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A case of type 2 autoimmune pancreatitis with spontaneous remission

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Abstract

A 70-year-old man with epigastric pain was referred to our hospital. Computed tomography and magnetic resonance imaging showed the diffusely enlarged pancreas compared to his normal pancreas 6 months prior to presentation. Serum levels of IgG4 and amylase were normal, while C-reactive protein was slightly elevated. Endoscopic ultrasound-guided fine-needle biopsy of the pancreas revealed acinar–ductal metaplasia with neutrophil infiltration and without infiltration of IgG4-positive plasma cells. After the clinical diagnosis of type 2 autoimmune pancreatitis (AIP), his symptoms spontaneously improved without steroid therapy. Three months later, radiological findings showed improved pancreas size and serological findings. The pathological diagnosis of type 2 AIP using endoscopic ultrasound-guided fine-needle biopsy is challenging, particularly for proving granulocyte epithelial lesions. This was a valuable type 2 AIP case in which the images before, at the time of onset, and at the time of spontaneous remission were evaluated.

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Abbreviations

AIP:

Autoimmune pancreatitis

ICDC:

International Consensus Diagnostic Criteria

LPSP:

Lymphoplasmacytic sclerosing pancreatitis

GELs:

Granulocyte epithelial lesions

IBD:

Inflammatory bowel disease

IPMN:

Intraductal papillary mucinous neoplasm

MRCP:

Magnetic resonance cholangiopancreatography

CeCT:

Contrast-enhanced computed tomography

MRI:

Magnetic resonance imaging

EUS-FNB:

Endoscopic ultrasound-guided fine-needle biopsy

EUS-FNA:

Endoscopic ultrasound-guided fine-needle aspiration

ADM:

Acinar–ductal metaplasia

References

  1. Shimosegawa T, Chari ST, Frulloni L, et al. International consensus diagnostic criteria for autoimmune pancreatitis: guidelines of the International Association of Pancreatology. Pancreas. 2011;40:352–8.

    Article  PubMed  Google Scholar 

  2. Yoshida K, Toki F, Takeuchi T, et al. Chronic pancreatitis caused by an autoimmune abnormality. Proposal of the concept of autoimmune pancreatitis. Dig Dis Sci. 1995;40:1561–8.

    Article  CAS  PubMed  Google Scholar 

  3. Kawaguchi K, Koike M, Tsuruta K, et al. Lymphoplasmacytic sclerosing pancreatitis with cholangitis: a variant of primary sclerosing cholangitis extensively involving pancreas. Hum Pathol. 1991;22:387–95.

    Article  CAS  PubMed  Google Scholar 

  4. Zamboni G, Lüttges J, Capelli P, et al. Histopathological features of diagnostic and clinical relevance in autoimmune pancreatitis: a study on 53 resection specimens and 9 biopsy specimens. Virchows Arch. 2004;445:552–63.

    Article  PubMed  Google Scholar 

  5. Notohara K, Burgart LJ, Yadav D, et al. Idiopathic chronic pancreatitis with periductal lymphoplasmacytic infiltration: clinicopathologic features of 35 cases. Am J Surg Pathol. 2003;27:1119–27.

    Article  PubMed  Google Scholar 

  6. Hamano H, Kawa S, Horiuchi A, et al. High serum IgG4 concentrations in patients with sclerosing pancreatitis. N Engl J Med. 2001;344:732–8.

    Article  CAS  PubMed  Google Scholar 

  7. Umehara H, Okazaki K, Masaki Y, et al. A novel clinical entity, IgG4-related disease (IgG4RD): general concept and details. Mod Rheumatol. 2012;22:1–14.

    Article  CAS  PubMed  Google Scholar 

  8. Stone JH, Khosroshahi A, Deshpande V, et al. Recommendations for the nomenclature of IgG4-related disease and its individual organ system manifestations. Arthr Rheum. 2012;64:3061–7.

    Article  CAS  Google Scholar 

  9. Deshpande V, Zen Y, Chan JK, et al. Consensus statement on the pathology of IgG4-related disease. Mod Pathol. 2012;25:1181–92.

    Article  PubMed  Google Scholar 

  10. Chari ST, Kloeppel G, Zhang L, et al. Histopathologic and clinical subtypes of autoimmune pancreatitis: the Honolulu consensus document. Pancreas. 2010;39:549–54.

    Article  CAS  PubMed  Google Scholar 

  11. Levy MJ, Reddy RP, Wiersema MJ, et al. EUS-guided trucut biopsy in establishing autoimmune pancreatitis as the cause of obstructive jaundice. Gastrointest Endosc. 2005;61:467–72.

    Article  PubMed  Google Scholar 

  12. Mizuno N, Bhatia V, Hosoda W, et al. Histological diagnosis of autoimmune pancreatitis using EUS-guided trucut biopsy: a comparison study with EUS-FNA. J Gastroenterol. 2009;44:742–50.

    Article  CAS  PubMed  Google Scholar 

  13. Banks PA, Bollen TL, Dervenis C, et al. Classification of acute pancreatitis–2012: revision of the Atlanta classification and definitions by international consensus. Gut. 2013;62:102–11.

    Article  PubMed  Google Scholar 

  14. Imai K, Matsubayashi H, Fukutomi A, et al. Endoscopic ultrasonography-guided fine needle aspiration biopsy using 22-gauge needle in diagnosis of autoimmune pancreatitis. Dig Liver Dis. 2011;43:869–74.

    Article  PubMed  Google Scholar 

  15. Sah RP, Chari ST, Pannala R, et al. Differences in clinical profile and relapse rate of type 1 versus type 2 autoimmune pancreatitis. Gastroenterology. 2010;139:140–8 (quiz e12-3).

    Article  PubMed  Google Scholar 

  16. Kurita A, Yasukawa S, Zen Y, et al. Comparison of a 22-gauge Franseen-tip needle with a 20-gauge forward-bevel needle for the diagnosis of type 1 autoimmune pancreatitis: a prospective, randomized, controlled, multicenter study (COMPAS study). Gastrointest Endosc. 2020;91:373-81.e2.

    Article  PubMed  Google Scholar 

  17. Ishikawa T, Kawashima H, Ohno E, et al. Usefulness of endoscopic ultrasound-guided fine-needle biopsy for the diagnosis of autoimmune pancreatitis using a 22-gauge Franseen needle: a prospective multicenter study. Endoscopy. 2020;52:978–85.

    Article  PubMed  Google Scholar 

  18. Notohara K, Kamisawa T, Fukushima N, et al. Guidance for diagnosing autoimmune pancreatitis with biopsy tissues. Pathol Int. 2020;70:699–711.

    Article  PubMed  Google Scholar 

  19. Detlefsen S, Joergensen MT, Mortensen MB. Microscopic findings in EUS-guided fine needle (SharkCore) biopsies with type 1 and type 2 autoimmune pancreatitis. Pathol Int. 2017;67:514–20.

    Article  PubMed  Google Scholar 

  20. Matsumoto Y, Shimizu A, Ogawa K, et al. Late-onset type-2 autoimmune pancreatitis with two mass lesions diagnosed by endoscopic ultrasound-guided fine-needle aspiration. Clin J Gastroenterol. 2021;14:899–904.

    Article  PubMed  Google Scholar 

  21. Ollo D, Terraz S, Arnoux G, et al. Biliary involvement in type 2 autoimmune pancreatitis. Case Rep Gastroenterol. 2019;13:200–6.

    Article  PubMed  PubMed Central  Google Scholar 

  22. Yoon SB, Moon SH, Song TJ, et al. Endoscopic ultrasound-guided fine needle aspiration versus biopsy for diagnosis of autoimmune pancreatitis: systematic review and comparative meta-analysis. Dig Endosc. 2021;33:1024–33.

    Article  PubMed  Google Scholar 

  23. Hayashi H, Miura S, Fujishima F, et al. Utility of endoscopic ultrasound-guided fine-needle aspiration and biopsy for histological diagnosis of type 2 autoimmune pancreatitis. Diagnostics. 2022;12:2464.

    Article  PubMed  PubMed Central  Google Scholar 

  24. Hart PA, Levy MJ, Smyrk TC, et al. Clinical profiles and outcomes in idiopathic duct-centric chronic pancreatitis (type 2 autoimmune pancreatitis): the Mayo Clinic experience. Gut. 2016;65:1702–9.

    Article  CAS  PubMed  Google Scholar 

  25. Maire F, Le Baleur Y, Rebours V, et al. Outcome of patients with type 1 or 2 autoimmune pancreatitis. Am J Gastroenterol. 2011;106:151–6.

    Article  CAS  PubMed  Google Scholar 

  26. Notohara K. Histological features of autoimmune pancreatitis and IgG4-related sclerosing cholangitis with a correlation with imaging findings. J Med Ultrason. 2001;2021(48):581–94.

    Google Scholar 

  27. Hart PA, Kamisawa T, Brugge WR, et al. Long-term outcomes of autoimmune pancreatitis: a multicentre, international analysis. Gut. 2013;62:1771–6.

    Article  PubMed  Google Scholar 

  28. Kamisawa T, Chari ST, Giday SA, et al. Clinical profile of autoimmune pancreatitis and its histological subtypes: an international multicenter survey. Pancreas. 2011;40:809–14.

    Article  CAS  PubMed  Google Scholar 

  29. Iwamoto Y, Tatsumi F, Kohara K, et al. Dramatic recovery without steroid therapy and withdrawal from insulin therapy in a subject with hyperglycemic and hyperosmolar syndrome and depletion of insulin secretory capacity induced by type 2 autoimmune pancreatitis: a case report. Medicine (Baltimore). 2022;101: e28609.

    Article  PubMed  Google Scholar 

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Authors and Affiliations

  1. Division of Gastroenterology and Hepatology, The Third Department of Internal Medicine, Kansai Medical University, 2-5-1, Shinmachi, Hirakata, Osaka, 573-1010, Japan

    Takashi Ito, Tsukasa Ikeura, Masataka Masuda, Koh Nakamaru, Shinji Nakayama, Masaaki Shimatani, Makoto Takaoka & Makoto Naganuma

  2. Department of Anatomic Pathology, Kurashiki Central Hospital, Okayama, Japan

    Kenji Notohara

  3. Kansai Medical University Kori Hospital, Neyagawa, Osaka, Japan

    Kazuichi Okazaki

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  1. Takashi Ito
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  2. Tsukasa Ikeura
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  3. Kenji Notohara
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  4. Masataka Masuda
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  5. Koh Nakamaru
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  9. Kazuichi Okazaki
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  10. Makoto Naganuma
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Contributions

All authors contributed to this case report. The first draft of the manuscript was written by Takashi Ito and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.

Corresponding author

Correspondence to Tsukasa Ikeura.

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Conflict of interest

Takashi Ito, Tsukasa Ikeura, Kenji Notohara, Masataka Masuda, Koh Nakamaru, Shinji Nakayama, Masaaki Shimatani, Makoto Takaoka, Kazuichi Okazaki, and Makoto Naganuma declare that they have no conflict of interest.

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Ito, T., Ikeura, T., Notohara, K. et al. A case of type 2 autoimmune pancreatitis with spontaneous remission. Clin J Gastroenterol 16, 297–302 (2023). https://doi.org/10.1007/s12328-022-01753-y

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  • DOI: https://doi.org/10.1007/s12328-022-01753-y

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