CASe RePORT
Stem Cell Transplant in Severe Glanzmann
Thrombasthenia in an Adult Patient
Mani Ramzi, Mehdi Dehghani, Shirin Haghighat, Hourvash Haghighi Nejad
Abstract
Glanzmann thrombasthenia is an inherited auto-somal
recessive disorder characterized by normal platelet
count but lack of platelet aggregation due to absence of
platelet glycoprotein IIb/IIIa. The disease usually is
associated with mild bleeding, but severe fatal
hemorrhage may occur. Allogeneic stem cell transplant
is the only curative method of treatment. A literature
search showed 18 previously reported cases of
Glanzmann thrombasthenia treated with allogeneic
hematopoietic stem cell transplant. We report an
18-year-old woman with severe Glanzmann
thrombasthenia who was treated with allogeneic
hematopoietic stem cell transplant from her sister.
After 24-month follow-up, the patient was well, had no
bleeding tendency, and had mild chronic skin graft-
versus-host disease.
Key words: Graft-versus-host disease, Transplant,
Inherited, Glanzmann
Introduction
Glanzmann thrombasthenia is a rare inherited
autosomal recessive disorder characterized by
normal platelet count but lack of platelet aggregation
due to absence of platelet glycoprotein IIb/IIIa. The
clinical course of the disease usually can be mild but
sometimes, may be severe and may be characterized
by repeated life-threatening hemorrhage, with severity
varying between patients.
1
The clinical course may be
unpredictable.
2
Therapy is based on supportive care including
hemostatic procedures, antifibrinolytic agents, desmo-
pressin for minor hemorrhage, and transfusion with
packed red blood cells or platelet concentrates for
major hemorrhage.
3-5
The main complication of
platelet concentrate transfusion is the occurrence of
alloimmunization against human leukocyte antigens
(HLAs) and the glycoprotein IIb/IIIa complex. In this
condition, patients become refractory to platelet
transfusions and have a poor prognosis.
Allogeneic hematopoietic stem cell transplant for
severe Glanzmann thrombasthenia was initially
reported in 1985.
6
Since then, hematopoietic stem
cell transplant has been reported sporadically.
7-11
A
literature search showed 18 cases of Glanzmann
thrombasthenia treated with allogeneic hematopoietic
stem cell transplant.
12
We report an 18-year-old woman who presented
with severe, repeated bleeding. She was treated
successfully with allogeneic hematopoietic stem cell
transplant in our center from her HLA-matched
sister. At 2-year posttransplant follow-up, the patient
was well, asymptomatic, and had mild chronic skin
graft-versus-host disease (GVHD).
Case Report
An 18-year-old woman was diagnosed at age 1 year,
after gum bleeding, with Glanzmann thrombasthenia.
She was referred to our center due to refractory
bleeding and frequent hospitalization because of
bleeding. She had a history of gastrointestinal
bleeding at age 11 years that required blood
transfusion for the first time. Since then, she had
frequent epistaxis leading to severe anemia and
requiring blood and platelet transfusion. During the
past several years, she continued to need frequent
platelet and blood transfusions, and recently
required recombinant factor VII to stop severe and
uncontrolled bleeding events. Her bleeding became
frequent, and she required many hospitalizations
Copyright © Başkent University 2016
Printed in Turkey. All Rights Reserved.
From the Hematology Research Center, Department of Hematology, Medical Oncology, and
Stem Cell Transplantation, Shiraz University of Medical Sciences, Shiraz, Iran
Corresponding author: Mani Ramzi, Professor of Hematology and Medical Oncology,
Department of Hematology, Medical Oncology, and Stem Cell Transplantation, Shiraz
University of Medical Sciences, Namazee Hospital, Zand Boulevard, Shiraz, Iran
Phone: +98 713 647 4301 Fax: +98 713 647 4301 E-mail: ramzim@sums.ac.ir
Experimental and Clinical Transplantation (2016) 6: 688-690
DOI: 10.6002/ect.2014.0165