Cyclosporine-Induced Pain Syndrome in a Child Undergoing Hematopoietic Stem Cell Transplant

Lavoratore, Sara R; Navarro, Oscar M.; Grunebaum, Eyal; Ali, Muhammad; Koo, Alicia; Schechter, Tal; Gassas, Adam; Doyle, John; Dupuis, L. Lee

doi:10.1345/aph.1l641

Cited by 20 publications

(12 citation statements)

References 23 publications

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“…The onset of CIPS is variable. It has been d as early as 3 days following the intravenous infusion of cyclosporine [7] and as late as 8 years, as seen in our patient. Similarly, association with the trough levels of the CIs is variable too.…”

supporting

confidence: 73%

Calcineurin-inhibitor induced pain syndrome in a lung transplant patient

2013

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supporting

confidence: 73%

Calcineurin-inhibitor induced pain syndrome in a lung transplant patient

2013

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“…CIPS is hypothesized to result from calcineurin-induced vascular changes that disturb bone perfusion and permeability, which leads to intraosseous vasoconstriction and BM edema [18,19]. This rare syndrome has been described in organ transplant recipients [32][33][34][35], where CIs must be maintained at a high concentration, whereas several cases of CIPS have been reported for patients undergoing HSCT [18,19,[36][37][38][39]. Moreover, in our study, 11 of 13 patients with HHV6-associated encephalitis/myelitis developed CIPS-like dysesthesia and/or pruritus.…”

Section: Discussionmentioning

confidence: 99%

High Incidence of Human Herpes Virus 6-Associated Encephalitis/Myelitis following a Second Unrelated Cord Blood Transplantation

Mori

Miyamoto

Nagafuji

et al. 2010

Biology of Blood and Marrow Transplantation

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Human herpes virus (HHV)6-associated limbic encephalitis and/or myelitis is one of the life-threatening central nervous system complications following allogeneic hematopoietic stem cell transplantation (HSCT). Recent reports have shown significant correlations of these complications with unrelated cord blood transplantation (UCBT). We retrospectively analyzed 228 allogeneic HSCT recipients in our single institution; 13 patients (5.7%) were diagnosed with HHV6-associated encephalitis/myelitis. This complication was documented in 8 of 51 UCBT recipients (15.7%) and 5 of 177 recipients (2.8%) transplanted with bone marrow or peripheral blood stem cells, indicating a higher incidence of this complication occurring in UCBT recipients (P = .0005). In addition, HHV6-associated encephalitis/myelitis occurred more frequently in recipients who underwent 2 or more HSCTs (7 of 59 recipients [11.9%]), compared to those who received only 1 HSCT (6 of 169 recipients [3.6%], P = .018). Of note, the incidence of this complication increased to 28.6% (6 of 21 recipients), when the analysis was restricted to a second or more UCBT recipients. All 13 patients presented preengraftment immune response prior to the onset of encephalitis. Two patients manifested typical symptoms at the onset of HHV6-associated encephalitis/myelitis, such as memory dysfunction, disorientation, and consciousness disturbance. However, 4 patients presented only with dysesthesia and pruritus, described as typical manifestations of patients with calcineurin-inhibitor-induced pain syndrome (CIPS), and the remaining 7 showed both symptoms, indicating that CIPS-like symptoms might be manifestations of HHV6-associated myelitis. Thus, physicians should be alert to this rare but often fatal complication, particularly for those who receive 2 or more HSCTs using UCB.

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“…Jain and colleagues 18 reported that patients with hepatic dysfunction had a longer half-life of tacrolimus (corresponding to a lower clearance rate and higher trough plasma concentration); and the presence of CIPS is correlated to a high blood concentration of calcineurin inhibitors. 12,19 This may explain why the incidence of CIPS is high in our present study involving OLT patients; hence, specific attention should be paid to the application of tacrolimus in patients with liver dysfunction in clinical practice. In our study, the incidence of ONFH was only 1.7%, and the diagnosis was made about 1 year after transplant.…”

Section: Discussionmentioning

confidence: 83%

Immunosuppressant-Related Hip Pain After Orthotopic Liver Transplant

He²,

Fu³

et al. 2012

Exp Clin Transplant

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Objectives: Immunosuppressant-related hip pain can greatly affect a patient's mobility and increase the number of total hip arthroplasties. We investigated risk factors and causes of hip pain after orthotopic liver transplant. Materials and Methods:The medical records of 175 adult orthotopic liver transplant patients, who were followed-up for more than 2 years, were retrospectively reviewed. Data collected from the records included primary disease, medications, biochemical results, Child-Turcotte-Pugh score, death, rejection, and complications related to liver transplant. Results: A total of 11 patients (6.3%) complained of hip pain, which was diagnosed as calcineurininhibitor-induced pain syndrome in 4 patients (2.3%), osteonecrosis of the femoral head in 3 patients (1.7%), and osteoporosis in 2 patients (1.1%). The incidence of calcineurin-inhibitorinduced pain syndrome was related to the dosage of tacrolimus (P > .05) but independent of methylprednisolone use. The occurrence of osteonecrosis of the femoral head was independent of the dosage and early withdrawal of methylprednisolone (P > .05). Patients with methylprednisolone withdrawal within 6 months had significantly longer survival than those using methylprednisolone for more than 6 months (50 ± 15 vs 41 ± 18 mo; P = .007). Conclusions: Calcineurin-inhibitor-induced pain syndrome and osteonecrosis of the femoral head are main causes of hip pain in adult orthotopic liver transplant patients. Osteonecrosis of the femoral head was not common, but the incidence of hip pain owing to calcineurin-inhibitor-induced pain syndrome was relatively high in orthotopic liver transplant patients. Early withdrawal of methylprednisolone could benefit the patients' survival.

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Cyclosporine-Induced Pain Syndrome in a Child Undergoing Hematopoietic Stem Cell Transplant

Abstract: Interventions aimed at reducing pain associated with CIPS may include the initiation of calcium-channel blocker therapy and conversion to an alternative calcineurin inhibitor.

Cited by 20 publications

References 23 publications

Calcineurin-inhibitor induced pain syndrome in a lung transplant patient

Calcineurin-inhibitor induced pain syndrome in a lung transplant patient

High Incidence of Human Herpes Virus 6-Associated Encephalitis/Myelitis following a Second Unrelated Cord Blood Transplantation

Immunosuppressant-Related Hip Pain After Orthotopic Liver Transplant

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