Regression of syndesmophyte after bone marrow transplantation for acute myeloid leukemia in a patient with ankylosing spondylitis: a case report

Abstract

IntroductionDisease progression of ankylosing spondylitis has been considered irreversible. However, we report a case of spontaneous regression of syndesmophyte development following allogeneic peripheral blood stem cell transplantation in a patient with acute myeloid leukemia, who was also diagnosed as having ankylosing spondylitis. To the best of our knowledge, this is the first case report presenting the partial radiologic regression of syndesmophytes.Case presentationA 39-year-old man with active ankylosing spondylitis achieved clinical remission and partial radiological regression of cervical spine syndesmophytes following a peripheral blood stem cell transplantation for acute myeloid leukemia. Our patient received an allogeneic peripheral blood stem cell transplantation following a pre-transplantation conditioning regimen of total body irradiation and cyclophosphamide. The donor was a human leukocyte antigen-matched 29-year-old man. Our patient has remained asymptomatic and has received no medication for ankylosing spondylitis for nearly three years.ConclusionsSeveral explanations are proposed for the regression of syndesmophytes and clinical improvement in active ankylosing spondylitis observed in our patient, including changes in bone remodeling and immune reconstitution following stem cell transplantation, the effect of immunosuppressive agents, or fluctuation in the natural course of ankylosing spondylitis although further studies are required. The regression of syndesmophytes in ankylosing spondylitis in this case raises the possibility that stem cell transplantation might contribute to the development of a novel therapeutic strategy for treatment of the disease.

Role of stem cells in spondyloarthritis: Pathogenesis, treatment and complications

“…In another case report, a 39-year-old man with active AS and concomitant acute myeloid leukaemia (AML) was given pre-transplantation total body irradiation and cyclophosphamide followed by allogeneic peripheral blood SCT. He experienced clinical remission and partial radiological regression of cervical spine syndesmophytes, and remained medication-and symptom-free for three years after the transplantation [75].…”

Stem Cell Transplantation as a Possible Strategy for Treating Standard Therapy-Resistant Ankylosing Spondylitis

“…Суммарно описано четыре ТСК больным АС, из них в 1 случае трансплантация выполнена больному с сочетанием лимфомы и АС [12], в 2 случаях -больным АС без онкоге-матологических заболеваний [13,14], в 1 случае -пациенту с сочетанием АС и миелоидного лейкоза [15]. У 3 больных АС достигнута безлекарственная ремиссия [12,13,15]: у больного с сочетанием лимфомы и АС, у пациента с соче-танием АС и миелоидного лейкоза и у больного АС без со-путствующих заболеваний.…”

“…У 3 больных АС достигнута безлекарственная ремиссия [12,13,15]: у больного с сочетанием лимфомы и АС, у пациента с соче-танием АС и миелоидного лейкоза и у больного АС без со-путствующих заболеваний.…”

“…Аллогенная ТСК, при которой пациенту вводится ко-стный мозг или стволовые клетки периферической крови от родственного или неродственного совместимого донора, применялась в 2 случаях [14,15], аутологичная трансплан-тация, при которой реципиент получает предварительно заготовленный собственный костный мозг или гемопоэти-ческие клетки периферической крови -также в 2 случаях [12,13].…”

“…[15]. У 39-летнего мужчины, в течение 20 лет страдавшего АС, был обнаружен острый миелоидный лейкоз.…”