Dermatomyositis in a kidney transplant patient: a case report
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His medical history was significant for monoclonal gammopathy of undetermined significance diagnosed five months earlier, with no signs of progression to overt myeloma, and subclinical hypothyroidism, medicated with levothyroxine and appropriately controlled. No history of recent trauma or unusual physical activity was identified. There was no personal or family history of known autoimmune or muscle diseases. Further chronic medication included azathioprine 50mg id, CsA 75mg bid, prednisolone 5mg id, furosemide 40mg id, omeprazole 20mg id and dipyridamole 75mg bid. He denied intake of other pharmaceutical drugs (particularly statins), illicit drugs or herbal therapies. Physical observation revealed unremarkable cardiac, pulmonary and abdominal examinations. Slight generalized erythroderma, periorbital oedema with heliotrope rash (Figure 1), psoriasiform changes of the scalp (Figure 2) and Gottron papules were evident upon close inspection. Neurological exam revealed generalized muscular atrophy and proximal muscle strength grade 4/5. Laboratory results at admission revealed normal-range haemoglobin, white blood cell count, platelets, blood urea nitrogen, creatinine, serum electrolytes, thyroid and parathyroid hormone studies, and albumin. Creatine phosphokinase (CPK) was increased at 1,319U/L (normal <171U/L), as was alanine transaminase (ALT) 63U/L (normal <45U/L), aspartate aminotransferase (AST) 123U/L (normal <35U/L), lactate dehydrogenase (LDH) 317U/L (normal 125-220U/L), aldolase 14.9U/L (normal <7.6U/L) and C-reactive protein 0.56mg/dL (normal <0.5mg/dL). Urinalysis was normal. Viral serologies were negative for hepatitis B surface antigen, hepatitis C antibody, <span class="elsevierStyleItalic">Human Immunodeficiency Virus</span> 1 and 2 antibodies, Epstein-Barr virus and cytomegalovirus antigenaemia. Azathioprine was discontinued due to presumed drug-induced hepatotoxicity, while CsA and prednisolone were continued. Persistently elevated CPK (>1,000U/L) and other muscle enzymes, progressively worsening clinical symptoms after five days of optimal hydration status and diuresis, and onset of dysphagia and dysphonia directed an investigation for inflammatory myopathies. Complement levels were normal and autoimmunity testing revealed positive antinuclear antibodies (+), normal anti-double-stranded DNA antibodies, positive anti-cytoskeleton fibres antibodies (+++) and positive anti-vimentin antibodies (+++), with negative myositis-associated antibodies (Jo-1, Mi-2, Ku, PM-Scl 100, SRP, PL-7, PL-12, EJ, OJ and SSA52). Electromyography displayed evidence of muscle fibre lesion in the proximal muscles of the lower limbs, characterised by normal insertional and spontaneous activity, but presence of polyphasic motor unit action potentials of short duration and low amplitude. Skin biopsy was compatible with dermatomyositis. Muscle biopsy revealed no mononuclear inflammatory infiltrates, but intense and diffuse immunoexpression of Major Histocompatibility Complex (MHC) class I in the sarcolemma of studied tissue fibres. Studies undertaken for secondary causes were negative and there was no evidence of interstitial lung disease on respiratory function tests. Oral prednisolone was increased to 1mg/kg/day and he was started on daily physiotherapy. An optimal clinical and laboratorial response (at discharge: CPK 75U/L, ALT 36U/L, AST 27U/L, LDH 226U/L, aldolase 7.8U/L) permitted discharge after 51 days of hospitalization. He is currently on prednisolone 5mg (weaned over nine months) and developed steroid-induced diabetes mellitus with need for insulin therapy. His muscle strength is much improved, with no evident skin alterations, dysphagia or dysphonia.</p><p class="elsevierStylePara">A common set of criteria for the diagnosis of dermatomyositis and polymyositis was proposed by Bohan and Peter in 1975, where four of five conditions are necessary for a definitive diagnosis: (i) characteristic skin findings, (ii) proximal muscle weakness, (iii) elevated muscle enzyme levels, (iv) abnormal electromyography, and (v) abnormal findings on muscle biopsy.<span class="elsevierStyleSup">5</span> In our patient, all criteria were fulfilled to diagnose dermatomyositis. Regarding the classification scheme proposed by the 119th European Neuromuscular Centre international workshop,<span class="elsevierStyleSup">6</span> our patient falls in the probable dermatomyositis category by not fulfilling the perifascicular atrophy criterion on muscle biopsy. Classic histological findings, especially identification of mononuclear cellular infiltrates, remain the basis of the diagnosis, however these can be absent even in the presence of disease. MHC class I expression on the sarcolemma has been proven to be upregulated in IMM and is a valid test for the diagnosis of these myopathies since they are not affected by potential sampling errors.<span class="elsevierStyleSup">7</span> Common myositis-specific autoantibodies present with a frequency ranging from 1-30% and were negative in this case, but are not required for definitive diagnosis of inflammatory myopathies. There is an increased risk for malignancies,<span class="elsevierStyleSup">5</span> and age-appropriate cancer screening tests should be performed. Major therapeutic aims include elimination of muscle inflammation, restoration of muscle strength and prevention of chronic muscle disease in order to reduce morbidity and improve quality of life.<span class="elsevierStyleSup">5</span> Immunosuppressive therapy entails corticosteroids at a dose of 1mg/kg/day.<span class="elsevierStyleSup">5,8</span> Concurrent administration of methotrexate, azathioprine or mycophenolate mofetil may be started,<span class="elsevierStyleSup">5</span> but may be reserved for patients who fail treatment with glucocorticoids alone. In retrospect, we believe that the patient´s worsening symptoms during the earlier stages of hospitalization were actually due to the discontinuation of azathioprine, which was probably responsible for ameliorating the underlying condition. Due to the improved clinical and biochemical status with the increased dosage of prednisolone and physiotherapy, in conjunction with presumed azathioprine-induced liver toxicity, we selected not to resume azathioprine, and the patient currently remains asymptomatic.</p><p class="elsevierStylePara">This case reflects an interesting case of dermatomyositis, since the concept of an inflammatory or autoimmune myopathy seems difficult to fathom in a patient on regular immunosuppressive therapy for the past 20 years, particularly with a regimen including corticosteroids, CsA and azathioprine.</p><p class="elsevierStylePara">Long-term follow-up data on patients with IMM are scarce. Studies on the pathophysiology of IMM in patients on chronic immunosuppressive regimens are warranted.</p><p class="elsevierStylePara"> </p><p class="elsevierStylePara"><span class="elsevierStyleBold">Conflict of interest</span></p><p class="elsevierStylePara"> </p><p class="elsevierStylePara">The authors declare that there is no conflict of interest associated with this manuscript.</p><p class="elsevierStylePara"><a href="grande/12278_16025_50710_en_f112278.jpg" class="elsevierStyleCrossRefs"><img src="12278_16025_50710_en_f112278.jpg" alt="Heliotrope rash."></img></a></p><p class="elsevierStylePara">Figure 1. Heliotrope rash.</p><p class="elsevierStylePara"><a href="grande/12278_16025_50711_en_f212278.jpg" class="elsevierStyleCrossRefs"><img src="12278_16025_50711_en_f212278.jpg" alt="Psoriasiform scalp."></img></a></p><p class="elsevierStylePara">Figure 2. Psoriasiform scalp.</p>" "pdfFichero" => "P1-E565-S4505-A12278-EN.pdf" "tienePdf" => true "PalabrasClave" => array:1 [ "en" => array:3 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec439308" "palabras" => array:1 [ 0 => "Immunosuppression" ] ] 1 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec439310" "palabras" => array:1 [ 0 => "Kidney transplant" ] ] 2 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec439312" "palabras" => array:1 [ 0 => "Dermatomyositis" ] ] ] ] "multimedia" => array:2 [ 0 => array:8 [ "identificador" => "fig1" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "copyright" => "Elsevier España" "figura" => array:1 [ 0 => array:4 [ "imagen" => "12278_16025_50710_en_f112278.jpg" "Alto" => 611 "Ancho" => 1370 "Tamanyo" => 561992 ] ] "descripcion" => array:1 [ "en" => "Heliotrope rash." ] ] 1 => array:8 [ "identificador" => "fig2" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "copyright" => "Elsevier España" "figura" => array:1 [ 0 => array:4 [ "imagen" => "12278_16025_50711_en_f212278.jpg" "Alto" => 733 "Ancho" => 1388 "Tamanyo" => 499108 ] ] "descripcion" => array:1 [ "en" => "Psoriasiform scalp." ] ] ] "bibliografia" => array:2 [ "titulo" => "Bibliography" "seccion" => array:1 [ 0 => array:1 [ "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib1" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:3 [ "referenciaCompleta" => "Miller FW. 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| 2023 March | 64 | 23 | 87 |
| 2023 February | 35 | 21 | 56 |
| 2023 January | 40 | 19 | 59 |
| 2022 December | 68 | 31 | 99 |
| 2022 November | 38 | 33 | 71 |
| 2022 October | 51 | 38 | 89 |
| 2022 September | 50 | 29 | 79 |
| 2022 August | 54 | 35 | 89 |
| 2022 July | 45 | 46 | 91 |
| 2022 June | 57 | 34 | 91 |
| 2022 May | 35 | 41 | 76 |
| 2022 April | 49 | 37 | 86 |
| 2022 March | 74 | 38 | 112 |
| 2022 February | 73 | 47 | 120 |
| 2022 January | 70 | 28 | 98 |
| 2021 December | 68 | 30 | 98 |
| 2021 November | 34 | 25 | 59 |
| 2021 October | 52 | 39 | 91 |
| 2021 September | 36 | 38 | 74 |
| 2021 August | 49 | 30 | 79 |
| 2021 July | 60 | 28 | 88 |
| 2021 June | 35 | 27 | 62 |
| 2021 May | 36 | 34 | 70 |
| 2021 April | 108 | 50 | 158 |
| 2021 March | 67 | 32 | 99 |
| 2021 February | 50 | 12 | 62 |
| 2021 January | 38 | 19 | 57 |
| 2020 December | 49 | 15 | 64 |
| 2020 November | 42 | 13 | 55 |
| 2020 October | 28 | 18 | 46 |
| 2020 September | 36 | 12 | 48 |
| 2020 August | 51 | 10 | 61 |
| 2020 July | 49 | 10 | 59 |
| 2020 June | 23 | 17 | 40 |
| 2020 May | 43 | 11 | 54 |
| 2020 April | 37 | 13 | 50 |
| 2020 March | 37 | 11 | 48 |
| 2020 February | 34 | 14 | 48 |
| 2020 January | 51 | 25 | 76 |
| 2019 December | 48 | 35 | 83 |
| 2019 November | 52 | 20 | 72 |
| 2019 October | 19 | 15 | 34 |
| 2019 September | 27 | 18 | 45 |
| 2019 August | 21 | 14 | 35 |
| 2019 July | 40 | 21 | 61 |
| 2019 June | 30 | 14 | 44 |
| 2019 May | 49 | 18 | 67 |
| 2019 April | 81 | 23 | 104 |
| 2019 March | 34 | 22 | 56 |
| 2019 February | 35 | 12 | 47 |
| 2019 January | 41 | 19 | 60 |
| 2018 December | 133 | 36 | 169 |
| 2018 November | 114 | 10 | 124 |
| 2018 October | 120 | 19 | 139 |
| 2018 September | 65 | 8 | 73 |
| 2018 August | 70 | 11 | 81 |
| 2018 July | 52 | 13 | 65 |
| 2018 June | 61 | 13 | 74 |
| 2018 May | 73 | 22 | 95 |
| 2018 April | 69 | 7 | 76 |
| 2018 March | 58 | 12 | 70 |
| 2018 February | 59 | 10 | 69 |
| 2018 January | 60 | 6 | 66 |
| 2017 December | 76 | 7 | 83 |
| 2017 November | 57 | 12 | 69 |
| 2017 October | 33 | 6 | 39 |
| 2017 September | 57 | 11 | 68 |
| 2017 August | 81 | 9 | 90 |
| 2017 July | 74 | 2 | 76 |
| 2017 June | 81 | 11 | 92 |
| 2017 May | 73 | 13 | 86 |
| 2017 April | 65 | 7 | 72 |
| 2017 March | 68 | 3 | 71 |
| 2017 February | 51 | 9 | 60 |
| 2017 January | 27 | 7 | 34 |
| 2016 December | 68 | 6 | 74 |
| 2016 November | 87 | 5 | 92 |
| 2016 October | 122 | 10 | 132 |
| 2016 September | 138 | 2 | 140 |
| 2016 August | 177 | 3 | 180 |
| 2016 July | 177 | 3 | 180 |
| 2016 June | 118 | 0 | 118 |
| 2016 May | 153 | 0 | 153 |
| 2016 April | 105 | 0 | 105 |
| 2016 March | 97 | 0 | 97 |
| 2016 February | 98 | 0 | 98 |
| 2016 January | 111 | 0 | 111 |
| 2015 December | 105 | 0 | 105 |
| 2015 November | 89 | 0 | 89 |
| 2015 October | 91 | 0 | 91 |
| 2015 September | 83 | 0 | 83 |
| 2015 August | 70 | 0 | 70 |
| 2015 July | 72 | 0 | 72 |
| 2015 June | 42 | 0 | 42 |
| 2015 May | 49 | 0 | 49 |
| 2015 April | 13 | 0 | 13 |
Show all
