Dermatomyositis in a kidney transplant patient: a case report
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.pdf" "tienePdf" => true "PalabrasClave" => array:1 [ "en" => array:3 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec433356" "palabras" => array:1 [ 0 => "Immunosuppression" ] ] 1 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec433358" "palabras" => array:1 [ 0 => "Kidney transplant" ] ] 2 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec433360" "palabras" => array:1 [ 0 => "Dermatomyositis" ] ] ] ] "bibliografia" => array:2 [ "titulo" => "Bibliografía" "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. New approaches to the assessment and treatment of the idiopathic inflammatory myopathies. 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Nat Clin Pract Rheumatol 2007;3:172-80. <a href="http://www.ncbi.nlm.nih.gov/pubmed/17334340" target="_blank">[Pubmed]</a>" "contribucion" => array:1 [ 0 => null ] "host" => array:1 [ 0 => null ] ] ] ] 3 => array:3 [ "identificador" => "bib4" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:3 [ "referenciaCompleta" => "Miller FW. Inflammatory myopathies: polymyositis, dermatomyositis, and related conditions. In: Koopman W, Moreland L, eds. Arthritis and Allied Conditions: A Textbook of Rheumatology. 15th edition. Philadelphia, PA: Lippincott Williams and Wilkins; 2005. p. 1593-620." "contribucion" => array:1 [ 0 => null ] "host" => array:1 [ 0 => null ] ] ] ] 4 => array:3 [ "identificador" => "bib5" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:3 [ "referenciaCompleta" => "Ernste FC, Reed AM. Idiopathic inflammatory myopathies: current trends in pathogenesis, clinical features, and up-to-date treatment recommendations. Mayo Clin Proc 2013;88(1):83-105. <a href="http://www.ncbi.nlm.nih.gov/pubmed/23274022" target="_blank">[Pubmed]</a>" "contribucion" => array:1 [ 0 => null ] "host" => array:1 [ 0 => null ] ] ] ] 5 => array:3 [ "identificador" => "bib6" "etiqueta" => "6" "referencia" => array:1 [ 0 => array:3 [ "referenciaCompleta" => "Hoogendijk JE, Amato AA, Lecky BR, Choy EH, Lundberg IE, Rose MR, et al. 119th ENMC international workshop: trial design in adult idiopathic inflammatory myopathies, with the exception of inclusion body myositis, 10-12 October 2003, Naarden, The Netherlands. Neuromuscul Disord 2004;14(5):337-45." "contribucion" => array:1 [ 0 => null ] "host" => array:1 [ 0 => null ] ] ] ] 6 => array:3 [ "identificador" => "bib7" "etiqueta" => "7" "referencia" => array:1 [ 0 => array:3 [ "referenciaCompleta" => "Van der Pas J, Hengstman GJ, ter Laak HJ, Borm GF, van Engelen BG. Diagnostic value of MHC class I staining in idiopathic inflammatory myopathies. 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| 2021 Octubre | 73 | 46 | 119 |
| 2021 Septiembre | 70 | 44 | 114 |
| 2021 Agosto | 54 | 45 | 99 |
| 2021 Julio | 76 | 43 | 119 |
| 2021 Junio | 52 | 23 | 75 |
| 2021 Mayo | 86 | 41 | 127 |
| 2021 Abril | 211 | 56 | 267 |
| 2021 Marzo | 103 | 34 | 137 |
| 2021 Febrero | 108 | 19 | 127 |
| 2021 Enero | 68 | 34 | 102 |
| 2020 Diciembre | 88 | 19 | 107 |
| 2020 Noviembre | 67 | 27 | 94 |
| 2020 Octubre | 49 | 16 | 65 |
| 2020 Septiembre | 55 | 17 | 72 |
| 2020 Agosto | 44 | 16 | 60 |
| 2020 Julio | 52 | 8 | 60 |
| 2020 Junio | 50 | 18 | 68 |
| 2020 Mayo | 55 | 18 | 73 |
| 2020 Abril | 49 | 20 | 69 |
| 2020 Marzo | 51 | 14 | 65 |
| 2020 Febrero | 48 | 18 | 66 |
| 2020 Enero | 77 | 32 | 109 |
| 2019 Diciembre | 67 | 23 | 90 |
| 2019 Noviembre | 71 | 20 | 91 |
| 2019 Octubre | 45 | 7 | 52 |
| 2019 Septiembre | 59 | 34 | 93 |
| 2019 Agosto | 36 | 18 | 54 |
| 2019 Julio | 38 | 18 | 56 |
| 2019 Junio | 35 | 28 | 63 |
| 2019 Mayo | 41 | 27 | 68 |
| 2019 Abril | 102 | 50 | 152 |
| 2019 Marzo | 46 | 33 | 79 |
| 2019 Febrero | 39 | 20 | 59 |
| 2019 Enero | 37 | 26 | 63 |
| 2018 Diciembre | 118 | 56 | 174 |
| 2018 Noviembre | 144 | 35 | 179 |
| 2018 Octubre | 125 | 26 | 151 |
| 2018 Septiembre | 86 | 23 | 109 |
| 2018 Agosto | 61 | 27 | 88 |
| 2018 Julio | 59 | 22 | 81 |
| 2018 Junio | 71 | 18 | 89 |
| 2018 Mayo | 61 | 19 | 80 |
| 2018 Abril | 84 | 13 | 97 |
| 2018 Marzo | 80 | 16 | 96 |
| 2018 Febrero | 61 | 13 | 74 |
| 2018 Enero | 61 | 10 | 71 |
| 2017 Diciembre | 80 | 12 | 92 |
| 2017 Noviembre | 69 | 15 | 84 |
| 2017 Octubre | 42 | 10 | 52 |
| 2017 Septiembre | 85 | 16 | 101 |
| 2017 Agosto | 104 | 19 | 123 |
| 2017 Julio | 74 | 16 | 90 |
| 2017 Junio | 74 | 23 | 97 |
| 2017 Mayo | 79 | 24 | 103 |
| 2017 Abril | 68 | 14 | 82 |
| 2017 Marzo | 57 | 10 | 67 |
| 2017 Febrero | 99 | 11 | 110 |
| 2017 Enero | 60 | 13 | 73 |
| 2016 Diciembre | 82 | 6 | 88 |
| 2016 Noviembre | 122 | 18 | 140 |
| 2016 Octubre | 160 | 11 | 171 |
| 2016 Septiembre | 308 | 3 | 311 |
| 2016 Agosto | 312 | 7 | 319 |
| 2016 Julio | 282 | 14 | 296 |
| 2016 Junio | 170 | 0 | 170 |
| 2016 Mayo | 196 | 0 | 196 |
| 2016 Abril | 179 | 0 | 179 |
| 2016 Marzo | 128 | 0 | 128 |
| 2016 Febrero | 171 | 0 | 171 |
| 2016 Enero | 159 | 0 | 159 |
| 2015 Diciembre | 150 | 0 | 150 |
| 2015 Noviembre | 121 | 0 | 121 |
| 2015 Octubre | 162 | 0 | 162 |
| 2015 Septiembre | 117 | 0 | 117 |
| 2015 Agosto | 116 | 0 | 116 |
| 2015 Julio | 125 | 0 | 125 |
| 2015 Junio | 89 | 0 | 89 |
| 2015 Mayo | 118 | 0 | 118 |
| 2015 Abril | 30 | 0 | 30 |
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