A case of intralymphatic histiocytosis mimicking lymphangioma circumscriptum associated with a puncture wound caused by a thorn

Abstract

A 83-year-old Japanese female presented with a skin lesion on her right fifth finger, which had developed after she had pricked her finger on a Sudachi thorn 2 months earlier. A physical examination revealed a light brown, slightly papillomatotic plaque on the right fifth finger. A histopathological examination of a biopsy specimen demonstrated numerous dilated vessels in the dermis, and some of them showed the internal aggregation of mononuclear cells. These cells were positive for CD68, and the surrounding vessels expressed D2-40. A tissue culture of the skin showed no fungal growth. Based on these findings, we diagnosed her with intralymphatic histiocytosis (IH). The patient was monitored without any specific treatment. At 7 months after her initial visit, there were no significant changes, but her right fifth finger exhibited lymphedema. It is well known that IH is associated with chronic rheumatoid arthritis, but other conditions, such as artificial joint replacement, infection, malignancy, and trauma, have also been reported to be associated with IH. In our case, the prick of a Sudachi thorn may have triggered the IH, but there have been no reports of similar cases.