Letter to the Editor
Successfull simultaneous pancreas kidney transplantation in a patient with congenital partial lipodystrophy
Trasplante simultáneo de riñón y páncreas exitoso en paciente con lipodistrofia parcial congénita
Luis Leon
, Pablo Uva, Elena Minue, Roxana Pilotti, Ignacio Cabrera, Alejandro Giunippero, Antonio Gallo, Francisco Osella, Fernanda Toniolo, Eduardo Chuluyan, Domingo Casadei
Corresponding author
Trasplante de Páncreas, Instituto de Nefrología - Nephrology, Buenos Aires, Argentina
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Significance of early kidney biopsy" "tienePdf" => "en" "tieneTextoCompleto" => "en" "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "451" "paginaFinal" => "453" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de Sjogren y nefropatía mixta. La importancia de la precocidad en la biopsia renal" ] ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1106 "Ancho" => 1508 "Tamanyo" => 428094 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Renal biopsy. Glomeruli with mesangial and endocapillary proliferation in which some hyaline pseudothrombi of different sizes are observed (arrows). 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"paginaFinal" => "451" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Luis Leon, Pablo Uva, Elena Minue, Roxana Pilotti, Ignacio Cabrera, Alejandro Giunippero, Antonio Gallo, Francisco Osella, Fernanda Toniolo, Eduardo Chuluyan, Domingo Casadei" "autores" => array:11 [ 0 => array:4 [ "nombre" => "Luis" "apellidos" => "Leon" "email" => array:1 [ 0 => "lleon@intramed.net" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Pablo" "apellidos" => "Uva" ] 2 => array:2 [ "nombre" => "Elena" "apellidos" => "Minue" ] 3 => array:2 [ "nombre" => "Roxana" "apellidos" => "Pilotti" ] 4 => array:2 [ "nombre" => "Ignacio" "apellidos" => "Cabrera" ] 5 => array:2 [ "nombre" => "Alejandro" "apellidos" => "Giunippero" ] 6 => array:2 [ "nombre" => "Antonio" "apellidos" => "Gallo" ] 7 => array:2 [ "nombre" => "Francisco" "apellidos" => "Osella" ] 8 => array:2 [ "nombre" => "Fernanda" "apellidos" => "Toniolo" ] 9 => array:2 [ "nombre" => "Eduardo" "apellidos" => "Chuluyan" ] 10 => array:2 [ "nombre" => "Domingo" "apellidos" => "Casadei" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Trasplante de Páncreas, Instituto de Nefrología - Nephrology, Buenos Aires, Argentina" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Trasplante simultáneo de riñón y páncreas exitoso en paciente con lipodistrofia parcial congénita" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Lipodystrophies (LDs) are a heterogeneous group of genetic and acquired disorders that mainly affect women. They are characterised by total or partial loss of subcutaneous adipose tissue (lipoatrophy), which may be associated with accumulation of fat (lipohypertrophy) in different regions of the body. Patients with LDs have extreme insulin resistance, hyperglycaemia, severe hypertriglyceridaemia, low HDL levels and fatty liver. The seriousness of these metabolic complications is correlated with the extent of fat loss.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">There are 2 hereditary forms:<ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">-</span><p id="par0015" class="elsevierStylePara elsevierViewall">Generalised lipodystrophy, which has an autosomal recessive inheritance pattern and is characterised by total loss of subcutaneous adipose tissue, and</p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">-</span><p id="par0020" class="elsevierStylePara elsevierViewall">Congenital partial lipodystrophy, the more common form, which has an autosomal dominant inheritance pattern and is linked to partial loss of adipose tissue.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">3</span></a></p></li></ul></p><p id="par0025" class="elsevierStylePara elsevierViewall">Several researchers have noted that the metabolic abnormalities observed are due to a failure in the functions of regulation of fatty acid storage and release of adipose tissue. There are also genes involved in the effector actions of insulin, and in adipocyte proliferation and differentiation. Different candidate genes have been analysed, such as the insulin receptor gene, the insulin receptor substrate 1 gene and the beta-2 adrenergic receptor gene.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">4</span></a> The lack of subcutaneous adipose tissue deposits is due to the low concentration of leptin. Hypoleptinaemia alters the hunger/satiety signals in the central nervous system and causes hyperphagia. Then, caloric excess causes fat to accumulate in the muscles and liver and triggers severe insulin resistance, diabetes with high insulin requirements and hypertriglyceridaemia, which creates a high risk of pancreatitis. Patients with LDs also have fatty liver, myocardiopathies and proteinuric nephropathies.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">A 41-year-old female patient with a diagnosis of congenital partial lipodystrophy, diabetes diagnosed at age 15, being treated with insulin aspart (115<span class="elsevierStyleHsp" style=""></span>IU/day), with poor glycaemic control, who had bilateral proliferative retinopathy and end-stage renal disease secondary to membranoproliferative glomerulonephritis type I (by renal punch biopsy [RPB]), in haemodialysis three times per week since April 2012. In addition, she had mixed dyslipidaemia being treated with atorvastatin 20<span class="elsevierStyleHsp" style=""></span>mg/day and fenofibrate 200<span class="elsevierStyleHsp" style=""></span>mg/day and HTN. Physical examination (PE): dry weight 66.500<span class="elsevierStyleHsp" style=""></span>kg; height 170<span class="elsevierStyleHsp" style=""></span>cm; BMI: 23.01; BP 160/70.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Her limbs looked thin, with loss of subcutaneous fat in the buttocks, thighs and upper limbs. Hypotrophic breasts, slight hepatomegaly, acanthosis nigricans on the neck and hirsutism.</p><p id="par0040" class="elsevierStylePara elsevierViewall">It was decided to place her on the waiting list for a double kidney–pancreas transplant, considering her CKD in dialysis and her diabetes without insulin resistance, given her C-peptide level below one and her expected treatment with recombinant human leptin, requested for compassionate use. She received a double kidney–pancreas transplant on 07/09/2015, with an intestinal bypass. She also received induction with ATG-Fresenius (anti-human T-lymphocyte immunoglobulin from rabbits) (15<span class="elsevierStyleHsp" style=""></span>mg/kg divided into 5 doses), antibacterial prophylaxis for 5 days with piperacillin/tazobactam, an antifungal agent for one month, an anti-cytomegalovirus (CMV) agent (for 3 months) and an anti-pneumocystis pneumonia (PCP) agent for 2 years. Maintenance immunosuppression was added with belatacept (<span class="elsevierStyleItalic">de novo</span>) and tacrolimus/Deltisona (methylprednisolone)/mycophenolate sodium. The indication for belatacept was due to the patient's vascular/metabolic risk and to initial efforts to decrease the dose of tacrolimus (relative to the standard dose) to later suspend it in the long term. The grafts functioned immediately; neither insulin nor haemodialysis was required. In the postoperative period, the patient had intercurrent intestinal obstruction with an anastomosis fistula, which required conversion to a new Roux-en-Y duodenojejunostomy. She also had a low-output pancreatic fistula, which closed without complications. Currently, the patient has normal renal and pancreatic function, with the following values: fructosamine 237<span class="elsevierStyleHsp" style=""></span>μmol/l (normal level [NL] 285<span class="elsevierStyleHsp" style=""></span>μmol/l), HbA1c 4.6% (NL 2.5%–5.9%), C-peptide 1.8<span class="elsevierStyleHsp" style=""></span>ng/ml (NL 0.5–2<span class="elsevierStyleHsp" style=""></span>ng/ml), creatinine 0.8<span class="elsevierStyleHsp" style=""></span>mg/dl (NL 0.8–1.4<span class="elsevierStyleHsp" style=""></span>mg/dl) and no proteinuria. Tacrolimus levels of 6<span class="elsevierStyleHsp" style=""></span>ng/ml are being maintained.</p><p id="par0045" class="elsevierStylePara elsevierViewall">This is the first report of a double kidney–pancreas transplant with a favourable course in a patient with congenital partial lipodystrophy and a new immunosuppressant regimen.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of interest" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Leon L, Uva P, Minue E, Pilotti R, Cabrera I, Giunippero A, et al. Trasplante simultáneo de riñón y páncreas exitoso en paciente con lipodistrofia parcial congénita. Nefrologia. 2016;36:450–451.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Therapeutic use of recombinant leptin" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "D. Araujo-Vilar" 1 => "C. Guillin-Amarelle" 2 => "S. Sanchez Iglesias" 3 => "A. Castro" 4 => "F.F. 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Barrio Castellanos" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Libro" => array:3 [ "fecha" => "2012" "paginaInicial" => "415" "editorial" => "Editorial Panamericana" ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0045" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Lipodistrofia parcial adquirida y diabetes" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "A. Paglione" 1 => "N. Ferrari" 2 => "G. Berg" 3 => "G. Fretchel" 4 => "M. Taverna" 5 => "V. Fasulo" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Medicina (Buenos Aires)" "fecha" => "2001" "volumen" => "61" "paginaInicial" => "81" "paginaFinal" => "84" ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Partial and generalized lipodystrophy: comparison of baseline characteristics and response to metreleptin" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "T. Diker-Cohen" 1 => "E. Cochran" 2 => "P. Gorden" 3 => "R. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 1 | 4 | 5 |
| 2024 October | 35 | 38 | 73 |
| 2024 September | 51 | 23 | 74 |
| 2024 August | 57 | 59 | 116 |
| 2024 July | 37 | 31 | 68 |
| 2024 June | 46 | 28 | 74 |
| 2024 May | 47 | 29 | 76 |
| 2024 April | 44 | 35 | 79 |
| 2024 March | 31 | 21 | 52 |
| 2024 February | 29 | 37 | 66 |
| 2024 January | 46 | 25 | 71 |
| 2023 December | 34 | 26 | 60 |
| 2023 November | 28 | 29 | 57 |
| 2023 October | 70 | 25 | 95 |
| 2023 September | 118 | 32 | 150 |
| 2023 August | 31 | 27 | 58 |
| 2023 July | 33 | 24 | 57 |
| 2023 June | 35 | 23 | 58 |
| 2023 May | 35 | 29 | 64 |
| 2023 April | 14 | 11 | 25 |
| 2023 March | 26 | 16 | 42 |
| 2023 February | 21 | 20 | 41 |
| 2023 January | 26 | 27 | 53 |
| 2022 December | 40 | 43 | 83 |
| 2022 November | 34 | 32 | 66 |
| 2022 October | 37 | 49 | 86 |
| 2022 September | 28 | 25 | 53 |
| 2022 August | 35 | 50 | 85 |
| 2022 July | 28 | 36 | 64 |
| 2022 June | 28 | 28 | 56 |
| 2022 May | 25 | 32 | 57 |
| 2022 April | 25 | 36 | 61 |
| 2022 March | 30 | 30 | 60 |
| 2022 February | 28 | 34 | 62 |
| 2022 January | 18 | 29 | 47 |
| 2021 December | 28 | 42 | 70 |
| 2021 November | 25 | 34 | 59 |
| 2021 October | 31 | 33 | 64 |
| 2021 September | 24 | 30 | 54 |
| 2021 August | 26 | 35 | 61 |
| 2021 July | 40 | 24 | 64 |
| 2021 June | 22 | 25 | 47 |
| 2021 May | 30 | 33 | 63 |
| 2021 April | 46 | 47 | 93 |
| 2021 March | 38 | 39 | 77 |
| 2021 February | 50 | 15 | 65 |
| 2021 January | 21 | 19 | 40 |
| 2020 December | 28 | 12 | 40 |
| 2020 November | 41 | 24 | 65 |
| 2020 October | 18 | 15 | 33 |
| 2020 September | 33 | 10 | 43 |
| 2020 August | 21 | 12 | 33 |
| 2020 July | 20 | 11 | 31 |
| 2020 June | 23 | 12 | 35 |
| 2020 May | 32 | 20 | 52 |
| 2020 April | 19 | 16 | 35 |
| 2020 March | 20 | 13 | 33 |
| 2020 February | 27 | 21 | 48 |
| 2020 January | 23 | 14 | 37 |
| 2019 December | 29 | 21 | 50 |
| 2019 November | 28 | 22 | 50 |
| 2019 October | 20 | 14 | 34 |
| 2019 September | 27 | 16 | 43 |
| 2019 August | 20 | 12 | 32 |
| 2019 July | 20 | 17 | 37 |
| 2019 June | 29 | 23 | 52 |
| 2019 May | 29 | 20 | 49 |
| 2019 April | 36 | 28 | 64 |
| 2019 March | 38 | 17 | 55 |
| 2019 February | 15 | 15 | 30 |
| 2019 January | 31 | 23 | 54 |
| 2018 December | 85 | 38 | 123 |
| 2018 November | 143 | 17 | 160 |
| 2018 October | 136 | 23 | 159 |
| 2018 September | 64 | 10 | 74 |
| 2018 August | 41 | 31 | 72 |
| 2018 July | 51 | 18 | 69 |
| 2018 June | 48 | 12 | 60 |
| 2018 May | 67 | 16 | 83 |
| 2018 April | 86 | 11 | 97 |
| 2018 March | 83 | 9 | 92 |
| 2018 February | 76 | 11 | 87 |
| 2018 January | 78 | 4 | 82 |
| 2017 December | 97 | 9 | 106 |
| 2017 November | 57 | 22 | 79 |
| 2017 October | 36 | 9 | 45 |
| 2017 September | 41 | 16 | 57 |
| 2017 August | 45 | 11 | 56 |
| 2017 July | 51 | 10 | 61 |
| 2017 June | 49 | 15 | 64 |
| 2017 May | 52 | 8 | 60 |
| 2017 April | 44 | 5 | 49 |
| 2017 March | 41 | 20 | 61 |
| 2017 February | 27 | 7 | 34 |
| 2017 January | 20 | 6 | 26 |
| 2016 December | 30 | 6 | 36 |
| 2016 November | 33 | 7 | 40 |
| 2016 October | 51 | 6 | 57 |
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