Systemic Lupus Erythematosus in Twin Sisters Following Ten Years of Hyperglobulinemic Purpura (Waldenström)

Zaharia, L. Irina; Hill, Jennifer; Loeb, Ellen; MacLellan, Adam; Khan, Aftab Ahmad; Hill, Nicola

doi:10.1111/j.0954-6820.1976.tb06760.x

Cited by 17 publications

(3 citation statements)

References 15 publications

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“…1 Diagnosis of HGPW may prompt screening for hematological and rheumatological disease owing to underlying associations, especially Sjogren's syndrome, lupus erythematosus and rheumatoid arthritis. 1,6,7,12,13 Less common associations include granulomatous disorders like sarcoidosis and tuberculosis (Table 2). Notably, our patient had an underlying diagnosis of myeloma but no other known connective tissue diseases.…”

Section: Discussionmentioning

confidence: 99%

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Hypergammaglobulinemic purpura of Waldenström—Unusual and impressive case in a patient with myeloma: A case report

DeBiasio

Cyr

Petkiewicz

et al. 2022

SAGE Open Medical Case Reports

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Background: Hypergammaglobulinemic purpura of Waldenström is an uncommon disease, which presents mostly in women on the lower extremities. It is sometimes associated with underlying immune dysregulation. Sjögren syndrome is the most common association; however, rare occurrences of the self-resolving syndrome with lymphoma or myeloma have been reported. Case Summary: We describe an unusual and impressive presentation of hypergammaglobulinemic purpura of Waldenström in an elderly female patient with myeloma. Notably, the patient did not have any concurrent connective tissue diseases. Despite her florid presentation, her hypergammaglobulinemic purpura of Waldenström spontaneously resolved within a few days. Conclusion: Hypergammaglobulinemic purpura of Waldenström is a self-resolving but recurrent syndrome, which may be associated with autoimmune disorders or rarely myeloma. Early diagnosis of the syndrome may avoid unnecessary treatment interventions and should prompt screening for underlying diseases.

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Section: Discussionmentioning

confidence: 99%

“…[3][4][5]8 Therefore, screening for underlying disorders at the initial presentation of HGPW is important. Henoch-Schonlein purpura 14 Lymphoma 8 Myeloma 3-5 Rheumatoid arthritis 6 Sarcoidosis [14][15][16][17] Sjögren's syndrome 6,7 Systemic lupus erythematosus 12,13 Tuberculosis 14,18…”

Section: Discussionmentioning

confidence: 99%

Hypergammaglobulinemic purpura of Waldenström—Unusual and impressive case in a patient with myeloma: A case report

DeBiasio

Cyr

Petkiewicz

et al. 2022

SAGE Open Medical Case Reports

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“…[11] and Zaharia et al . [12] emphasize that associated diseases can develop 10 or more years after the onset of HGP and cannot ever be completely excluded. Sanchez-Guerrero et al .…”

Section: Discussionmentioning

confidence: 99%

Clinical profile of cutaneous manifestations with and without hematologic disease: A comparative study

Pande

Kharkar

2014

Indian Dermatol Online J

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Aim:The aim was to study the clinical profile of cutaneous manifestations of hematologic disorders and to compare it with that of non-hematologic disorders.Materials and Methods:Cutaneous manifestations of hematologic diseases fall in seven well-defined categories. A total of 153 outpatients with skin manifestations fitting in these categories were enrolled in a comparative study of 1-year duration. Clinical profile of these cutaneous manifestations was studied and any underlying hematologic disorder was ruled out with the help of a hematologist. Difference in the clinical profile of cutaneous manifestations with and without hematologic diseases was studied.Result:Of the 26,174 outpatients during the study period, 153 had cutaneous manifestations fitting in the categories of hematologic disorders. Of these 153 patients, 33 had hematologic disease as the cause of their cutaneous manifestation (21.57%), whereas 78.42% had no hematologic disorder. Disorders of hemostasis formed the largest group (36%) followed by cutaneous deposits/infiltrations (15%), vesiculobullous disorders (6%), and cutaneous vasculitis (9%) were least commonly associated with hematologic disorders.Conclusion:Hematologic diseases are associated with complex array of cutaneous manifestations. The incidence of hematologic disease–associated cutaneous manifestations was 0.13%. Findings of this study will help dermatologists and physicians with the early recognition of cutaneous signs of hematologic disorders.

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A revised estimate of twin concordance in systemic lupus erythematosus

Deapen

Escalante

Weinrib

et al. 1992

Arthritis & Rheumatism

683

372

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Objective. Based on a small clinical series and previously published case reports, concordance for systemic lupus erythematosus (SLE) among monozygous (MZ) twins has been reported to be as high as 69%. Using a larger and less biased sample, we provide another estimate of this percentage.Methods. We established a registry of twins with SLE, based upon self-reports and information provided by the patients' physicians. We used DNA fingerprinting to validate the reported zygosity in a sample of these twins.Results. firmed by DNA fingerprinting in a subsample of 15 self-described MZ twins and 7 self-described DZ twins. All individuals had correctly predicted their zygosity.Conclusion. MZ concordance for SLE is similar to that for other autoimmune diseases and is much lower than previously believed.Concordance for disease in pairs of twins provides evidence of the relative etiologic contributions of genetic and environmental factors, as well as the timing of these effects. Low rates of concordance for disease over a lifetime, especially among monozygotic (MZ) twins, suggests a greater influence of environment; a large difference in concordance between MZ and dizygotic (DZ) twins suggests a greater influence of genetic determinants. However, for most diseases, there are no population-based twin concordancy data.Although the estimated annual incidence rate for systemic lupus erythematosus (SLE) is 5CL75 per million (1,2), no population-based studies of concordance rates in twins have been published. One case series reported that 69% of the MZ twin pairs were concordant (3), and anecdotal reports describe a similar proportion of concordance among MZ twin pairs. These reports also suggest that a much lower proportion of DZ twin pairs are concordant. In the study presented here, we used a larger sample size and an ascertainment mechanism that is less overtly biased in an effort to derive more meaningful data concerning the etiology of SLE.

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Systemic Lupus Erythematosus in Twin Sisters Following Ten Years of Hyperglobulinemic Purpura (Waldenström)

Cited by 17 publications

References 15 publications

Hypergammaglobulinemic purpura of Waldenström—Unusual and impressive case in a patient with myeloma: A case report

Hypergammaglobulinemic purpura of Waldenström—Unusual and impressive case in a patient with myeloma: A case report

Clinical profile of cutaneous manifestations with and without hematologic disease: A comparative study

A revised estimate of twin concordance in systemic lupus erythematosus

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