Disarticulation of a knee joint in a haemophiliac with high inhibitor titre

Chuansumrit, Ampaiwan; Hathirat, P; Keorochana, S; Tardtong, P; Pintadit, P; Rueangwetsawat, Y; Isarangkura, P

doi:10.1111/j.1365-2516.1996.tb00027.x

Cited by 5 publications

(6 citation statements)

References 7 publications

(1 reference statement)

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“…The successful disarticulation of the knee joint was previously reported in 1996 [8]. At 8 years of age, he had an infected compound fracture of both the tibia and ®bula and exhibited the highest inhibitor titre of 3450 BU.…”

Section: Case Reportmentioning

confidence: 78%

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Immune tolerance in a haemophilia A patient with high inhibitor using locally prepared lyophilized cryoprecipitate

et al. 2000

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The immune tolerance in a 12-year-old haemophilia A patient was carried out at the Faculty of Medicine, Ramathibodi Hospital, Bangkok in 1998. His inhibitor titres ranged from 10 to 3450 Bethesda units (BU). He suffered from serious bleeding episodes requiring prolonged hospitalization and the disarticulation of the left knee joint. After obtaining informed consent, locally prepared lyophilized cryoprecipitate (LC), heat treated at 60 degrees C for 25 h, was given in a dose of 13 units kg-1 body weight of factor VIII three times per week. His inhibitor was increased from 15 to 580 BU within the first 4 weeks of immune tolerance. Finally, it was decreased to 40 BU in the 36th week. The only adverse effect was seroconversion of anti-hepatitis C virus after receiving 108 bottles of LC for 36 weeks. In conclusion, the locally prepared LC was able to control the bleeding episodes in a haemophilia A patient with high inhibitor. To our knowledge, this is the first reported case in Thailand.

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Section: Case Reportmentioning

confidence: 78%

“…At 8 years of age, he had an infected compound fracture of both the tibia and ®bula and exhibited the highest inhibitor titre of 3450 BU. The successful disarticulation of the knee joint was previously reported in 1996 [8]. He walked using crutches and an arti®cial leg.…”

Section: Case Reportmentioning

confidence: 99%

Immune tolerance in a haemophilia A patient with high inhibitor using locally prepared lyophilized cryoprecipitate

et al. 2000

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“…Haemophiliac treatment and medication in less developed countries are far behind those in more developed countries. Moreover, the management of bleeding in haemophiliacs with inhibitors is more difficult due to limited supplies of blood products such as prothrombin complex concentrate, factor VIII concentrate and recombinant activated factor VII (rFVIIa) [1]. Recently, several studies have shown the efficacy of rFVIIa in controlling bleeding episodes [2,3].…”

Section: Patients Receiving Rfviia For Controlling Bleeding Episodesmentioning

confidence: 99%

A single high dose of recombinant factor VIIa combining adjuvant therapy for controlling bleeding episodes in haemophiliacs with inhibitors

et al. 2001

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“…In cases of life-saving conditions among patients with severe bleeding episodes, plasma exchange was performed before bolus administration of factor VIII or IX concentrate accordingly. [9][10][11][12] Bypassing agents of rFVIIa and aPCC [13][14][15][16] have been available in 2000 and 2003, respectively.…”

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confidence: 99%

Real‐world evidence on health resource use among patients with haemophilia and inhibitor exhibiting severe bleeding episodes

et al. 2020

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Objective This study aimed to explore real‐world evidence on health resource use (HRU) spending on patients with haemophilia and inhibitor. Materials and methods Medical records from 1990 to 2019 of patients with haemophilia and inhibitor from three comprehensive haemophilia treatment centres were retrospectively retrieved. Results In all, 31 patients with haemophilia (A = 30, B = 1) and inhibitor ≥5 BU were included. The mean initial inhibitor of 95.4 BU was detected at the mean age of 6.7 years. The mean number of annual hospitalisations was 3.9. A total of 795 bleeding episodes (major =125, minor =670) were evaluated. The treatment included bypassing agents or plasma exchange before administering high‐dose factor VIII concentrate and intervention or surgery. Six patients succumbed to bleeding at the mean age of 17.2 years. Nineteen surviving patients experienced multiple morbidity except six patients with successful and partially successful immune tolerance induction (ITI). The mean (SD) annual total medical consumption for episodic treatment and successful ITI per patient with haemophilia A were 30,804 (81,332) USD and 55,531 (100,566) USD, respectively. Only episodic treatment was paid by the government budget for limited amounts of bypassing agents. Conclusion Management for patients with haemophilia and inhibitor exhibiting severe bleeding is challenging for medical personnel in countries having limited resources over decades. The real‐world data will be used to negotiate with the government to increase budget for adequate bypassing agents or nonreplacement therapy and to include ITI in the national haemophilia treatment.

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Disarticulation of a knee joint in a haemophiliac with high inhibitor titre

Cited by 5 publications

References 7 publications

Immune tolerance in a haemophilia A patient with high inhibitor using locally prepared lyophilized cryoprecipitate

Immune tolerance in a haemophilia A patient with high inhibitor using locally prepared lyophilized cryoprecipitate

A single high dose of recombinant factor VIIa combining adjuvant therapy for controlling bleeding episodes in haemophiliacs with inhibitors

Real‐world evidence on health resource use among patients with haemophilia and inhibitor exhibiting severe bleeding episodes

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