Post on 22-Feb-2021
• 45 yo female with a hx of migraines & excessive
sun exposure
• Presents with complaints of a very pruritic rash on
her abdomen, buttocks, & lower extremities.
• 2 days prior, took acetaminophen-butalbital-
caffeine & consumed 2 new cooking ingredients:
Malanda (Xanthosoma sagittifolium) & boniato
(Ipomoea batatas)
• Pt denies any prior occurrences or any other
associated symptoms.
• On exam, patient presented with multiple
erythematous, hyperpigmented linear streaks
scattered on bilateral legs, buttocks, & inferior
abdomen consistent with flagellate erythema.
Excoriations were diffusely present.
• Histology: a dense, perivascular lymphocytic
infiltrate with very few eosinophils & marked
dermal edema. Melanin diffusely scattered within
epidermal basal layer but not within the dermis.
No iron dermal deposition.
• Treatment: Stop all recent medications & cooking
ingredients, 40mg of IM triamcinolone acetonide,
triamcinolone acetonide 0.1% topical cream BID x
2 wks, & fexofenadine 180mg PO QD
• 2 wks after visit, the patient cooked & consumed
food containing malanga & boniato again. She
experienced diffuse pruritus, but denied any rash.
Pruritus was relieved with diphenhydramine.
• At 3 wk follow-up, pt showed improvement of rash
& pruritus, & was instructed to continue her
fexofenadine.
INTRODUCTION CLINICAL & HISTOPATHOLOGICAL IMAGES TREATMENT
REFERENCES
1. Moulin, G., B. Fiere, and A. Beyvin, [Cutaneous pigmentation caused by bleomycin].
Bull Soc Fr Dermatol Syphiligr, 1970. 77(2): p. 293-6.
2. Nousari, H.C., et al., "Centripetal flagellate erythema": a cutaneous manifestation
associated with dermatomyositis. J Rheumatol, 1999. 26(3): p. 692-5.
3. Bolognia, J., Jorizzo, J. L., & Schaffer, J. V., Dermatology. 2012, Philadelphia:
Elsevier Saunders.
4. James W, B.T., Elston D, Andrews Diseases of the Skin Clinical Dermatology. 11th
ed. 2011: Saunders Elsevier.
5. Callen, J.P. and R.L. Wortmann, Dermatomyositis. Clin Dermatol, 2006. 24(5): p.
363-73.
6. Suzuki, K., et al., Persistent plaques and linear pigmentation in adult-onset Still's
disease. Dermatology, 2001. 202(4): p. 333-5.
7. Hanada, K. and I. Hashimoto, Flagellate mushroom (Shiitake) dermatitis and
photosensitivity. Dermatology, 1998. 197(3): p. 255-7.
8. Yamamoto, T. and K. Nishioka, Flagellate erythema. Int J Dermatol, 2006. 45(5): p.
627-31.
9. Fernandez-Obregon, A.C., K.P. Hogan, and M.K. Bibro, Flagellate pigmentation from
intrapleural bleomycin. A light microscopy and electron microscopy study. J Am Acad
Dermatol, 1985. 13(3): p. 464-8.
10. Wright, A.L., S.S. Bleehen, and A.E. Champion, Reticulate pigmentation due to
bleomycin: light- and electron-microscopic studies. Dermatologica, 1990. 180(4): p.
255-7.
11. Eungdamrong, J. and B. McLellan, Flagellate erythema. Dermatol Online J, 2013.
19(12): p. 20716.
12. Scheiba, N., M. Andrulis, and P. Helmbold, Treatment of shiitake dermatitis by
balneo PUVA therapy. J Am Acad Dermatol, 2011. 65(2): p. 453-5.
Ryan Schuering DO1, Gregory Bartos OMS III 2, Francisco Kerdel MD 3,4, Stanley Skopit DO, MSE, FAOCD, FAAD5
1 PGY-3 Dermatology Residency Training Program, LCH/LECOM, South Miami, FL, 2 OMS III Nova Southeastern University (NSU) College of Medicine, 3 Department of Dermatology, Florida International University, Miami, FL, 4 Florida Academic Dermatology Center, LCH,
South Miami, Fl, 5Program Director, Dermatology Residency Training Program, LCH/LECOM
A Rare Etiology of Flagellate Erythema: A Case Report & Review
• Discontinuation of offending agent
• Treatment for flagellate erythema is mostly
symptomatic: pruritus may be targeted with topical
corticosteroids & oral antihistamines while
hyperpigmentation usually resolves spontaneously
within 1-8 weeks.3
• Areas of lasting hyperpigmentation have been
treated with intense pulse light therapy & Erbium
1540nm non-ablative laser.11
• Erythematous papules from shiitake consumption
have been targeted with short-term balneo-PUVA
therapy showing complete clearance of itch &
healing of lesions.12
• Evaluation for systemic etiology such as
dermatomyositisCASE PRESENTATION• Flagellate erythema is a rare cutaneous phenomenon described as linear erythematous streaks with pruritus &
hyperpigmentation. Known etiologies are bleomycin, dermatomyositis, adult-onset stills disease, & shiitake dermatitis. Our
patient did not fall into any common etiological category & historically was newly exposed to Butalbital-acetominophen-
caffeine, malanga, & boniato prior to onset. A thorough literature search on these three compounds showed no evidence of
flagellate erythema as an adverse reaction.
• Bleomycin, an antitumor medication, is used as treatment with certain malignancies. Flagellate erythema has been reported
as an adverse effect of bleomycin with an incidence rate of 10-20%.3 The precise mechanism remains unknown although
some speculate that bleomycin induces generalized pruritus leading to scratching. The scratching allows for the drug to exit
blood vessels & reacts toxically with the skin.
• Dermatomyositis is an inflammatory myositis with cutaneous manifestations. Well characterized cutaneous manifestations
are heliotrope rash, Gottron’s papules, periungal telangiectasia, & shawl sign. Flagellate erythema has been reported in
association with disease activity & may precede muscle symptoms.4 Dermatomyositis has a 15-25% increased risk for
malignancy.5
• Adult-onset Still’s disease is an inflammatory disease comprised of high spiking fevers, arthralgia, hyperferritinemia,
hepatosplenomegaly & rash. The characteristic rash is a salmon maculopapular erythema that appears during high fevers.
Persistent erythematous plaques suggesting flagellate erythema have been reported in few cases.6
• Shiitake dermatitis, AKA toxicoderma, is caused by the consumption of undercooked shiitake mushrooms. Incidence is
highest in China & Japan where the mushroom is commonly grown & consumed. Flagellate erythema originates from
significant pruritus & the Koebner phenomenon leading to linear grouping of non-pigmented papules. The rash improves on
its own within two weeks.7
DISCUSSION
CONCLUSION
Clinical Finding Histology Pearls
Bleomycin Linear streaks located on trunk and/or
shoulders. It is unique that these linear
streaks are hyperpigmented, & devoid of
inflammation.8
- Epidermis shows increased melanin pigment,
hyperkeratosis with focal parakeratosis,
irregular acanthosis, spongiosis, & exocytosis
of lymphocytes.
- Dermis shows edema, vasodilation &
perivascular lymphocytic infiltration.9,10
- Patient will have started
chemotherapy regimen within
last 6 months.
- Flagellate erythema is not a
sufficient cause to stop cancer
therapy
Dermatomyositis Reddish, Linear streaks reflecting strong
inflammation commonly on back, lack
brown hyperpigmentation seen with
Bleomycin.2
- Epidermis shows mild atrophy with
vacuolization of the basal layer.
- Dermis shows lymphocytic infiltration in upper
dermis & moderate edema in papillary dermis8
- Elevated creatine kinase &
ESR/CRP
- Look for heliotrope rash,
Gottron’s papules, muscle
weakness
- Screen for malignancies
Adult-Onset
Still’s Disease
Persistent plaques with linear
pigmentation with or without coalescent
erythematous plaques
- Mild perivascular infiltration of mononuclear
cells & neutrophil, dyskeratotic cells in the
epidermis
- Monitor blood count
- Monitor cardia function
- Serial LFT’s & lipids
Shiitake
Mushrooms
Widespread, disseminated, very small
erythematous papules, no pigmentation,
truncal involvement.7
- Epidermis shows elongation of rete ridges,
spongiosis & spongiotic bullae, with infiltration
of inflammatory cells. The dermis shows
edema, & superficial & intermediate
perivascular infiltrates of mononuclear cells
- Recent preparation of
mushrooms or visit to Japanese
restaurant
- Avoid sun exposure due to
photosensitive lesions
• Flagellate erythema is a dermatosis comprised of
hyperpigmented, pruritic, linear, & erythematous
streaks.
• It has been described in association with bleomycin
use1, dermatomyositis2, adult-onset stills disease3,
& shiitake mushroom consumption4.
• The patient presented here did not encounter or
meet the criteria for any of the known etiologies.
• The recognition of this rare diagnostic clue is
paramount in discovering its underlying condition
as it may have significant health implications for the
patient.
• Flagellate erythema has been reported in
association with several systemic diseases &
chemical agents.
• A thorough history & evaluation is important in
determining the underlying cause.
• Our patient did not appear to have the history or
clinical features to indicate any of the known
causes for flagellate erythema.
• Thus, this case possibly demonstrates a novel
cause of flagellate erythemadue to consumption of
malanga (Xanthosoma sagittifolium) and boniato
(Ipomoea batatas).
A B C D EFigure A-D Erythematous, hyperpigmented linear streaks on the anterior, lateral, & posterior aspects of the lower extremities & anterior aspect
of the lower abdomen. Figure D Left lateral lower extremity, punch biopsy site. Figure E A dense, perivascular lymphocytic infiltrate with very
few eosinophils & marked dermal edema.