Propylthiouracil-Associated Leukocytoclastic Necrotizing Cutaneous Vasculitis

Abstract

The purpose of this case report and review of the literature is to provide an exploration of the clinical symptoms, diagnosis, prevention, and management of propylthiouracil (PTU)-associated vasculitis in the intensive care setting. A PubMed search of the available literature was conducted using the MeSH search terms "propylthiouracil" and "vasculitis." The literature search returned 121 articles. Twenty-five were excluded because they were not in English. Fifty-nine case reports or case studies describing PTU-associated vasculitis were included. Data extracted from each case study included patient age, sex, autoimmune markers, laboratory tests, length of time on PTU, treatment for vasculitis, and patient outcomes. The authors reviewed 128 cases of PTU-associated vasculitis. The majority were women (8.8:1 F:M ratio), and the most common presenting symptoms were rash (51.6%), fever (46.9%), and arthralgia (43.8%). In addition to discontinuing PTU, the most common treatment was steroids (71.9%). Eight patients (6.3%) progressed to end-stage renal disease; two (1.6%) required intubation for respiratory failure; and five (3.9%) died of various organ systems failure related to vasculitis development. A high index of suspicion for vasculitis should be maintained, especially when presented with skin manifestations in the presence of PTU therapy. Screening with myeloperoxidase-antinuclear cytoplasmic antibodies is most sensitive. Positive screening should prompt a thorough clinical investigation. In cases of severe skin manifestations, the focus should be on aggressive wound care. Our case report is unique, not only in the size and extent of cutaneous involvement, but also as the first description of mortality secondary to cutaneous manifestations.

Cutaneous leukocytoclastic vasculitis in a patient treated with carboplatin for uterine carcinoma

“…Namely, Lee et al reported a case of LCV caused by another anti-cancer drug, everolimus. The patient presented with no systemic manifestations of vasculitis and did not receive any immunosuppressive therapy, but he needed skin grafting [4]. Our colleagues Grasic et al presented a similar case with everolimus connected to LCV, where the patient needed only treatment discontinuation and local measures, as in our case [7].…”

“…Leukocytoclastic vasculitis is a histopathological term used to describe a variety of types of small vessel vasculitis and mainly refers to the skin, although there are types of vasculitis that can affect other regions of the body. By using the positive results from direct immunofluorescence examination, we may categorize the type of LCV based on the detection of immune complex deposits in the vessel wall [2,4]. LCV presents approximately seven to ten days after exposure to the causative agent.…”

Bad bites? A case report of rapidly progressive painful necrotic lower limb lesions from propylthiouracil‐associated vasculitis

“…In severe cases, like in our patient, further immunosuppression is required. Late presentation can be life‐threatening …”

“…PTU vasculitis occurs more commonly in females, and the most common symptoms are rash, fever and arthralgia . It can affect any organ system.…”

Propylthiouracil-induced ANCA-negative cutaneous small vessel vasculitis

“…Usually it is limited to the skin in a form of cutaneous leukocytoclastic vasculitis, but may also affect organs including kidneys and lungs. Some fatal cases have been reported even with limited cutaneous disease [11]. Discontinuation of PTU should be a first step in the treatment and could lead to complete resolution of symptoms.…”