More immune dysregulation: Sarcoidosis and chronic graft-versus-host disease after allogeneic stem cell transplant

Manalo, Iviensan F.; Miller, India A.; Davis, Loretta S.

doi:10.1016/j.jdcr.2016.01.008

Cited by 6 publications

(5 citation statements)

References 10 publications

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“…An additional 15 cases of sarcoidosis following HSCT have been reported in the literature, with ve patients having received an auto HSCT [18, 21,22], while ten had received an allo HSCT [18, [23][24][25][26][27][28][29][30]. Although the total populations of the previously published case reports are unknown, the overall low incidences in the allogeneic transplantation group appear consistent between our cohort and the published data.…”

Section: Patient Characteristicssupporting

confidence: 81%

Sarcoidosis following Hematopoietic Stem Cell Transplantation: clinical characteristics and HLA associations

Wurm-Kuczera

Büntzel

König

et al. 2021

Preprint

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Purpose: Extrinsic factors and genetic predisposition contribute to the etiology of sarcoidosis, converging in a phenotype of altered immune response associated with multisystemic inflammatory granulomatous tissue infiltration. Immunological reconstitution after hematopoietic stem cell transplantation (HSCT) may represent a unique window for the pathogenesis of the disease. We describe the incidence, clinicopathological features, and HLA associations of sarcoidosis after HSCT in a single-center cohort of patients, together with data from previously published cases. Methods: We retrospectively analyzed clinical characteristics and HLA haplotypes from allogeneic (allo) or autologous (auto) HSCT patients from January 2001 through May 2021 at the University Medicine Goettingen (UMG), and data from 15 previously published cases. Results: 13 patients had received an allo HSCT, and six patients an auto HSCT, including four at our center (three allo HSCT, one auto HSCT). Sarcoidosis occurred after a median interval of 20 and 7 months, respectively. The predominant HLA allele associated with sarcoidosis was HLA DRB1*03:01. Sarcoidosis involved the respiratory tract in 15 patients (three unknown, one without pulmonary involvement), and it was associated with graft-versus-host disease in seven of 13 patients receiving allo HSCT. None of the donors or patients had a history of sarcoidosis before transplantation. Disease manifestations resolved by standard glucocorticoid treatment without long-term sequelae. Conclusion: Sarcoidosis may occur at low frequency during reconstitution of the immune system after HSCT. HLA allele associations reflect the associations observed in the general population, particularly with DRB1*03:01. Further insights into the interplay between T cell reconstitution and the development of sarcoidosis could also provide novel approaches to an improved understanding of the pathogenesis in sarcoidosis.

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Section: Patient Characteristicssupporting

confidence: 81%

Sarcoidosis following Hematopoietic Stem Cell Transplantation: clinical characteristics and HLA associations

Wurm-Kuczera

Büntzel

König

et al. 2021

Preprint

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“…Two of the patients were diagnosed with chronic GVHD at the same time the diagnosis of sarcoidosis was made, and there was a known history of sarcoidosis in the donor in three of the cases. None of the patients reported treatment with rituximab prior to the diagnosis of sarcoidosis 13 . Another association, the sarcoidosis‐lymphoma syndrome, describes the diagnosis of lymphoma in patients with already known sarcoidosis.…”

Section: Discussionmentioning

confidence: 99%

Scar sarcoidosis following rituximab therapy

Vesely

Thomas

Rudnick

et al. 2020

Dermatologic Therapy

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Drug-induced sarcoidosis has been rarely described but it constitutes a potential side effect of immunomodulatory medications. We report a case of rituximab-induced scar sarcoidosis and review similar published cases. Although there is evidence of B-cell dysregulation in the pathogenesis of sarcoidosis, the use of rituximab for this disease needs to be carefully evaluated based on reports of worsening and de novo development of sarcoidosis after rituximab therapy.

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“…Although granulomatous diseases are not considered for chronic GVHD diagnosis according to the 2014 NIH consensus, there are reports in the literature of sarcoidosis with skin involvement following allogeneic HSCT. Manalo et al described a case of cutaneous and pulmonary sarcoidosis occurring concurrently with lichen sclerosus-like chronic GVHD [ 75 ]. Recently, Kinsella et al reported a case of folliculotropic mycosis fungoides in the post-HSCT setting, first diagnosed as cutaneous sarcoidosis based on clinical-histopathological correlation.…”

Section: Clinical Manifestationsmentioning

confidence: 99%

Cutaneous Graft-Versus-Host Disease: Diagnosis and Treatment

et al. 2017

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Graft-versus-host disease (GVHD) is an immunological reaction and a frequent complication following allogeneic hematopoietic stem cell transplantation. It is associated with high mortality rates and may have a significant negative impact on the patient’s quality of life, particularly in the chronic-stage setting. Many different organs can be involved, which leads to a wide range of clinical manifestations. In this context, dermatologists play a key role by diagnosing and treating GVHD from the outset since cutaneous features are not just the most common but are also usually the presenting sign. Several skin-direct therapies are available and may be indicated as monotherapy or adjuvant treatment in order to allow faster tapering and withdrawal of systemic immunosuppression. Treatment of steroid-refractory patients remains a challenge and, to date, no consensus has been reached for one single agent in second-line therapy. This article aims to review skin involvement as well as provide and update discussion on therapeutic options for both acute and chronic cutaneous GVHD.

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More immune dysregulation: Sarcoidosis and chronic graft-versus-host disease after allogeneic stem cell transplant

Cited by 6 publications

References 10 publications

Sarcoidosis following Hematopoietic Stem Cell Transplantation: clinical characteristics and HLA associations

Sarcoidosis following Hematopoietic Stem Cell Transplantation: clinical characteristics and HLA associations

Scar sarcoidosis following rituximab therapy

Cutaneous Graft-Versus-Host Disease: Diagnosis and Treatment

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