Solitary mastocytoma arising from a tight infant identification bracelet

Wei-Liang Koh; Yong-Kwang Tay; Chee-Seng Sim

doi:10.4103/ijpd.IJPD_85_17

CASE REPORT

Year : 2018 | Volume : 19 | Issue : 3 | Page : 258-260

Solitary mastocytoma arising from a tight infant identification bracelet

Wei-Liang Koh¹, Yong-Kwang Tay¹, Chee-Seng Sim²
¹ Department of Dermatology, Changi General Hospital, Singapore
² Department of Pathology, Changi General Hospital, Singapore

Date of Web Publication

28-Jun-2018

Correspondence Address:
Dr. Wei-Liang Koh
Department of Dermatology, Changi General Hospital, 2 Simei Street 3, Singapore 529889
Singapore

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/ijpd.IJPD_85_17

Abstract

Mastocytosis is characterized by a proliferation of mast cells in the skin and extracutaneous organs. Cutaneous mastocytoma is the second most common presentation of mastocytosis in children and has been reported to arise post-trauma. We report herein the second case of solitary mastocytoma resulting from application of an excessively tight infant identification bracelet and the first in the English literature.

Keywords: Infant identification bracelet, mastocytoma, trauma

How to cite this article:
Koh WL, Tay YK, Sim CS. Solitary mastocytoma arising from a tight infant identification bracelet. Indian J Paediatr Dermatol 2018;19:258-60

How to cite this URL:
Koh WL, Tay YK, Sim CS. Solitary mastocytoma arising from a tight infant identification bracelet. Indian J Paediatr Dermatol [serial online] 2018 [cited 2020 Aug 11];19:258-60. Available from: http://www.ijpd.in/text.asp?2018/19/3/258/217488

Introduction

Mastocytosis is characterized by a proliferation of mast cells in the skin and extracutaneous organs. In children, mastocytosis is usually limited to the skin and can present as urticaria pigmentosa, cutaneous mastocytomas, diffuse cutaneous mastocytosis, and very rarely, telangiectasia macularis eruptiva perstans. Cutaneous mastocytoma is the second most common presentation of mastocytosis in children.^[1] These typically present within 1 year of age, majority within the first 3 months, usually as a single lesion.^[2] This can be an indurated red-brown macule, papule, plaque, or tumor up to few centimeters in diameter. Darier's sign may be positive in 50% of patients.^[3] Solitary mastocytomas have been reported to arise post-trauma.^[4],[5],[6] We report the second case of a solitary mastocytoma resulting from application of an excessively tight infant identification bracelet and the first in the English literature.

Case Report

A 6-month-old healthy Chinese female infant was brought to our department by her parents for evaluation of a “keloid” over her left heel. According to the mother, there was a linear cut over the patient's left heel due to an excessively tight plastic identification bracelet that was placed at birth, which was removed 3 days after. The cut healed with the development of a raised lump, which gradually increased in size. There were no similar lumps elsewhere.

Examination revealed a 2 cm × 0.7 cm brownish firm plaque with overlying scales over the patient's left heel [Figure 1]. Darier's sign was negative. Review of systems was unremarkable. The differential diagnoses considered were keloid, fibrous hamartoma of infancy, infantile myofibroma, and cutaneous mastocytoma.

Figure 1: Brownish firm linear plaque with overlying scales located over the left heel

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Histology from a 3-mm punch biopsy showed a diffuse infiltrate of mast cells arrayed in sheets and strands within the upper and mid dermis, highlighted with Giemsa and toluidine blue stains [Figure 2] and [Figure 3]. A diagnosis of solitary cutaneous mastocytoma was made. On revisiting the history, the parents did not notice the presence of redness, swelling, or blistering over the lesion before. Advice on triggers avoidance to decrease chances of mast cell degranulation was given. Active nonintervention was suggested since the patient was asymptomatic.

Figure 2: Punch biopsy showing hyperplastic epidermis with acanthosis and hyperkeratosis. Diffuse infiltrate of mast cells arrayed in sheets and strands within the upper and mid dermis (H and E, ×40)

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Figure 3: Diffuse dermal infiltrate of mast cells highlighted by Giemsa stain (×600)

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Discussion

Solitary mastocytomas have been reported to arise post-trauma following hepatitis B vaccination at the vaccination site,^[4] constriction of thigh from a tightly wrapped umbilical cord,^[5] and underneath the identification bracelet on the wrist of a newborn infant.^[6] Mastocytomas are postulated to represent a reactive process where cytokines upregulated in wound healing such as stem cell factor (SCF), interleukin-3 (IL-3), and nerve growth factor stimulate mast cell proliferation.^[5] Of interest, mast cells in a newborn maybe more sensitive to SCF in the presence of IL-3 post-injury as anin vitro study has shown that cord blood mast cells express a higher density of c-kit (which codes for a membrane receptor for SCF), compared to those from the peripheral blood of adult donors.^[7]

Treatment-wise, advice regarding triggers avoidance should be given for all patients (e.g., avoidance of friction, pressure, certain medications with mast cell degranulating properties such as aspirin, morphine, and codeine). Asymptomatic cases can be observed as most tend to resolve spontaneously.^[1] Symptomatic cases can be treated with potent topical or intralesional corticosteroids, topical calcineurin inhibitors, oral antihistamines, sodium cromoglycate, ketotifen, or surgical excision.^[8],[9]

In our patient, we postulate that her solitary cutaneous mastocytoma might have arisen as a reactive process secondary to preceding trauma from the tight identification bracelet. A similar case was reported in the Spanish literature where a solitary mastocytoma appeared under the identification bracelet on the left wrist of a 2-day-old newborn infant, with Darier's sign being positive.^[6] Another possibility would be that a congenital mastocytoma was already present over the patient's left heel. The heel was an area that could be chronically irritated from friction, leading to a mastocytoma with false-negative Darier's sign due to constant triggering.^[10] We favor the former hypothesis as trauma-induced solitary mastocytomas have been reported in the literature with plausible mechanisms delineated, and the parents reiterated that there was no preceding lesion before the cut caused by the identification bracelet. Solitary cutaneous mastocytoma should be considered as a differential in an infant presenting with a post-trauma lump. We report to the best of our knowledge the second case of a solitary mastocytoma that arose post-trauma associated with a tight infant identification bracelet.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

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