Sarcoid- like Phenomenon - ustekinumab induced granulomatous reaction mimicking diffuse metastatic disease: a case report and review of the literature

J Med Case Rep. 2019 Jul 30;13(1):257. doi: 10.1186/s13256-019-2137-1.

Abstract

Background: The utilization of monoclonal antibodies has become more widespread over the past decade. However, the development of non-caseating granulomas with the use of monoclonal antibodies, such as ustekinumab, is not widely reported in the literature.

Case presentation: We report a case of a 50-year-old Caucasian male who presented complaining of weight loss and shortness of breath. He was receiving ustekinumab for refractory psoriasis but had no other significant medical comorbidities. On physical examination, reduced breath sounds on the right side were noted. Blood cultures were drawn on presentation and came back negative in 48 hours. A chest computed tomography scan revealed a large right lung mass in addition to right-sided pleural effusion. Therapeutic thoracocentesis was done; fluid cytology and analysis were negative for malignancy, acid-fast bacilli, or fungal infections. A positron emission tomography scan showed multifocal radiotracer uptake including within right lung mass, multiple bones, lymph nodes, liver and spleen. Biopsies showed hyalinized non-necrotizing granulomas. Immunohistochemical stains for AE1/AE3, cytokeratin 7 and 20, and thyroid transcription factor 1, were all negative. He was started on steroid therapy, and ustekinumab was discontinued and the follow-up computed tomography after a few months showed substantial improvement. However, over the course of next 4 months patient developed hepatic dysfunction and recurrent ascites and ultimately underwent transjugular intrahepatic portosystemic shunt placement. Furthermore, he was started on azathioprine and steroids were tapered. He improved clinically and was discharged from our hospital within a week.

Conclusions: This case highlights the need for careful consideration of patient medication history while evaluating the possible differential diagnoses that may contribute to a patient's presentation.

Keywords: Refractory psoriasis; Sarcoid-like reaction; Sarcoidosis; Ustekinumab.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Dermatologic Agents / administration & dosage
  • Dermatologic Agents / adverse effects*
  • Granuloma / chemically induced*
  • Humans
  • Lung Neoplasms / diagnosis
  • Male
  • Middle Aged
  • Pleural Effusion / diagnostic imaging
  • Positron-Emission Tomography
  • Psoriasis / drug therapy
  • Sarcoidosis / diagnosis
  • Tomography, X-Ray Computed
  • Ustekinumab / administration & dosage
  • Ustekinumab / adverse effects*

Substances

  • Dermatologic Agents
  • Ustekinumab