Juvenile xanthogranuloma: a case report and review of the literature

BMC Research Notes

Artina Pajaziti²  Syzana Rexhepi Hapçiu¹  Laura Pajaziti¹ 

[1] Clinic of Dermatovenerology, University of Prishtina, Rr. “Spitalit” p.n., 10000 Prishtina, Kosovo;University of Prishtina, Faculty of Medicine, 10000 Prishtina, Kosovo

关键词: Nodule;    Juvenile xanthogranuloma;   

Others  :  1134172
DOI  :  10.1186/1756-0500-7-174

 received in 2013-10-21, accepted in 2014-03-20,  发布年份 2014

【 摘 要 】

Background

Juvenile xanthogranuloma is a rare disorder which may be present at birth, or appears in infancy. It can also occur in adults of all ages; appears with lesions that may be solitary or multiple nodules several millimeters in diameter. The predilection sites are head and neck, but it may occur on the extremities and trunk also. There can also be involved internal organs such as lung, kidney, gastrointestinal tract, etc. The most frequent extracutaneous location is the eye.

Case presentation

We report a case of juvenile xanthogranuloma in a male child with onset in the fourth month of life. He presented with a nodule 8 millimeters in diameter, tan-orange in color, ulcerated in the centre, located on the left corner of the left eye. A biopsy without total excision was performed. After the biopsy, the nodule enlarged to 1.5 cm in diameter and became haemorrhagic. The histologic evaluation and immunohistochemistry analysis resulted in the diagnosis of juvenile xanthogranuloma. For aesthetic reasons the nodule was removed by surgical resection.

Conclusion

Juvenile xanthogranuloma is on a spectrum of histiocytic disorders, which is necessary to differentiate from maligniances in childhood by biopsy.

【 授权许可】

   
2014 Pajaziti et al.; licensee BioMed Central Ltd.

【 图 表 】

Figure 1.

【 参考文献 】

• [1]Dehner LP: Juvenile xanthogranuloma in the first decades of life. A clinicopathologic study of 174 cases with cutaneous an extracutaneous manifestations. Am J Surg Pathol 2003, 5:579-593. [PubMed]
• [2]Jung T, Emmert S, Günzl HJ, Neumann C, Rünger TM: Congenital manifestations of juvenile xanthogranuloma (large nodular form). Hautarzt 2000, 51:423-426. [PubMed]
• [3]Cypel TKS, Zuker RM MD: Juvenile xanthogranuloma: case report and review of the literature. Can J Plast Surg 2008, 16(3):175-177. Autumn
• [4]Hernandez-Martin A, Baselga E, Drolet BA, Esterly NB: Juvenile xanthogranuloma. J Am Acad Dermatol 1997, 36:335-367. [PubMed]
• [5]Chang MW: Update on juvenile xanthogranuloma: unusual cutaneous and systemic variants. Semin Cutan Med Surg 1999, 18:195-205. [PubMed]
• [6]Curtis T, Wheeler DT, Cibis GW, Law SK, Rowsey JJ, Brown LL, Roy H: Juvenile xanthogranuloma. [http://emedicine.medscape.com/article/1209681-overview webcite]
• [7]Sangüeza OP, Salmon JK, White CR Jr, Beckstead JH: Juvenile xanthogranuloma: a clinical, histopathologic and immunohistochemical study. J Cutan Pathol 1995, 22(4):327-335.
• [8]Liang S, Liu YH, Fang K: Juvenile xanthogranuloma with ocular involvement. Pediatr Dermatol 2009, 26(2):232-234.
• [9]Karcioglu ZA, Mullaney PB: Diagnosis and management of iris juvenile xanthogranuloma. J Pediatr Ophthalmol Strabismus 1997, 34(1):44-51. [Medline]
• [10]Adamson HG: Society intelligence: the dermatological society of London. Br J Dermatol 1905, 17:222.
• [11]Helwig EB, Hackney VC: Juvenile xanthogranuloma (nevoxanthoendothelioma). Am J Pathol 1954, 30:625-626.
• [12]Püttgen KB: Juvenile xanthogranuloma (JXG). [http://www.uptodate.com/contents/juvenile-xanthogranuloma webcite]
• [13]Sonoda T, Hashimoto H, Enjoji M: Juvenile xanthogranuloma. Clinicopathologic analysis and immunohistochemical study of 57 patients. Cancer 1985, 56:2280-2286. [PubMed]
• [14]Zelger B, Cerio R, Orchard G, Wilson-Jones E: Juvenile and adult xanthogranuloma. A histological and immunohistochemical comparison. Am J Surg Pathol 1994, 18:126-135. [PubMed]
• [15]Tomaszewski MM, Lipton GP: Unusual expression of S-100 protein in histiocytic neoplasms. J Cutan Pathol 1998, 25:129-135.