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MBBS, MD (Pathology),
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Bengaluru.
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Aug 2018




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Thanking you
With sincere regards
Dr. Rajendra Kumar Ghritlaharey, M.S., M. Ch., FAIS
Associate Professor,
Department of Paediatric Surgery, Gandhi Medical College & Associated
Kamla Nehru & Hamidia Hospitals Bhopal, Madhya Pradesh 462 001 (India)
E-mail: drrajendrak1@rediffmail.com
On May 11,2011




Dr. Shankar P.R.

"On looking back through my Gmail archives after being requested by the journal to write a short editorial about my experiences of publishing with the Journal of Clinical and Diagnostic Research (JCDR), I came across an e-mail from Dr. Hemant Jain, Editor, in March 2007, which introduced the new electronic journal. The main features of the journal which were outlined in the e-mail were extensive author support, cash rewards, the peer review process, and other salient features of the journal.
Over a span of over four years, we (I and my colleagues) have published around 25 articles in the journal. In this editorial, I plan to briefly discuss my experiences of publishing with JCDR and the strengths of the journal and to finally address the areas for improvement.
My experiences of publishing with JCDR: Overall, my experiences of publishing withJCDR have been positive. The best point about the journal is that it responds to queries from the author. This may seem to be simple and not too much to ask for, but unfortunately, many journals in the subcontinent and from many developing countries do not respond or they respond with a long delay to the queries from the authors 1. The reasons could be many, including lack of optimal secretarial and other support. Another problem with many journals is the slowness of the review process. Editorial processing and peer review can take anywhere between a year to two years with some journals. Also, some journals do not keep the contributors informed about the progress of the review process. Due to the long review process, the articles can lose their relevance and topicality. A major benefit with JCDR is the timeliness and promptness of its response. In Dr Jain's e-mail which was sent to me in 2007, before the introduction of the Pre-publishing system, he had stated that he had received my submission and that he would get back to me within seven days and he did!
Most of the manuscripts are published within 3 to 4 months of their submission if they are found to be suitable after the review process. JCDR is published bimonthly and the accepted articles were usually published in the next issue. Recently, due to the increased volume of the submissions, the review process has become slower and it ?? Section can take from 4 to 6 months for the articles to be reviewed. The journal has an extensive author support system and it has recently introduced a paid expedited review process. The journal also mentions the average time for processing the manuscript under different submission systems - regular submission and expedited review.
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KIST Medical College, P.O. Box 14142, Kathmandu, Nepal.
E-mail: ravi.dr.shankar@gmail.com
On April 2011
Anuradha

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Dr. Anuradha
E-mail: anuradha2nittur@gmail.com
On Jan 2020

Important Notice

Case report
Year : 2022 | Month : September | Volume : 16 | Issue : 9 | Page : WD01 - WD03 Full Version

Rowell Syndrome: A True Entity or a Diagnostic Challenge?


Published: September 1, 2022 | DOI: https://doi.org/10.7860/JCDR/2022/57837.16913
S Sivaramakrishnan, K Manoharan, G Sukanya, Richa Jotwani

1. Senior Resident, Department of Dermatology, Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu, India. 2. Professor and Head, Department of Dermatology, Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu, India. 3. Associate Professor, Department of Dermatology, Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu, India. 4. Junior Resident, Department of Dermatology, Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu, India.

Correspondence Address :
Dr. G Sukanya,
Sree Balaji Medical College and Hospital, CLC Works Road, Chromepet, Chennai, Tamil Nadu, India.
E-mail: drsukanyamathupal@gmail.com

Abstract

Lupus Erythematosus (LE) is an inflammatory condition of the connective tissue which manifests in variable forms. Rowell syndrome is a rare entity, with a female preponderance, where there is the presence of both LE and Erythema Multiforme (EM)-like lesions. Such patients are positive for antinuclear, anti-Ro/La and rheumatoid factor. Subcutaneous LE (SCLE) and EM can often share characteristics. The presence of lesions resembling both the conditions can pose a diagnostic difficulty, especially in the absence of complete clarity on the existence of Rowell syndrome. Present case is of a 47-year-old female, who presented with rashes over the face and trunk, along with photosensitivity. Cutaneous examination revealed multiple erythematous hyperpigmented plaques over the face with atypical targetoid lesions over the trunk and limbs. The diagnosis of Rowell’s syndrome was confirmed based on immunology and histopathology. This case has been reported to highlight the importance of delving into the various presentations of LE.

Keywords

Erythema multiforme-like, Hyperpigmented plaques, Limbs, Lupus erythematosus

Case Report

A 47-year-old female patient presented to the Dermatology Outpatient with a three month history of rashes over the face and trunk, associated with photosensitivity, which had aggravated in the past 10 days. There were similar lesions on the arms, painless oral ulceration over the hard palate, painful knee and hip joints. There was no such history of similar episodes in the past. She was not taking any medicines and no episodes of fever were reported.

General and systemic examinations were normal. Cutaneous examination showed multiple erythematous and hyperpigmented plaques with ill-defined borders on the forehead, lateral side of face, and upper lip region, resembling photosensitive LE lesions of subacute lupus erythematosus (Table/Fig 1), (Table/Fig 2). Raised atypical targetoid lesions (edematous and erythematous to hyperpigmented papules and plaques) were present on sun-exposed areas like the upper chest, back, upper limbs and scalp (Table/Fig 3). Few, small, erythematous, targetoid lesions (annular patches with rounded borders) were seen over the palms, resembling EM-like lesions (Table/Fig 4). A few dusky papules and macules were also present over the helix of the ear. The oral cavity also had hyperpigmented patches and an ulcerated, painless erosion was seen on the hard palate (Table/Fig 5).

Dermoscopic examination of the facial lesions showed white structureless areas [characteristic dermoscopic finding in Discoid Lupus Erythematosus (DLE)] with loss of hair follicles, multiple white and yellow dots, and multiple pigment globules, akin to DLE (Table/Fig 6). A provisional diagnosis of erythema multiforme, lupus erythematosus, and adverse drug reaction was considered and the patient was subjected to further investigations.

Complete haemogram showed signs of anaemia (hemoglobin – 8.2 gm/dL), reduced total counts (3600 cells/cu.mm) and elevated Erythrocyte Sedimentation Rate (ESR) (30 mm/hour). Antinuclear Antibody (ANA) profile, Anti-ds DNA, HBsAg, Anti-HCV, CRP, anti-Ro and anti-La antibodies were requested. Among these, ANA (1:100 dilution) and Anti-Ro antibody were found to be positive. Fine, speckled, nuclear pattern of ANA on Indirect Immunofluorescence (IIF) was seen. Histopathological study of a 3.5 mm punch biopsy, taken from a lesion on the lower back, showed necrotic keratinocytes with basal cell degeneration, pigment incontinence, dilated dermal capillaries with surrounding lympho-histiocytic mononuclear infiltration (Table/Fig 7). Based on the aforementioned clinical history, and histological and laboratory findings, a differential diagnosis of Subacute Cutaneous Lupus Erythematosus (SCLE) was proposed, but with the existence of EM-like lesions. Thus, given the simultaneous presence of both LE and EM-like lesions, along with a supporting ANA profile, a diagnosis of Rowell syndrome was ascertained.

The patient was started on sunscreen, topical mid-potent corticosteroid (mometasone furoate 0.1%) cream, once-daily application, intravenous dexamethasone 4 mg twice a day for a week, and oral hydroxychloroquine 200 mg twice a day. The patient showed gradual resolution of skin lesions and the steroid was then gradually tapered to 30 mg of oral prednisolone daily for one week, followed by 20 mg of oral prednisolone daily for one week and a maintenance dose of oral prednisolone 15 mg once daily was achieved. The patient was asked to continue hydroxychloroquine 200 mg twice daily along with topical agents. The patient is currently on follow-up on a fortnightly basis and is responding to treatment (Table/Fig 8).

Discussion

Rowell Syndrome is an uncommon condition characterised by the presence both LE and EM-like lesions in patients with a typical immunologic pattern. EM is characterised by the presence of target lesions which derive their name from the appearance of rings of different colours that is most commonly seen on the hands and extensor aspect of limbs. Classic EM occurs due to certain infectious causes like Mycoplasma pneumoniae, drugs like non steroidal anti-inflammatory drugs, antibiotics, anticonvulsants, or malignancies (1). LE is an autoimmune condition with variable presentation. Lesions of SCLE are widespread, asymmetrical in distribution without any scarring. It frequently involves the extensor surface of the arms, upper chest, upper back, shoulders and neck, where systemic involvement is usually absent but anti-Ro antibodies are frequently present (2). Psoriasiform and annular polycyclic are the predominant forms known, but other presentations have also been described. The histological similarity in both these conditions is the presence of lymphocytic exocytosis into the epidermis, inflammation at the dermoepidermal junction, vacuolar degeneration of basal epidermal cell layer along with oedema of the upper dermis (3).

In concordance with the case reports by Yachoui R and Cronin PM, (2) and Gallo L et al., (4), the index patient also had characteristic features – middle-aged woman with clinical features suggestive of both SCLE and EM. Till date, very few cases of Rowell syndrome have been reported. Although the disease is common in young adults, and the median age reported is 32, with instances of paediatric and elderly cases (4). It was first identified by Rowell in 1963 in four female patients with DLE, speckled immunofluorescence pattern, who were positive for Rheumatoid factor and anti-Ro/La antibodies.

The definition of RS remains a matter of debate, as a few scholars have opined that LE associated with EM may be an overlapping syndrome, a subtype of LE or merely a co-incidence (5). There was no identifiable precipitating factor for EM in the index patient.

The criteria by Zeitouni NC et al., to diagnose Rowell syndrome comprise major as well as minor criteria (6). Major criteria include SLE, DLE, or SCLE; EM-like lesions; and a speckled pattern of ANA positivity. Minor criteria consist of anti-Ro/anti-La antibody, a positive RF and chilblains (7). To diagnose RS, all three major criteria and atleast one minor criterion must be present (2). In this case, all three major criteria along and one minor criterion (positive anti-Ro antibody) were fulfilled, which clinched the diagnosis (Table/Fig 9).

Speckled ANA is considered to be a consistent finding of Rowell syndrome, which was positive in the index patient. Both LE and RS follow a similar line of management. Patients with Rowell syndrome generally have a favourable prognosis and show complete resolution of lesions within one year of starting therapy (7). Antimalarials, steroids, dapsone, azathioprine, and cyclosporine have shown to be beneficial (8). The index patient responded well to intravenous dexamethasone 4 mg twice a day and oral hydroxychloroquine 200 mg twice a day.

Conclusion

SCLE and Rowell syndrome have overlapping clinical, histological, and immunological findings. Index case also highlights the features shared by SCLE and EM, often making it difficult to diagnose. There have been very few reports of Rowell syndrome in literature and not much is known about this rare entity. Hence, this case has been reported to highlight the various features of Rowell syndrome and to bring into question of whether this condition is a possible designation of subacute lupus erythematosus or a true entity on its own.

References

1.
Branişteanu DE, Ianoşi SL, Dimitriu A, Stoleriu G, Oanta A, Branişteanu DC. Drug-induced Rowell syndrome, a rare and difficult to manage disease: A case report. Exp Ther Med. 2018;15(1):785-88.
2.
Yachoui R, Cronin PM. Systemic lupus erythematosus associated with erythema multiforme-like lesions. Case Rep Rheumatol. 2013;212145. [crossref] [PubMed]
3.
Aydogan K, Karadogan SK, Balaban Adim S, Tunali S. Lupus erythematosus associated with erythema multiforme: Report of two cases and review of the literature. J Eur Acad Dermatol Venereol. 2005;19(5):621-27. [crossref] [PubMed]
4.
Gallo L, Megna M, Festa B, Stellato P, di Pinto R, Fabbrocini G, et al. Rowell Syndrome: A diagnostic challenge. J Clin Aesthet Dermatol. 2020:(4):40-42.
5.
Lyon CC, Blewitt R, Harrison PV. Subacute cutaneous lupus erythematosus. Acta Derm Venereol (Stockh). 1998;78:57-59. [crossref] [PubMed]
6.
Zeitouni NC, Funaro D, Cloutier RA, Gagné E, Claveau J. Redefining Rowell’s syndrome. Br J Dermatol. 2000;142(2):343-46. [crossref] [PubMed]
7.
Andronache IT, Suta C, Ionescu C, Calistrat A, Suta M. Rowell syndrome-a controversial clinical entity. Rom J Rhematol. 2015:24:230-34. [crossref]
8.
Kadiru RA, Hegde SP, Mithun HK, Rao AC. Neuropsychiatric manifestations in an adolescent male with Rowell syndrome. J Postgrad Med. 2021;67(2):109. [crossref] [PubMed]

DOI and Others

DOI: 10.7860/JCDR/2022/57837.16913

Date of Submission: May 17, 2022
Date of Peer Review: Jun 17, 2022
Date of Acceptance: Jul 21, 2022
Date of Publishing: Sep 01, 2022

AUTHOR DECLARATION:
• Financial or Other Competing Interests: None
• Was informed consent obtained from the subjects involved in the study? Yes
• For any images presented appropriate consent has been obtained from the subjects. Yes

PLAGIARISM CHECKING METHODS:
• Plagiarism X-checker: May 28, 2022
• Manual Googling: Jul 14, 2022
• iThenticate Software: Aug 30, 2022 (8%)

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