- Department of Medicine, Albert Einstein Israeli Faculty of Health Sciences, São Paulo, Brazil.
- Department of Medicine, Catanduva Medical School (FAMECAUNIFIPA), Catanduva, Brazil.
- Department of Medicine, Santa Casa de Sao Paulo School of Medical Sciences, Sao Paulo, Brazil.
- Department of Medicine, São Leopoldo Mandic, Campinas, Brazil.
- Department of Neurosurgery, Hospital Saúde de Caxias do Sul, Caxias do Sul, Brazil.
- Department of Neurology, Pontifical Catholic University of São Paulo, Sorocaba, Brazil.
DOI:10.25259/SNI_918_2020
Copyright: © 2021 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.How to cite this article: Rafael Trindade Tatit1, Paulo Eduardo Albuquerque Zito Raffa2, Giovana Cassia de Almeida Motta3, André Alexandre Bocchi4, Júlia Loripe Guimaraes1, Paulo Roberto Franceschini5, Paulo Henrique Pires de Aguiar6. Rosai-Dorfman disease mimicking images of meningiomas: Two case reports and literature review. 21-Jun-2021;12:292
How to cite this URL: Rafael Trindade Tatit1, Paulo Eduardo Albuquerque Zito Raffa2, Giovana Cassia de Almeida Motta3, André Alexandre Bocchi4, Júlia Loripe Guimaraes1, Paulo Roberto Franceschini5, Paulo Henrique Pires de Aguiar6. Rosai-Dorfman disease mimicking images of meningiomas: Two case reports and literature review. 21-Jun-2021;12:292. Available from: https://surgicalneurologyint.com/surgicalint-articles/10905/
Abstract
Background: Rosai-Dorfman disease (RDD) is a rare non-Langerhans cell histiocytic proliferative disorder classically as a massive cervical lymphadenopathy. However, over the years, extranodal locations were confirmed with the central nervous system involvement in less than 5% of cases, which is marked as a significant differential diagnosis of meningiomas, with which they are widely confused due to the similarity of their radiological images.
Case Description: We report a 37-year-old man and 45-year-old man who were diagnosed with intracranial RDD but whose radiological images mimic meningiomas, requiring anatomopathological and tumor’s immunohistochemistry for definitive diagnosis. Moreover, a review of 184 publications with 285 cases of intracranial involvement of this disease was also performed, comparing these findings with those brought in the previous studies.
Conclusion: Intracranial Rosai-Dorfman tumors should always be remembered as differential diagnosis of meningiomas since they are similar radiologically and macroscopically. Once remembered and diagnosed, the lesion must be treated following the same pattern of resection done in meningiomas and, treatment’s differences will not occur in the surgical excision technique, but in complementary chemotherapy implementation, radiotherapy, and even with radiosurgery aid, depending on the case. Thus, it is possible to obtain better results than with just the isolated surgical procedure.
Keywords: Central nervous system, Histiocytosis, Magnetic resonance imaging, Meningioma, Rosai-Dorfman disease
INTRODUCTION
Rosai-Dorfman disease (RDD) is a rare non-Langerhans cell histiocytosis characterized by accumulation of activated histiocytes in the affected tissues. Widely heterogeneous and with a variety of clinical phenotypes, it may be present from the isolated form to the form in association with other diseases such as autoimmune,[
His first description dates back to 1965 by a French pathologist - Pierre Paul Louis Lucien Destombes,[
In intracranial RDD, the most involved structures are the suprasellar region, cerebral convexity, parasagittal region, cavernous sinus, and petroclival region,[
The present study reports two cases of RDD with intracranial involvement, one of them with follow-up of more than 15 years. A review of 184 publications with 285 cases of RDD with CNS involvement (CNS-RDD) was also performed, comparing these findings with those brought in the previous studies. For identifying the studies, the MeSH tool from PubMed database was used, using the keywords “Histiocytosis, Sinus” restrict to MeSH Major Topic (entry terms: histiocytoses, Sinus; Sinus Histiocytoses; Sinus Histiocytosis; RDD; Disease, Rosai-Dorfman; RDD; Sinus Histiocytosis with Massive Lymphadenopathy; DestombesRosai-Dorfman Syndrome; Destombes Rosai Dorfman Syndrome; and Syndrome, Destombes-Rosai-Dorfman) and the keyword “Central Nervous System;” no filter was used for languages, date of publication or type of study. In addition, manual searches were performed based on the studies found by the initial electronic search. All articles including new cases of the disease and containing basic information (sex, age, location of the pathology, and if there was isolated involvement of the CNS) were included in the study.
CASES REPORTS
First case report
Male patient, 37 years old, presented 4 years before with painless left supraclavicular adenomegaly, with progressive increase followed by intense pain in the left clavicle after physical activity. Imaging examinations demonstrated the presence of bone infiltration, supraclavicular and infraclavicular adenomegaly, as well as lesions in the orbit and cranial cap. Biopsy of supraclavicular lymph node confirmed lymphadenitis with massive sinus histiocytosis compatible with RDD, with immunohistochemical examination demonstrating CD68 and S100 positive and CD30 and CD1a negative. The patient initially presented an excellent response with corticoids using, noting significant regression of adenomegaly, and general improvement of symptoms. In the last year, however, he began to refer to migratory arthralgia with an increase in cervical adenomegalies, requiring the continuous use of corticoids and increased doses in exacerbations, and he presented with pulsatile headache which was often disabling. A skull MRI was performed which revealed an expansive lesion in the left frontal region [
Figure 2:
Magnetic resonance imaging at the post operated showed total resection of the left frontal tumor associated with edema, determining deletion of the local cortical grooves, compression of the frontal horn of the left lateral ventricle and contralateral midline deviation. (a) Axial gadolinium-enhanced image. (b) Axial T2-weighted.
Second case report
Male patient, 45 years old, receives specialized neurological care with convergent strabismus and complaint of diplopia, headache, ringing in the left ear and hypoacusis for 6 months. A gadolinium-contrasted MRI examination was requested, which demonstrated a lesion in the petroclival region invading the cavernous sinus with extension into the posterior fossa, with contrast uptake compatible with meningioma. MRI also showed that the lesion reached the cervical region, descending through the petroclival portion, and bordering the clivus [
Figure 3:
Magnetic resonance imaging preoperated showing expansive lesion in the left petroclival region with left to right mass effect, invading the cavernous sinus with extension into the posterior fossa and reach the cervical region, descending through the petroclival portion, bordering the clivus. Lesion with contrast enhances compatible with meningioma. (a) Coronal gadolinium-enhanced image (GEI). (b and c) Axial GEI.
DISCUSSION
According to 285 RDD literature reviewed cases [
Table 1:
Characteristics of CNS-RDD cases according to present and previous reports from references.[
Table 2:
Central nervous system involvement in 287 cases of Rosai-Dorfman disease.[
On the other hand, comparing the frequencies of intracranial or spinal involvement according to the systemic or non-systemic involvement of the disease, RDD with isolated CNS involvement, reported in 77% (n = 221) of all cases of CNS-RDD, showed that 84% (n = 186) of isolated cases of the CNS had exclusively intracranial involvement and only 10% (n = 23) had exclusively spinal involvement. As for systemics CNS-RDD cases, the exclusively intracranial involvement occurred in 56% (n = 37) of the cases, while the exclusively spinal involvement was present in 26% (n = 17) of them. Therefore, it should be noted that comparing RDD with isolated CNS involvement, systemic CNS-RDD has a lower prevalence of exclusively intracranial involvement and a greater involvement of the spinal cord; in 26% (n = 17) of the cases with systemic presentation there was exclusive involvement of the spinal and in approximately 18% (n = 12) of the cases there was intracranial and spinal involvement. Thus, it is interesting to note that when RDD has systemic involvement, spinal cord involvement is more frequent than in relation to RDD with exclusive CNS involvement, which may have different explanations, such as perhaps because of systemic disease focus origin, usually sinus and with massive lymphadenopathy in the region, be closer to the spinal cord, this will somehow facilitate the disease spread to this nearest neural structure. This would mean that, once systemic RDD was present, it could spread more easily to any location in the CNS, without maintaining the preferential intracranial involvement of CNS exclusive cases.
The typical radiological findings of intracranial RDD show dural-based, extra-axial, well-circumscribed masses mimicking meningioma with MRI usually reveals multiple well-defined, dural-based or intraventricular, extra-axial masses with possible perilesional cerebral edema.[
As for the two reported cases of RDD, they were very similar to the expected age group and sex grouping, according to the literature and our review. As for the location of the lesion, which can happen in many regions, including the supratentorial region, where meningiomas occur and in which one of them mimics, the two cases presented in this study are very representative, especially the second one, since at first moment it was thought that it was one of those. Furthermore, the involvement reaching the cervical portion of the second case is compatible with a higher prevalence location of spinal cord injuries according to previous studies.[
More importantly, surgical resection should follow the same pattern as meningiomas, since the texture of both is very similar, and it is extremely unlikely that with only radiological images the two pathologies can be differentiated before neurosurgical removal for anatomopathological analysis. At present, the best treatment for intracranial RDD involves surgical excision,[
CONCLUSION
Thus, we conclude that intracranial Rosai-Dorfman tumors should always be remembered as differential diagnosis of meningiomas, since they are similar radiologically and macroscopically. Once remembered and diagnosed, the lesion must be treated following the same pattern of resection done in meningiomas and, treatment’s differences will not occur in the surgical excision technique, but in complementary chemotherapy implementation, radiotherapy, and even with radiosurgery aid, depending on the case. Thus, it is possible to obtain better results than with just the isolated surgical procedure.
Declaration of patient consent
Patient’s consent not required as patients identity is not disclosed or compromised.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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