Annals of Hematology

, Volume 95, Issue 2, pp 279–285 | Cite as

Labial salivary gland biopsy for diagnosing immunoglobulin light chain amyloidosis: a retrospective analysis

  • Tomotaka Suzuki
  • Shigeru Kusumoto
  • Taro Yamashita
  • Arisa Masuda
  • Shiori Kinoshita
  • Takashi Yoshida
  • Fumiko Takami-Mori
  • Hisashi Takino
  • Asahi Ito
  • Masaki Ri
  • Takashi Ishida
  • Hirokazu Komatsu
  • Mitsuharu Ueda
  • Yukio Ando
  • Hiroshi Inagaki
  • Shinsuke Iida
Original Article

Abstract

Our goal was to evaluate the usefulness of labial salivary gland (LSG) biopsy for diagnosing immunoglobulin light chain (AL) amyloidosis, by comparing bone marrow and skin biopsies in the same patient population. This retrospective study included 34 consecutive patients who showed evidence of monoclonal proteins and symptoms considered to be due to amyloidosis, and who underwent a tissue biopsy from LSG between January 2005 and December 2012 at Nagoya City University Hospital. All samples of superficial tissues, including LSG, bone marrow, and skin, were independently evaluated as having amyloid deposits by a central review, which was blind to clinical information. An AL amyloidosis diagnosis was based on evidence of amyloid deposition in any biopsied tissue. Eighteen patients were diagnosed with AL amyloidosis. The sensitivity for detecting amyloid deposition was highest in biopsies of LSG at 89 %, followed by 77 % for bone marrow, and 72 % for skin. Amyloid deposition was detected in at least one superficial tissue of all the 18 patients. An LSG biopsy may be appropriate as a first-choice procedure to diagnose AL amyloidosis. Multiple biopsies of superficial tissues, including LSG, bone marrow, and skin, are recommended to increase the sensitivity for diagnosing AL amyloidosis.

Keywords

AL amyloidosis Labial salivary gland Bone marrow Skin Superficial tissue biopsy 

Notes

Compliance with ethical standards

Funding

This study was supported in part by the Amyloidosis Research Committee from the Ministry of Health, Labour and Welfare, Japan.

Conflict of interest

Shinsuke Iida has received research funding from Bristol-Myers Squibb Co., Chugai Pharmaceutical Co. Ltd., Taiho Pharmaceutical Co. Ltd., Celgene K.K., Kyowa Hakko Kirin Co. Ltd., Ono Pharmaceutical Co. Ltd., Eli Lilly Japan K.K., Nippon Kayaku Co. Ltd., and honoraria from Celgene K.K., Janssen Pharmaceutical K.K. and Ono Pharmaceutical Co. Ltd.

Takashi Ishida has received research funding from Kyowa Hakko Kirin Co., Ltd., Bayer Pharma AG, and Celgene K.K., and honoraria from Kyowa Hakko Kirin Co., Ltd.

Ethics approval and consent to participate

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards. For this type of retrospective study, formal consent was not required.

References

  1. 1.
    Gertz MA, Comenzo R, Falk RH, Fermand JP, Hazenberg BP, Hawkins PN, Merlini G, Moreau P, Ronco P, Sanchorawala V, Sezer O, Solomon A, Grateau G (2005) Definition of organ involvement and treatment response in immunoglobulin light chain amyloidosis (AL): a consensus opinion from the 10th International Symposium on Amyloid and Amyloidosis, Tours, France, 18–22 April 2004. Am J Hematol 79(4):319–328. doi: 10.1002/ajh.20381 PubMedCrossRefGoogle Scholar
  2. 2.
    Gertz MA (2014) Immunoglobulin light chain amyloidosis: 2014 update on diagnosis, prognosis, and treatment. Am J Hematol 89(12):1132–1140. doi: 10.1002/ajh.23828 PubMedCrossRefGoogle Scholar
  3. 3.
    Kyle RA, Spencer RJ, Dahlin DC (1966) Value of rectal biopsy in the diagnosis of primary systemic amyloidosis. Am J Med Sci 251(5):501–506PubMedCrossRefGoogle Scholar
  4. 4.
    Comenzo RL (2006) Systemic immunoglobulin light-chain amyloidosis. Clin Lymphoma Myeloma 7(3):182–185. doi: 10.3816/CLM.2006.n.056 PubMedCrossRefGoogle Scholar
  5. 5.
    Hachulla E, Janin A, Flipo RM, Saile R, Facon T, Bataille D, Vanhille P, Hatron PY, Devulder B, Duquesnoy B (1993) Labial salivary gland biopsy is a reliable test for the diagnosis of primary and secondary amyloidosis. A prospective clinical and immunohistologic study in 59 patients. Arthritis Rheum 36(5):691–697PubMedCrossRefGoogle Scholar
  6. 6.
    Delgado WA, Mosqueda A (1989) A highly sensitive method for diagnosis of secondary amyloidosis by labial salivary gland biopsy. J Oral Pathol Med 18(5):310–314PubMedCrossRefGoogle Scholar
  7. 7.
    Foli A, Palladini G, Caporali R, Verga L, Morbini P, Obici L, Russo P, Sarais G, Donadei S, Montecucco C, Merlini G (2011) The role of minor salivary gland biopsy in the diagnosis of systemic amyloidosis: results of a prospective study in 62 patients. Amyloid 18(Suppl 1):80–82. doi: 10.3109/13506129.2011.574354029 PubMedCrossRefGoogle Scholar
  8. 8.
    Sungur C, Sungur A, Ruacan S, Arik N, Yasavul U, Turgan C, Caglar S (1993) Diagnostic value of bone marrow biopsy in patients with renal disease secondary to familial Mediterranean fever. Kidney Int 44(4):834–836PubMedCrossRefGoogle Scholar
  9. 9.
    van G II, Hazenberg BP, Bijzet J, van Rijswijk MH (2006) Diagnostic accuracy of subcutaneous abdominal fat tissue aspiration for detecting systemic amyloidosis and its utility in clinical practice. Arthritis Rheum 54(6):2015–2021. doi: 10.1002/art.21902 CrossRefGoogle Scholar
  10. 10.
    Libbey CA, Skinner M, Cohen AS (1983) Use of abdominal fat tissue aspirate in the diagnosis of systemic amyloidosis. Arch Intern Med 143(8):1549–1552PubMedCrossRefGoogle Scholar
  11. 11.
    Gertz MA, Lacy MQ, Dispenzieri A, Hayman SR, Kumar SK, Dingli D, Ansell SM, Gastineau DA, Inwards DJ, Johnston PB, Litzow MR, Micallef IN, Porrata LF, Leung N, Hogan WJ, Buadi FK (2010) Autologous stem cell transplant for immunoglobulin light chain amyloidosis: a status report. Leuk Lymphoma 51(12):2181–2187. doi: 10.3109/10428194.2010.524329 PubMedCrossRefGoogle Scholar
  12. 12.
    Sacsaquispe SJ, Antunez-de Mayolo EA, Vicetti R, Delgado WA (2011) Detection of AA-type amyloid protein in labial salivary glands. Med Oral Patol Oral Cir Bucal 16(2):e149–152PubMedCrossRefGoogle Scholar
  13. 13.
    Stoopler ET, Vogl DT, Alawi F, Greenberg MS, Sollecito TP, Salazar G, Stadtmauer EA (2011) The presence of amyloid in abdominal and oral mucosal tissues in patients initially diagnosed with multiple myeloma: a pilot study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 111(3):326–332. doi: 10.1016/j.tripleo.2010.10.028 PubMedCrossRefGoogle Scholar
  14. 14.
    Rubinow A, Cohen AS (1978) Skin involvement in generalized amyloidosis. A study of clinically involved and uninvolved skin in 50 patients with primary and secondary amyloidosis. Ann Intern Med 88(6):781–785PubMedCrossRefGoogle Scholar
  15. 15.
    Lachmann HJ, Booth DR, Booth SE, Bybee A, Gilbertson JA, Gillmore JD, Pepys MB, Hawkins PN (2002) Misdiagnosis of hereditary amyloidosis as AL (primary) amyloidosis. N Engl J Med 346(23):1786–1791. doi: 10.1056/NEJMoa013354 PubMedCrossRefGoogle Scholar
  16. 16.
    Comenzo RL, Zhou P, Fleisher M, Clark B, Teruya-Feldstein J (2006) Seeking confidence in the diagnosis of systemic AL (Ig light-chain) amyloidosis: patients can have both monoclonal gammopathies and hereditary amyloid proteins. Blood 107(9):3489–3491. doi: 10.1182/blood-2005-10-4148 PubMedCrossRefGoogle Scholar
  17. 17.
    Vrana JA, Gamez JD, Madden BJ, Theis JD, Bergen HR 3rd, Dogan A (2009) Classification of amyloidosis by laser microdissection and mass spectrometry-based proteomic analysis in clinical biopsy specimens. Blood 114(24):4957–4959. doi: 10.1182/blood-2009-07-230722 PubMedCrossRefGoogle Scholar
  18. 18.
    Gilbertson JA, Theis JD, Vrana JA, Lachmann H, Wechalekar A, Whelan C, Hawkins PN, Dogan A, Gillmore JD (2015) A comparison of immunohistochemistry and mass spectrometry for determining the amyloid fibril protein from formalin-fixed biopsy tissue. J Clin Pathol 68(4):314–317. doi: 10.1136/jclinpath-2014-202722 PubMedCrossRefGoogle Scholar
  19. 19.
    Fernandez de Larrea C, Verga L, Morbini P, Klersy C, Lavatelli F, Foli A, Obici L, Milani P, Capello GL, Paulli M, Palladini G, Merlini G (2015) A practical approach to the diagnosis of systemic amyloidoses. Blood 125(4):2239–2244. doi: 10.1182/blood-2014-11-609883 PubMedCrossRefGoogle Scholar
  20. 20.
    Gillmore JD, Wechalekar A, Bird J, Cavenagh J, Hawkins S, Kazmi M, Lachmann HJ, Hawkins PN, Pratt G, Committee B (2015) Guidelines on the diagnosis and investigation of AL amyloidosis. Br J Haematol 168(2):207–218. doi: 10.1111/bjh.13156 PubMedCrossRefGoogle Scholar
  21. 21.
    Schonland SO, Hegenbart U, Bochtler T, Mangatter A, Hansberg M, Ho AD, Lohse P, Rocken C (2012) Immunohistochemistry in the classification of systemic forms of amyloidosis: a systematic investigation of 117 patients. Blood 119(2):488–493. doi: 10.1182/blood-2011-06-358507 PubMedCrossRefGoogle Scholar
  22. 22.
    Masouye I (1997) Diagnostic screening of systemic amyloidosis by abdominal fat aspiration: an analysis of 100 cases. Am J Dermatopathol 19(1):41–45PubMedCrossRefGoogle Scholar

Copyright information

© Springer-Verlag Berlin Heidelberg 2015

Authors and Affiliations

  • Tomotaka Suzuki
    • 1
    • 2
  • Shigeru Kusumoto
    • 1
  • Taro Yamashita
    • 3
  • Arisa Masuda
    • 1
  • Shiori Kinoshita
    • 1
  • Takashi Yoshida
    • 1
  • Fumiko Takami-Mori
    • 1
    • 2
  • Hisashi Takino
    • 4
  • Asahi Ito
    • 1
  • Masaki Ri
    • 1
  • Takashi Ishida
    • 1
  • Hirokazu Komatsu
    • 1
  • Mitsuharu Ueda
    • 3
  • Yukio Ando
    • 3
  • Hiroshi Inagaki
    • 4
  • Shinsuke Iida
    • 1
  1. 1.Department of Hematology & OncologyNagoya City University Graduate School of Medical SciencesNagoyaJapan
  2. 2.Department of HematologyToyokawa City HospitalToyokawaJapan
  3. 3.Department of NeurologyKumamoto University Graduate School of Medical SciencesKumamotoJapan
  4. 4.Department of Pathology and Molecular DiagnosticsNagoya City University Graduate School of Medical SciencesNagoyaJapan

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