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Fig 2. Miliarial gout. Hematoxylin-eosinestained sectionshowing granulomatous dermatitis with amorphouscrystalline material within the dermis.
J AM ACAD DERMATOLJULY 2014
e18 Letters
forearms, and thighs over the previous year (Fig 1).He reported that these lesions worsened withwarm weather and then gradually dissipated. Aflesh-colored nodule was present on the right elbow.He denied having pruritus or joint pain. Medicalhistory was significant for diabetes treatedwith metformin. Family history was negative forcutaneous disease.
Punch biopsy of a typical papule was performedand the specimen placed in formalin. Histologicexamination revealed granulomatous dermatitis withamorphous crystalline material consistent with gout(Fig 2). Serum uric acid level was within normallimits at 4.9 mg/dL (normal reference range, 3.5 to7.2 mg/dL).
Only a few cases of miliarial gout have beenpreviously reported. As with gouty arthritis, a normalserum uric acid level does not exclude thediagnosis.3 Similar to typical tophaceous gout,miliarial gout can be the initial manifestation ofgout. In fact, both of the referenced cases of miliarialgout occurred in the absence of a known history ofgouty arthritis. It is unclear whether these patientsare predisposed to the development of gouty arthritisin the future. Because there are so many clinicalvariants of tophaceous gout, patients may presentwith more than 1 variant. It is likely that our patient’slongstanding nodule on the elbow represents a moretypical manifestation of tophaceous gout. Althoughour patient’s histopathology was typical of topha-ceous gout, to actually fulfill clinical diagnosticcriteria, as established by the American College ofRheumatology,4 the biopsy specimen should havebeen submitted in absolute alcohol to demonstratemonosodium urate crystals by polarized lightmicroscopy.
Open access under CC BY-NC-ND license.
Allopurinol alone1 or in combination withcolchicine2 have been reported to improve miliarialgout, and our patient was referred to a rheumato-logist for management but was lost to follow-up.The prevalence of gout is reported to be between0.5% and 1%, and the incidence of gout is increasing,possibly due to an aging population.5 This caseillustrates the importance of considering atypicalmanifestations of common rheumatologic diseases.
Kenyatta A. Mireku, BS, Jessica R. Burgy, MD, andLoretta S. Davis, MD
Georgia Regents University, Augusta, Georgia
Funding sources: None.
Conflicts of interest: None declared.
Correspondence to: Jessica R. Burgy, MD, 1004Chafee Avenue, FH-100, Department of Derma-tology, Augusta, GA 30912-3190
E-mail: [email protected]
REFERENCES
1. Shukla R, Vender RB, Alhabeeb A, Salama S, Murphy F. Miliarial
gout (a new entity). J Cutan Med Surg 2007;11:31-4.
2. Aguayo RS, Baradad M, Soria X, Abal L, Sanmart�ın V, Egido R,
et al. Unilateral milia-type intradermal tophi associated with
underlying urate subcutaneous deposition: an uncommon
cutaneous presentation of gout. Clin Exp Dermatol 2013;38:
622-5.
3. McCarty DJ. Gout without hyperuricemia. JAMA 1994;271:
302-3.
4. Wallace SL, Robinson H, Masi AT, Decker JL, McCarty DJ, Yu TF.
Preliminary criteria for the classification of the acute arthritis of
primary gout. Arthritis Rheum 1977;20:895-900.
5. Adams PF, Hendershot GE, Marano MA. Centers for Disease
Control and Prevention/National Center for Health Statistics.
Current estimates from the National Health Interview Survey,
1996. Vital Health Stat 1999;10:1-203.
http://dx.doi.org/10.1016/j.jaad.2014.01.851
A case of dermatitis herpetiformis after amini-gastric bypass surgery
To the Editor: Celiac disease and dermatitisherpetiformis share the same strong HLA association,the presence of circulating IgA antitissue andantiepidermal transglutaminase antibodies, and thesame typical histologic features of villous atrophy ofthe small intestine.1 Both diseases can be triggeredby gluten overload and gastric surgery.2
We report a case of dermatitis herpetiformis aftermini-gastric bypass surgery with no clinical sign orsymptom of celiac disease. Mini-gastric bypass is amodification of the standard Roux-en-Y procedureusing a long gastric tube with an antecolic loopgastrojejunostomy. It has become a widely
mailto:[email protected]://refhub.elsevier.com/S0190-9622(13)01334-0/sref1http://refhub.elsevier.com/S0190-9622(13)01334-0/sref1http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref3http://refhub.elsevier.com/S0190-9622(13)01334-0/sref3http://refhub.elsevier.com/S0190-9622(13)01334-0/sref4http://refhub.elsevier.com/S0190-9622(13)01334-0/sref4http://refhub.elsevier.com/S0190-9622(13)01334-0/sref4http://refhub.elsevier.com/S0190-9622(13)01334-0/sref5http://refhub.elsevier.com/S0190-9622(13)01334-0/sref5http://refhub.elsevier.com/S0190-9622(13)01334-0/sref5http://refhub.elsevier.com/S0190-9622(13)01334-0/sref5http://dx.doi.org/10.1016/j.jaad.2013.12.032http://creativecommons.org/licenses/by-nc-nd/4.0/
Fig 1. Dermatitis herpetiformis. Grouped papulovesicleswith excoriations on the extensor surface of the shoulder.
Fig 2. Dermatitis herpetiformis. Direct immunofluores-cence studies on skin biopsy specimen showing granularIgA deposits in the dermal papillae.
J AM ACAD DERMATOLVOLUME 71, NUMBER 1
Letters e19
performed bariatric surgery because it offers similarefficacy with a simpler technique and a lower rate ofcomplicatons.3
A 40-year-old white woman presented for a 1-yearhistory of intermittent itching with symmetricallygrouped papulovesicles on the extensor surfaces ofthe elbows, the shoulders, and the buttocks, withexcoriations and hyperpigmentation (Fig 1). She wastreated with 0.05% fluocinonide cream with noimprovement.
Two years before this rash, she had a mini-gastricbypass for morbid obesity. She had no significantmedical history and no symptom or sign of celiacdisease before or after the surgery.
A hematoxylin-eosin stain showed subepidermalbullae and clefts in dermal papillae filled withneutrophils and a perivascular mononuclear infil-trate with neutrophils. Direct immunofluorescencestudies showed granular IgA deposits in the dermalpapillae (Fig 2).
Endomysial and transglutaminase antibodies,both IgG and IgM, were positive. All other bloodtests were within normal limits. The patient was
started on a gluten-free diet and dapsone (100 mgdaily) with clinical improvement of her symptomswithin a few days.
Bai et al2 reported 10 patients who developedsymptoms of celiac disease 2 months after differenttypes of peptic ulcer surgery. Kovaleski et al4
described a patient who developed dermatitisherpetiformis after a gastric stapling procedure anda second who developed dermatitis herpetiformisafter a gastrectomy with partial pancreatectomy andcolectomy.
It is known that dermatitis herpetiformis is linkedto gluten intolerance and has a clear relationship toceliac disease but the development of dermatitisherpetiformis without celiac disease is not unex-pected because the pathogenesis behind the skinand the intestinal lesions are unlikely to be fullyidentical. About 5% of celiac disease cases developdermatitis herpetiformis5 and the histologic intestinalchanges persist even when cutaneous symptoms arecontrolled with dapsone but normalize with strictavoidance of gluten. Patients with isolated celiacdisease do not have cutaneous IgA deposition,6
showing that this phenomenon is related todermatitis herpetiformis itself.
As suggested by Kovaleski et al,4 the triggeringfactor of dermatitis herpetiformis is probably thesurgery-induced enteral inflammation leading to across-reaction between cutaneous and intestinalantitransglutaminase antibodies. These antibodiesof mucosal origin can persist in circulation anddeposit in the skin.
In conclusion, dermatitis herpetiformis is a modelof autoimmune disease linking gastrointestinalmucosal inflammation to cutaneous condition.The exact mechanism by which gastrointestinalinflammation translates into cutaneous disease isunknown. Dermatitis herpetiformis should besuspected in any pruritic rash after mini-gastricbypass and other surgeries of the upper gastrointes-tinal tract.
Farid Stephan, MD, Roger Haber, MD, DianeMaalouf, MD, and Roland Tomb, MD, PhD
Department of Dermatology, Faculty of Medicine,Saint Joseph University, Beirut, Lebanon
Funding sources: None.
Conflicts of interest: None declared.
Correspondence to: Diane Maalouf, MD, Depart-ment of Dermatology, Faculty of Medicine,Saint Joseph University, Damas Street, PO Box17-5208, Achrafieh, Beirut, Lebanon
E-mail: [email protected]
mailto:[email protected]
J AM ACAD DERMATOLJULY 2014
e20 Letters
REFERENCES
1. Bolotin D, Petronic-Rosic V. Dermatitis herpetiformis, part I:
epidemiology, pathogenesis, and clinical presentation.
J Am Acad Dermatol 2011;64:1017-24.
2. Bai J,MoranC,MartinezC,Niveloni S, Crosetti E, Sambuelli A, et al.
Celiac sprue after surgery of the upper gastrointestinal tract:
report of 10 patients with special attention to diagnosis, clinical
behavior, and follow-up. J Clin Gastroenterol 1991;13:521-4.
3. Lee WJ, Yu PJ, Wang W, Chen TC, Wei PL, Huang MT.
Laparoscopic Roux-en-Y versus mini-gastric bypass for the
treatment of morbid obesity: a prospective randomized
controlled clinical trial. Ann Surg 2005;242:20-8.
4. Kovaleski C, Jaliman D, Kumar V, Valeski JE. Dermatitis herpeti-
formis following gastric surgery. Arch Dermatol 1997;133:113-4.
5. Rose C, Brocker EB, Zillikens D. Clinical, histological and
immunopathological findings in 32 patients with dermatitis
herpetiformis Duhring. J Dtsch Dermatol Ges 2010;8:265-71.
6. Karlsson IJ, Dahl MG, Marks JM. Absence of cutaneous IgA in
coeliac disease without dermatitis herpetiformis. Br J Dermatol
1978;99:621-5.
http://dx.doi.org/10.1016/j.jaad.2014.01.854
Subcutaneous Sweet syndrome associated withdisseminated nontuberculous mycobacterialinfection leading to the diagnosis ofadult-onset immunodeficiency
To the Editor: A 51-year-old woman without notablemedical history presented with multiple 1.5- to 2-cmtender, erythematous, and swollen nodules over thelegs, ankles, and soles (Fig 1) for 2 weeks. Fever up
Fig 1. Subcutaneous Sweet syndrome. A and Bnodules over bilateral legs and soles. C andD, Histpanniculitis. (C and D, Hematoxylin-eosin stain; o
to 398C with chills was also noted. The remainder ofthe physical examination revealed unremarkablefindings. Laboratory tests showed leukocytosis withneutrophilia (white blood cell count 18,280/uL withsegments:75.1%), increased C-reactive protein (14mg/dL), and elevated erythrocyte sedimentation rate(60 mm/h). Histopathological examination of theskin lesion showed neutrophilic lobular panniculitiswithout vasculitis or any micro-organisms seen inspecial stains (Fig 1). Blood chemistry, urinalysis,antistreptolysin O antibody, hepatitis B virussurface antigen, antihepatitis C virus antibody, anti-nuclear antibody, cryoglobulins, antineutrophiliccytoplasmic antibodies, and blood along withtissue cultures produced negative or normal results.Chest x-ray and chest computed tomographyshowed mediastinal and right hilar lymphadenopa-thy. Sputum cytology and culture were negative formalignant cells or infection. The patient declinedcomputed tomographyeguided lymph node biopsyfor investigation of inflammatory, infectious, orneoplastic diseases. The skin lesions graduallyimproved after cefazolin and diclofenac use.Because of uncertain origin of the lymphadenopathyand possible occult infection, corticosteroid was notfurther administered.
However, multiple tender erythematous nodulesover bilateral forearms occurred 1 month later and
, Multiple deep painful and erythematousopathological finding as neutrophilic lobularriginal magnifications: C, 340; D, 3400.)
http://refhub.elsevier.com/S0190-9622(13)01334-0/sref1http://refhub.elsevier.com/S0190-9622(13)01334-0/sref1http://refhub.elsevier.com/S0190-9622(13)01334-0/sref1http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref2http://refhub.elsevier.com/S0190-9622(13)01334-0/sref3http://refhub.elsevier.com/S0190-9622(13)01334-0/sref3http://refhub.elsevier.com/S0190-9622(13)01334-0/sref3http://refhub.elsevier.com/S0190-9622(13)01334-0/sref3http://refhub.elsevier.com/S0190-9622(13)01334-0/sref4http://refhub.elsevier.com/S0190-9622(13)01334-0/sref4http://refhub.elsevier.com/S0190-9622(13)01334-0/sref5http://refhub.elsevier.com/S0190-9622(13)01334-0/sref5http://refhub.elsevier.com/S0190-9622(13)01334-0/sref5http://refhub.elsevier.com/S0190-9622(14)00939-6/sref6http://refhub.elsevier.com/S0190-9622(14)00939-6/sref6http://refhub.elsevier.com/S0190-9622(14)00939-6/sref6http://dx.doi.org/10.1016/j.jaad.2014.01.854
A case of dermatitis herpetiformis after a mini-gastric bypass surgeryReferences