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LETTERS TO EDITOR
Year : 2018  |  Volume : 66  |  Issue : 3  |  Page : 866-868

Calvarial intraosseous leiomyoma: Another consideration in children with suspected esosinophilic granuloma


Department of Neurosurgery, Zhongshan City People's Hospital, Zhongshan, Guangdong, China

Date of Web Publication15-May-2018

Correspondence Address:
Dr. Wenjian Zheng
Department of Neurosurgery, Zhongshan City People's Hospital, 2nd Sunwen West Street, Zhongshan, Guangdong – 528403
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.232331

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How to cite this article:
Zheng W, Li L, Lin S. Calvarial intraosseous leiomyoma: Another consideration in children with suspected esosinophilic granuloma. Neurol India 2018;66:866-8

How to cite this URL:
Zheng W, Li L, Lin S. Calvarial intraosseous leiomyoma: Another consideration in children with suspected esosinophilic granuloma. Neurol India [serial online] 2018 [cited 2018 May 20];66:866-8. Available from: http://www.neurologyindia.com/text.asp?2018/66/3/866/232331




Sir,

An 11-year old boy complained of a painless, growing mass on the forehead for the past 3 months. He had no headache or visual symptoms. On examination, the mass was firm, non-tender, and non-pulsatile. His medical history was unremarkable. The magnetic resonance imaging (MRI) of the brain showed a 20-mm × 15 mm lesion, on the right frontal bone [Figure 1] and [Figure 2].
Figure 1: Magnetic resonance imaging (MRI) revealed a 20 mm×15 mm mass arising from the frontal bone extending outwards. (Transverse plane)

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Figure 2: Magnetic resonance imaging (MRI) revealed a 20 mm×15 mm mass arising from the frontal bone extending outwards. Intracranial involvement was not evident. (Coronal plane)

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Due to its rapid course and destruction of the bone, a diagnosis of eosinophilic granuloma (EG) was the first consideration. Total resection of the mass was performed. The mass was firm and hypo-vascular. It eroded the whole layer of the frontal bone but spared the dura. We drilled the adjacent bone 5 mm beyond the tumor edge. The bony defect was repaired with a small titanium plate.

Histological analysis showed that the specimen mainly contained spindle cells with positive immunohistochemistry staining for vimentin and smooth muscle actin (SMA), which was consistent with the diagnosis of a leiomyoma [Figure 3]. Three months after the surgery, computed tomography (CT) showed no evidence of local recurrence.
Figure 3: Histoathological findings showed a tumor composed of densely packed spindle cells with cigar-shaped nucleus, moderate amount of eosinophilic cytoplasm, surrounding a few blood vessels. (hematoxylin and eosin, original magnification ×200)

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Leiomyoma is the proliferation of spindle-shaped cells arranged in thick intersecting bundles. Until now, only 23 cases of intraosseous leiomyoma (IOL) in the orofacial region, and four cases of this entity in the skull have been reported.[1],[2],[3]v The age of onset of this tumor ranged from 8 to 15 years.

Bony destruction due to the presence of an IOL can be well-observed on a CT scan. Due to its rarity, it is usually not considered in the radiographic differential diagnosis. The more likely diagnostic considerations for this radiographic presentation include EG, dermoid cyst, fibrous dysplasia, hemangioma, and myofibroma. EG is a localized form of histiocytosis X. It has a similar age of onset, disease course, and imaging finding as an IOL. EG may, however, be accompanied by fever and leukocytosis, and the surgical finding could be different [Table 1]. Definitive diagnosis is made basically on histological examination and immunohistochemical staining. Leiomyomas are benign, well-differential smooth muscle tumors characterized by the positive immunoreactivity of SMA and vimentin within them.
Table 1: Differential diagnosis between IOL and EG

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Once the decision to undertake surgery for these tumors is made, EG requires total resection of the tumor and adjacent bone. The recurrence rate of leiomyomas, is however, notably rare. No recurrence was observed in patients harboring a leiomyoma even when they underwent only enucleation of the tumor.

Unfortunately, we failed to recognize the possibility of leiomyoma in our case. During surgical exploration, if tumor appearance is no typical of an EG, an intraoperative frozen section examination should be performed. This can avoid unnecessary skull bone removal and it consequent reconstruction. This fact is of great significance in children with a suspected EG. In the presence of a small defect, a cranioplasty is usually performed with harvested autologous bone chips. In the presence of whole-layer or larger defects, reconstruction should be made with titanium plates for better protection of the underlying structures and for the cosmetic effect provided by the latter method of reconstruction. The risk of implant deformation is noticeable in children due to the growth and maturation of their calvarium.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Jeung CU SS, Kim SH, Song KS. Primary leiomyoma of the temporal bone. J Korean Neurosurg Soc2004;35:2.  Back to cited text no. 1
    
2.
Wu Q, Liu C, Luo X, Fan Y, Zhou L, Liu Q. Primary leiomyoma of the parietal bone. Neurol India 2013;61:686-7.  Back to cited text no. 2
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3.
Vijayasaradhi M, Uppin SG, Sreedhar V, Sundaram C, Panigrahi MK. Frontal intradiploic angioleiomyoma. J Neurosurg Pediatr2008;2:266-8.  Back to cited text no. 3
[PUBMED]    


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