- Email: [email protected]
Cat-scratch disease with bulky lymphadenopathy. A case report
Accepted Manuscript Title: CAT SCRATCH DISEASE WITH BULKY LYMPHADENOPATHY.ACASE REPORT Author: Bidish Patel Pampa Ch Toi Debdutta Basu PII: DOI: Reference:
S2212-8328(15)00034-X http://dx.doi.org/doi:10.1016/j.pid.2015.06.001 PID 134
To appear in: Received date: Revised date: Accepted date:
5-11-2013 8-10-2014 18-6-2015
Please cite this article as: Bidish PatelPampa Ch ToiDebdutta Basu CAT SCRATCH DISEASE WITH BULKY LYMPHADENOPATHY.ACASE REPORT (2015), http://dx.doi.org/10.1016/j.pid.2015.06.001 This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
TITLE:CAT SCRATCH DISEASE WITH BULKY LYMPHADENOPATHY.ACASE REPORT TITLE PAGE:
Title of the article:
cr
Cat Scratch Disease With Bulky Lymphadenopathy .A Case Report.
ip t
Type of article: Case report
us
Authors: 1.Bidish Patel
an
Post graduate student(final year) Department of Pathology
M
JIPMER Puducherry-605006
d
India
te
Contact no-07639846049
Ac ce p
E mail id- [email protected] 2.Pampa Ch Toi (MD) Assistant Professor
Department of Pathology
JIPMER, Puducherry-605006 India
Contact no-09894327275 ,E mail id- [email protected]
Page 1 of 7
ip t
3.Debdutta Basu(MD)
cr
Professor Department of Pathology
us
JIPMER Puducherry-605006
an
India Contact no-09843158620
d
Corresponding Author: Pampa.Ch.Toi
Ac ce p
Puducherry-605006
te
Department of Pathology JIPMER
M
E mail [email protected]
Mob no-0989432727
E-mail:[email protected]
ABSTRACT
Cat-scratch disease (CSD) is an uncommon cause of lymphadenopathy in India. It occurs in children and presents as a regional lymphadenopathy. It is a benign ,self limiting disease caused by Bartonella henselae, a bacteria. Clinically a wide spectrum of differential diagnosis such as tuberculosis, Ebstein Barr virus infection, malignancies like lymphomas and metastases may be considered and should be ruled out before the treatment is decided. We report this case,as the patient presented with a very large sized lymph node, giving a clinical suspicion of lymphoma.
Page 2 of 7
Keywords :Cat-scratch disease; large sized lymphadenopathy; differential diagnosis, lymphoma; treatment;
ip t
INTRODUCTION
an
us
cr
Cat-scratch disease (CSD) is an uncommon cause of lymphadenopathy in India. It occurs commonly in children ,presenting as a regional lymphadenopathy.1,2 It is a benign ,self limiting disease caused by Bartonella henselae, a bacteria2.Clinically a wide spectrum of differential diagnosis may be considered and should be ruled out before the treatment is decided. These include tuberculosis, Ebstein Barr virus infection, malignancies like lymphomas and metastases. CSD should also be considered under the differential diagnosis so that appropriate diagnostic test can be done to rule out this cause of lymphadenopathy.1 We report this case, even though lymphadenopathy is a common mode of presentation, a very large sized lymph node is unusual giving a clinical suspicion of lymphoma. CASE SUMMARY
Ac ce p
te
d
M
We report a case of a 12 year boy who presented with a bosselated swelling in right side of neck measuring 8 X 7 cm in the upper deep cervical region for 3 months. It was associated with intermittent episodes of fever. The swelling was progressively increasing in size, tender and had ulcerated through the skin for a week resulting in discharge of foul smelling pus. No other lymphadenopathy anywhere was present.There was no history of weight loss, chronic cough or contact with tuberculosis patients. Lymphoma was the clinical diagnosis before biopsy. However tuberculosis, given its prevalence was also considered in the clinical differential diagnosis. The patient’s chest X-Ray and abdominal ultrasound were normal. Mantoux test was normal. His peripheral smear showed neutrophilia (78%).No atypical cells were seen. Culture of the pus swab taken from discharging sinus did not reveal any growth. Lymph node excision biopsy was done. Grossly, multiple foci of necrosis were noted. The Haematoxylin and Eosin stained sections were studied which showed partial effacement of architecture with large areas of suppurative necrosis along with few vague stellate shaped abscesses (fig1a). Rimming these abscesses were sheets of neutrophils, lymphocytes and epithelioid histiocytes (fig1b). Typical caseous necrosis or Langhans type giant cells were absent.No atypical lymphoid cell population was seen. Warthin-Starry and Ziehl Neelsen Stains were subsequently done. The Warthin-Starry stain was positive and showed clumps of tangled bacilli(fig1c). Ziehl Neelsen Stain for acid fast bacilli was negative. A diagnosis of cat scratch disease was given. A repeat history taking confirmed the presence of kittens in the boy’s home with whom he frequently played. The patient also gave history of a wooden splinter entering his finger a few weeks back.
Page 3 of 7
ip t
Although, suppuration could be explained due to the splinter injury the size of the swelling was considerable.The boy was started on oral antibiotics. In view of suppuration, the boy was started empirically on oral Amoxycillin at presentation and had completed the course by the time the biopsy was reported. His last presentation was at the time of collecting the report from the clinician by which time he was asymptomatic. Hence, no further antibiotics were prescribed DISCUSSION :
us
cr
An estimated 22,000 cases of CSD, with more than 2000 hospital admissions, are diagnosed every year in the United States.3 However,in India, there is a paucity of published literature leading us to the conclusion that either the disease is extremely uncommon or is underreported.
M
an
In 1931 Debré observed the occurrence of regional lymphadenopathy following cat scratches, then 20 years later published a report of “la maladie des griffe du chat”, establishing CSD as a clinical entity.4 It was not until 1993 that the causative organism was finally established as Bartonella henselae.5 These bacteria are Gram negative pleomorphic bacilli(rods,coccoid or L-shaped forms).The mode of transmission is through cat scratch,bite or lick.The cat flea Ctenocephalides felis is the source of infection for the cat.
Ac ce p
te
d
Depending on the level of immunocompetence,the bacteria may cause a benign,self limited illness lasting 6-12 weeks in the absence of treatment to a life threatening systemic disease.In the immunocompetent,within 3-10 days,skin erythema followed by vesiculopustular lesions develop at the site inoculation and regional lymphadenopathy follows in 1-3 weeks.A distinctive entity,Parinaud’s oculoglandular syndrome comprising preauricular lymphadenopathy and conjunctivitis may be seen in 4% of cases when conjunctiva or eyelids are the site of inoculation.6 In those with systemic involvement,hepatosplenic abscesses or granulomas,retinitis,seizures,encephalopathy,pneumonia,pleural effusion and osteomyelitis are the more common manifestations.5,7 The histopathological features are , a necrotizing granulomatous lymphadenitis with stellate zones of necrosis. Earliest changes include follicular hyperplasia with increased tingible body macrophages, clear monocytoid cells, vascular proliferation and pink,amorphous intercellular proteinaceous material deposits.Later microabscesses develop in subcapsular sinus and progressively enlarge to involve large zones.Neutrophil debris is often seen in necrotic areas .Finally,epithelioid granulomas rim the abscess with rare Langhans giant cells and form the classic stellate necrotizing granulomas. Suppuration may occur in upto 11.8% patients.6 The clinical differential diagnosis include bacterial suppurative lymphadenitis, Tuberculous lymphadenitis, Fungal lymphadenitis, Tularemia ,Kikuchi necrotizing lymphadenitis and High grade lymphomas. Histologically,the closest differential is Lymphogranuloma venereum(LGV). An appropriate clinical history, demonstration of organisms and serology are essential to
Page 4 of 7
distinguish the two.Warthin-Starry stain and Brown-Hopps Gram’s stain stain the organism while Brown-Brenn Gram’s stain does not. Indirect fluorescence and ELISA tests to detect antibodies are more sensitive than histopathology or routine pus culture.2
ip t
CONCLUSION :
us
cr
Acute to subacute suppurative lymphadenitis, irrespective of the large size of the lymphnodes in children must be approached with Cat scratch disease as one of the differentials, especially when there is a lack of clear cut evidence of bacterial or tuberculous infection. The clinician must also elicit the history of pet animals at home, routinely, for all children presenting with lymphadenopathy. This would allow early institution of appropriate treatment and avoid unnecessary invasive diagnostic procedures.
an
REFERENCES :
1. Weinspach SB, Tenenbaum T, Schönberger S, Schaper J, Engers R, Rueggeberg J etal.Cat
M
scratch disease--heterogeneous in clinical presentation: five unusual cases of an infection
d
caused by Bartonella henselae.Klin Padiatr. 2010;222:73-8
te
2. Ioachim HL,Medeiros LJ .Cat scratch lymphadenitis in Ioachim’s Lymph Node Pathology.4th
Ac ce p
Edition 2009.Wolters Kluwer Health-Lippincott Williams and Wilkins, Philadelphia.p110-14 3. Jackson LA, Perkins BA, Wenger JD. Cat scratch disease in the United States: An analysis of three national databases. Am J Public Health. 1993; 83: 1707–11 4. Debré R, Lamy M, Jammet ML, Costil L, Mozziconacci P. La maladie des griffes de chat. Bull Mem Soc Med Hop Paris. 1950; 66: 76–9 5. Opavsky MA. Cat Scratch disease : The story continues. Can J Infect Dis 1997;8:43-9 6. Carithers HA. Cat-scratch disease. An overview based on a study of 1200 patients. Am J Dis Child.1985; 139: 1124–133
Page 5 of 7
7 .Jacob J,Gaikwad A,Ghatge P.Cat Scratch disease presenting as Right lower arm
cr
ip t
swelling.Indian J Radiology and Imaging 2006; 16(3): 341-43.
us
LEGENDS OF FIGURES:
an
Figure1(a) Section showing Stellate abscess (H&E stain X 40)
Figure1(b) Section showing Stellate abscess, with granulomatous reaction ,epithelioid cells and neutrophils.(H&E stain X 400)
Ac ce p
te
d
M
Figure1(c)Section showing the bacilli in the background (Warthin Starry stain X oil immersion)
Page 6 of 7
Ac
ce
pt
ed
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an
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i
Figure
Page 7 of 7
S2212-8328(15)00034-X http://dx.doi.org/doi:10.1016/j.pid.2015.06.001 PID 134
To appear in: Received date: Revised date: Accepted date:
5-11-2013 8-10-2014 18-6-2015
Please cite this article as: Bidish PatelPampa Ch ToiDebdutta Basu CAT SCRATCH DISEASE WITH BULKY LYMPHADENOPATHY.ACASE REPORT (2015), http://dx.doi.org/10.1016/j.pid.2015.06.001 This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
TITLE:CAT SCRATCH DISEASE WITH BULKY LYMPHADENOPATHY.ACASE REPORT TITLE PAGE:
Title of the article:
cr
Cat Scratch Disease With Bulky Lymphadenopathy .A Case Report.
ip t
Type of article: Case report
us
Authors: 1.Bidish Patel
an
Post graduate student(final year) Department of Pathology
M
JIPMER Puducherry-605006
d
India
te
Contact no-07639846049
Ac ce p
E mail id- [email protected] 2.Pampa Ch Toi (MD) Assistant Professor
Department of Pathology
JIPMER, Puducherry-605006 India
Contact no-09894327275 ,E mail id- [email protected]
Page 1 of 7
ip t
3.Debdutta Basu(MD)
cr
Professor Department of Pathology
us
JIPMER Puducherry-605006
an
India Contact no-09843158620
d
Corresponding Author: Pampa.Ch.Toi
Ac ce p
Puducherry-605006
te
Department of Pathology JIPMER
M
E mail [email protected]
Mob no-0989432727
E-mail:[email protected]
ABSTRACT
Cat-scratch disease (CSD) is an uncommon cause of lymphadenopathy in India. It occurs in children and presents as a regional lymphadenopathy. It is a benign ,self limiting disease caused by Bartonella henselae, a bacteria. Clinically a wide spectrum of differential diagnosis such as tuberculosis, Ebstein Barr virus infection, malignancies like lymphomas and metastases may be considered and should be ruled out before the treatment is decided. We report this case,as the patient presented with a very large sized lymph node, giving a clinical suspicion of lymphoma.
Page 2 of 7
Keywords :Cat-scratch disease; large sized lymphadenopathy; differential diagnosis, lymphoma; treatment;
ip t
INTRODUCTION
an
us
cr
Cat-scratch disease (CSD) is an uncommon cause of lymphadenopathy in India. It occurs commonly in children ,presenting as a regional lymphadenopathy.1,2 It is a benign ,self limiting disease caused by Bartonella henselae, a bacteria2.Clinically a wide spectrum of differential diagnosis may be considered and should be ruled out before the treatment is decided. These include tuberculosis, Ebstein Barr virus infection, malignancies like lymphomas and metastases. CSD should also be considered under the differential diagnosis so that appropriate diagnostic test can be done to rule out this cause of lymphadenopathy.1 We report this case, even though lymphadenopathy is a common mode of presentation, a very large sized lymph node is unusual giving a clinical suspicion of lymphoma. CASE SUMMARY
Ac ce p
te
d
M
We report a case of a 12 year boy who presented with a bosselated swelling in right side of neck measuring 8 X 7 cm in the upper deep cervical region for 3 months. It was associated with intermittent episodes of fever. The swelling was progressively increasing in size, tender and had ulcerated through the skin for a week resulting in discharge of foul smelling pus. No other lymphadenopathy anywhere was present.There was no history of weight loss, chronic cough or contact with tuberculosis patients. Lymphoma was the clinical diagnosis before biopsy. However tuberculosis, given its prevalence was also considered in the clinical differential diagnosis. The patient’s chest X-Ray and abdominal ultrasound were normal. Mantoux test was normal. His peripheral smear showed neutrophilia (78%).No atypical cells were seen. Culture of the pus swab taken from discharging sinus did not reveal any growth. Lymph node excision biopsy was done. Grossly, multiple foci of necrosis were noted. The Haematoxylin and Eosin stained sections were studied which showed partial effacement of architecture with large areas of suppurative necrosis along with few vague stellate shaped abscesses (fig1a). Rimming these abscesses were sheets of neutrophils, lymphocytes and epithelioid histiocytes (fig1b). Typical caseous necrosis or Langhans type giant cells were absent.No atypical lymphoid cell population was seen. Warthin-Starry and Ziehl Neelsen Stains were subsequently done. The Warthin-Starry stain was positive and showed clumps of tangled bacilli(fig1c). Ziehl Neelsen Stain for acid fast bacilli was negative. A diagnosis of cat scratch disease was given. A repeat history taking confirmed the presence of kittens in the boy’s home with whom he frequently played. The patient also gave history of a wooden splinter entering his finger a few weeks back.
Page 3 of 7
ip t
Although, suppuration could be explained due to the splinter injury the size of the swelling was considerable.The boy was started on oral antibiotics. In view of suppuration, the boy was started empirically on oral Amoxycillin at presentation and had completed the course by the time the biopsy was reported. His last presentation was at the time of collecting the report from the clinician by which time he was asymptomatic. Hence, no further antibiotics were prescribed DISCUSSION :
us
cr
An estimated 22,000 cases of CSD, with more than 2000 hospital admissions, are diagnosed every year in the United States.3 However,in India, there is a paucity of published literature leading us to the conclusion that either the disease is extremely uncommon or is underreported.
M
an
In 1931 Debré observed the occurrence of regional lymphadenopathy following cat scratches, then 20 years later published a report of “la maladie des griffe du chat”, establishing CSD as a clinical entity.4 It was not until 1993 that the causative organism was finally established as Bartonella henselae.5 These bacteria are Gram negative pleomorphic bacilli(rods,coccoid or L-shaped forms).The mode of transmission is through cat scratch,bite or lick.The cat flea Ctenocephalides felis is the source of infection for the cat.
Ac ce p
te
d
Depending on the level of immunocompetence,the bacteria may cause a benign,self limited illness lasting 6-12 weeks in the absence of treatment to a life threatening systemic disease.In the immunocompetent,within 3-10 days,skin erythema followed by vesiculopustular lesions develop at the site inoculation and regional lymphadenopathy follows in 1-3 weeks.A distinctive entity,Parinaud’s oculoglandular syndrome comprising preauricular lymphadenopathy and conjunctivitis may be seen in 4% of cases when conjunctiva or eyelids are the site of inoculation.6 In those with systemic involvement,hepatosplenic abscesses or granulomas,retinitis,seizures,encephalopathy,pneumonia,pleural effusion and osteomyelitis are the more common manifestations.5,7 The histopathological features are , a necrotizing granulomatous lymphadenitis with stellate zones of necrosis. Earliest changes include follicular hyperplasia with increased tingible body macrophages, clear monocytoid cells, vascular proliferation and pink,amorphous intercellular proteinaceous material deposits.Later microabscesses develop in subcapsular sinus and progressively enlarge to involve large zones.Neutrophil debris is often seen in necrotic areas .Finally,epithelioid granulomas rim the abscess with rare Langhans giant cells and form the classic stellate necrotizing granulomas. Suppuration may occur in upto 11.8% patients.6 The clinical differential diagnosis include bacterial suppurative lymphadenitis, Tuberculous lymphadenitis, Fungal lymphadenitis, Tularemia ,Kikuchi necrotizing lymphadenitis and High grade lymphomas. Histologically,the closest differential is Lymphogranuloma venereum(LGV). An appropriate clinical history, demonstration of organisms and serology are essential to
Page 4 of 7
distinguish the two.Warthin-Starry stain and Brown-Hopps Gram’s stain stain the organism while Brown-Brenn Gram’s stain does not. Indirect fluorescence and ELISA tests to detect antibodies are more sensitive than histopathology or routine pus culture.2
ip t
CONCLUSION :
us
cr
Acute to subacute suppurative lymphadenitis, irrespective of the large size of the lymphnodes in children must be approached with Cat scratch disease as one of the differentials, especially when there is a lack of clear cut evidence of bacterial or tuberculous infection. The clinician must also elicit the history of pet animals at home, routinely, for all children presenting with lymphadenopathy. This would allow early institution of appropriate treatment and avoid unnecessary invasive diagnostic procedures.
an
REFERENCES :
1. Weinspach SB, Tenenbaum T, Schönberger S, Schaper J, Engers R, Rueggeberg J etal.Cat
M
scratch disease--heterogeneous in clinical presentation: five unusual cases of an infection
d
caused by Bartonella henselae.Klin Padiatr. 2010;222:73-8
te
2. Ioachim HL,Medeiros LJ .Cat scratch lymphadenitis in Ioachim’s Lymph Node Pathology.4th
Ac ce p
Edition 2009.Wolters Kluwer Health-Lippincott Williams and Wilkins, Philadelphia.p110-14 3. Jackson LA, Perkins BA, Wenger JD. Cat scratch disease in the United States: An analysis of three national databases. Am J Public Health. 1993; 83: 1707–11 4. Debré R, Lamy M, Jammet ML, Costil L, Mozziconacci P. La maladie des griffes de chat. Bull Mem Soc Med Hop Paris. 1950; 66: 76–9 5. Opavsky MA. Cat Scratch disease : The story continues. Can J Infect Dis 1997;8:43-9 6. Carithers HA. Cat-scratch disease. An overview based on a study of 1200 patients. Am J Dis Child.1985; 139: 1124–133
Page 5 of 7
7 .Jacob J,Gaikwad A,Ghatge P.Cat Scratch disease presenting as Right lower arm
cr
ip t
swelling.Indian J Radiology and Imaging 2006; 16(3): 341-43.
us
LEGENDS OF FIGURES:
an
Figure1(a) Section showing Stellate abscess (H&E stain X 40)
Figure1(b) Section showing Stellate abscess, with granulomatous reaction ,epithelioid cells and neutrophils.(H&E stain X 400)
Ac ce p
te
d
M
Figure1(c)Section showing the bacilli in the background (Warthin Starry stain X oil immersion)
Page 6 of 7
Ac
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ed
M
an
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i
Figure
Page 7 of 7