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Vol. 26. Num. 6.November - December 2015Pages 261-310
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Vol. 26. Num. 6.November - December 2015Pages 261-310
Case Report
DOI: 10.1016/j.neucir.2015.04.004
Chronic intradiploic hematomas of the skull without coagulopathy: Report of two cases
Hematoma crónico intradiploico sin coagulopatía: informe de 2 casos
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Mehmet Tokmaka,
Corresponding author
drmehmettokmak@gmail.com

Corresponding author.
, Erdinc Ozeka, Celal Iplikçioğlub
a Neurosurgery Department, Medipol University, Istanbul, Turkey
b Neurosurgery Department, Harran University, Urfa, Turkey
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Table 1. Reported intradiploic hematoma cases in the literature.
Abstract
Background

Chronic intradiploic hematoma was first described by Chorbski and Davis in 1934. To date, only twelve cases have been reported in the literature. Chronic intradiploic hematomas have also been described as non-neoplastic cysts of the diploe, traumatic cysts, and giant cell repetitive granulomas. The term chronic intradiploic hematoma was coined by Sato et al. in 1994.

Case description

Case 1: a 16-year-old male presented with a non-tender scalp swelling on the right fronto-orbital region. Computed tomography (CT) scans showed an intraosseous isodense lesion with surrounding sclerosis. Magnetic resonance imaging (MRI) results revealed an intradiploic mass with homogeneous cystic and abnormal signal intensities. Case 2: a 64-year-old male presented with a 6-month history of headaches and visual disturbances. CT scans showed an isodense lesion with surrounding sclerosis in the posterolateral left orbit. MRI results revealed a hypointense lesion on both the T1weighted and T2-weighted images of the posterolateral left orbit.

Conclusion

Although rarely seen, the presence of a benign chronic hematoma should be considered as part of the differential diagnosis for each intradiploic mass lesion. When taking the medical history, all patients with such mass lesions should be asked about previous minor or major head traumas. Due to the possibility of progressive growth, surgical excision of an intradiploic hematoma is recommended after radiological diagnosis of the condition.

Keywords:
Intradiploic space
Hematoma
Skull
Resumen
Antecedentes

El hematoma crónico intradiploico fue descrito por primera vez por Chorbski y Davis en 1934. Hasta la actualidad solo se han publicado 12 casos en la literatura. Los hematomas crónicos intradiploicos también se han descrito como quistes no neoplásicos del diploe, quistes traumáticos y granulomas repetitivos de células gigantes. El término hematoma crónico intradiploico fue acuñado por Sato et al. en 1994.

Descripción de los casos

Caso 1: Varón de 16años que consultó por hinchazón no dolorosa en región frontoorbitaria derecha. La tomografía computarizada mostró una lesión intraósea isodensa con esclerosis circundante. La resonancia magnética (RM) reveló una masa intradiploica con quiste homogéneo de señal anormal. Caso 2: Varón de 64años que consultó por historia de 6meses de dolor de cabeza y alteraciones visuales. La tomografía computarizada craneal mostró una lesión isodensa con esclerosis circundante en la región posterolateral de la órbita izquierda. La RM mostró una lesión hipointensa tanto en las secuencias TI como T2 de la porción posterolateral de la órbita izquierda.

Conclusión

A pesar de su rareza, la presencia de un hematoma crónico benigno debe considerarse en el diagnóstico diferencial de cada lesión masiva intradiploica. A los pacientes con este tipo de lesiones de masa se les debe interrogar sobre el antecedente de trauma craneal previo mayor o menor. Debido a la posibilidad de crecimiento progresivo, se recomienda la extirpación quirúrgica del hematoma intradiploico una vez realizado el diagnóstico radiológico de la enfermedad.

Palabras clave:
Espacio intradiploico
Hematoma
Cráneo
Full Text
Introduction

A chronic intradiploic hematoma is a rare type of lesion that usually presents as a slowly growing skull mass following a minor head injury. Although the pathogenesis is not clear, a benign reparative reaction to traumatic diploic bleeding is suspected to be the cause of the cyst formation. We present two cases of chronic intradiploic hematoma without coagulopathy after minor trauma.

Case reportsCase 1

A 16-year-old male presented with a non-tender scalp swelling on the right fronto-orbital region. He had a history of a minor head trauma in the same region two years earlier, and the aforementioned scalp swelling started one year after the trauma. A neurological examination was unremarkable. No pain was associated with the lesion; moreover, the lesion was not soft and showed no signs of inflammation. All hematological and biochemical tests were normal. X-rays of the cranium showed a large round osteolytic lesion in the right frontal-orbital bone with surrounding sclerosis (Fig. 1). Non-contrast CT scans showed an intraosseous isodense lesion with surrounding sclerosis, expansion of the diploe, and erosion of the inner and outer tables. MRI results revealed an intradiploic cystic mass that was hyperintense on both T1- and T2-weighted sequences (Fig. 2). No abnormalities were observed in the adjacent brain tissue. In surgery to treat the lesion, we noticed that the outer and inner tables were damaged by the cyst. A brownish fluid was evacuated from the cystic lesion, and the effected bone was totally curetted. Light microscopy of the specimen obtained from the cyst revealed fibrous material with hemosiderin-laden macrophages. Light microscopy of the curetted bone specimen showed fibroblastic reactions with hemosiderin-laden macrophages and lymphocytes (Fig. 3a and b). These features are consistent with an organizing hematoma. The postoperative course was uneventful.

Fig. 1.
(0.15MB).

Lateral plain X-ray showing a large round osteolytic lesion with surrounding sclerosis located on the right frontal orbital bone.

Fig. 2.
(0.14MB).

T1-weighted MRI demonstrating a hyperintense lesion in the right frontal region. Notice that there is no abnormality on the underlying brain.

Fig. 3.
(0.13MB).

(a) Magnified views of photomicrographs of the hematoma (after dyeing with hematoxylin eosin 10× (a), 40× (b)). Histopathological examination revealed fibroblastic reactions with hemosiderin-laden macrophages and lymphocytes.

Case 2

A 64-year-old male presented with a 6-month history of headaches and visual disturbances. The patient had suffered a minor head trauma one year before admission. Physical examination revealed swelling over the left frontal bone. The mass was non-pulsatile, and the patient reported no tenderness upon palpation. The neurological examination was normal, and the patient's hematological and biochemical profiles were unremarkable. X-rays of the skull were normal. Non-contrast CT showed an isodense lesion with surrounding sclerosis in the posterolateral of the left orbit. The bone defect was irregular and involved both the external and inner tables. MRI revealed a hypointense lesion on both the T1-weighted and T2-weighted images; this lesion had a diameter of 1.5cm and was located on the posterolateral of left orbital (Fig. 4). The patient underwent surgery, and the destroyed bone was curetted. Histopathology of the curetted material revealed fibroblastic reactions with old hemorrhagic foci in the calcified bone tissue (i.e., hemosiderin-laden macrophages) and some lymphocytes and cholesterol clefts, all of which are consistent with an organizing hematoma (Table 1). The postoperative course was uneventful.

Fig. 4.
(0.13MB).

T1-weighted MRI revealing a hypointense mass in the left fronto-orbital region.

Table 1.

Reported intradiploic hematoma cases in the literature.

Author/year  Age/sex  Features  Bone  Radiology/imagining  Histopathology  Treatment 
Mercado 1984  31 months/M  Minor head injury 10 months of age. Swelling noted 2 months later  Parietal  Inner table intact. Outer table expansion  Giant cell reparative granuloma  Excision and cranioplasty 
Palantinsky 1986  70 years/M  Minor head injury 40 years prior, scalp swelling noted after injury  Parietal  Inner and outer table eroded  Vascularized granulation tissue+ macrophages with vacuoles and hemosiderin. FB giant cells – chronic  Excision and cranioplasty 
Yuasa 1992  20 years/M  Minor head injury at 15 years of age. Incidental finding at later date  Parietal  Inner table expansion  Intradiploic hematoma. Collagenous tissue. Cystic lesion within diploe containing nothing – dense wall  Excision and cranioplasty 
Sato 1994  20 years/M  Minor head injury 4 years prior. Incidental finding at later date  Parietal  MRI: high intensity with low Central area on T1. T2 low intensity with high central area. Contrast enhancement noted.  Central cyst with xanthochromic fluid. Cyst wall consisted of non-neoplastic granulation tissue  Excision and cranioplasty 
Goel 1996  17 years/M  Minor head injury. Progressive scalp swelling/proptosis for 8 years with hemophilia  Frontal  CT: intradiploic lesion expanding frontal bone  Intraosseous hematoma  Evacuation of hematoma 
Uemura 1999  32 years/M  Minor head injury at 13 years of age with intermittent headaches  Frontal  MRI: intradiploic mass with heterogeneous intensity on T1  Organizing hematoma  Excision 
Yucesoy 1999  25 days/M  Birth trauma. Scalp swelling noted after difficult delivery  Parietal  CT: intraosseous isodense lesion with surrounding sclerosis  Granulation tissue and organizing hematoma  Excision 
Mobbs 2000  3 years/M  Major head injury 2 years prior  Parietal  CT: Inner table intact Outer table expansion  Organizing hematoma  Curettage 
Goel 2000  58 years/M  Minor head injury 24 years prior. Swelling/proptosis  Frontal  CT: intradiploic lesion Expanding frontal bone  Organizing hematoma  Excision 
Present case 1  16 years/M  Minor head injury 2 years prior. Swelling noted 1 year later  Frontal  CT: intraosseous isodense lesion with surrounding sclerosis  Organizing hematoma  Excision 
Present case 2  64 years/M  Minor head injury 1 year prior. Swelling noted after sclerosis  Frontal  CT: intraosseous isodense Lesion with surrounding admission  Organizing hematoma  Curettage 
Discussion

The chronic intradiploic hematoma lesion was first described by Chorbski and Davis in 19341 and was named by Sato et al.2 To date, only 12 cases of chronic intradiploic hematoma have been reported in the literature.1–11 Other names found in the literature for intradiploic hematoma include non-neoplastic cyst of diploe, traumatic cyst, and giant cell repetitive granuloma.4,6,9,10,12

The exact pathogenesis of the intradiploic hematoma is not clear. However, chronic intradiploic hematomas associated with anticoagulant use, birth trauma, and shunt surgery have been reported. Moreover, acute hematomas in the various layers of the scalp and the skull usually resolve spontaneously.1 However, trauma can initiate bleeding in the diploic space. If the resulting diploic hematoma is not absorbed, the surrounding connective tissue usually encapsulates the hematoma and creates a cyst.1 The natural history of this connective tissue can show various stages of differentiation, i.e., fibrous tissue, osteoid tissue, and sometimes osseous tissue, followed by the disappearance of all foreign elements.1,13 Intradiploic hematomas can cross the sutures, while subperiosteal hematomas cannot.

The literature reports of patients with chronic intradiploic hematomas usually describe some common features for the patients. The patients are usually young adult males, yet children with intradiploic hematomas have been also reported.3,4,7,8,11 The hematological and biochemical profiles of the patients in the literature were normal with exception in three patients who had a factor VIII deficiency, thereby causing progressive growth in the size of the hematoma after diagnosis.3,4,13 Only one patient had suffered a major head injury 2 years prior to the skull mass occurrence.7 Although the parietal and frontal bones were the most affected sites, temporal and occipital bones were involved in cases. The cases in this report shared similar features with the previously reported cases.

Skull films reveal the expansion of the diploe in cases of hyperostosis or the thinning of the inner and outer tables. The outer table of the skull was intact in all cases except three, including the two cases in the present report; in these three cases, both the outer and inner tables were destroyed.7 CT can reveal intradiploic masses of varying densities. However, Yuasa et al. stated that CT may not be sufficient to determine if the lesion is interosseous, epidural, or subdural.10 MRI examination was first used to diagnose intradiploic hematomas by Sato and colleagues.2 In contrast to CT, MRI provides more precise information, which allows for the determination of the type of bone lesion, and demonstrates the exact location of the lesion and extent of the lesion's effects on the adjacent structures. MRI reveals variable signal intensities for lesions within the diploic space. Contrast enhancement was observed in only one case.2 In both of the cases presented in this paper, the lesions appeared isodense in CT scans. However, the intradiploic mass was hyperintense in T1- and T2-weighted MRI images in case 1 in this report, but the intradiploic mass was hypointense in T1- and T2-weighted images for case 2 in this report. This variation is probably due to the age of and the organization of the hematoma.

A correct diagnosis could only be established after a histopathological examination, which can differentiate between an old hematoma and a cyst with a wall of granulation tissue or collagenous tissue. New bone formation also could be observed in some cases.4,6,9–12 Chronic intradiploic hematomas may radiologically or histologically resemble giant cell tumors, aneurysmal bone cysts, giant cell repetitive granulomas, or fibrous dysplasia. Some authors believe that intradiploic hematomas and the giant cell repetitive granulomas are produced by the same process.7 However, giant cell repetitive granulomas are usually seen in the mandible and considered as a local reparative reactive process following a traumatic intraosseous hemorrhage; furthermore, histological examinations show reactive osteoclast-like giant cells and fibroblastic cells.13,14

Total surgical removal of the hematoma is the treatment of choice for chronic diploic hematomas. No additional treatment is typically required. In some cases, reconstructive cranioplasty might be useful for either cosmetic or protective reasons. Since most of these lesions are in the frontal visible region, we recommend including cosmetic cranioplasty within the same surgical procedure. However, some authors preferred to preserve the intact inner table in cases when the inner table is thin and compressible.3

Conclusion

Although rarely seen, the presence of a benign chronic hematoma should be considered as part of the differential diagnosis in each case of an intradiploic mass lesion. All patients with such lesions should be asked about previous minor or major head traumas in the area under investigation. Due to the progressive growth possibility, we recommend surgical excision of the intradiploic hematoma after radiological diagnosis.

References
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J. Chorbski,L. Davis
Cyst formations of the skull
Surg Gynecol Obstet, 58 (1934), pp. 12-31
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K. Sato,T. Kubota,H. Kawano
Chronic diploic hematoma of the parietal bone. Case report
J Neurosurg, 80 (1994), pp. 1112-1115 http://dx.doi.org/10.3171/jns.1994.80.6.1112
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N. Dange,A. Mahore,K.M. Avinash,V. Joshi,J. Kawale,A. Goel
Chronic intradiploic hematoma in patients with coagulopathy
J Clin Neurosci, 17 (2010), pp. 1047-1049 http://dx.doi.org/10.1016/j.jocn.2009.11.030
[4]
R. Garza-Mercado,E. Cavazos,F. Hernandez-Batres
Giant cell reparative granuloma of the cranial vault: exceptional bone lesion
Neurosurgery, 15 (1984), pp. 228-232
[5]
A. Goel,K. Desai,S. Patil
Unusual intradiploic haematoma: case report
J Neurosurg, 93 (2000), pp. 895-896 http://dx.doi.org/10.3171/jns.2000.93.5.0895
[6]
A. Goel,T. Nadkarni,D. Mazumdar
Intradiploic haematoma behaving as a progressive mass lesion
Br J Neurosurg, 10 (1996), pp. 611-612
[7]
R.J. Mobbs,P.R. Gollapudi,J.W. Fuller
Intradiploic haemotoma after skull fracture: case report and literature review
Surg Neurol, 54 (2000), pp. 87-91
[8]
S. Nagi,H. Megdiche,H. Daoud,M. Bouchriha,R. Sebaï,L. Belghith
Intradiploic hematoma: report of two cases and review of literature
Tunis Med, 83 (2005), pp. 426-428
[9]
K. Uemura,K. Tsuboi,T. Nose
Intradiploic hematoma
Am J Neuroradiol, 18 (1997), pp. 397-398
[10]
H. Yuasa,H. Watanabe,Y. Uemura,S. Tokito
Intraosseous hematoma of the skull: case report
Neurosurgery, 30 (1992), pp. 776-778
[11]
K. Yucesoy,T. Mertol,H. Ozer,E. Ozer
An infantile intraosseous hematoma of the skull. Report of a case and review of the literature
Childs New Syst, 15 (1999), pp. 69-72
[12]
E. Palatinsky,G. Liccardo,N. Di Lorenzo
Chronic subperiosteal hematoma of the skull in an adult: a case report
Neurosurgery, 19 (1986), pp. 294-296
[13]
H.L. Jaffe
Giant-cell reparative granuloma, traumatic bone cyst, and fibrous (fibro-oseous) dysplasia of the jawbones
Oral Surg Oral Med Oral Pathol, 6 (1953), pp. 159-175
[14]
L.T. Austin Jr.,D.C. Dahlin,R.Q. Royer
Giant-cell reparative granuloma and related conditions affecting the jawbones
Oral Surg Oral Med Oral Pathol, 12 (1959), pp. 1285-1295
Copyright © 2014. Sociedad Española de Neurocirugía
Neurocirugía (English edition)

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