Brain Tumor Res Treat.  2024 Apr;12(2):148-151. 10.14791/btrt.2024.0018.

Pediatric Eosinophilic Granuloma Associated With Delayed Epidural Hematoma Following on Seizure: A Case Report

• Cho HJ
• Park SC
• Lee JM
• Jung NY
• Park JB

• KMID: 2555865
• DOI: http://doi.org/10.14791/btrt.2024.0018

Abstract

Eosinophilic granuloma (EG), a subtype of Langerhans cell histiocytosis (LCH), the monostotic form, is a rare condition characterized by a solitary bone lesion. It is even more unusual for this condition to be accompanied by an epidural hematoma (EDH). This case is unique in that it is the first to involve delayed EDH following a seizure. We describe a remarkable example of EG accompanied by an EDH and consider the rarity of this comorbidity. A 32-month-old boy developed a rapidly growing skull mass following a minor head injury. During surgical preparation for a biopsy, the patient experienced a single convulsion. Imaging following the seizure revealed an EDH in the vicinity of the mass. The mass was excised and confirmed to be an EG, but with positive margins. The patient underwent chemotherapy after systemic skeletal evaluation, in accordance with the LCH III protocol established by the Histiocytosis Society. EG is a rare neoplasm that typically presents as a painless growth on the skull that gradually enlarges over time. The correlation between EG and EDH is exceedingly uncommon, with only a few documented cases. This case study underscores the significance of considering EG in the differential diagnosis of an expanding cranium mass, even when associated with EDH. Prompt diagnosis and treatment can prevent serious complications and improve patient outcomes.

Keyword

Histiocytosis; Langerhans-cell; Eosinophilic granuloma; Cerebral hemorrhage; Seizures; Case report

Figure

• 

  Fig. 1 Preoperative radiologic findings. A: CT showing an osteolytic mass. B: T1-enhanced signal showing a markedly enlarged cystic lesion on the follow-up MRI scan taken 3 weeks after the fall.

• 

  Fig. 2 Gross image and histopathological analysis. A: The tumor is a tender osteolytic lesion with a crimson gelatinous hematoma appearance upon macroscopic inspection. B: During the pathological analysis, CD1a labeling shows affirmation and S-100 shows faint positivity. Affirmative edges are detected on the cryogenic biopsy.

Reference

1. Lee YS, Kwon JT, Park YS. Eosinophilic granuloma presenting as an epidural hematoma and cyst. J Korean Neurosurg Soc. 2008; 43:304–306. PMID: 19096637.

2. Mosiewicz A, Rola R, Jarosz B, Trojanowska A, Trojanowski T. Langerhans cell histiocytosis of the parietal bone with epidural and extracranial expansion - case report and a review of the literature. Neurol Neurochir Pol. 2010; 44:196–203. PMID: 20496290.

3. Mut M, Cataltepe O, Bakar B, Cila A, Akalan N. Eosinophilic granuloma of the skull associated with epidural haematoma: a case report and review of the literature. Childs Nerv Syst. 2004; 20:765–769. PMID: 15024599.

4. Chen HC, Shen WC, Chou DY, Chiang IP. Langerhans cell histiocytosis of the skull complicated with an epidural hematoma. AJNR Am J Neuroradiol. 2002; 23:493–495. PMID: 11901027.

5. Lee KW, McLeary MS, Zuppan CW, Won DJ. Langerhans’ cell histiocytosis presenting with an intracranial epidural hematoma. Pediatr Radiol. 2000; 30:326–328. PMID: 10836596.

6. Martínez-Lage JF, Bermúdez M, Martínez-Barba E, Fuster JL, Poza M. Epidural hematoma from a cranial eosinophilic granuloma. Childs Nerv Syst. 2002; 18:74–76. PMID: 11935249.

7. Manaka S, Izawa M, Nawata H. Skull tumor simulating sinus pericranii: case report. J Neurosurg. 1977; 46:671–673. PMID: 845656.

8. Cho DY, Liau WR, Chiang IP. Eosinophilic granuloma with acute epidural hematoma: a case report. Pediatr Neurosurg. 2001; 35:266–269. PMID: 11741122.

9. Albadr FB, Elgamal EA, Alorainy IA, Hassan HH. Fluid-fluid level in Langerhans cell histiocytosis of the skull. Neurosciences (Riyadh). 2006; 11:47–49. PMID: 22266503.

10. Bhat AR, Jain AK, Kirmani AR, Nizami F. Pathological intracranial extradural hematoma in a 10-year-old child. J Pediatr Neurosci. 2010; 5:164–166. PMID: 21559170.

11. Pawar RV, Hagiwara M, Milla S, Wisoff J, George AE. Eosinophilic granuloma presenting as post-traumatic scalp hematoma with epidural hemorrhage: a case report. Neuroradiol J. 2011; 24:767–771. PMID: 24059774.

12. Bakhaidar MG, Alghamdi FA, Baeesa SS. Spontaneous extradural hemorrhage due to Langerhans cell histiocytosis of the skull in a child: a rare presentation. J Pediatr Neurosci. 2016; 11:52–55. PMID: 27195034.

13. Sadashiva N, Baruah S, Rao S, Mahadevan A, Rao KN, Vazhayil V, et al. Eosinophilic granuloma of skull with fluid level and epidural hematoma: a case report and review of the literature. Pediatr Neurosurg. 2017; 52:41–45. PMID: 27595542.

14. Al-Mousa A, Altarawneh M, Alqatawneh O, Bashir Z, Al-Dwairy S, Shtaya A. Eosinophilic granuloma of the skull presenting as non-traumatic extradural haematoma in children. Int J Gen Med. 2020; 13:1229–1234. PMID: 33262638.

15. Ueno K, Katayama K, Mizukami A, Nomura Y, Watanabe R, Sasaki T, et al. Spontaneous remission of skull Langerhans cell histiocytosis that had developed by repeated head injury: illustrative case. J Neurosurg Case Lessons. 2023; 6:CASE2327. PMID: 37728298.

16. Tazi A, Hiltermann JN, Vassallo R. Adult lung histiocytosis. Weitzman S, Egeler RM, editors. Histiocytic disorders of children and adults: basic science, clinical features and therapy. 1st ed. Cambridge: Cambridge University Press;2005. p. 187–207.

17. D’Ambrosio N, Soohoo S, Warshall C, Johnson A, Karimi S. Craniofacial and intracranial manifestations of Langerhans cell histiocytosis: report of findings in 100 patients. AJR Am J Roentgenol. 2008; 191:589–597. PMID: 18647937.

18. Rawlings CE 3rd, Wilkins RH. Solitary eosinophilic granuloma of the skull. Neurosurgery. 1984; 15:155–161. PMID: 6483131.