Article
Long term follow up of topical mustine treatment for cutaneous
Langerhans cell histiocytosis
Peter H Hoegera, Vasanta R Nandurib, John I Harpera, David A Athertona, Jon Pritchardc
a Department of
Dermatology and Institute of Child Health, Great Ormond Street Hospital
for Children NHS Trust, London, UK, b Department of
Haematology/Oncology, Great Ormond Street Hospital for Children, c Department of Surgery, Great
Ormond Street Hospital for Children
Correspondence to: Dr P H Hoeger, University of Hamburg, Department of Dermatology, Division of Paediatric Dermatology, Martinistr. 52, D-20246 Hamburg, Germany email: hoeger{at}uke.uni-hamburg.de
Accepted 16 November
1999
BACKGROUND AND OBJECTIVES
Skin
lesions in Langerhans cell histiocytosis (LCH) are often painful and
difficult to treat. Topical application of nitrogen mustard (0.02%
mechlorethamine hydrochloride, mustine), an alkylating cytostatic
agent, has been shown to be effective. There is, however, concern about
potentially harmful long term side effects.
STUDY DESIGNIn a retrospective
study 20 children with LCH (average extent of initial skin involvement:
16.4% body surface) were followed up for an average of 8.3 years after
completion of topical mustine therapy. They had received a total of 34 courses (mean duration 14.2 weeks) of topical mustine. Disease status
on follow up was assessed according to the Histiocyte Society classification.
RESULTSAfter mustine was
introduced, 16 patients were able to discontinue systemic steroids
and/or chemotherapy. Topical mustine was well tolerated in 18 patients,
but two developed irritant dermatitis. On follow up, the disease was
inactive in 10 patients. Among the children with active disease, six
had mild skin disease and four had progressive disease, two of them
with skin lesions unresponsive to mustine treatment. Scars confined to
areas of formerly active skin disease were found in six patients. There was no evidence of premalignant or malignant skin disease in the treated areas.
CONCLUSIONTopical mustine is an
effective and safe treatment for skin disease in most children with
LCH. Residual scarring was probably a result of the disease itself
rather than to mustine. Although no evidence of skin cancer was found
in this study, continued long term follow up is advisable.
Keywords: cutaneous LCH; topical chemotherapy; long term follow up
© 2000 by Archives of Disease in Childhood
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