Saito M, Ogasawara R, Izumiyama K, Mori A, Kondo T, Tanaka M, Morioka M, Ieko M. Acquired hemophilia A in solid cancer: Two case reports and review of the literature. World J Clin Cases 2018; 6(14): 781-785 [PMID: 30510943 DOI: 10.12998/wjcc.v6.i14.781]
Corresponding Author of This Article
Makoto Saito, MD, PhD, Department of Internal Medicine and Hematology, Aiiku Hospital, Minami 4 Nishi 25 Chuo-ku, Sapporo 0640804, Hokkaido, Japan. ikyoku@aiiku-hp.or.jp
Research Domain of This Article
Medicine, Research & Experimental
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Makoto Saito, Reiki Ogasawara, Koh Izumiyama, Akio Mori, Takeshi Kondo, Masanori Tanaka, Masanobu Morioka, Department of Internal Medicine and Hematology, Aiiku Hospital, Sapporo 0640804, Japan
Masahiro Ieko, Department of Internal Medicine, Health Sciences University of Hokkaido, Toubetsu 0610293, Japan
Author contributions: Saito M, Ogasawara R, Izumiyama K, Mori A, Kondo T, Tanaka M, Morioka M and Ieko M collected the patient’s clinical data; Saito M designed and wrote the report.
Informed consent statement: Consent was obtained from each patient or family (wife) for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest in this work.
CARE Checklist (2016) statement: I have prepared this report according to the guidelines of the CARE Checklist (2016).
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: Makoto Saito, MD, PhD, Department of Internal Medicine and Hematology, Aiiku Hospital, Minami 4 Nishi 25 Chuo-ku, Sapporo 0640804, Hokkaido, Japan. ikyoku@aiiku-hp.or.jp
Telephone: +81-11-5632211
Received: August 20, 2018 Peer-review started: August 21, 2018 First decision: October 11, 2018 Revised: October 21, 2018 Accepted: October 22, 2018 Article in press: October 22, 2018 Published online: November 26, 2018
ARTICLE HIGHLIGHTS
Case characteristics
60s two Japanese men with solid cancer [one is gastric cancer, the other is hepatocellular carcinoma (HCC)] had a marked bleeding tendency.
Clinical diagnosis
Progressed anemia due to severe hemorrhage, requiring blood transfusion.
Differential diagnosis
Hemorrhagic disease, such as immune thrombocytopenia or disseminated intravascular coagulation.
Laboratory diagnosis
In both patients, hemoglobin level reached < 7 g/dL, prolonged APTT of 94 s, and FVIII activity was reduced to 3.1%. The inhibitor titer was 7.59 and 57.1 BU/mL respectively, compatible with acquired hemophilia A (AHA). In the second patients, hepatitis C virus antibodies were positive and the levels of alpha-fetoprotein and protein induced by vitamin K absence-II were 1862 ng/mL and 210 mAU/mL, respectively.
Imaging diagnosis
Endoscopic examination in the first case revealed intestinal bleeding from the site of the anastomosis. Abdominal computed tomography scan in the second patients revealed HCC (5.5 cm in diameter).
Pathological diagnosis
In the first case, resected stomach and intraperitoneal cytology identified gastric cancer.
Treatment
Immunological treatments (prednisone and cyclophosphamide in case 1, and rituximab alone in case 2) were administered instead of bypassing agents. Oral tegafur/gimeracil/oteracil was administered in case 1, and transcatheter arterial chemoembolization was performed in case 2.
Related reports
Recently, a systematic review described a large number of AHA patients with cancer.
Term explanation
AHA patients with cancer are more likely to exhibit recurrent hemorrhage and are less likely to achieve a complete response with eradication of the neutralizing autoantibodies.
Experiences and lessons
Besides hemostatic therapy and immunological treatments, successful treatment of AHA patients with cancer requires the concurrent treatment of the underlying malignancy.
