Langerhans cell histiocytosis infiltration in cerebrospinal fluid: A case report

Ghosal, Nandita; Kapila, Kusum; Kakkar, Sunita; Verma, Kusum

doi:10.1002/1097-0339(200102)24:2<123::aid-dc1023>3.0.co;2-u

Cited by 9 publications

(8 citation statements)

References 11 publications

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“…30,31,53,58 Rarely, CSF cytology will reveal Langerhans cells. 27 As is the case in extracranial LCH, cerebral parenchymal lesions usually respond well to surgical excision, conventional radiation, radiosurgery, and chemotherapy. 15,21,30,31,40,48,53 One patient in our series, who was diagnosed when she was 1 year old with multisystem LCH, developed a large frontotemporal mass 7 years later, which was proven based on biopsy findings to be LCH.…”

Section: Cerebral Parenchymal Lchmentioning

confidence: 97%

Craniospinal Langerhans cell histiocytosis in children: 30 years' experience at a single institution

Davidson

McComb²,

Bowen³

et al. 2008

PED

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Object The goal of this study was to review a large series of patients with Langerhans cell histiocytosis (LCH) who had craniospinal lesions to assess the long-term course, outcome, and efficacy of treatment of the disease. Methods Forty-four patients with LCH who presented to a single pediatric neurosurgical department between 1976 and 2006 were retrospectively reviewed. Results This series included 29 boys and 15 girls, ranging in age from 2 months to 13 years, with a mean follow-up duration of 4.5 years. Twenty-seven patients (61%) had unifocal bone lesions, 12 (27%) had multifocal bone disease, 2 (5%) had solitary hypothalamic–pituitary axis lesions, and 3 (7%) had multiple organ involvement at presentation. Five (19%) of the 27 patients with unifocal bone disease and 4 (33%) of the 12 patients with multifocal bone disease had delayed development of new bone lesions during the follow-up period. The time to development of new bone lesions ranged from 1 month to 1 year. Two of the 3 patients with multiple-organ LCH died. Patient age ≤ 2 years at the time of initial presentation was a risk factor for both initial multifocality and eventual dissemination. In all patients with initial multifocal bone involvement or later dissemination of unifocal bone disease, LCH was controlled by chemotherapy, except for 2 who were treated by surgery alone. Three patients had histological evidence of spontaneous resolution of their lesions. Conclusions Patients with unifocal LCH can be effectively treated with surgery alone. Very young patients are more likely to have multifocal disease and disseminations, and will usually require chemotherapy to control their disease. Spontaneously regressing lesions need not be resected; however, a biopsy procedure can be performed for diagnostic purposes.

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Section: Cerebral Parenchymal Lchmentioning

confidence: 97%

Craniospinal Langerhans cell histiocytosis in children: 30 years' experience at a single institution

Davidson

McComb²,

Bowen³

et al. 2008

PED

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“…We were unable to find literature specifically addressing the significance of this finding; classically it is said to be associated with meningitis due to fungi or tuberculosis [ 4 ]. None of the other case reports we studied, including one with Langerhans cells in the CSF, reported this finding [ 5 ]. One series has shown elevations of neurofilament protein light chain, glial fibrillary acid protein and total tau protein in the CSF of patients with LCH affecting the brain [ 6 ].…”

Section: Discussionmentioning

confidence: 82%

Langerhans Cell Histiocytosis in an Adult with Involvement of the Calvarium, Cerebral Cortex and Brainstem: Discussion of Pathophysiology and Rationale for the Use of Intravenous Immune Globulin

Dardis¹,

Aung²,

Shapiro³

et al. 2015

Case Rep Neurol

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We report a case of Langerhans cell histiocytosis in a 64-year-old male who presented with symptoms and signs of brain involvement, including seizures and hypopituitarism. The diagnosis was confirmed with a biopsy of a lytic skull lesion. The disease affecting the bone showed no sign of progression following a short course of cladribine. Signs of temporal lobe involvement led to an additional biopsy, which showed signs of nonspecific neurodegeneration and which triggered status epilepticus. Lesions noted in the brainstem were typical for the paraneoplastic inflammation reported in this condition. These lesions improved after treatment with cladribine. They remained stable while on treatment with intravenous immune globulin.

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“…The cytologic examination of CSF in LCH patients can be normal or show only lymphocytic meningitis, and thus direct CSF involvement by LCH is probably underdiagnosed. 6,7 The only report of such case in the international literature was published in 2001 by Ghosal and colleagues, who speculate that this unusual localization could be due to involvement of the choroid plexus or to direct invasion of a skull lesion through the dura. 6 Other serous cavities, such as the pleura, can be involved by LCH.…”

Section: Case Reportmentioning

confidence: 99%

Diagnosis of Langerhans cell histiocytosis on cytological examination of cerebrospinal fluid: Report of the first case

Tommasino

Cardamone

Tortora

et al. 2022

Diagnostic Cytopathology

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Langerhans cell histiocytosis (LCH) is a disease of unknown etiology characterized by a proliferation of histiocytic cells resembling dendritic Langerhans cells. LCH can be unifocal or multifocal, with one-or many-organ involvement. The serous fluids are rarely involved. Cytological diagnosis of LCH is possible and relies on recognition of the typical cytomorphological features and subsequent immunocytochemical confirmation. Given the possibility of multisystem involvement, after diagnosing LCH it is necessary to carry out staging exams such as a bone survey, abdominal ultrasound, complete blood count, screening for diabetes insipidus and pulmonary function tests.We present the first case of LCH where the diagnosis was reached on cytological material from the cerebrospinal fluid. To the best of our knowledge, this is the first such case reported in the international literature to date. The morphological and immunocytochemical characteristics of our case are described, and the relevant literature is reviewed

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Langerhans cell histiocytosis infiltration in cerebrospinal fluid: A case report

Cited by 9 publications

References 11 publications

Craniospinal Langerhans cell histiocytosis in children: 30 years' experience at a single institution

Craniospinal Langerhans cell histiocytosis in children: 30 years' experience at a single institution

Langerhans Cell Histiocytosis in an Adult with Involvement of the Calvarium, Cerebral Cortex and Brainstem: Discussion of Pathophysiology and Rationale for the Use of Intravenous Immune Globulin

Diagnosis of Langerhans cell histiocytosis on cytological examination of cerebrospinal fluid: Report of the first case

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