A Solitary Variant of Congenital Self‐healing Reticulohistiocytosis: Solitary Hasimoto‐Prltzker Disease

Abstract

Four neonates had solitary, congenital, rapidly growing, spontaneously ulcerating tumors of the face, trunk, and extremities. No extracutaneous involvement was found, and all lesions spontaneously involuted. Mononuclear cells of the cutaneous infiltrate were Langerhans' cells. These findings expand the spectrum of congenital self-healing reticulohistiocytosis.

Late manifestation of diabetes insipidus in “pure” cutaneous Langerhans cell histiocytosis

“…This entity appears to be characterised by four features [2,14]: (1) congenital or perinatal occurrence of papules, nodules or papulovesicules; (2) in an otherwise healthy child lacking visceral involvement; (3) histopathology demonstrating an LC in®ltrate; and (4) spontaneous regression of skin lesions without recurrence.…”

“…While LCH con®ned to the bones or lung has long been recognized [4], it was not until 1973 that pure cutaneous LCH was described [9]. Solitary forms, such as Langerhans cell (LC) histiocytoma [2], and disseminated forms of cutaneous LCH can be distinguished. The latter were termed pure cutaneous histiocytosis X [2,23] or congenital self-healing reticulohistiocytosis (Hashimoto-Pritzker disease in recognition of the original description [9]).…”

“…Solitary forms, such as Langerhans cell (LC) histiocytoma [2], and disseminated forms of cutaneous LCH can be distinguished. The latter were termed pure cutaneous histiocytosis X [2,23] or congenital self-healing reticulohistiocytosis (Hashimoto-Pritzker disease in recognition of the original description [9]). It is now generally accepted that this entity represents a form of LCH, and congenital self-healing LCH (CSHLCH) is the preferred designation [1,15].…”

Solitary cutaneous dendritic cell tumor in a child: Role of dendritic cell markers for the diagnosis of skin Langerhans cell histiocytosis

“…Several cases of a solitary form with a single cutaneous tumor have been published. [5][6][7] Solitary CSHRH seems to contribute to approximately 25% of cases. 8 Review of the literature reveals a variety of designations for this entity that do not necessarily have to be present by birth, but may appear during early childhood (Table I).…”

Congenital Solitary Histiocytoma: A Variant of Hashimoto-Pritzker Histiocytosis

“…Ultrastructural electron microscopy study demonstrated the presence of Birbeck granules in 0–40% of the histiocytes [5, 6]. In 1986, Berger et al [7] reported the first case of a rare form of this congenital self-healing reticulohistiocytosis that occurred in a newborn, who had a solitary cutaneous nodule and no other involvement. Since then, about 30 similar cases have been reported.…”