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Graft-Versus-Host Disease, Pediatric


Basics


Description


Graft-versus-host disease (GVHD) is a multiorgan inflammatory process that develops when immunologically competent T lymphocytes from a histoincompatible donor are infused into an immunocompromised host unable to reject them. Divided into acute and chronic, historically based on time of presentation but best delineated by clinicopathologic findings.  
  • Acute: develops within 100 days after allogeneic hematopoietic stem cell transplant (HSCT); affects skin, GI tract, and/or liver
  • Chronic: develops 100 days after allogeneic HSCT; presents with diverse features resembling autoimmune syndromes
  • Chronic subtypes
    • Progressive: extension of acute GVHD
    • Quiescent: after resolution of acute GVHD
    • De novo: no prior acute GVHD

Epidemiology


  • Acute GVHD (grades II-IV): 10-80% of patients receiving T-cell-replete HSCT
    • 35-45% for human leukocyte antigen (HLA)-identical related donor bone marrow
    • 60-80% if 1-antigen HLA-mismatched unrelated donor bone marrow or peripheral stem cells
    • 35-65% if 2-antigen HLA-mismatched unrelated umbilical cord blood
  • Chronic GVHD: most common cause of late morbidity and mortality of allogeneic HSCT
    • 15-25% if HLA-identical related marrow
    • 40-60% if HLA-matched unrelated marrow
    • 54-70% if HLA-matched unrelated peripheral stem cells
    • 20% if unrelated umbilical cord blood
  • Flare-ups triggered by infection (usually viral)

Risk Factors


  • HLA disparity (both major and minor antigens)
  • Older donor or recipient age
  • Stem cell source and dose
    • Highest risk: with peripheral stem cells
    • Lowest risk: with umbilical cord blood
  • Donor leukocyte infusions
  • Reactivation of viruses (e.g., HHV-6, CMV)
  • T-cell depletion decreases incidence.
  • Acute GVHD-specific
    • Higher intensity conditioning regimen
    • Prior pregnancies in female donors
    • Gender mismatch
  • Chronic GVHD specific
    • Severity of acute GVHD
    • Malignancy as indication for transplantation
    • Use of total-body irradiation
    • Type of immunosuppressive prophylaxis

Genetics
  • HLA gene complex on chromosome 6; inherited as haplotype
  • Full siblings: 25% chance HLA identical
  • Minor histocompatibility antigen differences likely account for GVHD in HLA-identical sibling stem cell transplants.

General Prevention


  • Transfusion: irradiation of all cellular blood products for patients at risk
  • Stem cell transplantation
    • Selection of a histocompatible donor
    • Immunosuppression (gold standard): cyclosporine or tacrolimus with a short course of methotrexate
    • Other options: corticosteroids, sirolimus, mycophenolate mofetil, and low-dose cyclophosphamide
    • Donor T-cell depletion with anti-T-cell antibodies ex vivo in graft or in vivo in recipient

Pathophysiology


  • Acute GVHD: interaction of donor and host innate and adaptive immune responses
    • Severity related to degree of HLA mismatch
    • 3 phases ending in "cytokine storm"
      • Tissue damage by conditioning regimen
      • Priming and activation of donor T cells
      • Infiltration of activated T cells into skin, GI tract, and liver resulting in apoptosis
  • Chronic GVHD: findings similar to autoimmune disorders: donor T cells directed against host antigens, donor T-cell autoreactivity, B-cell dysregulation, regulatory T-cell deficiency; marked collagen deposition in target organs and lack of T-cell infiltration

Etiology


  • Hematopoietic stem cell transplantation
  • Transfusion of nonirradiated blood products to immunodeficient hosts: caused by viable donor lymphocytes engrafting in the recipient
  • Transfusion of nonirradiated blood from a donor homozygous for 1 of the recipient's HLA haplotypes (1st- or 2nd-degree relative)
  • Intrauterine maternal-fetal transfusions and exchange transfusions in neonates
  • Solid organ grafts: contain immunocompetent T cells, into immunosuppressed recipient

Diagnosis


History


  • Acute GVHD
    • Rash: usually 1st manifestation; pruritus or burning sensation can precede rash.
    • Diarrhea, abdominal pain, and intestinal bleeding: unusual to precede skin disease
    • Anorexia, nausea, vomiting, and dyspepsia
    • Jaundice (liver involvement)
  • Chronic GVHD
    • Dry eyes and/or dry mouth, blurry vision, eye irritation, photophobia, eye pain
    • Difficulty swallowing or retrosternal pain
    • Sensitivity to mint, spicy foods, or tomatoes
    • Weight loss, failure to thrive, anorexia, nausea, vomiting, diarrhea
    • Dyspnea, wheezing, cough
    • Poor wound healing, especially post trauma
    • Joint stiffness, muscle cramps
  • Infections: pneumococcal sepsis, Pneumocystis jiroveci, fungal infections

Physical Exam


  • Acute/transfusion-associated GVHD
    • Skin (most common site)
      • Maculopapular rash of palms, soles; can become confluent erythroderma
      • Severe form: bullae formation, to even full-thickness necrosis
    • GI tract: weight loss; profuse, watery, often green, and often bloody diarrhea
    • Liver: jaundice; atypical findings: painful hepatomegaly, ascites, rapid weight gain
  • Chronic GVHD
    • Skin (involved in almost every patient)
      • Hyper- or hypopigmentation, xerosis (skin dryness), pruritus, scaling, patchy erythema, poikiloderma, skin atrophy; lichenoid, eczematous, and/or sclerodermatous changes
      • Advanced scleroderma: thickened, tight, and fragile skin
    • Hair: thin, fragile; premature graying
    • Scalp: dry or seborrheic
    • Nails: dystrophic, fragile; entire nail loss
    • Mouth: xerostomia, mucositis, ulcers; whitish, lacy plaques on tongue and buccal surfaces, may be painful; tight oral aperture
    • Esophageal strictures, stenosis, webs
    • Blood: thrombocytopenia, anemia, eosinophilia, autoantibodies, hypo- or hypergammaglobulinemia
    • Joints: stiffness, contractures, swelling
    • Muscles: eosinophilic fasciitis, myositis
    • Lung: bronchiolitis obliterans (obstructive), bronchiolitis obliterans organizing pneumonia (restrictive), pleural effusions
    • Other: pericardial effusions, pericarditis, cardiomyopathy, nephrotic syndrome, peripheral neuropathy, genital ulceration

Diagnostic Tests & Interpretation


Diagnosis is often made on clinical grounds.  
Lab
  • Complete blood count with differential and Coombs test: autoimmune thrombocytopenia (most common), hemolytic anemia, and neutropenia; eosinophilia, resolves with treatment
  • Blood smear: Howell-Jolly bodies due to functional asplenia of chronic GVHD
  • Elevated ALT/AST without hyperbilirubinemia
  • Vitamin D: often low; risk for osteoporosis
  • Urinalysis: may show protein, glucose, blood
  • Schirmer test: decreased tear production
  • Pulmonary function tests
  • Echocardiogram/electrocardiogram
  • Fluorescein microscopy: punctate keratopathy

Imaging
  • High-resolution chest CT: bronchiolitis obliterans
  • Barium swallow: strictures, webs

Diagnostic Procedures/Other
  • Endoscopy or colonoscopy with biopsy: apoptosis and loss of villi; GI tract GVHD
  • Skin biopsy: localized epidermal atrophy
  • Liver biopsy: bile duct damage reminiscent of primary biliary cirrhosis
  • Rule out viral/fungal infections
  • Analysis of pleural, pericardial fluid

Differential Diagnosis


  • Acute GVHD
    • Skin: drug reaction, chemoradiotherapy, viral exanthema, engraftment syndrome; TEN for grade IV skin GVHD
    • Liver: hepatic veno-occlusive disease, total parenteral nutrition, drug toxicity, bacterial sepsis, or viral infection
    • GI: diarrhea secondary to transplant conditioning regimen, infectious causes (e.g., Clostridium difficile, CMV, adenovirus), or opiate withdrawal
  • Chronic GVHD
    • Skin: keratosis pilaris, eczema, psoriasis

Alert
  • In chronic GVHD, do not give live vaccines; may lead to symptomatic infection
  • Sudden high fevers may indicate bacterial sepsis, may be overwhelming. Chronic GVHD patients often functionally asplenic; have profoundly impaired immune function

Treatment


Medication


  • Acute GVHD (grades II-IV)
    • 1st line: systemic steroids at 2 mg/kg/24 h for 7-14 days then quick taper; cyclosporine or tacrolimus if not taking as prophylaxis
    • 2nd line: mycophenolate mofetil, sirolimus (rapamycin), antithymocyte globulin, and etanercept (experimental)
    • Infliximab: steroid-refractory GI disease
    • Visceral organ involvement requires urgent start of 2nd-line therapy.
    • For isolated, mild skin GVHD, topical tacrolimus ointment and triamcinolone
  • Chronic GVHD
    • Steroids alone or with cyclosporine, sirolimus, tacrolimus, or mycophenolate mofetil
    • Goal: steroids <0.5 mg/kg alternating days; with cyclosporine or tacrolimus
  • Steroid-refractory GVHD
    • Mycophenolate mofetil, sirolimus, pentostatin (investigational)
    • Other options many off-label: antithymocyte globulin; rituximab; low-dose methotrexate in liver GVHD; thalidomide; hydroxychloroquine; imatinib; low-dose cyclophosphamide; etanercept; alefacept; alemtuzumab: high infection risk
  • Oral rinses with dexamethasone: oral GVHD
  • Ursodeoxycholic acid: hepatic GVHD

Additional Therapies


General Measures
  • Prophylaxis for P. jiroveci pneumonia and pneumococcal infection
  • Antifungal coverage if on multiple immunosuppressive agents
  • Hypogammaglobulinemia: IVIG
  • Monitor closely for viral reactivation.
  • Skin care: Lubricate dry skin with petroleum jelly. Protect skin from injury. Avoid sunburn.
  • Correct electrolyte imbalances for muscular aches and cramps.
  • Nutrition consults for malnutrition and wasting.
  • If chronic GVHD persists past 2-3 months or prednisone is needed at 1 mg/kg/day, alternative therapy should be used.
  • Hospitalization may be required for hydration, nutritional support, IV medications, monitoring, treatment of infections, and other supportive care.

Additional Therapies


  • Extracorporeal photophoresis: very effective for chronic skin GVHD; lower response rate if visceral organs involved
  • Psoralen plus ultraviolet A is of some benefit in skin GVHD (lichenoid, not sclerotic).
  • Mesenchymal stem cells (experimental)
  • Artificial tears for sicca syndrome
  • Physical therapy/range-of-motion exercises to prevent contractures
  • Inhaled corticosteroids and azithromycin (experimental) for bronchiolitis obliterans

Ongoing Care


Follow-up Recommendations


Patient Monitoring
  • Steroids: osteoporosis, diabetes
  • Calcineurin inhibitors: hypertension, renal dysfunction, hypomagnesemia
  • Sirolimus: hyperlipidemia, leukopenia, microangiopathic hemolytic anemia
  • Mycophenolate mofetil: GI discomfort, diarrhea, leukopenia

Prognosis


Prognosis of GVHD is based on severity.  
  • Acute GVHD: graded from I to IV based on organ involvement, percent of body surface area involved (skin), volume of diarrhea (gut), and/or elevation of serum bilirubin (liver). The higher the grade, the lower the long-term survival. Grade I survival is the same as for patients without GVHD; grade II, 60%; grade III, 25%; grade IV, 5-15%
  • Acute GVHD: 50-60% of patients respond to corticosteroids plus cyclosporine or tacrolimus.
  • Poor prognosis for survival: extensive skin involvement, progressive onset, GI involvement, thrombocytopenia, weight loss, and low Karnofsky performance status (40-60% survival)
  • 50% of patients still require therapy 5 years after diagnosis of chronic GVHD.

Complications


  • Mortality from GVHD after HSCT is usually related to infection.
  • Rarely, patients die of hepatic failure or abdominal catastrophe.
  • In transfusion-associated GVHD, death is usually from bone marrow aplasia with destruction of the host's marrow by donor lymphocytes.

Additional Reading


  • Carpenter  PA, MacMillan  ML. Management of acute graft versus host disease in children. Pediatr Clin North Am.  2010;57(1):273-295.  [View Abstract]
  • Jacobsohn  DA. Optimal management of chronic graft-versus-host disease in children. Br J Haematol.  2010;150(3):278-292.  [View Abstract]
  • Schlomchik  WD. Graft-versus-host disease. Nat Rev Immunol.  2007;7(5):340-357.  [View Abstract]
  • Wolff  D, Schleuning  M, von Harsdorf  S, et al. Consensus conference on clinical practice in chronic GVHD: second-line treatment of chronic graft-versus-host disease. Biol Blood Marrow Transplant.  2011;17(1):1-17.  [View Abstract]

Codes


ICD09


  • 279.50 Graft-versus-host disease, unspecified
  • 279.51 Acute graft-versus-host disease
  • 279.52 Chronic graft-versus-host disease
  • 279.53 Acute on chronic graft-versus-host disease
  • 996.85 Complications of transplanted bone marrow
  • 996.85 Complications of transplanted bone marrow

ICD10


  • D89.813 Graft-versus-host disease, unspecified
  • D89.810 Acute graft-versus-host disease
  • D89.811 Chronic graft-versus-host disease
  • D89.812 Acute on chronic graft-versus-host disease
  • T86.09 Other complications of bone marrow transplant

SNOMED


  • 234646005 Graft-versus-host disease (disorder)
  • 402355000 Acute graft-versus-host disease
  • 402356004 Chronic graft-versus-host disease

FAQ


  • Q: Do all patients with acute GVHD get chronic GVHD?
  • A: No. ~30% of patients <10 years of age who receive HLA-identical sibling HSCT will get acute GVHD, whereas only 13% will develop chronic GVHD. Chronic GVHD can develop in a patient who did not have acute GVHD; the prognosis is much more favorable than for the progressive form.
  • Q: Do all severe chronic GVHD patients die?
  • A: No. Occasionally, the GVHD will "burn out." This is rare, and the process by which it happens is not understood.
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