In patients with NF1, PN are prevalent, progressive, and debilitating1–3

It is estimated that NF1 affects approximately 1 in 3000 people worldwide.1,2
PN, benign nerve sheath tumors, are a common manifestation of NF1 that may progress over time.2
Up to 50% icon. Variable PN location icon. Early action is crucial icon. 8% to 13% icon.
*Using whole-body MRI.

PN of any size and location can cause significant clinical complications that interrupt daily life7
Example of PN on the back.
Example of PN on the cervical spine.
Example of PN on the arm.§
Image reproduced from Rai VR, et al. 20248 (https://pubmed.ncbi.nlm.nih.gov/39279083/), made available under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/).

Image reproduced from Gross AM, et al. 20184 (https://doi.org/10.1093/neuonc/noy067), public domain in the US. 

§Image reproduced from Antônio JR, 20139 (http://dx.doi.org/10.1590/abd1806-4841.20132125), made available under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/).
Advanced imaging plays an essential role in diagnosis and disease management.1,4
Whole-body MRI is well-suited for imaging PN crossing anatomic planes and for evaluating the tumor burden in patients with NF1 PN.10
Physical impact icon. Cognitive impact icon. Psychosocial impact icon.
Impaired well-being icon. Limitations in day-to-day activities icon. Indirect costs icon.
Pain is a substantial morbidity for patients with NF1 PN.5,7
The pain can be:
  • Chronic, episodic, or both16
  • Diffuse or localized to the PN site16
From a retrospective chart review of 41 patients (aged 3 to 25) with 57 distinct PN with at least 7 years of clinical data and with volumetric MRI measurements from 2 different time points who were enrolled in HCP Natural History Study of patients with NF1. The study aims to characterize NF1-related manifestations.4

#Study of 60 children and adolescents with NF1 PN aged 6 to 18 years who were enrolled in a natural history protocol at the NCI. Caregivers and adolescents completed the IPI and BASC-II-P scales assessing the impact of NF1 PN.17

||CASSIOPEA was a retrospective study of 78 French children (aged ≥3 to <18 years) with NF1 PN referred to a specialist NF1 center or whose case underwent MDT review, confirming that the symptomatic PN was inoperable. The study was conducted between January 1, 2013, and December 31, 2019. A key exclusion criterion was prior or ongoing treatment with a MEK inhibitor as the study aimed to provide information on the natural history of NF1.18
PN may not be operable or fully resectable due to1:
  • Proximity to vital organs
  • Poorly defined margins
  • Challenging locations such as the head, neck, spine, or trunk
  • Hypervascularity, which can increase the risk of bleeding during surgery
Those who undergo surgery face
the risk of surgical complications, including19,20:
  • Delayed healing
  • Bleeding
  • Hematomas
  • Necrosis
Postoperative complications include19,22:
  • Permanent neurological deficits
  • Functional impairment
  • Nerve damage
**Based on an observational, cross-sectional study using a one-time survey among pediatric NF1 PN patients (n=61) aged 8 to 18 years as well as caregivers of patients (n=82) aged 2 to 7 years with NF1 PN from December 2020 to January 2021.19
Rates of PN regrowth after surgery range from 20% to 68%, depending on the extent of resection6,21,‡‡
In 2 retrospective analyses6,21††:
††Based on a retrospective review of pediatric inpatient and outpatient records of 121 patients who had 302 procedures on 168 tumors over a 20-year period at a single large pediatric referral center and a retrospective review of 96 pediatric patients who had 186 subtotal or partial resections on 130 tumors over a 10-year follow-up period at an NF center.6,21
††In the 2012 study by Prada CE et al, subtotal resection was defined as 50% to 80% excision.6

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Adult Data

Pediatric Data

Kylie, age 19, leaning on a fence and smiling at the camera. Kylie takes Koselugo for NF1 PN.
Korf BR, Rubenstein AE. Neurofibromatosis: A handbook for patients, families, and health care professionals. 2nd ed. Thieme Medical Publishers; 2005. Anderson JL, Gutmann DH. Neurofibromatosis type 1. In: Islam MP, Roach SE, eds. Handbook of Clinical Neurology. 3rd series; Vol 132. Elsevier B.V.; 2015:75-86. Miller DT, Freedenberg D, Schorry E, et al. Health supervision for children with neurofibromatosis type 1. Pediatrics. 2019;143(5):e20190660. doi:10.1542/peds.2019-0660 Gross AM, Singh G, Akshintala S, et al. Association of plexiform neurofibroma volume changes and development of clinical morbidities in neurofibromatosis 1. Neuro Oncol. 2018;20(12):1643-1651. doi:10.1093/neuonc/noy067 Copley-Merriman C, Yang X, Juniper M, Amin S, Yoo HK, Sen SS. Natural history and disease burden of neurofibromatosis type 1 with plexiform neurofibromas: a systematic literature review. Adolesc Health Med Ther. 2021;12:55-66. doi:10.2147/AHMT.S303456 Prada CE, Rangwala FA, Martin LJ, et al. Pediatric plexiform neurofibromas: impact on morbidity and mortality in neurofibromatosis type 1. J Pediatr. 2012;160(3):461-467. doi:10.1016/j.jpeds.2011.08.051 Yoo HK, Porteous A, Ng A, et al. Impact of neurofibromatosis type 1 with plexiform neurofibromas on the health-related quality of life and work productivity of adult patients and caregivers in the UK: a cross-sectional survey. BMC Neurol. 2023;23(1):419. doi:10.1186/s12883-023-03429-7 Rai VR, Rathore H, Kumari M, Ibrahim MN, Riaz M, Parveen R. Neurofibromatosis type 1 (NF1) presenting with dichotomous pubertal presentation: a case series. J Pak Med Assoc. 2024;74(9):1703-1706. doi:10.47391/JPMA.10955 Antônio JR, Goloni-Bertollo EM, Trídico LA. Neurofibromatosis: chronological history and current issues. An Bras Dermatol. 2013;88(3):329-343. doi:10.1590/abd1806-4841.20132125 Ahlawat S, Fayad LM, Khan MS, et al. Current whole-body MRI applications in the neurofibromatoses: NF1, NF2, and schwannomatosis. Neurology. 2016;87(7)(suppl 1):S31-S39. doi:10.1212/WNL.0000000000002929 Avery RA, Katowitz JA, Fisher MJ, et al; OPPN Working Group. Orbital/periorbital plexiform neurofibromas in children with neurofibromatosis type 1: multidisciplinary recommendations for care. Ophthalmology. 2017;124(1):123-132. doi:10.1016/j.ophtha.2016.09.020 Hou Y, Allen T, Wolters PL, et al. Predictors of cognitive development in children with neurofibromatosis type 1 and plexiform neurofibromas. Dev Med Child Neurol. 2020;62(8):977-984. doi:10.1111/dmcn.14489 Foji S, Mohammadi E, Sanagoo A, Jouybari L. The patients' experiences of burden of neurofibromatosis: a qualitative study. Iran J Nurs Midwifery Res. 2021;26(4):342-348. doi:10.4103/ijnmr.IJNMR_178_20 Granström S, Friedrich RE, Langenbruch AK, Augustin M, Mautner VF. Influence of learning disabilities on the tumour predisposition syndrome NF1—survey from adult patients' perspective. Anticancer Res. 2014;34(7):3675-3681. Sanagoo A, Jouybari L, Koohi F, Sayehmiri F. Evaluation of QoL in neurofibromatosis patients: a systematic review and meta-analysis study. BMC Neurol. 2019;19(1):123. doi:10.1186/s12883-019-1338-y Fisher MJ, Blakeley JO, Weiss BD, et al. Management of neurofibromatosis type 1-associated plexiform neurofibromas. Neuro Oncol. 2022;24(11):1827-1844. doi:10.1093/neuonc/noac146 Wolters PL, Burns KM, Martin S, et al. Pain interference in youth with neurofibromatosis type 1 and plexiform neurofibromas and relation to disease severity, social-emotional functioning, and quality of life. Am J Med Genet A. 2015;167A(9):2103-2113. doi:10.1002/ajmg.a.37123 Wolkenstein P, Chaix Y, Entz Werle N, et al. French cohort of children and adolescents with neurofibromatosis type 1 and symptomatic inoperable plexiform neurofibromas: CASSIOPEA study. Eur J Med Genet. 2023;66(5):104734. doi:10.1016/j.ejmg.2023.104734 Yang X, Yoo HK, Amin S, et al. Clinical and humanistic burden among pediatric patients with neurofibromatosis type 1 and plexiform neurofibroma in the USA. Childs Nerv Syst. 2022;38(8):1513-1522. doi:10.1007/s00381-022-05513-8 Nguyen R, Ibrahim C, Friedrich RE, Westphal M, Schuhmann M, Mautner VF. Growth behavior of plexiform neurofibromas after surgery. Genet Med. 2013;15(9):691-697. doi:10.1038/gim.2013.30 Needle MN, Cnaan A, Dattilo J, et al. Prognostic signs in the surgical management of plexiform neurofibroma: the Children's Hospital of Philadelphia experience, 1974-1994. J Pediatr. 1997;131(5):678-682. doi:10.1016/s0022-3476(97)70092-1 Iheanacho I, Yoo HK, Yang X, Dodman S, Hughes R, Amin S. Epidemiological and clinical burden associated with plexiform neurofibromas in pediatric neurofibromatosis type-1 (NF-1): a systematic literature review. Neurol Sci. 2022;43(2):1281-1293. doi:10.1007/s10072-021-05361-5

IMPORTANT SAFETY INFORMATION
WARNINGS AND PRECAUTIONS
Left Ventricular Dysfunction. Koselugo can cause cardiomyopathy, defined as a decrease in left ventricular ejection fraction (LVEF) ≥10% below baseline. In the pediatric safety pool, Grade 2 LVEF decrease occurred, as well as decreased LVEF of ≥20% resulting in dose interruption and dose reduction. The median time to first occurrence of LVEF decrease was approximately 12 months. In the adult population, Grade 2 LVEF decrease occurred, with decreased LVEF resulting in dose interruption. The median time to first occurrence of LVEF decrease was approximately 4 months. Assess ejection fraction by echocardiogram prior to initiating treatment, every 3 months during the first year of treatment, every 6 months thereafter, and as clinically indicated. Withhold, reduce dose, or permanently discontinue Koselugo based on severity of adverse reaction. In patients who interrupt Koselugo for decreased LVEF, obtain an echocardiogram or a cardiac MRI every 3 to 6 weeks until resolution. Upon resolution of decreased LVEF, obtain an echocardiogram or a cardiac MRI every 2 to 3 months.

Ocular Toxicity. Koselugo can cause ocular toxicity, including retinal vein occlusion (RVO), retinal pigment epithelial detachment (RPED), and blurred vision. In the pediatric safety pool, blurred vision, photophobia, cataracts, ocular hypertension, and retinal tear occurred. Blurred vision resulted in dose interruption. RPED occurred in the pediatric population during treatment with Koselugo and resulted in permanent discontinuation. In the adult population, blurred vision and vitreous floaters occurred in patients receiving Koselugo. Conduct ophthalmic assessments prior to initiating Koselugo, at regular intervals during treatment, and for new or worsening visual changes. Permanently discontinue Koselugo in patients with RVO. Withhold Koselugo in patients with RPED, conduct ophthalmic assessments every 3 weeks until resolution, and resume Koselugo at a reduced dose.

Gastrointestinal Toxicity. Koselugo can cause gastrointestinal toxicities, including diarrhea and colitis. In the pediatric safety pool (N=134), diarrhea occurred in 59% of patients, in addition to diarrhea resulting in permanent discontinuation and dose interruption. In the adult population (N=71), diarrhea occurred in 42% of patients who received Koselugo, in addition to diarrhea resulting in dose interruption. The median time to first onset of diarrhea was approximately 2 months in the pediatric safety pool and 1 month in the adult population. Advise patients to start an anti-diarrheal agent (eg, loperamide) and to increase fluid intake immediately after the first episode of diarrhea. Withhold, reduce dose, or permanently discontinue Koselugo based on severity of adverse reaction.

Skin Toxicity. Koselugo can cause severe rashes, including dermatitis acneiform. In the pediatric safety pool (N=134), rash occurred in 68% of patients. The most frequent rashes included dermatitis acneiform (47%) and maculopapular rash (31%). Pruritus, alopecia, and eczema occurred. In the adult population (N=71), rash occurred in 85% of patients who received Koselugo. The most frequent rash included dermatitis acneiform (66%). Alopecia and pruritus occurred in patients who received Koselugo. Grade 3 rash and rash resulting in dose interruption and dose reduction occurred in both the pediatric safety pool and the adult population. Permanent discontinuation also occurred in the adult population. Monitor for severe skin rashes. Withhold, reduce dose, or permanently discontinue Koselugo based on severity of adverse reaction. 

Increased Creatine Phosphokinase (CPK). Koselugo can cause increased CPK, myalgia, and rhabdomyolysis. In the pediatric safety pool (N=134), increased CPK, based on laboratory data, occurred in 73% of patients, including Grade 3 or 4. In the adult population (N=71), increased CPK, based on laboratory data, occurred in 70% of patients who received Koselugo, including Grade 3 or 4. Increased CPK resulted in dose interruption and dose reduction in both the pediatric safety pool and adult population. Increased CPK concurrent with myalgia occurred in both populations, including one patient who permanently discontinued Koselugo for myalgia in the pediatric safety pool. Obtain serum CPK prior to initiating Koselugo, periodically during treatment, and as clinically indicated. If increased CPK occurs, evaluate for rhabdomyolysis or other causes. Withhold, reduce dose, or permanently discontinue Koselugo based on severity of adverse reaction.

Increased Levels of Vitamin E and Risk of Bleeding (Koselugo Capsules). Koselugo capsules contain vitamin E, which can inhibit platelet aggregation and antagonize vitamin K-dependent clotting factors. Supplemental vitamin E is not recommended if daily vitamin E intake (including the amount of vitamin E in Koselugo and supplement) will exceed the recommended or safe limits due to increased risk of bleeding. An increased risk of bleeding may occur in patients who are co-administered vitamin-K antagonists or anti-platelet antagonists with Koselugo capsules. Monitor for bleeding in these patients and increase international normalized ratio (INR) monitoring in patients taking a vitamin-K antagonist. Perform anticoagulant assessments more frequently and adjust the dose of vitamin-K antagonists or anti-platelet agents as appropriate. Koselugo oral granules do not contain vitamin E.

Embryo-Fetal Toxicity. Koselugo can cause fetal harm when administered during pregnancy. In animal studies, administration of selumetinib to mice during organogenesis caused reduced fetal weight, adverse structural defects, and effects on embryo-fetal survival at approximate exposures >5 times the human exposure at the clinical dose of 25 mg/m2 twice daily. Advise pregnant women of the potential risk to a fetus. Advise females of reproductive potential and males with female partners of reproductive potential to use effective contraception during treatment with Koselugo and for 1 week after the last dose.

ADVERSE REACTIONS
Common adverse reactions ≥40% in pediatric patients include vomiting, diarrhea, increased CPK, dry skin, paronychia, nausea, dermatitis acneiform, and pyrexia.
Common adverse reactions ≥40% in adult patients include rash (all), dermatitis acneiform, and diarrhea.

DRUG INTERACTIONS
Effect of Other Drugs on Koselugo
Concomitant use of Koselugo with a strong or moderate CYP3A4 inhibitor or fluconazole increased selumetinib plasma concentrations, which may increase the risk of adverse reactions. Avoid coadministration with Koselugo. If coadministration cannot be avoided, reduce Koselugo dosage.
Concomitant use of Koselugo with a strong or moderate CYP3A4 inducer decreased selumetinib plasma concentrations, which may reduce Koselugo efficacy. Avoid concomitant use with Koselugo.

SPECIAL POPULATIONS
Pregnancy & Lactation. Verify the pregnancy status of patients of reproductive potential prior to initiating Koselugo. Due to the potential for adverse reactions in a breastfed child, advise patients not to breastfeed during treatment with Koselugo and for 1 week after the last dose.

To report SUSPECTED ADVERSE REACTIONS, contact AstraZeneca at 1-800-236-9933 or at https://us-aereporting.astrazeneca.com or FDA at 1-800-FDA-1088 or www.fda.gov/medwatch.

INDICATION
KOSELUGO is indicated for the treatment of adult and pediatric patients 1 year of age and older with neurofibromatosis type 1 (NF1) who have symptomatic, inoperable plexiform neurofibromas (PN).

Please see full Prescribing Information for Koselugo (selumetinib) at https://alexion.com/Documents/koselugo_uspi.pdf.

BASC-II-P=Behavior Assessment System for Children-2nd Edition Parent Rating Scale; HCP=healthcare professional; IPI=Impact of Pediatric Illness; MDT=multidisciplinary team; MEK=mitogen-activated protein kinase kinase; MPNST=malignant peripheral nerve sheath tumor; MRI=magnetic resonance imaging; NCI=National Cancer Institute; NF1=neurofibromatosis type 1; PN=plexiform neurofibromas.