Fixed Drug Eruption-like lesions in an 8-year-old boy

presented by

Loh Liew Cheng MB ChB MRCP(1)

S M Wong MB ChB MRCP (2)

Kuala Lumpur, Malaysia

September 18, 2007

(1) Consultant Dermatologist, Subang Jaya Medical Centre, Kuala Lumpur, Malaysia

(2) Dermatology unit, Department of Medicine, University of Malaya Medical Centre, Kuala Lumpur, Malaysia

 
Abstract 8-year-old Indian boy was referred to the dermatology clinic from the paediatric department for a recurrent skin eruption. The rash appeared on the chest, neck and trunk, preceded by fever for one week. On examination, there were multiple annular, hyperpigmented lesions with erythematous rim on the chest, neck, and abdomen of a total of fewer than ten. Some lesions had bullae in the centre resembling target lesions. Is this erythema multiforme or fixed drug eruption?
Patient
8-year-old boy
Duration
12 months
Distribution
neck and trunk
History

8-year-old Indian boy was referred to the dermatology clinic from the paediatric department for a recurrent skin eruption. The rash appeared on the chest, neck and trunk, preceded by fever for one week. He was well with no other symptoms, nor past medical history. There was no history of cold sore either. Parents denied any drug, supplement ingestion or specific diet prior to the fever and rash. The rash was recurrent which started about one year ago and recurred two months ago. On each occasion, the rash was preceded by fever and resolved after ten days with residual post-inflamatory hyperpigmentation. The rash was neither itchy nor painful and the distribution was random. Both occasions he was seen at the paediatric clinic and had a full blood count and coagulation profile done which were normal.

Physical Examination
On examination in our clinic, there were multiple annular, hyperpigmented lesions with erythematous rim on the chest, neck, and abdomen of a total of fewer than ten. Some lesions had bullae in the centre resembling target lesions. He was well and afebrile. There was no evidence of lymphadenopathy and no lesions in the oral mucosa. Other systemic examination was normal.
Images

Laboratory Data

 

Histopathology

A skin biopsy was performed and histopathology result showed dyskeratotic keratinocytes and subepidermal micro vesicles seen with basal vacuolations. The dermis was infiltrated with mild chronic inflammatory cells. Focally, there were neutrophils in the tip of the papillary dermis. The findings were consistent with erythema multiforme.

Diagnosis

Erythema multiforme vs Fixed Drug Eruption

Reasons Presented

Though clinical pictures are more consistent with FDE, there were no history of any drug or supplement ingestion or specific diet and the distribution of the lesions were random on each occasion. Repeat full blood count showed slightly elevated lymphocytes (49%) and normal autoimmune screen (complements 3 & 4, anti-nuclear factor). However the blood serology for Herpes simplex virus (HSV) IgM was positive. A repeat of blood serology for HSV IgM and subsequent IgG showed a rising titre and positive respectively confirming herpes simplex infection.

Questions
  1. Can we diagnosis FDE if the distribution of lesions was random in each recurrence and the parents were adamant that there were no related drugs or food involved?
  2. Does herpes simplex serology have any association with the rash (there is no association of FDE with HSV found in the literatures)?

 

References

1. Malnick SD, Green L: Erythema multiforme and herpes simplex virus. Lancet 1990 Feb 3; 335(8684): 302.
2. Huff JC, Weston WL, Tonnesen MG: Erythema multiforme: a critical review of
characteristics, diagnostic criteria, and causes. J Am Acad Dermatol 1983 June 8(6): 763-75.
3. Bolognia J, Jorizzo JL, Rapini RP. Dermatology 2003 ed. Elsevier; p313-316.
4. Weston WL, Morelli JG. Herpes simplex virus in childhood erythema multiforme. Pediatrics. 1992; 89:32-34.
5. Tatnall FM, Schofield JK, Leigh IM. A double-blind, placebo-controlled trial of continuous acyclovir therapy in recurrent erythema multiforme. Br J Dermatol. 1995;132:267-70.

Keywords fixed drug eruption, erythema multiforme, herpes simplex infection
Comments from Faculty and Members

Maria Lorna Frez M.D., Clinical Associate Professor in Dermatology, University of the Philippines College of Medicine, Philippines General Hospital, Manila, Phillipines on September 20, 2007

Taking everything into consideration, I favor the diagnosis of erythema multiforme over fixed drug eruption. The highly pigmented character of the lesions may be due to the patient's Indian race. To answer the question, there is a variant of FDE called "wandering FDE" where recurrent lesions develop on varying, instead of fixed location. The positive serology for herpes simplex is compatible with EM.

David Elpern M.D. , Williamstown, Massachusetts, USA on September 20, 2007

This is a difficult case. The clinical pictures are small -- we might benefit from having larger photos. In addition to what was suggested, atypical erythema multiforme-like lesions have been described in textile contact dermatitis. All of this child's lesions (at least those described above) have occurred on areas that are contacted by clothing -- so, this may need to be considered in the differential diagnosis.

Lazarov A. Textile dermatitis in patients with contact sensitization in Israel: a 4-year prospective study. J Eur Acad Dermatol Venereol. 2004 Sep;18(5):531-7.

Dermatology Clinic, Meir Hospital, 44281, Kfar Saba, affiliated with the Sackler School of Medicine, Tel Aviv University, Israel. lazarov1@netvision.net.il

Although chronic dermatitis was the typical clinical presentation, less frequent forms such as purpuric, hyperpigmented and papulopustular lesions and atypical forms such as erythema multiforme-like, nummular-like lesions, lichenification and erythrodermia were observed in 24.4% of the cases."

Khalid Al Aboud M.D., Medical Director, Makkah, Saudi Arabia on September 20, 2007

There is no comments on blood vessels in the dermis. The presence of neutrophils in the dermis may point to urticarial vasculitis. However, the morphology of the lesion is only with fixed drug eruption. Given the scenario that, it recurs with fever, it might be related to antipyretics. Detailed history is needed from the child and his parents.

Khalifa Sharquie MD, PhD, Professor, Baghdad, Iraq on September 20, 2007

Whatever the history, the cilinical picture and histopathological findings are that of FDE. Sometimes drugs in milk like sulfa given to cows might be a cause even there is no history of drugs intake.There is no relation to herpes simplex antibody titer but fever and reaction of FDE might contibuted to rising antibody in previously infected individual with herpes.

Ibrahim Misk M.D., Jordan. on September September 20, 2007

Clinically consistent with Fixed Drug Eruption but at the same time I must respect the opinion of the histopathologist for erythema multiforme.

Muhsin A. A. Aldhalimi MD, Head, Department of Dermatology, College of Medicine, University of Kufa, Najaf, Iraq on September 21, 2007

Thank you very much for this nice presentation. It rises again the question of histological differentiation of EM, TEN and FDE and whether they represent a spectrum of same disease or they are separate entities. The histological findings cannot differentiate definitely between the two differential diagnoses. From clinical point of view, the diagnosis most probably fixed drug eruption. We notice many patients with typical FDE lesions who don't give positive drug history. The drug history in an 8-year old child cannot be excluded definitely. He may take any simple drug from the home pharmacy without notice of the parents or the drug taken with animal-origin foods that may contain sulfa or tetracycline that are commonly used by veterinarians.

Bhushan Kumar MD, Former Head of Dermatology, Postgraduate Institute of Medical Education & Research, Chandigarh, India on September 22, 2007

Recurrent erythema multiforme due to recurrent herpes simplex infection is a recognised entity. FDE lesions can occur at variable sites but post inflammatory hyperpigmentation of the previous lesions is always striking - which in this case is missing.

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