Quiz in Hematology

Headnote

Diagnosis: Melanoderma after Hematopoietic Stem Cell Transplantation

Hematopoetik Kök Hücre Nakli Sonrasi Gelisen Melanoderma

Keywords: Hematopoietic stem cell transplantation, Melanoderma, Skin findings, SCID

Anahtar Sözcükler: Hematopoetik kök hücre nakli, Melanoderma, Deri bulgulari, SCID

An 8-month-oldboy diagnosed with T-B+NK- SCID underwent peripheral blood hematopoietic stem cell transplantation (HSCT) from MSD without conditioning. However, he developed pancytopenia and became transfusion dependent by posttransplant 2nd month. Bone marrow aspiration/biopsy revealed an aplastic marrow with 98% donor chimerism. With a diagnosis of T-cell engraftment of the donor but no engraftment of the other lineages, a 2nd HSCT with conditioning (BU/FLU/ATG) was performed at post-transplant +23rd month from the same donor. Due to hyperferritinemia pre-transplant desferoxamine was given. On post-transplant day +2, he developed hyperpigmented patches (Figure 1). Platelet count was 22x109/L and aPTT and PT were normal. Platelet transfusion was given; however the lesions did not subside with the expected color change of ecchymoses. Skin biopsy from medial thigh was obtained (Figure 2).

Generalized hyperpigmentation, after conditioning, is a common finding after HSCT [1]. However, in our patient the lesions were patchy. There are few reports of melanoderma [1,2] after HSCT and in one, melanoderma was reported as a finding of chronic GvHD [2]. Based on the absence of clinical signs of GvHD and lack of typical histological evidence, the melanoderma in our patient was attributed to drugs used in conditioning. The transfusional iron loading may cause a generalized darkening of the skin; however in our patient the lesions were patchy and developed just after completion of the conditioning regimen and subsequent stem cell infusion. The patient did not develop acute or chronic GvHD signs throughout the follow-up. The lesions' color faded after engraftment gradually, although did not disappear totally. Currently, the patient is alive at post-HSCT 6th month.

Informed Consent: Informed consent has been obtained from the parents of the patient, Concept: Sule Ünal, Ilhan Tezcan, Safak Güçer, Meryem Seda Boyraz, Deniz Çagdas, Duygu Uçkan Çetinkaya, Design: Sule Ünal, Ilhan Tezcan, Safak Güçer, Meryem Seda Boyraz, Deniz Çagdas, Duygu Uçkan Çetinkaya, Data Collection or Processing: Sule Ünal, Ilhan Tezcan, Safak Güçer, Meryem Seda Boyraz, Deniz Çagdas, Duygu Uçkan Çetinkaya, Analysis or Interpretation: Sule Ünal, Ilhan Tezcan, Safak Güçer, Meryem Seda Boyraz, Deniz Çagdas, Duygu Uçkan Çetinkaya, Literature Search: Sule Ünal, Ilhan Tezcan, Safak Güçer, Meryem Seda Boyraz, Deniz Çagdas, Duygu Uçkan Çetinkaya, Writing: Sule Ünal, Ilhan Tezcan, Safak Güçer, Meryem Seda Boyraz, Deniz Çagdas, Duygu Uçkan Çetinkaya.

Conflict of Interest: The authors of this paper have no conflicts of interest, including specific financial interests, relationships, and/or affiliations relevant to the subject matter or materials included.

References

References

1. Aractingi S, Janin A, Devergie A, Bourges M, Socie G, Gluckman E. Histochemical and ultrastructural study of diffuse melanoderma after bone marrow transplantation. Br J Dermatol 1996;134:325-331.

2. Martin-Gorgojo A, Martín JM, Gavrilova M, Monteagudo C, Jordá-Cuevas E. Chronic graft-versus-host disease presenting with coexisting diffuse melanoderma and hypopigmented patches: A peculiar presentation. Eur J Dermatol. 2013;23:553- 555.