Evidence map›Paper›PMID 29118704›Full record

ArticleCase reports in oncology

Spontaneous Remission of Severe Systemic Langerhans Cell Histiocytosis with Bladder Involvement: A Case Study.

Isotta M Magaton, Alexandar Tzankov, Fatime Krasniqi, Christof Rottenburger, Rosanna Zanetti-Daellenbach, Peter Grendelmeier, Viola Heinzelmann-Schwarz, Michael Mayr, Fabienne D Schwab

Open access · goldAbstract readCase Reports
In one paragraph

Article in Case reports in oncology. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact, top 83% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 2 citations in OpenAlex.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

9 authors at 3 institutions in 1 country.

Isotta M MagatonHospital for Women, Clinic Fontana, Cantonal Hospital Graubünden, Chur, Switzerland.
Alexandar TzankovInstitute of Pathology, University Hospital Basel, Basel, Switzerland.
Fatime KrasniqiDepartment of Oncology, University Hospital Basel, Basel, Switzerland.
Christof RottenburgerClinic of Radiology and Nuclear Medicine, University Hospital Basel, Basel, Switzerland.
Rosanna Zanetti-DaellenbachDepartment of Gynecology and Gynecological Oncology, Claraspital, Basel, Switzerland.
Peter GrendelmeierDepartment of Pulmonology, University Clinic of Internal Medicine, Cantonal Hospital Baselland, Liestal, Switzerland.
Viola Heinzelmann-SchwarzDepartment of Gynecology and Gynecological Oncology, Hospital for Women, University Hospital Basel, Basel, Switzerland.
Michael MayrMedical Outpatient Clinic, University Hospital Basel, Basel, Switzerland.
Fabienne D SchwabDepartment of Gynecology and Gynecological Oncology, Hospital for Women, University Hospital Basel, Basel, Switzerland.
University Hospital of Basel · CHKantonsspital Graubünden · CHSt. Claraspital · CH

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundThe clinical presentation of Langerhans cell histiocytosis (LCH) is heterogeneous ranging from single-organ involvement to systemic disease causing substantial morbidity and mortality. We describe an unusual course of severe multisystem LCH with spontaneous remission. CASE PRESENTATION: We report on a 45-year-old Caucasian woman with cervical cancer, FIGO stage IVB. Five months after the end of combined radiochemotherapy and brachytherapy, the patient was readmitted because of severe dysuria. Sterile leukocyturia was seen, and cystoscopy revealed only 3 unspecific small mucosal lesions compatible with postradiation cystitis. Incidentally, a computed tomography (CT) scan of the body showed diffuse micronodular and cystic lesions in lungs and hypodense lesions in the liver. Biopsies revealed infiltrations of CD1a and Langerin (CD207)-positive histiocytes in the lung, liver, and bladder. Additionally, positron emission tomography-CT (PET-CT) was compatible with bone involvement. Retrospective analysis revealed that the increase in alkaline phosphatase might have been a surrogate of bone marrow infiltration with osseous activity. Repeated pneumothoraces occurred, and only one course of vinblastine-prednisolone could be applied. Despite ongoing tobacco consumption and without further therapy, PET-CT showed considerable remission 2 months later. However, despite stable remission, documented by serial PET and conventional CT scans, persistent infiltration of the bladder by Langerhans histiocytes could still be demonstrated 17 months later. Unfortunately, cervical cancer recurred and progressed.

conclusionMultisystem LCH may rapidly occur, may be oligosymptomatic and, even in high-risk cases, remission without specific therapy might occur. Whether alkaline phosphatase might be a surrogate to monitor osseous disease activity has to be further explored.

Indexed as

Alkaline phosphataseBladderCD1a-positive histiocytesCervical cancerLangerhans cell histiocytosisLangerinMultisystem diseaseRemissionRisk organsTherapy

Identifiers

PMID29118704
PMCPMC5662969
OpenAlexW2761204027

What OpenQuestion holds

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LicenceCC BY-NC
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.