Managing the exceptional: pyogenic granuloma as a manifestation of oral cGVHD

Chronic graft-versus-host disease (cGVHD) represents one of the major complications following hematopoietic stem cell transplantation (allo-HSCT), affecting up to 80% of patients within the oral cavity. While erosions and lichenification are classic, the appearance of pyogenic granulomas (PG) remains a rare manifestation, particularly in children, capable of severely altering quality of life through intense pain and functional discomfort.

This case report describes the diagnostic and therapeutic challenge posed by a 5-year-old patient, treated with a second haploidentical allo-HSCT for severe aplastic anemia, who developed ulcerated nodular lesions 270 days post-transplant. Following the initial failure of conservative management with topical corticosteroids (0.1% clobetasol propionate), the objective of this study is to detail the surgical management of these nodules and the specific strategies implemented to counter their recurrence.

Clinical analysis is based on the hypothesis that complete surgical excision, although constituting the gold standard treatment, can only be effective when combined with the elimination of local traumatic factors and stabilized control of systemic cGVHD. This case highlights the importance of rigorous histopathological diagnosis to differentiate these fibrovascular lesions from more common manifestations of oral cGVHD.

Clinical protocol and management

This case report documents the diagnostic and therapeutic management of a unique pediatric patient, a 5-year-old boy presenting with a rare form of oral chronic graft-versus-host disease (cGVHD). The patient had previously undergone a second haploidentical hematopoietic stem cell transplant (allo-HCT) to treat severe aplastic anemia.

The management protocol followed several precise chronological steps:

• Observation phase (D+270 post-transplantation): Identification of bilateral ulcerative lesions on the buccal mucosa and lateral borders of the tongue, progressing toward ulcerated nodular lesions.
• Initial conservative treatment: Topical application of 0.1% clobetasol propionate. Given the lack of clinical improvement and the progression of pain, an invasive approach was decided.
• Surgical procedure: Complete excision of the nodules in a surgical center, followed by a histopathological examination for diagnostic confirmation.
• Recurrence management: After observing a recurrence on the right buccal mucosa 7 days after surgery, a new protocol was established combining clobetasol 0.1% ointment with the use of acetate plates (protective splints) to eliminate local micro-trauma.

The final diagnosis of pyogenic granuloma was confirmed by histological analysis of the excised tissues.

Clinical and histopathological observations

Following a second haploidentical allo-SCT for severe aplastic anemia, clinical manifestations appeared at D+270 post-transplantation. The 5-year-old patient initially presented with painful ulcerated lesions on the bilateral buccal mucosa and the lateral borders of the tongue. These lesions progressed into ulcerated nodular formations.

Histopathological examination of the nodules after excision confirmed the diagnosis of pyogenic granuloma. This manifestation occurred in the context of oral chronic graft-versus-host disease (cGVHD).

Evolution and efficiency of interventions

The following table details the response to the various therapeutic strategies employed:

Follow-up and recurrence control

The study highlights significant tissue reactivity with the following observations:

• Recurrence delay: A recurrence appeared only 7 days after the surgical procedure on the right buccal mucosa.
• Therapeutic synergy: The failure of clobetasol alone, then surgery alone, highlights the importance of the combined approach. The use of acetate plates allowed for the neutralization of local traumatic factors, promoting the action of the topical corticosteroid.
• Final outcome: Adequate control of the underlying oral cGVHD, coupled with the elimination of mechanical irritants, led to a favorable clinical outcome with no further recurrence mentioned.

Clinical analysis and recurrence management

This case report highlights the diagnostic and therapeutic complexity of oral manifestations of chronic graft-versus-host disease (cGVHD) in children. Although cGVHD affects up to 80% of patients following hematopoietic stem cell transplantation, presentation as pyogenic granuloma (PG) remains rare. Clinically, the progression from simple ulcerations to painful nodules on post-transplantation day 270 underscores the need for continuous monitoring, even long after the transplant.

The initial failure of conservative treatment with 0.1% clobetasol propionate demonstrates that nodular lesions in a context of cGVHD can be refractory to topical steroids alone. The rapid recurrence observed seven days after surgical excision on the right buccal mucosa is a critical point: it suggests that the surgical procedure is not sufficient if local irritative factors persist or if systemic inflammation is not fully controlled.

The favorable outcome achieved after recurrence through the combination of clobetasol and acetate plates is significant. This combined approach indicates that mechanical protection against local trauma, coupled with anti-inflammatory treatment, is decisive in achieving the regression of recurrent lesions. The main limitation of these results lies in the unique nature of the reported case (n=1), which does not allow for the establishment of a universal protocol, but offers an effective rescue strategy for the practitioner facing a recurrence.

Summary of results

This case study documents the management of a 5-year-old child developing ulcerated pyogenic granulomas at D+270 following a bone marrow allograft (oral cGVHD). Faced with a post-surgical recurrence occurring in just 7 days, the application of 0.1% clobetasol propionate combined with the use of protective acetate plates achieved complete regression of the lesions.

In concrete terms, for the practitioner:

• Differential diagnosis: Systematically include pyogenic granuloma in your clinical assessment for any persistent nodular lesion in a transplant patient, beyond the classic lichenoid manifestations of cGVHD.
• Micro-trauma control: In cases of recurrence or locations exposed to friction, the fabrication of thermoformed acetate plates is a key strategy to isolate the wound and promote healing.
• Therapeutic optimization: Use these plates as transfer trays to increase the contact time of topical corticosteroids on the lesion, thus improving the effectiveness of the drug treatment compared to a simple application.

Technical lexicon of the study

Chronic Graft-Versus-Host Disease (cGVHD): Systemic immunological pathology occurring after allogeneic hematopoietic stem cell transplantation (allo-HCT), characterized by an attack on host tissues by donor immunocompetent cells. It affects the oral cavity in nearly 80% of patients.

Pyogenic granuloma: Benign reactive inflammatory lesion, clinically nodular and often ulcerated, presenting an exophytic vascular proliferation. In this clinical case, it appeared as a rare manifestation of oral cGVHD.

Haploidentical allo-HCT: Type of hematopoietic stem cell transplantation where the donor is a partially compatible family member (sharing a single HLA haplotype). The patient in the study underwent this protocol to treat severe aplastic anemia.

Clobetasol propionate 0.1%: Very high-potency topical corticosteroid used in the conservative management of inflammatory oral mucosal lesions related to cGVHD.

Acetate plates: Protection or maintenance devices used here in combination with clobetasol ointment to treat the recurrence of granuloma on the right buccal mucosa, promoting prolonged contact of the active ingredient.

Severe aplastic anemia: Severe bone marrow failure resulting in pancytopenia, constituting the initial indication for transplantation in the 5-year-old pediatric patient described in this report.

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