Isotretinoin-associated Sweet’s syndrome: a case report
© Moghimi et al.; licensee BioMed Central Ltd. 2014
Received: 7 August 2014
Accepted: 9 October 2014
Published: 17 October 2014
Sweet’s syndrome (SS) is characterized by various clinical symptoms, physical features, and pathological findings. Although cases of SS are very rare, there has been an increase in the incidence of drug-induced SS. Till date, there have been only few reported cases of isotretinoin-induced SS.
In this report, we describe the case of a 19-year-old girl who developed SS after systemic treatment with oral isotretinoin for nodulocystic acne.
The findings of this report emphasize the importance of evaluating isotretinoin as a possible, though uncommon, cause of SS and replacing it with another treatment if its involvement is suspected.
KeywordsSweet syndrome Isotretinoin Neutrophilic dermatosis Drug reaction
Sweet’s syndrome (SS) is characterized by painful erythematous skin lesions due to neutrophil infiltration in the upper dermis, fever, leukocytosis with a predominance of neutrophils, and increased erythrocyte sedimentation rate (ESR) ,. There are three clinical forms of SS: classical (idiopathic), malignancy associated, and drug induced . Classical SS usually develops in women aged between 30 and 40 years and is often preceded by upper respiratory or gastrointestinal tract infection, inflammatory bowel disease, and pregnancy. Malignancy-associated SS can occur as a paraneoplastic syndrome. Drug-induced SS most commonly occurs in patients who have been treated with granulocyte colony-stimulating factor . Although this form of SS is very rare, there has been an increase in the incidence of drug-induced SS . Here we describe the case of a 19-year-old girl who developed drug-induced SS after systemic treatment with oral isotretinoin (13-cis-retinoic acid) for nodulocystic acne and emphasize the importance of evaluating isotretinoin as a possible, though uncommon, cause of SS.
This is a report of rare case, however the report was approved by the Ethics Committee of Semnan University of Medical Sciences, Semnan, Iran.
Etiologically, SS may be associated with drugs, infections, and paraneoplastic or inflammatory conditions such as inflammatory bowel diseases and rheumatological diseases. When no triggering factor is detected, the disease is categorized as classic or idiopathic .
Drugs reported as inducing Sweet’s syndrome
Granulocytes growth factors
G-CSF (granulocyte-colony stimulating factor); GM-CSF (granulocyte monocyte colony stimulating factor); All trans-retinoic acid
Calmette-Guérin; Influenza; Streptococcus pneumonia; Small-pox
Doxycycin; Cyclines (minocycline, tetracycline and doxycycline); Quinolones: norfloxacin, ofloxacin; Nitrofurantoin; Streptogramin (quinupristin/dalfopristin); Trimethoprim-sulphamethoxazole
Proteasomes inhibitors (bortezomib); Tyrosine kinases inhibitors (imatinib)
Non-steroidal anti-inflammatory drugs
Diclofenac; Celecoxib; Rofecoxib
Amoxapine; Clozapine; Diazepam; Lormetazepam
Azathioprine; Carbamazepine; Furosemide; Hydralazine; Isotretinoin; Lenalidomide; Oral contraceptive; Levonorgestrel/ethynil oestradiol; gestodene/ethynil oestradiol; Propylthiouracil
Drug-induced SS is uncommon. Because of the lack of useful and appropriate criteria for its diagnosis, Walker and Cohen proposed five specific diagnostic criteria in 1996 . Thompson and Montarella performed a systematic review of literature on drug-induced SS and evaluated the degree of causal relationships in different case reports . In our case, we used the modified Naranjo criteria to diagnose drug-induced SS . The modified scale includes 10 criteria: presence of previous conclusive reports; temporary onset related to drug administration; temporary resolution of lesions after drug withdrawal or treatment with a specific antagonist; temporary recurrence of lesions due to drug readministration; presence of alternative causes (other than drugs) that could cause the reaction; appearance of lesions after placebo administration; drug detection in the blood (or other fluids) at concentrations known to be toxic; role of drug dosage in the worsening or improvement of the reaction; history of exposure to the same or similar drugs, followed by a similar reaction; and confirmation of an adverse event by any objective evidence . The case reported here had a causality score of 4 based on the above criteria (Possible). This score was not higher because no rechallenge was performed due to the severity of the symptoms. There were no signs of any inflammatory disease, neoplasia, or pregnancy. To the best of our knowledge, this is the third case of SS caused by isotretinoin and the second case of SS caused by isotretinoin therapy for acne vulgaris.
The first case was reported by Gyorfy et al in 2003. They reported the cases of two children who developed SS due to isotretinoin administration. However, the children were administered this drug to prevent neuroblastoma recurrence and dysplastic colon re-emergence after bone marrow transplantation and not for acne treatment .
The other case of isotretinoin-induced SS was reported by Ammar et al. in 2007 in a 19-year-old man who received isotretinoin for severe acne vulgaris that was resistant to standard topical acne treatment. After one week of the treatment, the patient developed SS. His condition was improved by continuing isotretinoin and by initiating corticosteroids. However, he was diagnosed as having ulcerative colitis two years later .
In drug-induced SS, a pre-existing underlying condition associated with SS should be excluded. Clinical criteria can help in distinguishing drug-induced SS from underlying conditions. Based on the observation made in this case, we emphasize the importance of evaluating isotretinoin as a possible, though uncommon, cause of SS and replacing it with other treatments if its involvement is suspected.
Written informed consent was obtained from the patient for the publication of this report.
JM and MP was managing the case.MP, DP and MA drafting the paper. JM and HH were managing the case and scientific editing of the manuscript. All authors read and approved the final manuscript.
Erythrocyte sedimentation rate
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