise on Total cholesterol 6.9 3.8 (mmol/L) postoperative day one. Haemoglobin levels LDL cholesterol (mmol/L) 4.1 1.7 decreased from 116 g/L (reference range 110–160) HDL cholesterol 1.5 1.0 preoperatively to 92 g/L postoperatively, though (mmol/L) did not require treatment. Myoglobinuria and a Triglycerides (mmol/L) 2.8 2.4 transiently elevated creatine kinase (6590 U/L) Insulin (mU/L) 24.5 9.7 were documented, with renal function remaining Leptin (ng/ml) 25.6 5.1 normal throughout and recorded abnormal values trending towards baseline prior to discharge on 500 mg metformin XR and 50 mcg thyroxine daily. postoperative day five. She weighed 58 kg and her leg circumferences had There was significant metabolic improvement three reduced considerably (Table 1). There was also months postoperatively ( ). She remained on Table 1 significant cosmetic improvement (Figure 2).

Australasian Journal of Plastic Surgery 68 Volume 2 Number 2 2019 Karunaratne, Langbart, Greenfield, Southwell-Keely: Large-volume liposuction in acquired partial lipodystrophy AJOPS | CASE REPORT

Three years later revision liposuction was Conclusion undertaken for a minimal increase in subcutaneous We report the successful treatment of acquired fat, cosmesis and gradually worsening biochemical partial lipodystrophy with large-volume markers, with total cholesterol 4.7 mmol/L, LDL liposuction. This resulted in significant biochemical cholesterol 2.5 mmol/L, HDL cholesterol 1.1 and aesthetic improvements for the patient. mmol/L, triglycerides 2.5 mmol/L and insulin 15.2 mU/L. Despite this, her weight remained stable at Disclosure 57 kg. Her metformin XR dose was increased to The authors have no financial or commercial 1000 mg daily and she commenced on rosuvastatin conflicts of interest to disclose. 10 mg daily. At surgery, 1700 mL was aspirated, and she progressed to discharge uneventfully. References 1 Garg A. Acquired and inherited lipodystrophies. N Engl J Discussion Med. 2004;350(12):1220–34. https://doi.org/10.1056/NEJM- ra025261 PMid:15028826 As few as 250 cases of acquired partial lipodystrophy 2 Mitchell SW. Singular case of absence of adipose have been reported,1 limiting the literature to matter in the upper half of the body. Am J Med Sci. individual case reports and small series. However, 1885;179:105. https://doi.org/10.1097/00000441-188507000- other lipodystrophy syndromes with similar 00006 clinical manifestations have been more thoroughly 3 Hussain I, Garg A. Lipodystrophy syndromes. Endocri- investigated due to higher prevalence.5,7 In these nol Metab Clin North Am. 2016;45(4):783–97. https://doi. studies and others, liposuction has improved the org/10.1016/j.ecl.2016.06.012 PMid:27823605 metabolic profile of patients, as well as making 4 Misra A, Peethambaram A, Garg A. Clinical features and metabolic and autoimmune derangements in acquired aesthetic enhancements that improve quality of partial lipodystrophy: report of 35 cases and review of the 5,7,8 life. literature. Medicine (Baltimore). 2004;83(1):18–34. https:// Large-volume liposuction has been defined as doi.org/10.1097/01.md.0000111061.69212.59 PMid:14747765 aspiration of ≥5000 mL, and recommendations of 5 Wolfort FG, Cetrulo CL Jr, Nevarre DR. Suction-assist- ed lipectomy for lipodystrophy syndromes attributed safe maximum aspirate volume vary significantly.9 to HIV-protease inhibitor use. Plast Reconstr Surg. Postoperative care can be complicated by significant 1999;104(6):1814–20; discussion 1821–22. https://doi. blood loss, venous thromboembolism, fluid shifts org/10.1097/00006534-199911000-00032 and electrolyte abnormalities.9 The key is adequate 6 Kandamany N, Munnoch A. Liposuction for lower limb li- fluid resuscitation and electrolyte replacement. A podystrophy in congenital analbuminaemia: a case report. . 2014;67(2):e54–57. https://doi. series of 631 cases found large-volume liposuction J Plast Reconstr Aesthet Surg org/10.1016/j.bjps.2013.09.001 PMid:24090727 could be safely undertaken if patient selection 7 Schmeller W, Hueppe M, Meier-Vollrath I. Tumescent is judicious, the operation is conducted by an liposuction in lipoedema yields good long-term results. Br J experienced surgeon and postoperative care is Dermatol. 2012;166(1):161–68. https://doi.org/10.1111/j.1365- meticulous.10 In our case, 18,950 mL was aspirated 2133.2011.10566.x PMid:21824127 from a patient with acquired partial lipodystrophy, 8 Boriani F, Villani R, Morselli PG. Metabolic effects of with biochemical improvements and no major large-volume liposuction for obese healthy women: a meta-analysis of fasting insulin levels. . postoperative complications. To our knowledge Aesthetic Plast Surg 2014;38(5):1050–56. https://doi.org/10.1007/s00266-014- this is the largest volume liposuction reported in 0386-3 PMid:25099498 this rare syndrome. The value of the procedure 9 Albin R, de Campo T. Large-volume liposuction in 181 was highlighted by the improvement in physical patients. Aesthetic Plast Surg. 1999;23(1):5–15. https://doi. appearance, biochemical markers and patient org/10.1007/s002669900235 PMid:10022931 satisfaction. Although we were concerned that 10 Commons GW, Halperin B, Chang CC. Large-volume removal of significant fat mass might worsen the liposuction: a review of 631 consecutive cases over 12 years. . 2001;108(6):1753–63; discussion metabolic profile (due to the lack of a ‘sink’ for Plast Reconstr Surg 1764–57. https://doi.org/10.1097/00006534-200111000-00051 storing fatty acids), this did not eventuate.

Australasian Journal of Plastic Surgery 69 Volume 2 Number 2 2019