Reactions 1157 - 23 Jun 2007

Antineoplastics

Trichodysplasia spinulosa in children: 2 casereports

Two boys developed trichodysplasia spinulosa duringtreatment with antineoplastics [specific drugs and dosages notclearly stated] for acute lymphoblastic leukaemia (ALL).

An 8-year-old boy, who was diagnosed with ALL 2 yearspreviously and had started receiving antineoplastics accordingto the Children’s Cancer Group Protocol (CCG 1961),presented with a widespread, follicular-based skin eruption;his antineoplastic treatment included a 1-year intensivetreatment phase and ongoing maintenance phase usingmethotrexate, mercaptopurine and vincristine. Examinationrevealed follicular papules over his face, limbs and trunk, witha central keratinous spicule in most of the papules. He hadmild alopecia of his eyebrows. Skin biopsy showed keratoticplugging of the infundibulum along with hair follicle dilatation;enlarged and abnormal eosinophilic cells containing irregulartrichohyaline granules were present. No dermal papillae andhair shafts were observed. Electron microscopy showed viralparticle of about 30nm diameter. At this time, he hadprolonged and recurrent respiratory tract infections; theprominence of these infections during maintenance phase wasmore than expected. Investigations showedhypogammaglobulinaemia, absent CD19-positive B cells andreduced humoral response to tetanus, pneumococcus anddiphtheria. His immunological response was considered to bean exaggerated response to the antineoplastic treatment. Ninemonths after the onset of skin eruption, he started receivinggammaglobulin. Treatment with salicylic acid, ammoniumlactate, tretinoin and acitretin did not improve his eruption.His antineoplastic treatment was completed 15 months aftereruption onset and, 3 months later, his IgG levels normalised.His skin eruption persisted for another 6 months and thenrapidly regressed.

A 6-year-old boy, who was diagnosed with ALL 2 yearspreviously, developed fine, follicular, keratotic skin eruptionon his trunk that progressed to involve his limbs and face;4 months before eruption onset, he had completed anantineoplastic treatment for ALL according to the New York IIprotocol. The differential diagnosis included lichen nitidis,keratosis pilaris and follicular mucinosis. Skin biopsy showedfollicles dilation and keratotic plugging of the infundibulum,with excessive inner root sheath epithelium; no hair shafts anddermal papillae were observed. He did not receive any specifictreatment for his skin eruption. The eruption persisted foranother 3 months and then gradually resolved after a total of9 months.Sadler GM, et al. Trichodysplasia spinulosa associated with chemotherapy foracute lymphocytic leukaemia. Australasian Journal of Dermatology 48: 110-114,No. 2, May 2007 - Australia 801076922

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Reactions 23 Jun 2007 No. 11570114-9954/10/1157-0001/$14.95 Adis © 2010 Springer International Publishing AG. All rights reserved