Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 26, 2020; 8(18): 4122-4127
Published online Sep 26, 2020. doi: 10.12998/wjcc.v8.i18.4122
Sweet syndrome as a paraneoplastic manifestation of cholangiocarcinoma: A case report
Camille Carneiro Lemaire, Ana Luisa Carvalho Portilho, Luciana V Pinheiro, Rafael Alves Vivas, Maíra Britto, Melaine Montenegro, Luiz Felipe de Farias Rodrigues, Sérgio Arruda, André Castro Lyra, Lourianne Nascimento Cavalcante
Camille Carneiro Lemaire, Ana Luisa Carvalho Portilho, Department of Internal Medicine, Escola Bahiana de Medicina e Saúde Pública, Salvador 40290-000, Bahia, Brazil
Luciana V Pinheiro, Melaine Montenegro, Department of Internal Medicine, Hospital Geral Roberto Santos, Salvador 41180-780, Bahia, Brazil
Rafael Alves Vivas, Department of Surgery, Hospital Geral Roberto Santos, Salvador 41180-780, Bahia, Brazil
Maíra Britto, Department of Gastroenterology and Hepatology, Hospital Geral Roberto Santos, Salvador 41180-780, Bahia, Brazil
Luiz Felipe de Farias Rodrigues, Department of Radiology, Hospital Geral Roberto Santos, Salvador 41180-780, Bahia, Brazil
Sérgio Arruda, Pathological Anatomy, Fiocruz - Bahia, Universidade Estadual da Bahia, Salvador 40000-000, Bahia, Brazil
André Castro Lyra, Department of Medicine, Division of Gastroenterology and Hepatology, Federal University of Bahia and Gastro-Hepatology Service, Salvador 40295-050, Bahia, Brazil
Lourianne Nascimento Cavalcante, Department of Medicine, Division of Gastroenterology and Hepatology, Federal University of Bahia and SED-CHD Hospital Geral Roberto Santos, Salvador 40295-050, Bahia, Brazil
Author contributions: Lemaire CC, Portilho ALC, Pinheiro LV, Vivas RA, Britto M, and Montenegro M collected the patient’s clinical data; Rodrigues LFF analyzed the computed tomography images; Arruda S performed the anatomopathological analysis and provided the photos of the slides; Lyra AC and Cavalcante LN reviewed the data and wrote the paper.
Informed consent statement: The written informed consent was provided.
Conflict-of-interest statement: All authors worked on this case and none of them have conflict of interest.
CARE Checklist (2016) statement: The authors read the CARE checklist (2016) and the case report was prepared and revised in accordance with the CARE checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (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/
Corresponding author: Lourianne Nascimento Cavalcante, MD, PhD, Associate Professor, Associate Specialist, Department of Medicine, Division of Gastroenterology and Hepatology, Federal University of Bahia and SED-CHD Hospital Geral Roberto Santos, Av. Reitor Miguel Calmon, S/N - Vale do Canela, Salvador 40295-050, Bahia, Brazil. lourianne@gmail.com
Received: April 19, 2020
Peer-review started: April 19, 2020
First decision: July 25, 2020
Revised: August 2, 2020
Accepted: August 29, 2020
Article in press: August 29, 2020
Published online: September 26, 2020
Processing time: 155 Days and 5.9 Hours
Abstract
BACKGROUND

Sweet’s syndrome, also known as acute febrile neutrophilic dermatosis, is a rare skin disorder that may be associated with cancer.

CASE SUMMARY

A 58-year-old female presented with a cholestatic syndrome and significant weight loss three months before admission. Five months earlier, she had abruptly developed skin lesions with erythematous papules that evolved to erythematous blisters. Clinical evaluation and laboratory tests confirmed hepatic cholangiocarcinoma. Skin lesions histopathological findings showed neutrophilic dermatosis, massive edema, fibrin, necrosis, and elastosis. These results, in association with the macroscopic aspects of the findings, led to the diagnosis of paraneoplastic Sweet’s syndrome due to cholangiocarcinoma. As staging was consistent with an advanced tumor without a cure perspective, we opted to perform percutaneous biliary drainage, and subsequently, palliative care. Eventually, after a few weeks, the patient died.

CONCLUSION

In conclusion, the diagnosis of the underlying disease-causing Sweet’s syndrome must be accurate, and patients need to be followed-up, as neoplasia such as cholangiocarcinoma may be a later manifestation.

Keywords: Cholangiocarcinoma; Paraneoplastic syndrome; Sweet syndrome; Case report; Bullous lesions; Cholestasis

Core Tip: Sweet's syndrome preceding solid neoplasia, especially cholangiocarcinoma, is a rare condition. Only two cases of Sweet's syndrome associated with cholangiocarcinoma have been reported. Bullous skin lesions should be evaluated with a broad differential diagnosis, including the possibility of preceding the diagnosis of neoplasia. The evaluation and follow-up of patients with this condition is essential, seeking diagnosis and treatment in early stages of the neoplastic lesion.