A rare presentation of Erdheim Chester disease in a pediatric patient subsequently cured on the LCH III protocol
Background Erdheim Chester disease (ECD) is very rare in pediatrics with no standard treatment guidelines. Here we present the case of a pediatric ECD patient who was cured with a Langerhan cell histiocytosis (LCH) directed chemotherapy protocol. Aim The aim of the report was to publish this rare pr...
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| Veröffentlicht in: | Cancer Reports 2021-02, Vol.4 (1), p.e1304-n/a |
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| creator | Gupta, Aditya Kumar M, Abdul Wajid Meena, Jagdish P. ArunRaj, Sreedharan T. Mridha, Asit Naranje, Priyanka Kumar, Rakesh Seth, Rachna |
| description | Background
Erdheim Chester disease (ECD) is very rare in pediatrics with no standard treatment guidelines. Here we present the case of a pediatric ECD patient who was cured with a Langerhan cell histiocytosis (LCH) directed chemotherapy protocol.
Aim
The aim of the report was to publish this rare presentation of ECD in pediatrics and highlight the complete response obtained to treatment.
Methods
The details of the patient were extracted by a retrospective review of her clinical records.
Results (Case)
An 11 years old girl presented with fever and bone pain. On investigating she had multiple lytic bony lesions scattered throughout her skeleton. A biopsy from one of the bone lesions confirmed the diagnosis to be ECD. ECD is very rare in pediatrics and this case adds to the existing list of 11 previously reported ones. Also, worth mention is the fact that the child presented with isolated skeletal involvement in form of multiple osteolytic lesions. The child was started on the LCH‐III protocol on which she achieved a cure.
Conclusion
Lytic bone lesions in a child may be present in ECD. A subset of ECD may have good response to LCH like chemotherapy. |
| doi_str_mv | 10.1002/cnr2.1304 |
| format | Article |
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Erdheim Chester disease (ECD) is very rare in pediatrics with no standard treatment guidelines. Here we present the case of a pediatric ECD patient who was cured with a Langerhan cell histiocytosis (LCH) directed chemotherapy protocol.
Aim
The aim of the report was to publish this rare presentation of ECD in pediatrics and highlight the complete response obtained to treatment.
Methods
The details of the patient were extracted by a retrospective review of her clinical records.
Results (Case)
An 11 years old girl presented with fever and bone pain. On investigating she had multiple lytic bony lesions scattered throughout her skeleton. A biopsy from one of the bone lesions confirmed the diagnosis to be ECD. ECD is very rare in pediatrics and this case adds to the existing list of 11 previously reported ones. Also, worth mention is the fact that the child presented with isolated skeletal involvement in form of multiple osteolytic lesions. The child was started on the LCH‐III protocol on which she achieved a cure.
Conclusion
Lytic bone lesions in a child may be present in ECD. A subset of ECD may have good response to LCH like chemotherapy.</description><identifier>ISSN: 2573-8348</identifier><identifier>EISSN: 2573-8348</identifier><identifier>DOI: 10.1002/cnr2.1304</identifier><identifier>PMID: 33025727</identifier><language>eng</language><publisher>United States: John Wiley & Sons, Inc</publisher><subject>Autoimmune diseases ; Bones ; Cancer ; Case Report ; Case Reports ; Chemotherapy ; Children ; Conflicts of interest ; Diabetes ; Diseases ; Erdheim Chester disease ; Fever ; Health aspects ; Interferon ; Kinases ; LCH‐III protocol ; Medical prognosis ; Medical records ; Mutation ; Patients ; Pediatrics ; Radiation therapy ; Remission (Medicine) ; Steroids ; Tomography</subject><ispartof>Cancer Reports, 2021-02, Vol.4 (1), p.e1304-n/a</ispartof><rights>2020 The Authors. published by Wiley Periodicals LLC.</rights><rights>2020 The Authors. Cancer Reports published by Wiley Periodicals LLC.</rights><rights>COPYRIGHT 2021 John Wiley & Sons, Inc.</rights><rights>2021. This work is published under http://creativecommons.org/licenses/by/4.0/ (the "License"). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4254-8d23b16780b1a265ac70486f822097b3591843cebf717c491cb2acd69d00217b3</citedby><cites>FETCH-LOGICAL-c4254-8d23b16780b1a265ac70486f822097b3591843cebf717c491cb2acd69d00217b3</cites><orcidid>0000-0001-6024-9762 ; 0000-0002-3730-8944</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7941579/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7941579/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,723,776,780,860,881,1411,11541,27901,27902,45550,45551,46027,46451,53766,53768</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/33025727$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Gupta, Aditya Kumar</creatorcontrib><creatorcontrib>M, Abdul Wajid</creatorcontrib><creatorcontrib>Meena, Jagdish P.</creatorcontrib><creatorcontrib>ArunRaj, Sreedharan T.</creatorcontrib><creatorcontrib>Mridha, Asit</creatorcontrib><creatorcontrib>Naranje, Priyanka</creatorcontrib><creatorcontrib>Kumar, Rakesh</creatorcontrib><creatorcontrib>Seth, Rachna</creatorcontrib><title>A rare presentation of Erdheim Chester disease in a pediatric patient subsequently cured on the LCH III protocol</title><title>Cancer Reports</title><addtitle>Cancer Rep (Hoboken)</addtitle><description>Background
Erdheim Chester disease (ECD) is very rare in pediatrics with no standard treatment guidelines. Here we present the case of a pediatric ECD patient who was cured with a Langerhan cell histiocytosis (LCH) directed chemotherapy protocol.
Aim
The aim of the report was to publish this rare presentation of ECD in pediatrics and highlight the complete response obtained to treatment.
Methods
The details of the patient were extracted by a retrospective review of her clinical records.
Results (Case)
An 11 years old girl presented with fever and bone pain. On investigating she had multiple lytic bony lesions scattered throughout her skeleton. A biopsy from one of the bone lesions confirmed the diagnosis to be ECD. ECD is very rare in pediatrics and this case adds to the existing list of 11 previously reported ones. Also, worth mention is the fact that the child presented with isolated skeletal involvement in form of multiple osteolytic lesions. The child was started on the LCH‐III protocol on which she achieved a cure.
Conclusion
Lytic bone lesions in a child may be present in ECD. A subset of ECD may have good response to LCH like chemotherapy.</description><subject>Autoimmune diseases</subject><subject>Bones</subject><subject>Cancer</subject><subject>Case Report</subject><subject>Case Reports</subject><subject>Chemotherapy</subject><subject>Children</subject><subject>Conflicts of interest</subject><subject>Diabetes</subject><subject>Diseases</subject><subject>Erdheim Chester disease</subject><subject>Fever</subject><subject>Health aspects</subject><subject>Interferon</subject><subject>Kinases</subject><subject>LCH‐III protocol</subject><subject>Medical prognosis</subject><subject>Medical records</subject><subject>Mutation</subject><subject>Patients</subject><subject>Pediatrics</subject><subject>Radiation therapy</subject><subject>Remission (Medicine)</subject><subject>Steroids</subject><subject>Tomography</subject><issn>2573-8348</issn><issn>2573-8348</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2021</creationdate><recordtype>article</recordtype><sourceid>24P</sourceid><sourceid>BENPR</sourceid><recordid>eNp1kU1v1DAQhiMEolXpgT-ALHGBw279ldi-IK2iQldagYTgbDnOpOsqiYOdFO2_Z5YtVUFCPnjkeeZ9ZzxF8ZrRNaOUX_kx8TUTVD4rznmpxEoLqZ8_ic-Ky5zvKKVMV4Ib8bI4E4JimqvzYtqQ5BKQKUGGcXZziCOJHblO7R7CQOo95BkSaUMGl4GEkTgyQRvcnIInExZgGclLk-HHgmF_IH5J0BLUmfdAdvUN2W63aBDn6GP_qnjRuT7D5cN9UXz_eP2tvlntvnza1pvdykteypVuuWhYpTRtmONV6byiUled5pwa1YjSMC2Fh6ZTTHlpmG-4821lWvwThsBF8eGkOy3NAK3H1pLr7ZTC4NLBRhfs35kx7O1tvLfKSFYqgwLvHgRSxMnybIeQPfS9GyEu2XKJrppRUyL69h_0Li5pxPGsoIZyzksjkFqfqFvXgw1jF9HX42lhCD6O0AV83ygpOKtKprDg_anAp5hzgu6xe0btcff2uHt73D2yb56O-0j-2TQCVyfgJ7oc_q9k689f-W_JX-GWt20</recordid><startdate>202102</startdate><enddate>202102</enddate><creator>Gupta, Aditya Kumar</creator><creator>M, Abdul Wajid</creator><creator>Meena, Jagdish P.</creator><creator>ArunRaj, Sreedharan T.</creator><creator>Mridha, Asit</creator><creator>Naranje, Priyanka</creator><creator>Kumar, Rakesh</creator><creator>Seth, Rachna</creator><general>John Wiley & Sons, Inc</general><general>John Wiley and Sons Inc</general><scope>24P</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>IAO</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>PIMPY</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0001-6024-9762</orcidid><orcidid>https://orcid.org/0000-0002-3730-8944</orcidid></search><sort><creationdate>202102</creationdate><title>A rare presentation of Erdheim Chester disease in a pediatric patient subsequently cured on the LCH III protocol</title><author>Gupta, Aditya Kumar ; M, Abdul Wajid ; Meena, Jagdish P. ; ArunRaj, Sreedharan T. ; Mridha, Asit ; Naranje, Priyanka ; Kumar, Rakesh ; Seth, Rachna</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4254-8d23b16780b1a265ac70486f822097b3591843cebf717c491cb2acd69d00217b3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2021</creationdate><topic>Autoimmune diseases</topic><topic>Bones</topic><topic>Cancer</topic><topic>Case Report</topic><topic>Case Reports</topic><topic>Chemotherapy</topic><topic>Children</topic><topic>Conflicts of interest</topic><topic>Diabetes</topic><topic>Diseases</topic><topic>Erdheim Chester disease</topic><topic>Fever</topic><topic>Health aspects</topic><topic>Interferon</topic><topic>Kinases</topic><topic>LCH‐III protocol</topic><topic>Medical prognosis</topic><topic>Medical records</topic><topic>Mutation</topic><topic>Patients</topic><topic>Pediatrics</topic><topic>Radiation therapy</topic><topic>Remission (Medicine)</topic><topic>Steroids</topic><topic>Tomography</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Gupta, Aditya Kumar</creatorcontrib><creatorcontrib>M, Abdul Wajid</creatorcontrib><creatorcontrib>Meena, Jagdish P.</creatorcontrib><creatorcontrib>ArunRaj, Sreedharan T.</creatorcontrib><creatorcontrib>Mridha, Asit</creatorcontrib><creatorcontrib>Naranje, Priyanka</creatorcontrib><creatorcontrib>Kumar, Rakesh</creatorcontrib><creatorcontrib>Seth, Rachna</creatorcontrib><collection>Wiley Online Library Open Access</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Gale Academic OneFile</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Publicly Available Content Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Cancer Reports</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Gupta, Aditya Kumar</au><au>M, Abdul Wajid</au><au>Meena, Jagdish P.</au><au>ArunRaj, Sreedharan T.</au><au>Mridha, Asit</au><au>Naranje, Priyanka</au><au>Kumar, Rakesh</au><au>Seth, Rachna</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>A rare presentation of Erdheim Chester disease in a pediatric patient subsequently cured on the LCH III protocol</atitle><jtitle>Cancer Reports</jtitle><addtitle>Cancer Rep (Hoboken)</addtitle><date>2021-02</date><risdate>2021</risdate><volume>4</volume><issue>1</issue><spage>e1304</spage><epage>n/a</epage><pages>e1304-n/a</pages><issn>2573-8348</issn><eissn>2573-8348</eissn><abstract>Background
Erdheim Chester disease (ECD) is very rare in pediatrics with no standard treatment guidelines. Here we present the case of a pediatric ECD patient who was cured with a Langerhan cell histiocytosis (LCH) directed chemotherapy protocol.
Aim
The aim of the report was to publish this rare presentation of ECD in pediatrics and highlight the complete response obtained to treatment.
Methods
The details of the patient were extracted by a retrospective review of her clinical records.
Results (Case)
An 11 years old girl presented with fever and bone pain. On investigating she had multiple lytic bony lesions scattered throughout her skeleton. A biopsy from one of the bone lesions confirmed the diagnosis to be ECD. ECD is very rare in pediatrics and this case adds to the existing list of 11 previously reported ones. Also, worth mention is the fact that the child presented with isolated skeletal involvement in form of multiple osteolytic lesions. The child was started on the LCH‐III protocol on which she achieved a cure.
Conclusion
Lytic bone lesions in a child may be present in ECD. A subset of ECD may have good response to LCH like chemotherapy.</abstract><cop>United States</cop><pub>John Wiley & Sons, Inc</pub><pmid>33025727</pmid><doi>10.1002/cnr2.1304</doi><tpages>4</tpages><orcidid>https://orcid.org/0000-0001-6024-9762</orcidid><orcidid>https://orcid.org/0000-0002-3730-8944</orcidid><oa>free_for_read</oa></addata></record> |
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| source | Wiley Online Library Open Access; DOAJ Directory of Open Access Journals; Wiley Online Library Journals Frontfile Complete; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; PubMed Central |
| subjects | Autoimmune diseases Bones Cancer Case Report Case Reports Chemotherapy Children Conflicts of interest Diabetes Diseases Erdheim Chester disease Fever Health aspects Interferon Kinases LCH‐III protocol Medical prognosis Medical records Mutation Patients Pediatrics Radiation therapy Remission (Medicine) Steroids Tomography |
| title | A rare presentation of Erdheim Chester disease in a pediatric patient subsequently cured on the LCH III protocol |
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