Sodium valproate-induced cutaneous pseudolymphoma followed by recurrence with carbamazepine

Abstract

We report a patient with a sodium valproate-induced cutaneous pseudolymphoma, presenting with an erythematous papule, histologically mimicking a non-epidermotropic T-cell lymphoma. Polymerase chain reaction study of the skin biopsy revealed monoclonal rearrangement of the T-cell receptor gamma gene. Withdrawal of sodium valproate was followed by regression of the lesion, but 5 months after substitution by carbamazepine, two further papules appeared, with similar histological features and a T-cell clone identical to the initial one. Carbamazepine was stopped and the lesions disappeared without relapse over a 4-year follow-up. Sodium valproate is very rarely responsible for a hypersensitivity syndrome, and our case is the first report of sodium valproate-induced cutaneous pseudolymphoma. The recurrence with carbamazepine may be due to a common effect on T-cell lymphocyte function. The return of the same monoclonal population shows that the recurrence of monoclonal T cells may be observed in benign conditions and is not an exclusive hallmark of cutaneous lymphoma.

Carbamazepine is an inhibitor of histone deacetylases

“…A recent case report described a patient with cutaneous pseudolymphoma due to VPA that resolved after cessation of the drug but recurred after instituting CBZ therapy. The authors of the report showed that the same clonal T-cell receptor rearrangement that had been detected after VPA re-emerged after administration of CBZ (Cogrel et al, 2001).…”

Cutaneous Pseudolymphoma Induced by <i>Cimicifuga racemosa</i>

“…As observed in our patient, complete resolution of PL usually occurs after drug withdrawal. Problems may arise from the fact that drug-induced PL may mimic the histological pattern of malignant lymphoma [3]. Since the clinical and histological features may be ambiguous and sometimes not conclusive, resolution after drug withdrawal is the main criterion for the diagnosis of drug-induced cutaneous PL [3].…”

“…Problems may arise from the fact that drug-induced PL may mimic the histological pattern of malignant lymphoma [3]. Since the clinical and histological features may be ambiguous and sometimes not conclusive, resolution after drug withdrawal is the main criterion for the diagnosis of drug-induced cutaneous PL [3]. In our case, although the patient was not re-exposed to the drug to confirm the reaction, the temporal relationship of the cutaneous lesions with the drug use and the lack of recurrence after 6 months of follow-up strongly suggest the diagnosis of drug-induced PL rather than spontaneous resolution.…”

“…In many cases, histopathological findings are not unambiguous for the diagnosis of PL versus lymphoma. Therefore, resolution after drug withdrawal is considered to be the gold standard for the diagnosis of drug-induced cutaneous PL [3]. …”

Acute Hypersensitivity Syndrome Caused by Valproic Acid: A Review of the Literature and a Case Report

“…Another case report described a patient who, after resolution of symptoms caused by carbamazepine-induced AHS, was given VPA and immediately developed life-threatening AHS. 14 In addition, Cogrel and colleagues 15 reported an AHS-like condition with VPA treatment. The patient recovered after the VPA was discontinued.…”