Lingual ulceration associated with retinoic acid syndrome during treatment of acute promyelocytic leukemia

Abstract

It is important for dermatologists to recognize this cutaneous complication of ATRA as it poses many differential diagnoses in neutropenic patients.

Rare labial ulcer related to the use of all‐trans retinoic acid in a patient with acute promyelocytic leukemia

“…The manifestation of labial and/or scrotal ulcers is an early sign of RAS before the onset of more severe events of the syndrome. In previously reported cases, oral lesions appeared between days 5 and 30 after the initiation of ATRA therapy . In the present case, the labial ulcer appeared on day 8.…”

“…This leads the medical team to introduce empirical treatment for fungal, bacterial and/or viral infections, which are usually the most common causes of lesions in these patients. However, these medications do not promote the improvement of such lesions . In the present case, given the very low platelet and leukocyte counts at the time of the onset of the oral ulcer, the medical team choose to initiate empirical treatment for fungal, viral, and bacterial infection.…”

“…Painful ulcerations are associated with ATRA, manly in the genital region. Based on case reports, ulcers seem to occur more in white, middle‐aged male patients . Two cases of ulcers located in the oral region associated with the use of ATRA have previously been described .…”

“…The diagnosis of this type of ulcer should involve clinical aspects with recurrent characteristics in different reports found in the literature, such as the time of onset, the presence of pain, fever and a black eschar over the ulcer . Histological analysis is useful for ruling out other conditions, such as fungal or viral infection, since this type of lesion does not have a specific histology beyond ulceration and areas of necrosis with the presence of inflammation and without leukemic cells . It is worth mentioning that biopsy is often postponed due to the patient's systemic condition.…”

“…However, it is believed that the cause is the oxidative metabolism of neutrophils through the production of superoxide . Studies have associated the appearance of ulcers with increased leukocyte counts, supporting the correlation between the activation of inflammatory cytokines and tissue injury . Bearing in mind this proposed mechanism of development, it is reasonable to expect such ulcers to regress due to the use of dexamethasone, despite the many reports of the suspension of ATRA therapy in such cases.…”