Skip to main content

Advertisement

SpringerLink
Log in

An unfavorable and a successful pregnancy outcome during and after treatment of gamma heavy chain disease

  • Case Report
  • Published:
International Journal of Hematology Aims and scope Submit manuscript

Abstract

Gamma heavy chain disease (gHCD) is a rare B-cell lymphoproliferative disorder that mostly occurs after childbearing age. Here we report the first case of gHCD in a pregnant patient that was diagnosed in the second trimester, and another pregnancy in the same patient after initial treatment for gHCD. The former pregnancy ended in intrauterine fetal death, believed to be caused by insufficient maternal blood flow due to multiple placental infarcts. The latter pregnancy course was uneventful. Although we cannot rule out the possibility that the poor outcome of the former pregnancy was due to an unfortunate complication independent of gHCD, the courses of these pregnancies suggest that non-lymphomatous gamma heavy chain may have a significant impact on pregnancy and that its removal by treatment may improve outcomes.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4

Similar content being viewed by others

Data availability

The data that support the findings of this study are not openly available due to reasons of sensitivity and are available from the corresponding author upon reasonable request. Data are located in controlled access data storage at Saitama City Hospital.

References

  1. Bianchi G, Anderson KC, Harris NL, Sohani AR. The heavy chain diseases: clinical and pathologic features. Oncology (Williston Park). 2014;28:45–53.

    PubMed  Google Scholar 

  2. Wahner-Roedler DL, Witzig TE, Loehrer LL, Kyle RA. Gamma-heavy chain disease: review of 23 cases. Medicine (Baltimore). 2003;82:236–50.

    Article  CAS  PubMed  Google Scholar 

  3. Bieliauskas S, Tubbs RR, Bacon CM, Eshoa C, Foucar K, Gibson SE, et al. Gamma heavy-chain disease: defining the spectrum of associated lymphoproliferative disorders through analysis of 13 cases. Am J Surg Pathol. 2012;36:534–43.

    Article  PubMed  PubMed Central  Google Scholar 

  4. Alaggio R, Amador C, Anagnostopoulos I, Attygalle AD, Araujo IBO, Berti E, et al. The 5th edition of the World Health Organization Classification of Haematolymphoid Tumours: Lymphoid Neoplasms. Leukemia. 2022;36:1720–48.

  5. Lishner M, Avivi I, Apperley JF, Dierickx D, Evens AM, Fumagalli M, et al. Hematologic malignancies in pregnancy: management guidelines from an international consensus meeting. J Clin Oncol. 2016;34:501–8.

    Article  CAS  PubMed  Google Scholar 

  6. Evens AM, Advani R, Press OW, Lossos IS, Vose JM, Hernandez-Ilizaliturri FJ, et al. Lymphoma occurring during pregnancy: antenatal therapy, complications, and maternal survival in a multicenter analysis. J Clin Oncol. 2013;31:4132–9.

    Article  PubMed  Google Scholar 

  7. Noy A, Schöder H, Gönen M, Weissler M, Ertelt K, Cohler C, et al. The majority of transformed lymphomas have high standardized uptake values (SUVs) on positron emission tomography (PET) scanning similar to diffuse large B-cell lymphoma (DLBCL). Ann Oncol. 2009;20:508–12.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  8. Schöder H, Noy A, Gönen M, Weng L, Green D, Erdi YE, et al. Intensity of 18fluorodeoxyglucose uptake in positron emission tomography distinguishes between indolent and aggressive non-Hodgkin’s lymphoma. J Clin Oncol. 2005;23:4643–51.

    Article  PubMed  Google Scholar 

  9. Gregory ECW, Valenzuela CP, Hoyert DL. Fetal Mortality: United States, 2021. Natl Vital Stat Rep. 2023;72:1–21.

    PubMed  Google Scholar 

  10. Korteweg FJ, Erwich J, Holm JP, Ravise JM, van der Meer J, Veeger N, Timmer A. Diverse placental pathologies as the main causes of fetal death. Obstet Gynecol. 2009;114:809–17.

    Article  PubMed  Google Scholar 

  11. Maggen C, Dierickx D, Cardonick E, Mhallem Gziri M, Cabrera-Garcia A, Shmakov RG, et al. Maternal and neonatal outcomes in 80 patients diagnosed with non-Hodgkin lymphoma during pregnancy: results from the International Network of Cancer, infertility and pregnancy. Br J Haematol. 2021;193:52–62.

    Article  CAS  PubMed  Google Scholar 

  12. Lee EJ, Ahn KH, Hong SC, Lee EH, Park Y, Kim BS. Rituximab, cyclophosphamide, doxorubicin, vincristine, and prednisone (R-CHOP) chemotherapy for diffuse large B-cell lymphoma in pregnancy may be associated with preterm birth. Obstet Gynecol Sci. 2014;57:526–9.

    Article  PubMed  PubMed Central  Google Scholar 

  13. Mittel RJ, Tobin MC, Kulik J. Neonatal B-cell suppression after in utero exposure to R-CHOP. Ann Allergy Asthma Immunol. 2013;111:221–2.

    Article  CAS  PubMed  Google Scholar 

  14. Padberg S, Mick I, Frenzel C, Greil R, Hilberath J, Schaefer C. Transient congenital dilated cardiomyopathy after maternal R-CHOP chemotherapy during pregnancy. Reprod Toxicol. 2017;71:146–9.

    Article  CAS  PubMed  Google Scholar 

  15. Tincani A, Dall’Ara F, Lazzaroni MG, Reggia R, Andreoli L. Pregnancy in patients with autoimmune disease: A reality in 2016. Autoimmun Rev. 2016;15:975–7.

    Article  CAS  PubMed  Google Scholar 

Download references

Acknowledgements

The authors would like to thank the patient, her family, and the team of Saitama City Hospital.

Author information

Authors and Affiliations

  1. Department of Internal Medicine, Saitama City Hospital, 2460 Mimuro, Saitama-City, Saitama, 336-8522, Japan

    Masataka Hosoi, Kaito Takikawa, Yuya Arai, Misato Kikuchi & Takuro Watanabe

  2. Department of Obstetrics and Gynecology, Saitama City Hospital, Saitama, Japan

    Kazunori Ueno & Reina Ooka

  3. Department of Diagnostic Pathology, Saitama City Hospital, Saitama, Japan

    Jun Kikuchi & Jun Miyauchi

  4. Department of Pathology, Saitama Medical Center, Saitama Medical University, Saitama, Japan

    Jun Kikuchi

  5. Department of Obstetrics and Gynecology, Keio University School of Medicine, Tokyo, Japan

    Reina Ooka

Authors
  1. Masataka Hosoi
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Kazunori Ueno
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Jun Kikuchi
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Jun Miyauchi
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Reina Ooka
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Kaito Takikawa
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Yuya Arai
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. Misato Kikuchi
    View author publications

    You can also search for this author in PubMed Google Scholar

  9. Takuro Watanabe
    View author publications

    You can also search for this author in PubMed Google Scholar

Contributions

Masataka Hosoi, Kazunori Ueno, Reina Ooka, and Takuro Watanabe were involved in treatment of the patient. Masataka Hosoi, Kazunori Ueno, and Jun Kikuchi drafted the manuscript. Jun Kikuchi and Jun Miyauchi provided pathological information and diagnosis validation. Jun Miyauchi, Kaito Takikawa, Yuya Arai, Misato Kikuchi, and Takuro Watanabe revised the manuscript critically for important intellectual content. All authors contributed to the writing of the final manuscript and approved the version of the manuscript to be submitted.

Corresponding author

Correspondence to Masataka Hosoi.

Ethics declarations

Conflict of interest

The authors declare that they have no conflict of interest.

Ethics approval

This case report is an observational study. The Saitama City Hospital Human Subjects Review Committee has confirmed that no ethical approval is required.

Patient consent

The patient and her husband orally consented to publication of the case.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

New findings: We report a pregnant case diagnosed with gamma heavy chain disease (gHCD) in the second trimester. There is no obtainable information, to our knowledge, about pregnancy woman with gHCD. Thus, this is the first report describing a pregnancy course of a mother with gHCD. Surprisingly, the fetal growth seemed far worse than that of the most previously reported mothers with lymphoma. We also report another pregnancy course of same patient, after completion of initial treatment, in complete serological remission. This time, the pregnancy course was uneventful, suggesting that removal of gamma heavy chain by treatment may improve pregnancy outcomes.

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Hosoi, M., Ueno, K., Kikuchi, J. et al. An unfavorable and a successful pregnancy outcome during and after treatment of gamma heavy chain disease. Int J Hematol (2024). https://doi.org/10.1007/s12185-024-03769-1

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • DOI: https://doi.org/10.1007/s12185-024-03769-1

Keywords

Search

Navigation