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28 August 2012 EMA/COMP/426291/2012 Committee for Orphan Medicinal Products (4- hydroxyphenyl)acetyl]amino}butanoic acid for the treatment of progressive familial intrahepatic cholestasis On 17 July 2012, orphan designation (EU/3/12/1028) was granted by the European Commission to Albireo AB, Sweden, for 2,3,4,5- tetrahydro-1,2,5-benzothiadiazepin-8-(4- hydroxyphenyl)acetyl]amino}butanoic acid for the treatment of progressive familial intrahepatic cholestasis. What is progressive familial intrahepatic cholestasis? Progressive familial intrahepatic cholestasis is an inherited condition that causes progressive liver disease, which normally leads to liver failure. In progressive familial intrahepatic cholestasis, a fluid produced by the liver called bile, which helps digestion, is not transported normally to the intestine. This results in bile acids, essential components of bile, building up in liver cells and becoming toxic to the liver and causing cirrhosis (scarring). Symptoms usually begin in infancy and include severe itching, jaundice (yellowing of the skin and eyes), delayed growth, pancreatitis (inflammation of the pancreas) and bleeding inside the stomach or the gut. At later stages, high blood pressure in the vein that supplies blood to the liver and liver failure will appear. Progressive familial intrahepatic cholestasis is chronically debilitating due to early appearance of the symptoms and liver problems. It is also life-threatening due to severe liver damage. What is the estimated number of patients affected by the condition? At the time of designation, progressive familial intrahepatic cholestasis affected not more than 0.2 in 10,000 people in the European Union (EU) *. This is equivalent to not more than 10,000 people, and is below the ceiling for orphan designation, which is 5 people in 10,000. This is based on the information provided by the sponsor and the knowledge of the Committee for Orphan Medicinal Products (COMP). * Disclaimer: For the purpose of the designation, the number of patients affected by the condition is estimated and assessed on the basis of data from the European Union (EU 27), Norway, Iceland and Liechtenstein. This represents a population of 506,300,000 (Eurostat 2011). 7 Westferry Circus Canary Wharf London E14 4HB United Kingdom Telephone +44 (0)20 7418 8400 Facsimile +44 (0)20 7523 7040 E-mail info@ema.europa.eu Website www.ema.europa.eu An agency of the European Union European Medicines Agency, 2012. Reproduction is authorised provided the source is acknowledged.

What treatments are available? At the time of orphan designation, no satisfactory treatments were authorised in the EU for this condition. Rifampicin was used to treat the itchiness, ursodeoxycholic acid was used to treat some types of progressive familial intrahepatic cholestasis and vitamin supplements were given to children to prevent deficiencies. Patient with end-stage liver disease underwent liver transplantation. How is this medicine expected to work? The medicine is expected to block some proteins on the surface of intestinal cells called ileal sodiumdependent bile acid transporters. These bile acid transporters are involved in the transport of bile acids from the intestines back to the liver, where they are recycled to produce more bile, a process known as bile acid re-uptake. By blocking the transporters, the medicine is expected to reduce the entry of bile acids into liver cells, and thereby prevent their accumulation and the toxicity in the liver cells. This is expected to help improve the symptoms of the disease. What is the stage of development of this medicine? At the time of submission of the application for orphan designation, the evaluation of the effects of the medicine in experimental models was ongoing. At the time of submission, no clinical trials with the medicine in patients with progressive familial intrahepatic cholestasis had been started. At the time of submission, the medicine was not authorised anywhere in the EU for progressive familial intrahepatic cholestasis or designated as an orphan medicinal product elsewhere for this condition. In accordance with Regulation (EC) No 141/2000 of 16 December 1999, the COMP adopted a positive opinion on 13 June 2012 recommending the granting of this designation. Opinions on orphan medicinal product designations are based on the following three criteria: the seriousness of the condition; the existence of alternative methods of diagnosis, prevention or treatment; either the rarity of the condition (affecting not more than 5 in 10,000 people in the EU) or insufficient returns on investment. Designated orphan medicinal products are products that are still under investigation and are considered for orphan designation on the basis of potential activity. An orphan designation is not a marketing authorisation. As a consequence, demonstration of quality, safety and efficacy is necessary before a product can be granted a marketing authorisation. EMA/COMP/426291/2012 Page 2/6

For more information Sponsor s contact details: Albireo AB Arvid Wallgrens Backe 20 S-413 46 Gothenburg Sweden Telephone: +46 31 741 14 80 Telefax: +46 31 82 02 23 E-mail: info@albireopharma.com For contact details of patients organisations whose activities are targeted at rare diseases see: Orphanet, a database containing information on rare diseases which includes a directory of patients organisations registered in Europe. European Organisation for Rare Diseases (EURORDIS), a non-governmental alliance of patient organisations and individuals active in the field of rare diseases. EMA/COMP/426291/2012 Page 3/6

Translations of the active ingredient and indication in all official EU languages 1, Norwegian and Icelandic Language Active ingredient Indication English Bulgarian Czech 2,3,4,5-tetrahydro- 1,2,5- benzothiadiazepin-8-(4- hydroxyphenyl)acetyl]amino}butanoic acid (2S)-2-{[(2R)-2-[({[3,3-дибутил-7-(метилтио)-1,1- диоксидо-5-фенил- 2,3,4,5-тетрахидро-1,2,5-бензотиадиазепин-8- ил]окси}ацетил)амино]-2- (4-хидроксифенил)ацетил]амино}бутаноева киселина (4-hydroxyphenyl)acetyl]amino}butanová kyselina Treatment of progressive familial intrahepatic cholestasis Лечение на прогресивна фамилна интрахепатална холестаза Léčba progresivní familiární intrahepatální cholestázy Danish Dutch Estonian Finnish French (4-hydroxyphenyl)acetyl]amino}butansyre (4-hydroxyphenyl)acetyl]amino}butanoïczuur (2S)-2-{[(2R)-2-[({[3,3-dibutüül-7-(methüülthio)-1,1- dioksüido-5-fenüül- 2,3,4,5-tetrahüdro-1,2,5-benzothiadiazepiin-8- üül]oksü}atsetüül)amino]-2- (4-hüdroksüfenüül)atsetüül]amino}butanoiinhape (2S)-2-{[(2R)-2-[({[3,3-dibutyyli-7-(metyylitio)-1,1- dioksido-5-fenyyli- 2,3,4,5-tetrahydro-1,2,5-bentsotiadiatsepiini-8- yl]oksi}asetyyli)amino]-2- (4-hydroksifenyyli)asetyyli]amino}butaanihappo (4-hydroxyphenyl)acetyl]amino} acide butanoïque Behandling af progressiv familiær intrahepatisk kolestase Behandeling van progressieve familiale intrahepatische cholestase Progresseeruva perekondliku Intrahepaatilisee kolestaasi ravi Etenevän familiaalisen intrahepaattisen kolestaasin hoito Traitement de la cholestase intrahépatique familiale progressive 1 At the time of designation EMA/COMP/426291/2012 Page 4/6

Language Active ingredient Indication German Greek Hungarian Italian Latvian Lithuanian Maltese Polish Portuguese Romanian (4-hydroxyphenyl)acetyl]amino}butanoic acid (2S)-2-{[(2R)-2-[({[3,3-διβουτυλο-7-(μεθυλθειο)-1,1- διοξιδο-5-φαινυλο- 2,3,4,5-τετραϋδρο-1,2,5-βενζοθειαδιαζεπαμινο-8- yl]oxy}ακετυλ)αμινο]-2- (4-υδροξυφαινυλο)ακετυλ]αμινο}βουτανοϊκό οξύ (2S)-2-{[(2R)-2-[({[3,3-dibutil-7-metiltio)-1,1-dioxid-5 fenil- (4 hidroxifenil) acetil]amino}butánsav (4-hydroxyphenyl)acetyl]amino}butanoic acid (2S)-2-{[(2R)-2-[({[3,3-dibutil-7-(metiltio)-1,1- dioksido-5-fenil- 2,3,4,5-tetrahidro-1,2,5-benzotiadiazepīns-8- il]oksi}acetil)amino]-2- (4-hidroksifenil)acetil]amino}butānskābe (2S)-2-{[(2R)-2-[({[3,3-dibutil-7-(metiltio)-1,1- dioksido-5-fenil- il]oksi}acetil)amino]-2- (4-hidroksifenil)acetil]amino}sviesto rūgštis (4-hydroxyphenyl)acetyl]amino}butanoic acid dioksydo-5-fenyl- 2,3,4,5-tetrahydro-1,2,5-benzotiadiazepin-8- yl]oksyy}acetyl)amino]-2- (4-hydroksyfenyl)acetyl]amino}butanowy kwas Ácido (2S)-2-{[(2R)-2-[({[3,3-dibutil-7-(metiltio)-1,1- dióxido-5-fenil- 2,3,4,5-tetrahidro-1,2,5-benzotiadiazepina-8- (4-hidroxifenil)acetil]amino}butanoico Acid (2S)-2-{[(2R)-2-[({[3,3-dibutil-7-(metiltio)-1,1- dioxido-5-fenil- Behandlung progressiver familiärer intrahepatischer Cholestase Θεραπεία της προοδευτικής οικογενούς ενδοηπατικής χολόστασης Progresszív familiáris intrahepaticus cholestasis kezelése Trattamento della colestasi intraepatica progressiva familiare Progresīvās ģimenes intrahepatiskās holestāzes ārstēšana Progresuojančios paveldimos intrahepatinės cholestazės gydymas Kura tal-kolestasi intraepatika li tintiret progressiva Leczenie postępującej rodzinnej cholestazy wewnątrzwątrobowej Tratamento da colestase intra-hepática familiar progressiva Tratamentul colestazei familiale intrahepatice EMA/COMP/426291/2012 Page 5/6

Language Active ingredient Indication Slovak Slovenian Spanish Swedish Norwegian Icelandic (4-hidroxifenil)acetil]amino}butanoic dioxido-5-fenyl- 2,3,4,5-tetrahydro-1,2,5-benzotiadiazepín-8- (4-hydroxyfenyl)acetyl]amino}butánová kyselina (2S)-2-{[(2R)-2-[({[3,3-dibutil-7-(metiltio)-1,1- dioksido-5-fenil- il]oksi}acetil)amino]-2- (4-hidroksifenil)acetil]amino}maslena kislina Ácido (2S)-2-{[(2R)-2-[({[3,3-dibutil-7-(metiltio)-1,1- dioxido-5-fenil- (4-hidroxifenil)acetil]amino}butanoico dioxido-5-fenyl- 2,3,4,5-tetrahydro-1,2,5-benzotiadiazepin-8- (4-hydroxyfenyl)acetyl]amino}butansyra dioksido-5-fenyl- 2,3,4,5-tetrahydro-1,2,5-benzotiadiazepin-8- yl]oksy}acetyl)amino]-2- (4-hydroksyfenyl)acetyl]amino}butansyre (2S)-2-{[(2R)-2-[({[3,3-díbútýl-7-(methýlthíó)-1,1- díoxídó-5-phenýl-2,3,4,5-tetrahýdró- 1,2,5- benzóthíadíazepín-8-ýl]oxý}acetyýl)amínó]-2-(4- hýdróxýphenýl)acetýl]amínó}bútanóic sýra progresive Liečba progresívnej familiárnej intrahepatálnej cholestázy Zdravljenje progresivne familiarne intrahepatične holestaze El tratamiento de la colestasis intrahepática familiar progresiva Behandling av progressiv familjär intrahepatisk kolestas Behandling av progressiv familiær intrahepatisk kolestase Meðferð á ágengriættgenginni innanlifrar gallteppu EMA/COMP/426291/2012 Page 6/6