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Chronic GVHD manifesting as parotitis after allogeneic hematopoietic SCT

Bone Marrow Transplantation volume 44, pages 821–822 (2009)Cite this article

GVHD is artificially divided into acute and chronic forms depending on its date of onset. Although there may be a continuum of clinical findings, chronic GVHD can affect numerous organs that are not typically affected by acute GVHD. Chronic GVHD involving the salivary glands and/or lacrymal glands, which clinically manifests as xerophthalmia and/or xerostomia similar to Sjögren's syndrome, occurs commonly after hematopoietic cells transplantation, particularly with transplants using mobilized PBSCs. However, de novo chronic GVHD manifesting as parotitis and submandibular lymphadenopathy is underrepresented in the literature. Here, we describe a patient with severe aplastic anemia, who received an allogeneic hematopoietic cell transplant that developed recurrent parotitis and submandibular lymphadenopathy >8 months after the procedure, which may have been a manifestation of chronic GVHD.

The patient is a 37-year-old Nepalese female, who developed RBC and plt transfusion-dependent pancytopenia in 2003. A BM biopsy was consistent with a diagnosis of severe aplastic anemia, and she received treatment in her homeland with prednisone and CYA for >2 years without response. She presented to the NHLBI in 2006 where a diagnosis of severe aplastic anemia was confirmed. She received a trial of CYA and horse antithymocyte globulin without response and subsequently underwent a T-cell replete G-CSF-mobilized PBSC transplant from her HLA identical sibling on 21 March 2007 after conditioning with fludarabine, CY (Cytoxan) and horse antithymocyte globulin.1 She received CYA and mini-dose MTX as GVHD prophylaxis. Neutrophil engraftment occurred on day +20 and by day +30 short tandem repeat-based PCR studies revealed that she had achieved 100% donor chimerism in myeloid and T-cell lineages. On day +85, she developed acute GVHD of the gastrointestinal tract associated with watery diarrhea, which resolved completely with corticosteroid therapy. On day +219, she developed new onset thrombocytopenia possibly related to chronic GVHD, which prompted treatment with mycophenolate mofetil (an evaluation for plt auto-antibodies was not pursued). On day +263, the patient was admitted to the hospital for evaluation of multiple new symptoms including symmetric, diffuse swelling of the neck and cheeks, facial edema, fever, a nonproductive cough and was noted to have increased blood eosinophils (18.2% with an absolute eosinophil count of 1.966 K cells/μl). Her medications on admission included CYA, mycophenolate mofetil, valtrex, atovaquone, amlodipine, magnesium and lamivudine. She had no earlier history of mumps or sick contact exposure.

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References

  1. Srinivasan R, Takahashi Y, McCoy JP, Espinoza-Delgado I, Dorrance C, Igarashi T et al. Overcoming graft rejection in heavily transfused and allo-immunised patients with bone marrow failure syndromes using fludarabine-based haematopoietic cell transplantation. Br J Haematol 2006; 133: 305–314.

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Acknowledgements

We thank Drs Sumi Vasu, Steven Pavletic and Ram Srinivasan for their valuable input.

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Authors and Affiliations

  1. National Institutes of Health/National Cancer Institute-National Heart, Lung, and Blood Institute (NIH/NCI-NHLBI), Bethesda, MD, USA

    D Z Drew, T Donohue, C Ramos, L Cook, R Goodwin, N Patronas, D Arrington, M Ramanathan & R Childs

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  1. D Z Drew
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  2. T Donohue
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  3. C Ramos
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  4. L Cook
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  5. R Goodwin
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  6. N Patronas
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  7. D Arrington
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  8. M Ramanathan
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  9. R Childs
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Correspondence to R Childs.

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Drew, D., Donohue, T., Ramos, C. et al. Chronic GVHD manifesting as parotitis after allogeneic hematopoietic SCT. Bone Marrow Transplant 44, 821–822 (2009). https://doi.org/10.1038/bmt.2009.79

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