Adult Langerhans cell histiocytosis and immunomodulatory drugs: Review and analysis of thirty-four case reports

Table 2 List of case reports on immunomodulatory drugs in langerhans cell histiocytosis

Ref.	Publication year	Age	Sex	SS-LCH (1) MS-LCH (2)	Previous therapies	IMiDS dose1	Duration (mo)	Out come
Gnassia et al[27]	1987	32	F	1	No	50	60	NAD
Viraben et al[43]	1988	48	M	1	NA	300	1	NAD
Bensaid et al[19]	1992	29	F	2	No	100	3	ADR
Misery et al[33]	1993	67	M	1	No	200	3	NAD
Thomas et al[42]	1993	24	F	2	No	50	3	ADS
Thomas et al[42]	1993	59	F	2	Prednisone	50	18	ADS
Dallafior et al[23]	1995	65	F	1	Cladribin	200-100	10	NAD
Meunier et al[32]	1995	66	M	1	No	100	2	NAD
Bouyssou-Gauthier et al[21]	1996	68	M	2	Cyclophosph-mide/steroids	NA	NA	ADS
Gerlach et al[26]	1998	73	F	2	Etoposide/topi-cal mustard	200	12	ADR
Lair et al[29]	1998	33	F	2	RT/surgery	100	28	ADS
Claudon et al[22]	2002	44	F	2	Metotrexate/ste-roids	100	4	ADR
Kolde et al[28]	2002	23	M	2	No	500	3	ADS
Kolde et al[28]	2002	61	F	2	Prednisone	500	16	ADS
Mortazavi et al[35]	2002	29	M	2	No	100	2	ADS
Santillan et al[38]	2003	33	F	1	RT/surgery	100	12	NAD
Padula et al[36]	2004	31	F	1	RT/surgery	NA	NA	NAD
Sander et al[37]	2004	38	M	1	Cladribin	200	6	NAD
Mauro et al[31]	2005	73	F	2	CVP	100	7	ADR
Alioua et al[17]	2006	43	F	2	No	100	12	ADR
Moravvej et al[34]	2006	27	M	1	PUVA	100	3	ADR
Wollina et al[44]	2006	38	M	2	Cladribin	200	9	NAD
Broekaert et al[20]	2007	57	F	2	No	50	24	ADR
McClain et al[10]	2007	45	F	1	Vinorelbine/ prednisone	100	22	NR/PD
McClain et al[10]	2007	31	F	2	Vinorelbine/ prednisone/RT	100	0,5	NR/PD
McClain et al[10]	2007	21	F	2	Vinorelbine/ prednisone/RT	100	5	NAD
McClain et al[10]	2007	46	F	2	Vinorelbine/ prednisone/RT	100	12	NAD
Li et al[30]	2010	27	M	1	No	150	5	NAD
Fernandes et al[25]	2011	60	F	1	No	100	4	NAD
Shahidi-Dadras et al[39]	2011	20	M	1	Topical steroid	200	6	NAD
Szturz et al[41]	2012	35	M	2	Cladribin/ Cyclophospha-mide methilpredniso-lone/RT/CHO-EP/BEAM	Lena 25	9	ADR
El-safadi et al[24]	2012	59	F	1	Surgery/RT/ MTX	Lena 10	19	NAD
Subramaniyan et al[40]	2015	71	M	1	No	200	10	NAD
Ruiz Beguerie et al[18]	2017	56	F	1	No	150	12	NAD

¹Dose in mg/d. MS-LCH: Multisystem Langerhans cell histiocytosis; SS-LCH: Single-system Langerhans cell histiocytosis; RT: Radiotherapy; NAD: Nonactive disease; ADR: Active disease regressive; ADS: Active disease stable; NR/PD: Nonresponse/progression disease; MTX: Methotrexate; Lena 10: Lenalidomide 10 mg; Lena 25: Lenalidomide 25 mg; CVP: Cyclophosphamide, vincristine, prednisone; CHOEP: Cyclophosphamide, doxorubicin, vincristine, etoposide, and prednisone; BEAM: Carmustine, etoposide, cytarabine, and melphalan; PUVA: Photochemotherapy; NA: Not available; F: Female; M: Male; IMiDs: Immunomodulatory drugs.