Ann Dermatol.  2014 Apr;26(2):250-253. 10.5021/ad.2014.26.2.250.

Solitary Congenital Erosion in a Newborn: Report of a Solitary Congenital Self-Healing Reticulohistiocytosis

• Kim JE
• Kim BJ
• Kang H

• KMID: 2171664
• DOI: http://doi.org/10.5021/ad.2014.26.2.250

Abstract

Congenital self-healing reticulohistiocytosis (CSHRH) is a rare, cutaneous, self-limited form of Langerhans cell histiocytosis. Whereas multiple lesions are common, a solitary lesion is rare. A 14-day-old neonate presented with a solitary, 5-mm, oval, reddish, and eroded papule with crust on the left thigh that had existed since birth. No systemic involvement was found. Histopathology revealed dense infiltration of large histiocytes with scattered eosinophils and lymphocytes in the dermis. Immunohistochemistry showed S-100 and CD1a positivity. Two months later, the skin lesion involuted spontaneously, without evidence of recurrence and extracutaneous involvement. On the basis of the characteristic clinical course and typical histopathological findings, a diagnosis of solitary CSHRH was made.

Keyword

Hashimoto-Pritzker syndrome; Histiocytosis; Langerhans-cell

MeSH Terms

Cytochrome P-450 CYP1A1
Dermis
Diagnosis
Eosinophils
Histiocytes
Histiocytosis
Histiocytosis, Langerhans-Cell
Humans
Immunohistochemistry
Infant, Newborn*
Lymphocytes
Parturition
Recurrence
Skin
Thigh
Cytochrome P-450 CYP1A1

Figure

• 

  Fig. 1 Clinical photographs of congenital self-healing reticulohistiocytosis. (A) A solitary, 5-mm, oval, firm, reddish, and eroded papule with crust on the left anterior thigh at initial presentation. (B) After 2 months, the lesion clinically resolved with hypertrophic scar.

• 

  Fig. 2 Histopathological findings of the lesion. (A) Focal erosion and crusting of the epidermis, indistinct dermoepidermal junction, and a dense dermal infiltrate in the upper-and mid-dermis are seen. (B) A dense dermal infiltrate consists of large and round histiocytic cells with various nuclei in shapes and sizes, from irregularly round to oval or kidney-shaped without atypia (A: H&E, ×40; B: H&E, ×400).

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  Fig. 3 Immunohistochemically, the histiocytic cells showed a positive reaction for S-100 (A) and CD1a (B) (A: S-100, ×100; B: CD1a, ×100).

Reference

1. Hashimoto K, Pritzker MS. Electron microscopic study of reticulohistiocytoma. An unusual case of congenital, self-healing reticulohistiocytosis. Arch Dermatol. 1973; 107:263–270.

2. Shin N, Kang MJ, Cho S, Whang KK, Hahm JH. A case of congenital self-healing reticulohistiocytosis. Ann Dermatol. 2000; 12:193–196.

3. Berger TG, Lane AT, Headington JT, Hartmann K, Burrish G, Levin MW, et al. A solitary variant of congenital self-healing reticulohistiocytosis: solitary Hashimoto-Pritzker disease. Pediatr Dermatol. 1986; 3:230–236.

4. Chun SI, Song MS. Congenital self-healing reticulohistiocytosis--report of a case of the solitary type and review of the literature. Yonsei Med J. 1992; 33:194–198.

5. Shin MS, Park HJ, Choi YJ, Park MY. A case of a solitary type of congenital self-healing reticulohistiocytosis. Korean J Dermatol. 2008; 46:411–413.

6. Writing Group of the Histiocyte Society. Histiocytosis syndromes in children. Lancet. 1987; 329:208–209.

7. Ersoy-Evans S, Gursoy T, Yigit S, Akcoren Z, Sahin S. Solitary congenital self-healing reticulohistiocytosis in monozygotic twins. Pediatr Dermatol. 2006; 23:273–275.

8. Zunino-Goutorbe C, Eschard C, Durlach A, Bernard P. Congenital solitary histiocytoma: a variant of Hashimoto-Pritzker histiocytosis. A retrospective study of 8 cases. Dermatology. 2008; 216:118–124.

9. Bernstein EF, Resnik KS, Loose JH, Halcin C, Kauh YC. Solitary congenital self-healing reticulohistiocytosis. Br J Dermatol. 1993; 129:449–454.

10. Masouyé I, Chavaz P, Salomon D, Balderer J, Carraux P, Saurat JH. Solitary Langerhans cell histiocytoma: an unusual form of Hashimoto-Pritzker histiocytosis? Pediatr Dermatol. 1990; 7:299–302.

11. Jensen ML, Bygum A, Clemmensen O, Fenger-Gron J. Congenital self-healing reticulohistiocytosis - an important diagnostic challenge. Acta Paediatr. 2011; 100:784–786.

12. Dorjsuren G, Kim HJ, Jung JY, Bae BG, Lee JH. Solitary type of congenital self-healing reticulohistiocytosis. Ann Dermatol. 2011; 23:Suppl 1. S4–S7.

13. Satter EK, High WA. Langerhans cell histiocytosis: a case report and summary of the current recommendations of the Histiocyte Society. Dermatol Online J. 2008; 14:3.