Tubulointerstitial nephritis and sclerosing cholangitis associated with autoimmune pancreatitis
Nefritis tubulointersticial y colangitis esclerosante asociadas a pancreatitis autoinmune
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Nieto-Ríos, Gustavo A. Zuluaga-Valencia" "autores" => array:5 [ 0 => array:4 [ "nombre" => "Arbey" "apellidos" => "Aristizabal-Alzate" "email" => array:1 [ 0 => "arbeyaristi@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] 1 => array:3 [ "nombre" => "Carolina" "apellidos" => "Muñoz-Grajales" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] 2 => array:3 [ "nombre" => "Catalina" "apellidos" => "Ocampo-Kohn" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] 3 => array:3 [ "nombre" => "John F." "apellidos" => "Nieto-Ríos" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] 4 => array:3 [ "nombre" => "Gustavo A." "apellidos" => "Zuluaga-Valencia" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] ] "afiliaciones" => array:1 [ 0 => array:3 [ "entidad" => "Sección de Nefrología, Hospital Pablo Tobón Uribe, Medellín, Antioquia, Colombia, " "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "affa" ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Nefritis tubulointersticial y colangitis esclerosante asociadas a pancreatitis autoinmune" ] ] "textoCompleto" => "<p class="elsevierStylePara"><span class="elsevierStyleBold">To the Editor</span>:<span class="elsevierStyleBold"> </span></p><p class="elsevierStylePara">Autoimmune pancreatitis (AIP) is a form of chronic pancreatitis caused by an autoimmune inflammatory process with lymphocyte infiltration and fibrosis that lead to organ dysfunction,1 related to high levels of IgG4 and anti-carbonic anhydrase II antibodies.2,3 This disease frequently produces extra-pancreatic manifestations as well, such as sclerosing cholangitis and tubulointerstitial nephritis.4</p><p class="elsevierStylePara">Sclerosing cholangitis associated with AIP produces imaging test results and a clinical presentation similar to that of primary sclerosing cholangitis (PSC), but has a dramatic response to steroid treatment.5</p><p class="elsevierStylePara">Here, we describe the case of a patient with repeated episodes of pancreatitis and cholangitis who was managed as a case of PSC with no response, and who developed tubulointerstitial nephritis with renal biopsy findings suggestive of an autoimmune process, with resolution of gastrointestinal and renal manifestations through the administration of steroids.</p><p class="elsevierStylePara"> </p><p class="elsevierStylePara"><span class="elsevierStyleBold">CASE REPORT</span></p><p class="elsevierStylePara"> </p><p class="elsevierStylePara">Our patient was a 37-year old male who sought treatment in March 2006 for jaundice, fever, and abdominal pain; we first suspected an episode of cholangitis, but an endoscopic retrograde cholangiopancreatography and p-ANCA tests due to suspected PSC were negative, leading to the suspicion of microlithiasis.</p><p class="elsevierStylePara">In May of 2006, we performed an endoscopic sphincterotomy. Eight days later, the patient showed another episode of cholangitis. We considered the possibility of acalculous gallbladder disease as the cause for the recurring cholangitis; a cholecystokinin scintigraphy was compatible with this diagnosis, and we performed a laparoscopic cholecystectomy, but 15 days later the patient returned with yet another episode of cholangitis.</p><p class="elsevierStylePara">We returned to the suspected diagnosis of PSC, and performed a liver biopsy that revealed acute cholangitis with minimal foci of fibrosis. In early 2007, we administered a magnetic resonance cholangiography that revealed constrictions that were compatible with the diagnosis of PSC, with no possibility of performing a surgical intervention.</p><p class="elsevierStylePara">We managed the patient as a case of PSC, administering ursodeoxycholic acid and low doses of antibiotics (ciprofloxacin), and yet the patient continued to suffer repeated episodes of cholangitis.</p><p class="elsevierStylePara">In October 2007, the patient sought treatment for fever and abdominal pain; we started treatment with ciprofloxacin and requested an abdominal contrast tomography based on the patient’s creatinine value of 8.7mg/dl. In May 2007, the patient’s creatinine value was 1.2mg/dl.</p><p class="elsevierStylePara">The patient was evaluated in nephrology, and the only finding was paleness.</p><p class="elsevierStylePara">Laboratory analyses revealed creatinine: 7.6mg/dl, blood urea nitrogen (BUN): 46, normal sodium and potassium levels, pH: 7.32, bicarbonate: 16, Hb: 9.7g/dl, urinalysis with glycosuria (50mg/dl) and no hyperglycaemia.</p><p class="elsevierStylePara">A renal ultrasound revealed normally sized kidneys with increased bilateral echogenicity.</p><p class="elsevierStylePara">The patient was diagnosed with acute renal failure secondary to tubulointerstitial nephritis due to the consumption of quinolones.</p><p class="elsevierStylePara">After antibiotic treatment was removed and the patient was hydrated on the following day, creatinine decreased to 5.5mg/dl and BUN to 36mg/dl. Serum complement was normal, anti-nuclear antibodies (ANA) and serological tests for syphilis (VDRL) and human immunodeficiency virus (HIB) were negative; 24-hour proteinuria was 580mg. The patient was discharged with a creatinine value of 2.2mg/dl.</p><p class="elsevierStylePara">Twenty days later, the patient returned again for treatment for fever, diarrhoea, and oedema. Upon hospitalisation the patient had a creatinine value of 15mg/dl, potassium at 5.8mEq/l, and urine cytochemistry revealed leukocyturia, proteinuria (25mg/dl), glycosuria (50mg/dl), and haematuria (erythrocytes: 6 per field). A physical examination revealed no pathological findings. We considered this to be an exacerbation of the previous case of renal failure; due to the suspicion of tubulointerstitial nephritis, we started treatment with prednisone and took a renal biopsy.</p><p class="elsevierStylePara">The renal biopsy revealed: acute tubulointerstitial nephritis; immunofluorescence revealed: IgG ++ (interstitial), IgA and IgM +++ (interstitial), k and lambda chains: absent, C3: +++ peripheral, M and Bowman’s capsule, and absence of C1q. We interpreted these findings as histological changes corresponding to acute tubulointerstitial nephritis due to hypersensitivity to medications as opposed to autoimmune.</p><p class="elsevierStylePara">We continued to treat the patient with steroids, and upon discharge, creatinine was at 3.6mg/dl.</p><p class="elsevierStylePara">Since starting the patient on steroids, no further episodes of cholangitis or pancreatitis were produced, which led to the diagnosis of AIP with sclerosing cholangitis and tubulointerstitial nephritis as extra-pancreatic complications.</p><p class="elsevierStylePara">We did not measure IgG4 levels, since the patient had already received steroid treatment.</p><p class="elsevierStylePara">The first outpatient follow-up consultation revealed creatinine at 1.6mg/dl. The steroid treatment was progressively decreased, and the patient is currently on a regimen of 5mg prednisone every other day indefinitely. Follow-up measurements of creatinine revealed values ranging from 1.4mg/dl-1.7mg/dl.</p><p class="elsevierStylePara">The last follow-up session in December 2010 showed the patient’s creatinine value to be 1.43mg/dl, and no new episodes of cholangitis or pancreatitis have occurred.</p><p class="elsevierStylePara"> </p><p class="elsevierStylePara"><span class="elsevierStyleBold">DISCUSSION</span></p><p class="elsevierStylePara"> </p><p class="elsevierStylePara">In 1961, Starles made the first description of chronic pancreatitis with autoimmune manifestations; later, in 1995, this form of pancreatitis was labelled by Yoshida as “autoimmune pancreatitits”.6</p><p class="elsevierStylePara">Although the number of reports of this disease has recently increased, and some studies report a prevalence of 5% among patients with chronic pancreatitis, the true incidence of AIP remains unknown.7</p><p class="elsevierStylePara">AIP is frequently associated with rheumatoid arthritis, Sjögren’s syndrome, and inflammatory bowel disease; it is also common to encounter hypergammaglobulinaemia and elevated IgG4 levels, anti-carbonic anhydrase II, and lactoferrin auto-antibodies, which suggests an autoimmune disease, although its pathogenesis is still unkown.1-3</p><p class="elsevierStylePara">The extra-pancreatic autoimmune manifestations of AIP include sclerosing sialadenitis, retroperitoneal fibrosis, interstitial pneumonitis, sclerosing cholangitis, and tubulointerstitial nephritis.4,8</p><p class="elsevierStylePara">As regards sclerosing cholangitis associated with this type of pancreatitis, diagnosis is hindered by the fact that this condition shares many imaging test, cholangiography, and clinical findings with PSC; several authors have reported that the appearance of this disease in patients older than 60 years of age, with elevated levels of IgG4, and a dramatic response to steroid treatment all favour the diagnosis of AIP-associated sclerosing cholangitis as opposed to PSC.5</p><p class="elsevierStylePara">Various diagnostic criteria have been suggested for AIP; these include the revised Japan criteria, which place special emphasis on imaging test findings, the Mayo Clinic criteria (HISORT), which involve the use of histological, imaging test, and serology findings, as well as the manifestations in other organs and response to steroids, and the Italian criteria, which give greater importance to histological findings; however, no unified international consensus exists regarding which diagnostic criteria to use.9</p><p class="elsevierStylePara">As yet, few cases of tubulointerstitial nephritis have been reported in association with AIP; generally, these are observed in male patients older than 50 years of age, with mononuclear cell infiltrates that are positive for IgG4 in the renal interstitium, and with clinical and serological (decreased IgG4 levels) evidence of response to steroid treatment.8,10</p><p class="elsevierStylePara">Despite the lack of information regarding IgG4 levels in our patient, there were elements to suggest sclerosing cholangitis and tubulointerstitial nephritis associated with AIP: frequent episodes of cholangitis with magnetic resonance cholangiography findings that initially produced the suspicion of PSC, but the patient showed little response to normal treatment and had no auto-antibodies (p-ANCA, ANA), which are present in 85% of all patients with this pathology; this was supported by the repeated episodes of pancreatitis with evidence of diffuse growth of the pancreas, and resolution of symptoms through treatment with steroids.</p><p class="elsevierStylePara">The renal biopsy findings, specifically those from immunofluorescence testing, favoured the diagnosis of an autoimmune mechanism responsible for producing the tubulointerstitial nephritis observed in this patient.</p><p class="elsevierStylePara"><span class="elsevierStyleBold"> </span></p><p class="elsevierStylePara"><span class="elsevierStyleBold">Conflicts of interest</span></p><p class="elsevierStylePara"> </p><p class="elsevierStylePara">The authors state that they have no potential conflicts of interest related to the content of this article.</p><p class="elsevierStylePara"> </p>" "pdfFichero" => "P1-E544-S3783-A11185-EN.pdf" "tienePdf" => true "bibliografia" => array:2 [ "titulo" => "Bibliography" "seccion" => array:1 [ 0 => array:1 [ "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib1" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:3 [ "referenciaCompleta" => "Finkelberg DL, Sahani D, Deshpande V, Brugge WR. 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Am J Kidney Dis 2007;50(3):455-62. <a href="http://www.ncbi.nlm.nih.gov/pubmed/17720525" target="_blank">[Pubmed]</a>" "contribucion" => array:1 [ 0 => null ] "host" => array:1 [ 0 => null ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/20132514/0000003200000006/v0_201502091602/X2013251412002084/v0_201502091603/en/main.assets" "Apartado" => array:4 [ "identificador" => "35437" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor - Brief Case Reports" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/20132514/0000003200000006/v0_201502091602/X2013251412002084/v0_201502091603/en/P1-E544-S3783-A11185-EN.pdf?idApp=UINPBA000064&text.app=https://revistanefrologia.com/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/X2013251412002084?idApp=UINPBA000064" ]
| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 6 | 4 | 10 |
| 2024 October | 49 | 44 | 93 |
| 2024 September | 45 | 27 | 72 |
| 2024 August | 70 | 54 | 124 |
| 2024 July | 63 | 30 | 93 |
| 2024 June | 63 | 48 | 111 |
| 2024 May | 58 | 34 | 92 |
| 2024 April | 47 | 30 | 77 |
| 2024 March | 36 | 18 | 54 |
| 2024 February | 39 | 36 | 75 |
| 2024 January | 26 | 27 | 53 |
| 2023 December | 23 | 21 | 44 |
| 2023 November | 32 | 26 | 58 |
| 2023 October | 29 | 25 | 54 |
| 2023 September | 30 | 38 | 68 |
| 2023 August | 46 | 12 | 58 |
| 2023 July | 39 | 29 | 68 |
| 2023 June | 44 | 17 | 61 |
| 2023 May | 42 | 32 | 74 |
| 2023 April | 28 | 17 | 45 |
| 2023 March | 29 | 24 | 53 |
| 2023 February | 24 | 24 | 48 |
| 2023 January | 29 | 20 | 49 |
| 2022 December | 44 | 21 | 65 |
| 2022 November | 36 | 24 | 60 |
| 2022 October | 39 | 36 | 75 |
| 2022 September | 48 | 27 | 75 |
| 2022 August | 39 | 45 | 84 |
| 2022 July | 46 | 41 | 87 |
| 2022 June | 30 | 25 | 55 |
| 2022 May | 28 | 35 | 63 |
| 2022 April | 46 | 40 | 86 |
| 2022 March | 45 | 45 | 90 |
| 2022 February | 56 | 45 | 101 |
| 2022 January | 36 | 25 | 61 |
| 2021 December | 44 | 41 | 85 |
| 2021 November | 40 | 35 | 75 |
| 2021 October | 33 | 34 | 67 |
| 2021 September | 36 | 35 | 71 |
| 2021 August | 36 | 36 | 72 |
| 2021 July | 28 | 30 | 58 |
| 2021 June | 26 | 21 | 47 |
| 2021 May | 31 | 22 | 53 |
| 2021 April | 54 | 41 | 95 |
| 2021 March | 53 | 24 | 77 |
| 2021 February | 26 | 19 | 45 |
| 2021 January | 28 | 18 | 46 |
| 2020 December | 18 | 14 | 32 |
| 2020 November | 21 | 16 | 37 |
| 2020 October | 21 | 24 | 45 |
| 2020 September | 32 | 8 | 40 |
| 2020 August | 19 | 11 | 30 |
| 2020 July | 18 | 7 | 25 |
| 2020 June | 23 | 16 | 39 |
| 2020 May | 19 | 11 | 30 |
| 2020 April | 29 | 14 | 43 |
| 2020 March | 18 | 8 | 26 |
| 2020 February | 27 | 20 | 47 |
| 2020 January | 42 | 17 | 59 |
| 2019 December | 30 | 22 | 52 |
| 2019 November | 31 | 20 | 51 |
| 2019 October | 22 | 9 | 31 |
| 2019 September | 20 | 21 | 41 |
| 2019 August | 20 | 12 | 32 |
| 2019 July | 32 | 19 | 51 |
| 2019 June | 29 | 8 | 37 |
| 2019 May | 24 | 8 | 32 |
| 2019 April | 58 | 25 | 83 |
| 2019 March | 40 | 20 | 60 |
| 2019 February | 31 | 25 | 56 |
| 2019 January | 33 | 15 | 48 |
| 2018 December | 64 | 31 | 95 |
| 2018 November | 61 | 11 | 72 |
| 2018 October | 56 | 13 | 69 |
| 2018 September | 64 | 13 | 77 |
| 2018 August | 39 | 14 | 53 |
| 2018 July | 43 | 6 | 49 |
| 2018 June | 38 | 12 | 50 |
| 2018 May | 47 | 19 | 66 |
| 2018 April | 51 | 6 | 57 |
| 2018 March | 33 | 12 | 45 |
| 2018 February | 35 | 4 | 39 |
| 2018 January | 28 | 10 | 38 |
| 2017 December | 47 | 13 | 60 |
| 2017 November | 37 | 12 | 49 |
| 2017 October | 42 | 8 | 50 |
| 2017 September | 40 | 8 | 48 |
| 2017 August | 44 | 9 | 53 |
| 2017 July | 44 | 14 | 58 |
| 2017 June | 30 | 12 | 42 |
| 2017 May | 50 | 6 | 56 |
| 2017 April | 42 | 6 | 48 |
| 2017 March | 23 | 2 | 25 |
| 2017 February | 27 | 5 | 32 |
| 2017 January | 21 | 7 | 28 |
| 2016 December | 49 | 5 | 54 |
| 2016 November | 62 | 5 | 67 |
| 2016 October | 87 | 4 | 91 |
| 2016 September | 95 | 3 | 98 |
| 2016 August | 158 | 3 | 161 |
| 2016 July | 171 | 9 | 180 |
| 2016 June | 110 | 0 | 110 |
| 2016 May | 110 | 0 | 110 |
| 2016 April | 79 | 0 | 79 |
| 2016 March | 84 | 0 | 84 |
| 2016 February | 86 | 0 | 86 |
| 2016 January | 83 | 0 | 83 |
| 2015 December | 121 | 0 | 121 |
| 2015 November | 82 | 0 | 82 |
| 2015 October | 66 | 0 | 66 |
| 2015 September | 74 | 0 | 74 |
| 2015 August | 79 | 0 | 79 |
| 2015 July | 72 | 0 | 72 |
| 2015 June | 46 | 0 | 46 |
| 2015 May | 47 | 0 | 47 |
| 2015 April | 5 | 0 | 5 |
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