Intestinal metaplasia of gallbladder diagnosed after laparoscopic cholecystectomy in patient with Dubin-Johnson syndrome; Does hyperbilirubinemia protect against gallbladder cancer development?
Öz
This manuscript describes the incidental diagnosis of Dubin–Johnson Syndrome (DJS), a rare autosomal recessive disorder characterized by impaired hepatic bilirubin excretion and the hallmark appearance of a grossly black liver, in a 34-year-old female who underwent laparoscopic cholecystectomy for symptomatic cholelithiasis. The patient presented with right upper quadrant abdominal pain and mild hyperbilirubinemia. Intraoperatively, the liver was noted to have a strikingly dark pigmentation, prompting a biopsy. Histopathological analysis confirmed the diagnosis of DJS. Additionally, examination of the resected gallbladder revealed focal intestinal metaplasia—an established premalignant lesion associated with an increased risk of gallbladder carcinoma. This report underscores the rare coexistence of DJS and gallstone disease and confirms that the presence of DJS does not complicate laparoscopic cholecystectomy or predispose to postoperative cholestasis. To our knowledge, this is the first documented case of intestinal metaplasia of the gallbladder in a patient with DJS. These findings highlight the importance of routine histopathological evaluation of cholecystectomy specimens, even in cases where DJS is discovered incidentally, to facilitate early detection of potential premalignant changes.
Anahtar Kelimeler
Dubin Johnson Syndrome Intestinal Metaplasia Gallbladder Cancer Laparoscopic Cholecystectomy Bilirubin cytoprotection.
Etik Beyan
Informed consent is available in the patient's file.
Destekleyen Kurum
None
Teşekkür
The authors declare no conflicts of interest related to this manuscript. Thank you for considering this manuscript for possible publication.
Kaynakça
- 1. Kim JH, Kang MW, Kim S, Han JW, Jang JW, Choi JY, et al. Genotype-Phenotype Association in ABCC2 Exon 18 Missense Mutation Leading to Dubin-Johnson Syndrome: A Case Report. Int J Mol Sci. 2022;23(24):16168.
- 2. Rastogi A, Krishnani N, Pandey R. Dubin-Johnson syndrome--a clinicopathologic study of twenty cases. Indian J Pathol Microbiol. 2006;49(4):500-504.
- 3. Sticova E, Elleder M, Hulkova H, Luksan O, Sauer M, Wunschova-Moudra I, et al. Dubin-Johnson syndrome coinciding with colon cancer and atherosclerosis. World J Gastroenterol. 2013;19(6):946-950.
- 4. Siddiqui AH, Alsabe MR, Tehseen Z, Hatamleh MI, Taslim S, Abdelrahman A, et al. Dubin-Johnson Syndrome: A Case Report. Cureus. 2023;15(3):e36115. 5. Renault M, Nowicki M. Persistent cholestasis following cholecystectomy: A case of Dubin–Johnson syndrome. J Pediatr. 2010;157(3):167.
- 6. Wang B, Yang S, Hu X, Zhang Y. Laparoscopic cholecystectomy for cholecystolithiasis with Dubin-Johnson syndrome. JGH Open. 2019;3(6):532-533.
- 7. Kumar H, Kini H, Tiwari A. Histological evaluation of 400 cholecystectomy specimens. J Pathol Nep [Internet]. 2015;5(10):834-840.
- 8. Poget M, Salvatori Chappuis V, Carbó Descamps F, Saadi A. Gallbladder mucocele caused by intestinal metaplasia in lithiatic cholecystitis: A case report and literature review of a rare association. Int J Surg Case Rep. 2024;116:109405.
- 9. Tashiro K, Yamauchi M, Takao Y, Ogoshi E, Yamazaki Y, Aoki T. A case of Gallbladder cancer with Dubin-Johnson Syndrome. Nihon Rinsho Geka Gakkai Zasshi. 2000;61(10):2453-2457.
- 10. Maruhashi T, Soga J, Fujimura N, Idei N, Mikami S, Iwamoto Y et al. Hyperbilirubinemia, Augmentation of Endothelial Function, and Decrease in Oxidative Stress in Gilbert Syndrome. Circulation. 2012;126(5):598-603.
- 11. Inoguchi T, Nohara Y, Nojiri C, Nakashima N. Association of serum bilirubin levels with risk of cancer development and total death. Sci Rep. 2021;11(1):13224.
- 12. Shin JW, Kim N, Minh NT, Chapagain DD, Jee SH. Serum bilirubin subgroups and cancer risk: Insights with a focus on lung cancer. Cancer Epidemiol. 2025;94:102727.
- 13. Ihira H, Nakano S, Yamaji T, Katagiri R, Sawada N, Inoue M, et al. Plasma albumin, bilirubin, and uric acid and the subsequent risk of cancer: a case-cohort study in the Japan Public Health Center-based Prospective Study. Am J Epidemiol. 2024;193:1460–1469.
- 14. Yoon HS, Shu XO, Shidal C, Wu J, Blot WJ, Zheng W et al. Associations of Pre-Diagnostic Serum Levels of Total Bilirubin and Albumin with Lung Cancer Risk: Results from the Southern Community Cohort Study. Front Oncol. 2022;12:895479.
- 15. Yi F, Tao S, Wu H. Bilirubin metabolism in relation to cancer. Front Oncol. 2025;15:1570288.
Dubin-Johnson sendromlu bir olguda laparoskopik kolesistektomi sonrası saptanan safra kesesi inestinal metaplazisi: Hiperbilirubineminin safra kesesi kanseri gelişimine karşı koruyucu etkisi olabilir mi?
Öz
Bu yazıda, semptomatik kolelitiazis nedeniyle laparoskopik kolesistektomi uygulanan 34 yaşındaki bir kadında, nadir görülen ve otozomal resesif kalıtım gösteren Dubin–Johnson Sendromu’nun (DJS) rastlantısal tanısı anlatılmaktadır. DJS, hepatik bilirubin atılımında bozulma ve karakteristik olarak belirgin siyah renkli karaciğer görünümü ile tanımlanan bir hastalıktır. Hasta, sağ üst kadranda karın ağrısı ve hafif hiperbilirubinemi ile başvurmuştur. Operasyon sırasında karaciğerin belirgin koyu pigmentasyonu fark edilmiş ve bu durum biyopsi yapılmasına neden olmuştur. Histopatolojik inceleme sonucunda DJS tanısı doğrulanmıştır. Ek olarak, çıkarılan safra kesesinin incelenmesinde odak intestinal metaplazi saptanmıştır; bu lezyon, safra kesesi karsinomu gelişme riski ile ilişkilendirilen bilinen premalign bir değişikliktir. Bu olgu, DJS ile safra taşı hastalığının nadir birlikteliğini vurgulamakta ve DJS varlığının laparoskopik kolesistektomiye engel oluşturmadığını ya da postoperatif kolestaza yatkınlık yaratmadığını ortaya koymaktadır. Bildiğimiz kadarıyla, DJS tanısı almış bir hastada safra kesesi intestinal metaplazisinin rapor edildiği ilk olgudur. Bu bulgular, DJS tanısı rastlantısal olarak konmuş olsa bile, olası premalign değişikliklerin erken saptanabilmesi açısından kolesistektomi materyallerinin rutin histopatolojik değerlendirilmesinin önemini ortaya koymaktadır.
Anahtar Kelimeler
Dubin-Johnson sendromu İntestinal metaplazisi Safra kesesi kanseri Laparoskopik kolesistektomi Bilirübin sitoproteksiyonu
Etik Beyan
Hastanın onamı dosyasında mevcuttur.
Destekleyen Kurum
YOK
Teşekkür
Yazarlar, bu makale ile ilgili herhangi bir çıkar çatışması bulunmadığını beyan etmektedir. Bu çalışmayı olası yayın için değerlendirdiğiniz için teşekkür ederiz.
Kaynakça
- 1. Kim JH, Kang MW, Kim S, Han JW, Jang JW, Choi JY, et al. Genotype-Phenotype Association in ABCC2 Exon 18 Missense Mutation Leading to Dubin-Johnson Syndrome: A Case Report. Int J Mol Sci. 2022;23(24):16168.
- 2. Rastogi A, Krishnani N, Pandey R. Dubin-Johnson syndrome--a clinicopathologic study of twenty cases. Indian J Pathol Microbiol. 2006;49(4):500-504.
- 3. Sticova E, Elleder M, Hulkova H, Luksan O, Sauer M, Wunschova-Moudra I, et al. Dubin-Johnson syndrome coinciding with colon cancer and atherosclerosis. World J Gastroenterol. 2013;19(6):946-950.
- 4. Siddiqui AH, Alsabe MR, Tehseen Z, Hatamleh MI, Taslim S, Abdelrahman A, et al. Dubin-Johnson Syndrome: A Case Report. Cureus. 2023;15(3):e36115. 5. Renault M, Nowicki M. Persistent cholestasis following cholecystectomy: A case of Dubin–Johnson syndrome. J Pediatr. 2010;157(3):167.
- 6. Wang B, Yang S, Hu X, Zhang Y. Laparoscopic cholecystectomy for cholecystolithiasis with Dubin-Johnson syndrome. JGH Open. 2019;3(6):532-533.
- 7. Kumar H, Kini H, Tiwari A. Histological evaluation of 400 cholecystectomy specimens. J Pathol Nep [Internet]. 2015;5(10):834-840.
- 8. Poget M, Salvatori Chappuis V, Carbó Descamps F, Saadi A. Gallbladder mucocele caused by intestinal metaplasia in lithiatic cholecystitis: A case report and literature review of a rare association. Int J Surg Case Rep. 2024;116:109405.
- 9. Tashiro K, Yamauchi M, Takao Y, Ogoshi E, Yamazaki Y, Aoki T. A case of Gallbladder cancer with Dubin-Johnson Syndrome. Nihon Rinsho Geka Gakkai Zasshi. 2000;61(10):2453-2457.
- 10. Maruhashi T, Soga J, Fujimura N, Idei N, Mikami S, Iwamoto Y et al. Hyperbilirubinemia, Augmentation of Endothelial Function, and Decrease in Oxidative Stress in Gilbert Syndrome. Circulation. 2012;126(5):598-603.
- 11. Inoguchi T, Nohara Y, Nojiri C, Nakashima N. Association of serum bilirubin levels with risk of cancer development and total death. Sci Rep. 2021;11(1):13224.
- 12. Shin JW, Kim N, Minh NT, Chapagain DD, Jee SH. Serum bilirubin subgroups and cancer risk: Insights with a focus on lung cancer. Cancer Epidemiol. 2025;94:102727.
- 13. Ihira H, Nakano S, Yamaji T, Katagiri R, Sawada N, Inoue M, et al. Plasma albumin, bilirubin, and uric acid and the subsequent risk of cancer: a case-cohort study in the Japan Public Health Center-based Prospective Study. Am J Epidemiol. 2024;193:1460–1469.
- 14. Yoon HS, Shu XO, Shidal C, Wu J, Blot WJ, Zheng W et al. Associations of Pre-Diagnostic Serum Levels of Total Bilirubin and Albumin with Lung Cancer Risk: Results from the Southern Community Cohort Study. Front Oncol. 2022;12:895479.
- 15. Yi F, Tao S, Wu H. Bilirubin metabolism in relation to cancer. Front Oncol. 2025;15:1570288.
Ayrıntılar
| Birincil Dil | İngilizce |
|---|---|
| Konular | Genel Cerrahi |
| Bölüm | Editöre Mektup |
| Yazarlar | |
| Gönderilme Tarihi | 8 Eylül 2025 |
| Kabul Tarihi | 2 Ekim 2025 |
| Yayımlanma Tarihi | 1 Nisan 2026 |
| DOI | https://doi.org/10.4717/jsa.1780311 |
| IZ | https://izlik.org/JA96YB29HD |
| Yayımlandığı Sayı | Yıl 2026 Cilt: 19 |
Amaç ve Kapsam
The aim of the Journal of Surgical Arts (Cerrahi Sanatlar Dergisi) is to publish high-quality research articles, review articles on current topics, and rare case reports in the field of surgery. Additionally, expert opinions, letters to the editor, scientific letters, and manuscripts on surgical techniques are accepted for publication, and various manuscripts on medicine and surgery history, ethics, surgical education, and forensic medicine fields are included in the journal.
Medicine, Surgery, Emergency medicine
Yazım Kuralları
The editorial and publication processes of the journal are shaped in accordance with the guidelines of the International Council of Medical Journal Editors (ICMJE), the World Association of Medical Editors (WAME), the Council of Science Editors (CSE), the Committee on Publication Ethics (COPE), the European Association of Science Editors (EASE), and National Information Standards Organization (NISO). The journal conforms to the Principles of Transparency and Best Practice in Scholarly Publishing (doaj.org/bestpractice).
Originality, high scientific quality, and citation potential are the most important criteria for a manuscript to be accepted for publication. Manuscripts submitted for evaluation should not have been previously presented or already published in an electronic or printed medium. The submission of previous reviewer reports will expedite the evaluation process. Manuscripts that have been presented in a meeting should be submitted with detailed information on the organization, including the name, date, and location of the organization.
Manuscripts submitted to the Journal of Surgical Arts will go through a peer-review process. The Editor is the final authority in the decision-making process for all submissions.
An approval of research protocols by the Ethics Committee in accordance with international agreements (World Medical Association Declaration of Helsinki âEthical Principles for Medical Research Involving Human Subjects,â amended in October 2013, www.wma.net) is required for experimental, clinical, and drug studies and for some case reports.
Journal of Surgical Arts requires and encourages the authors and the individuals involved in the evaluation process of submitted manuscripts to disclose any existing or potential conflicts of interests, including financial, consultant, and institutional, that might lead to potential bias or a conflict of interest. Any financial grants or other support received for a submitted study from individuals or institutions should be disclosed to the Editorial Board.
When submitting a manuscript to Journal of Surgical Arts, authors accept to assign the copyright of their manuscript to the Journal. If rejected for publication, the copyright of the manuscript will be assigned back to the authors. When using previously published content, including figures, tables, or any other material in both print and electronic formats, authors must obtain permission from the copyright holder. Legal, financial and criminal liabilities in this regard belong to the author(s).
Journal of Surgical Arts allows reuse and remixing of contents as category "CC BY" in accordance with a Creative Commons license or other types of license with similar conditions. Journal allows the readers to read, download, copy, distribute, print, search, or link to the full texts of its articles and allows readers to use them for any other lawful purpose.
Etik İlkeler ve Yayın Politikası
The aim of the Journal of Surgical Arts (Cerrahi Sanatlar Dergisi) is to publish high-quality research articles, review articles on current topics, and rare case reports in the field of surgery. Additionally, expert opinions, letters to the editor, scientific letters, and manuscripts on surgical techniques are accepted for publication, and various manuscripts on medicine and surgery history, ethics, surgical education, and forensic medicine fields are included in the journal.
Ücret Politikası
The journal doesn't have an article processing charge (APC) or any submission charges.
Dizinler
Diğer Dizinler
Dergi Kurulları
Baş Editör
Hepatopankreatobiliyer cerrahi, Bariatrik cerrahi ve Laparoskopik cerrahi
Alan Editörü
Cerrahi Sanatlar Dergisi (ISSN: 1308-0709), aşağıdaki veri tabanlarında yer almakta veya indekslenmektedir: OJS/PKP, CROSSREF, EBSCO Host, Google Scholar, CiteFactor, MIAR Index, EuroPub Database, CABELLS, Dergipark (TÜBİTAK), Turkish Citation Index, Sobiad, Asos Index, İdeal Index ve Akademik Index.
Not: Cerrahi Sanatlar Dergisi, "Üniversitelerarası Konsey" (YÖK, Türkiye) tarafından tanımlanan uluslararası veritabanları (EBSCO, vb.) tarafından indekslenen dergiler arasındadır.
Gönderim: Makalenizi gönderirken, barındırma ve makale takip hizmeti sağlayıcısı olan DergiPark'a (Cerrahi Sanatlar Dergisi » Ana Sayfa (dergipark.org.tr)) yönlendirileceksiniz. Seçtiğiniz kullanıcı adı ve şifreyle kolayca giriş yapabilirsiniz.