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Case Reports
. 2016;45(6):20150440.
doi: 10.1259/dmfr.20150440. Epub 2016 Jul 25.

Haemophilic pseudotumour in two parts of the maxilla: case report

Ah-Young Kwon  1 , Kyung-Hoe Huh  1 , Won-Jin Yi  1 , Khanthaly Symkhampha  2 , Min-Suk Heo  1 , Sam-Sun Lee  1 , Soon-Chul Choi  1
Affiliations
Case Reports

Haemophilic pseudotumour in two parts of the maxilla: case report

Ah-Young Kwon et al. Dentomaxillofac Radiol. 2016.

Abstract

Haemophilic pseudotumour is a rare disease that occurs most often in femur, tibia, ilium or pelvic bone of a patient with haemophilia. Thus far, there have been only 31 reported cases in jaw bones and paranasal sinuses. Among them, the mandible is a more common site than the maxilla or paranasal sinuses. Here, we report a case of haemophilic pseudotumour in two parts of the maxilla. Contrast-enhanced CT showed an expansive and thinly corticated lesion with fluid attenuation at the left anterior maxilla which seemed like a post-operative maxillary cyst, ameloblastoma or odontogenic cyst. In addition, the thickened left palatal process of the maxilla seemed like fibrous dysplasia or intraosseous vascular malformation. Since haemophilic pseudotumour is not pathognomonic in radiological findings, when a patient who suffered from haemophilia or had taken anticoagulating agents has jaw lesion, haemophilic pseudotumour should be included in a differential diagnosis.

Keywords: diagnostic imaging; haemophilia B; maxilla; pseudotumour.

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Figures

Figure 1
Figure 1
(a) A periapical radiograph shows periapical rarefaction and periodontitis on the left maxillary first and second molars. (b) A cropped panoramic radiograph shows the elevation of the left hard plate and floor of the nasal cavity (double headed arrows). The left medial border of maxillary sinus is displaced in the direction of the nasal cavity (arrows). The oblique line in the left maxillary sinus is also observed (arrowheads).
Figure 2
Figure 2
(a) Coronal contrast-enhanced CT scan with bone window setting demonstrates different heights of both nasal floors (double-headed arrows). The lobulated and thinly corticated lesion pushes the lateral border of the nasal cavity (arrows). (b) Axial contrast-enhanced CT scan shows the expansive lesion with fluid attenuation in the anteromedial wall of the left maxillary sinus. The lesion compresses the nasolacrimal duct posteriorly (arrow). These radiographic findings mimic a post-operative maxillary cyst, ameloblastoma or odontogenic cyst such as keratocystic odontogenic tumour. (c) On the axial contrast-enhanced CT scan with bone window setting, another part of the lesion with slight altered trabecular bone pattern is observed on the palatal process of the maxilla. The nasopalatine duct is dislocated to the right side (arrow). The lesion mimics fibrous dysplasia or intraosseous vascular malformation. (d) On the sagittal contrast-enhanced CT scan with bone window setting, the cystic lesion A is adjacent to the intraosseous lesion B, and P is the pterygoid plate. (e) Axial contrast-enhanced CT scan with bone window setting demonstrates the septum in the left maxillary sinus which appears on the panoramic radiograph as an oblique line (arrowheads).
Figure 3
Figure 3
Photomicrograph of a histopathological slide. (a) Soft-tissue lesion. Under the mucosal epithelium of the maxillary sinus (arrow), haemorrhage surrounded by a fibrous capsule was observed [haematoxylin and eosin (H&E) stain, ×100]. (b) Intraosseous lesion. Between the bone tissues, the foci of the haemorrhage were scattered within the fibrous connective tissue (H&E stain, ×40).
Figure 4
Figure 4
Post-operative panoramic radiograph shows no pathological change at the operative site.
See this image and copyright information in PMC

References

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