Chronic Bullous Disease of Childhood in a Patient with Acute Lymphoblastic Leukemia

Abstract

Linear IgA disease is characterized by the presence of linear IgA deposits in the basement membrane zone of the skin, and circulating basement membrane zone antibodies are detected in 80% of cases. The disease occurs in both adults and children, and is designated adult linear IgA disease in the former and chronic bullous disease of childhood (CBDC) in the latter. We describe a 5-year-old boy with acute lymphoblastic leukemia in remission, in whom CBDC developed after treatment with trimethoprim/sulfamethoxazole (cotrimoxazole). To our knowledge, this is the first reported case of possible drug-induced CBDC.

Childhood linear IgA bullous disease induced by trimethoprim-sulfamethoxazole

“…The lack of oral and conjuctival involvement, as in our case, is suggestive of druginduced LABD. 3,5,6 However, mucosal lesions can be noted in up to 40% of drug-induced cases, which is still lower than the 80% involvement observed in the classical LABD. 7,11 Childhood LABD has been mostly associated with infections or drug ingestion and rarely with other conditions such as ulcerative colitis and autoimmune lymphoproliferative disease.…”

“…The age of onset in children ranges from 1 to 10 years, with 64% of childhood cases presenting by the age of 6 to 8 years. 1,3 Clinically, drug-induced LABD may mimic bullous pemphigoid, erythema multiforme, dermatitis herpetiformis and toxic epidermal necrolysis. 1,4,5 In our patient, the initial eruption was erythema multiforme-like.…”

“…The suspected causative drugs in the reported pediatric cases are NSAID and antibiotics such as amoxicillin-clavulanic acid and TMP-SMX. 1,3 To our knowledge, there have been 3 reports of LABD suspected to be induced by TMP-SMX in the TMP-SMX and penicillin V were immediately stopped. The eruption kept progressing for 3 days then became stable after adding topical corticosteroids.…”

“…3,10,11 The only pediatric possible TMP-SMX-induced LABD was a 5-year-old child presenting acute lymphoblastic leukemia (ALL) in remission who received prophylactic treatment with TMP-SMX. 3 Our patient is the first case of LABD induced by TMP-SMX that was confirmed by a rechallenge. Adverse cutaneous drug reactions related to TMP-SMX are almost exclusively due to the SMX component and are more frequent in children receiving full dose therapy rather than prophylactic dose.…”

“…1,2 LABD and chronic bullous disease of childhood are now considered to be a single entity. 3 The diagnosis of LABD is confirmed by detecting a linear band of IgA deposit at the basement membrane. 2 As many as two-thirds of LABD are related to drug ingestion, particularly certain antibiotics, non-steroidal anti-inflammatory drugs (NSAID) and diuretics.…”