Journal of Clinical Pediatrics ›› 2023, Vol. 41 ›› Issue (3): 181-186.doi: 10.12372/jcp.2022.22e1691
• Expert Review • Previous Articles Next Articles
FANG Yongjun, WEI Yuting, XUE Yao
Received:
2022-12-20
Online:
2023-03-15
Published:
2023-03-10
FANG Yongjun, WEI Yuting, XUE Yao. Advance in lysosomal storage disorders treated with allogeneic hematopoietic stem cell transplantation[J].Journal of Clinical Pediatrics, 2023, 41(3): 181-186.
"
LSD | 药物治疗 方案 | allo-HSCT | ||||
---|---|---|---|---|---|---|
适应证 | 移植时机 | 供体选择 | 预处理方案 | 潜在不良事件 | ||
黏多糖贮积症 | ERT、基因治疗、药物伴侣、减少底物 | 绝对适应证:年龄<2.5岁的MPSI重型(MPS IH)患者 相对适应证:年龄>2.5岁的MPS IH型患者,MPS Ⅰ非重型、MPS Ⅱ、Ⅳ、Ⅵ、Ⅶ型患者 | 尽早移植,移植年龄小于2周岁为佳 | 首选非携带、同胞全相供者,次选非亲缘脐血 | 清髓性方案(BuCy,联合或不联合ATG) | 植入失败、移植相关并发症,无法改善移植前形成的病变,对合并神经受损患者疗效欠佳 |
戈谢病 | ERT、基因治疗、药物伴侣、减少底物 | Ⅰ型儿童期发病者为最佳适应证,对于Ⅱ型和Ⅲ型患者,建议在神经症状出现前行HSCT | 尽早移植,明显肝功和神经受损等症状出现前 | 首选非携带、同胞全相供者,次选非亲缘脐血 | 清髓性方案(BuCy,联合或不联合ATG) | GVHD、感染、植入失败或死亡等移植后相关并发症 |
尼曼-匹克病 | ERT、基因治疗、减少底物 | 无绝对适应证,无神经系统受损或早期症状者可考虑行HSCT | 尽早移植,建议未出现神经系统改变前行HSCT | 首选无携带、HLA全相合同供者,次选优非亲缘脐血 | 清髓性方案(BuCy,联合或不联合ATG) | 疗效个体差异性大,风险较大,改善神经系统受损症状的效果及预后不确定 |
异染性脑白质营养不良 | ERT、基因治疗 | 部分无症状晚婴型、轻微早期症状的青少年或成人型MLD | 早期移植,建议症状发生前进行 | 首选未携带变异、HLA全相合同供者,次优选非亲缘脐血 | 清髓性方案(BuCy,联合或不联合ATG) | 移植相关病死率较高,无法改善周围神经系统受累 |
[1] |
Meikle PJ, Hopwood JJ, Clague AE, et al. Prevalence of lysosomal storage disorders[J]. JAMA, 1999, 281(3): 249-254.
doi: 10.1001/jama.281.3.249 |
[2] | Chin SJ, Fuller M. Prevalence of lysosomal storage disorders in Australia from 2009 to 2020[J]. Lancet Reg Health West Pac, 2022, 19: 100344. |
[3] |
Martina JA, Raben N, Puertollano R. SnapShot: lysosomal storage diseases[J]. Cell, 2020, 180(3): 602-602.
doi: S0092-8674(20)30098-2 pmid: 32032518 |
[4] |
Marques ARA, Saftig P. Lysosomal storage disorders - challenges, concepts and avenues for therapy: beyond rare diseases[J]. J Cell Sci, 2019, 132(2): jcs221739.
doi: 10.1242/jcs.221739 |
[5] |
Platt FM, d'Azzo A, Davidson BL, et al. Lysosomal storage diseases[J]. Nat Rev Dis Primers, 2018, 4(1): 27.
doi: 10.1038/s41572-018-0025-4 pmid: 30275469 |
[6] |
Moro E. Lysosomal storage disorders: molecular basis and therapeutic approaches[J]. Biomolecules, 2021, 11(7): 964.
doi: 10.3390/biom11070964 |
[7] |
Suarez-Guerrero JL, Gómez Higuera PJ, Arias Flórez JS, et al. Mucopolysaccharidosis: clinical features, diagnosis and management[J]. Rev Chil Pediatr, 2016, 87(4): 295-304.
doi: 10.1016/j.rchipe.2015.10.004 pmid: 26613630 |
[8] |
Khan SA, Peracha H, Ballhausen D, et al. Epidemiology of mucopolysaccharidoses[J]. Mol Genet Metab, 2017, 121(3): 227-240.
doi: S1096-7192(17)30206-8 pmid: 28595941 |
[9] |
Mistry PK, Lopez G, Schiffmann R, et al. Gaucher disease: progress and ongoing challenges[J]. Mol Genet Metab, 2017, 120(1-2): 8-21.
doi: S1096-7192(16)30323-7 pmid: 27916601 |
[10] |
Nguyen Y, Stirnemann J, Belmatoug N. Gaucher disease: a review[J]. Rev Med Interne, 2019, 40(5): 313-322.
doi: S0248-8663(18)31185-8 pmid: 30638965 |
[11] |
Vanier MT. Niemann-Pick diseases[J]. Handb Clin Neurol, 2013, 113: 1717-1721.
doi: 10.1016/B978-0-444-59565-2.00041-1 pmid: 23622394 |
[12] |
van Rappard DF, Boelens JJ, Wolf NI. Metachromatic leukodystrophy: disease spectrum and approaches for treatment[J]. Best Pract Res Clin Endocrinol Metab, 2015, 29(2): 261-273.
doi: 10.1016/j.beem.2014.10.001 pmid: 25987178 |
[13] |
Fernández-Pereira C, San Millán-Tejado B, Gallardo-Gómez M, et al. Therapeutic approaches in lysosomal storage diseases[J]. Biomolecules, 2021, 11(12): 1775.
doi: 10.3390/biom11121775 |
[14] |
Parenti G, Andria G, Ballabio A. Lysosomal storage diseases: from pathophysiology to therapy[J]. Annu Rev Med, 2015, 66: 471-86.
doi: 10.1146/annurev-med-122313-085916 pmid: 25587658 |
[15] |
Boelens JJ, Aldenhoven M, Purtill D, et al. Outcomes of transplantation using various hematopoietic cell sources in children with Hurler syndrome after myeloablative conditioning[J]. Blood, 2013, 121(19): 3981-2987.
doi: 10.1182/blood-2012-09-455238 pmid: 23493783 |
[16] |
Afroze B, Brown N. Ethical issues in managing lysosomal storage disorders in children in low and middle income countries[J]. Pak J Med Sci, 2017, 33(4): 1036-1041.
doi: 10.12669/pjms.334.12975 pmid: 29067088 |
[17] |
Ohashi T. Gene therapy for lysosomal storage diseases and peroxisomal diseases[J]. J Hum Genet, 2019, 64(2): 139-143.
doi: 10.1038/s10038-018-0537-5 pmid: 30498239 |
[18] |
Biffi A, Montini E, Lorioli L, et al. Lentiviral hematopoietic stem cell gene therapy benefits metachromatic leuko-dystrophy[J]. Science, 2013, 341(6148): 1233158.
doi: 10.1126/science.1233158 |
[19] |
Aldenhoven M, Kurtzberg J. Cord blood is the optimal graft source for the treatment of pediatric patients with lysosomal storage diseases: clinical outcomes and future directions[J]. Cytotherapy, 2015, 17(6): 765-774.
doi: S1465-3249(15)00693-3 pmid: 25840940 |
[20] |
Sessa M, Lorioli L, Fumagalli F, et al. Lentiviral haemopoietic stem-cell gene therapy in early-onset metachromatic leukodystrophy: an ad-hoc analysis of a non-randomised, open-label, phase 1/2 trial[J]. Lancet, 2016, 388(10043): 476-487.
doi: 10.1016/S0140-6736(16)30374-9 pmid: 27289174 |
[21] |
Aldenhoven M, Kurtzberg J. Cord blood is the optimal graft source for the treatment of pediatric patients with lysosomal storage diseases: clinical outcomes and future directions[J]. Cytotherapy, 2015, 17(6): 765-774.
doi: S1465-3249(15)00693-3 pmid: 25840940 |
[22] |
D’Avanzo F, Rigon L, Zanetti A, et al. Mucopoly-saccharidosis type ii: one hundred years of research, diagnosis, and treatment[J]. Int J Mol Sci, 2020, 21(4): 1258.
doi: 10.3390/ijms21041258 |
[23] |
Coutinho MF, Santos JI, Alves S. Less is more: substrate reduction therapy for lysosomal storage disorders[J]. Int J Mol Sci, 2016, 17(7): 1065.
doi: 10.3390/ijms17071065 |
[24] |
Friso A, Tomanin R, Salvalaio M, et al. Genistein reduces glycosaminoglycan levels in a mouse model of mucopolysaccharidosis type II[J]. Br J Pharmacol, 2010, 159(5): 1082-1091.
doi: 10.1111/j.1476-5381.2009.00565.x |
[25] |
Vellodi A, Young E, Cooper A, et al. Long-term follow-up following bone marrow transplantation for Hunter disease[J]. J Inherit Metab Dis, 1999, 22(5): 638-648.
doi: 10.1023/a:1005525931994 pmid: 10399096 |
[26] |
Wang J, Luan Z, Jiang H, et al. Allogeneic hematopoietic stem cell transplantation in thirty-four pediatric cases of mucopolysaccharidosis - a ten-year report from the China Children Transplant Group[J]. Biol Blood Marrow Transplant, 2016, 22(11): 2104-2108.
doi: 10.1016/j.bbmt.2016.08.015 |
[27] | Köhn AF, Grigull L, du Moulin M, et al. Hematopoietic stem cell transplantation in mucopolysaccharidosis type IIIA: a case description and comparison with a genotype-matched control group[J]. Mol Genet Metab Rep, 2020, 23: 100578. |
[28] |
Ringdén O, Remberger M, Svahn BM, et al. Allogeneic hematopoietic stem cell transplantation for inherited disorders: experience in a single center[J]. Transplantation, 2006, 81(5): 718-725.
doi: 10.1097/01.tp.0000181457.43146.36 pmid: 16534474 |
[29] | 唐湘凤, 卢伟, 井远方, 等. 非血缘脐血或单倍体来源的造血干细胞移植治疗戈谢病的临床研究[J]. 中国小儿血液与肿瘤杂志, 2020, 25(4): 195-199. |
[30] | Pan J, Geng Z, Jiang H. Allogeneic hematopoietic stem cell transplantation for the treatment of Niemann-Pick disease type A: a case report[J]. Zhongguo Dang Dai Er Ke Za Zhi, 2013, 15(9): 782-784. |
[31] | 陈姣, 刘小梅, 肖娟, 等. 异基因造血干细胞移植治疗尼曼匹克病B型1例[J]. 中国小儿血液与肿瘤杂志, 2021, 26(1): 48-50. |
[32] |
Quarello P, Spada M, Porta F, et al. Hematopoietic stem cell transplantation in Niemann-Pick disease type B monitored by chitotriosidase activity[J]. Pediatr Blood Cancer, 2018, 65(2): doi: 10.1002/pbc.26811.
doi: 10.1002/pbc.26811 |
[33] |
van Egmond ME, Pouwels PJ, Boelens JJ, et al. Improvement of white matter changes on neuroimaging modalities after stem cell transplant in metachromatic leukodystrophy[J]. JAMA Neurol, 2013, 70(6): 779-782.
doi: 10.1001/jamaneurol.2013.629 pmid: 23608771 |
[34] |
Beschle J, Döring M, Kehrer C, et al. Early clinical course after hematopoietic stem cell transplantation in children with juvenile metachromatic leukodystrophy[J]. Mol Cell Pediatr, 2020, 7(1): 12.
doi: 10.1186/s40348-020-00103-7 pmid: 32910272 |
[35] |
Boucher AA, Miller W, Shanley R, et al. Long-term outcomes after allogeneic hematopoietic stem cell transplantation for metachromatic leukodystrophy: the largest single-institution cohort report[J]. Orphanet J Rare Dis, 2015, 10: 94.
doi: 10.1186/s13023-015-0313-y pmid: 26245762 |
[36] |
Boelens JJ, Rocha V, Aldenhoven M, et al. Risk factor analysis of outcomes after unrelated cord blood transplantation in patients with hurler syndrome[J]. Biol Blood Marrow Transplant, 2009, 15(5): 618-625.
doi: 10.1016/j.bbmt.2009.01.020 |
[37] |
van Rood JJ, Stevens CE, Smits J, et al. Reexposure of cord blood to noninherited maternal HLA antigens improves transplant outcome in hematological malignancies[J]. Proc Natl Acad Sci U S A, 2009, 106(47): 19952-19957.
doi: 10.1073/pnas.0910310106 |
[1] | ZOU Liping. Childhood encephalopathy: a group of diseases associated with various diseases [J]. Journal of Clinical Pediatrics, 2023, 41(9): 641-643. |
[2] | ZHANG Weihua, ZOU Liping, REN Haitao, GUAN Hongzhi. Beware of the pitfalls in diagnosis and treatment of autoimmune encephalitis in children [J]. Journal of Clinical Pediatrics, 2023, 41(9): 644-649. |
[3] | HOU Chi, CHEN Wenxiong, LIAO Yinting, WU Wenxiao, TIAN Yang, ZHU Haixia, PENG Bingwei, ZENG Yiru, WU Wenlin, CHEN Zongzong, LI Xiaojing. Clinical analysis of autoimmune glial fibrillary acidic protein astrocytopathy in children [J]. Journal of Clinical Pediatrics, 2023, 41(9): 656-660. |
[4] | YANG Yating, CAI Yuehao, FANG Qiong, CHEN Lang, CHEN Qiaobin, LIN Zhi, WU Feifei, LIN Meng. Clinical analysis of idiopathic and symptomatic occipital lobe epilepsy in children [J]. Journal of Clinical Pediatrics, 2023, 41(9): 668-673. |
[5] | HOU Ruolin, WU Jing, LI Ling. Pediatric autoimmune encephalitis with brain MRI showing meningeal thickening and enhancement [J]. Journal of Clinical Pediatrics, 2023, 41(9): 674-679. |
[6] | WU Yuefang, SUN Yanling, WU Wanshui, DU Shuxu, LI Miao, SUN Liming. Analysis of prognostic factors and survival status of group 4 medulloblastoma in children [J]. Journal of Clinical Pediatrics, 2023, 41(9): 686-691. |
[7] | SUN Juan, LI Haiying, JIA Peisheng, WANG Huaili. Clinical analysis of fulminant myocarditis in 12 children [J]. Journal of Clinical Pediatrics, 2023, 41(9): 692-696. |
[8] | Reviewer: WANG Chenhui, Reviser: YANG Hui. Research progress on early screening and diagnosis of Crohn's disease in children [J]. Journal of Clinical Pediatrics, 2023, 41(9): 708-714. |
[9] | SHEN Nan, DU Bailu. Strategies for the diagnosis, treatment, and management of invasive fungal infections in children with hematologic neoplasms [J]. Journal of Clinical Pediatrics, 2023, 41(8): 571-577. |
[10] | XU Beixue, LIU Quanbo. Clinical analysis of 195 children with invasive pulmonary fungal infection [J]. Journal of Clinical Pediatrics, 2023, 41(8): 584-588. |
[11] | CHEN Hongyu, LIU Zihao, WANG Heping, LIAO Cuijuan, LI Li, WANG Wenjian, LAI Jianwei. Role of nontypeable Haemophilus influenzae biofilms in chronic pulmonary infection in children [J]. Journal of Clinical Pediatrics, 2023, 41(8): 589-593. |
[12] | KANG Lei, GUO Fang, LI Lifang, BAI Xinfeng, CHENG Caiyun, XU Meixian. Value of metagenomic next-generation sequencing in children with visceral leishmaniasis associated with hemolytic histiocytosis [J]. Journal of Clinical Pediatrics, 2023, 41(8): 594-598. |
[13] | SUN Zhicai, LIU Yuling, LI Xiaolin, PAN Xiaofen. Clinical analysis of 15 children with primary nephrotic syndrome complicated with adrenal crisis [J]. Journal of Clinical Pediatrics, 2023, 41(8): 610-612. |
[14] | WANG Hongxia, PAN Xiang, LU Jun. Report a case of α-ketoadipic aciduria caused by compound heterozygous variant of DHTKD1 gene [J]. Journal of Clinical Pediatrics, 2023, 41(8): 624-628. |
[15] | XI Bixin, HU Qun, LIU Aiguo. Research advances of the bronchiolitis obliterans syndrome following allogeneic hematopoietic stem cell transplant in children [J]. Journal of Clinical Pediatrics, 2023, 41(8): 629-633. |
|