Case Report
Donovanosis causing lymphadenitis, mastoiditis, and meningitis in a child Nadia Ahmed, Ashendri Pillay, Melissa Lawler, Raziya Bobat, Moherndran Archary Lancet 2015; 385: 2644 Department of Genitourinary Medicine, Mortimer Market Centre, London, UK (N Ahmed MB); Department of Paediatric Infectious Diseases, King Edward VIII Hospital, KwaZulu-Natal, South Africa (A Pillay MB, M Lawler MB, M Archary MB); and Department of Paediatrics, University of KwaZulu-Natal, South Africa (Raziya Bobat MB) Correspondence to: Dr Nadia Ahmed, Department of Genitourinary Medicine, Mortimer Market Centre, London WC1E 6JB, UK [email protected]
An 8-month-old girl presented to King Edward VIII Hospital, KwaZulu-Natal, in March, 2014, with a 3 month history of left ear pain, fever, and progressive neck swelling. She was born by vaginal delivery to an HIV-positive mother who was virologically suppressed on antiretroviral therapy, and she had no medical or drug history. On examination she had a large left-sided parotid abscess (figure) with small left cervical and submandibular lymphadenopathy and a left lower motor neuron facial nerve palsy. The tympanic membrane could not be seen because of distortion of the external auditory canal. Laboratory tests showed raised inflammatory markers; HIV DNA PCR negative. Our initial diagnosis was bacterial parotid abscess so we started intravenous coamoxiclav. She underwent an incision and drainage and biopsy samples of the abscess wall were sent for bacterial, mycobacterial, and fungal culture. The left cervical and intraparotid lymph nodes showed caseation. Brain CT showed extensive erosion of the left mastoid with bilateral lymphadenitis and a collection in the left parapharyngeal space, suggestive of lymphadenopathy with necrosis and suppuration due to tuberculosis. Lumbar puncture confirmed meningitis (polymorphs 28 cells per mm³, lymphocytes 136 cells per mm³, total protein 0·46 g/L, glucose 2·6 mmol/L). We started intravenous ceftriaxone, oral antituberculosis treatment, and intravenous steroids. Bacterial cultures grew Staphylococcus aureus but mycobacterial and fungal investigations were negative. Histology samples showed sheets of histiocytes with collections of microabscesses within which large numbers of Donovan bodies were highlighted on Giemsa and Warthin starry stains. The features were in keeping with granuloma inguinale of the parotid region. We made a diagnosis of Donovanosis (granuloma inguinale)
of the middle ear with secondary parotid, cervical, and parapharyngeal lymphadenitis, mastoiditis, and meningitis, and changed the antibiotics to oral clarithromycin and intravenous amikacin. The neck lesion healed and the facial palsy resolved 2 weeks after changing antibiotics. Donovanosis is seen in small endemic foci in the tropics. KwaZulu-Natal and Mpumalanga in South Africa, Papua New Guinea, parts of India, and Brazil are all regarded as hotspots, and major epidemics were reported in Durban 1988–1971.1 Granuloma inguinale is a bacterial infection caused by Klebsiella granulomatis (Calymmatobacterium) and is regarded as a sexually transmitted infection. Vertical transmission is rarely documented. The incubation period is unknown but has been reported up to 1 year after transmission.2 Donovanosis typically presents as a papule or nodule that ulcerates into one of four classic appearances: ulcerogranulomatous lesions (as in our patient), hypertrophic or verrucous lesions, necrotic or dry lesions, or sclerotic or cicatricial lesions. Only 6% of cases occur elsewhere than the genitals, and we found only two previous reports of granuloma inguinale in the neck.3,4 Reports in children are rare.1 Our patient’s differential diagnosis, in view of the atypical and extensive nature of infection, included bacterial infection, tuberculosis, and squamous cell carcinoma. Treatment for donovanosis is azithromycin until the lesions are healed. Addition of an aminoglycoside should be considered in patients co-infected with HIV, pregnant women, and cases who do not respond to initial treatment. Alternatives include co-trimoxazole, doxycyline, ciprofloxacin, or gentamicin. We treated our patient with a macrolide and an aminoglycoside in view of the extensive nature of the disease. The mother confirmed she had had untreated vaginal ulcers during pregnancy and delivery. She and her partner were subsequently treated for genital ulcer disease according to guidelines of syndromic management of sexually transmitted infections in South Africa. At last follow-up in May, 2015, the lesions were completely healed with only residual scarring and she had no neurological sequelae. Contributors All authors cared for the patient. NA, RB, and MA wrote the report. Written consent to publication was obtained.
Figure: Donovanosis Left-sided parotid abscess with lesions typical of donovanosis (granuloma inguinale) showing intraparotid lymph node abscess with sinus formation (blue arrow) and cervical lymph node abscess with sinus (green arrow). 2644
References 1 O’Farrel N. Donovanosis. Sex Transm Infect 2002; 78: 452–57. 2 Ramachander M, Jayalaxmi S, Pankaja P. A study of donovanosis at Guntur. Indian J Dermatol Venereol 1967; 33: 237–44. 3 Rajam RV, Rangiah PN. Monograph series no 24. Geneva: WHO 1954; 24: 1–72. 4 Govender D, Hadley GP, Donnellan R. Granuloma inguinale (donovanosis) presenting as a neck mass in an infant. Pediatr Surg Int 1999; 15: 129–31. www.thelancet.com Vol 385 June 27, 2015
Donovanosis causing lymphadenitis, mastoiditis, and meningitis in a child Nadia Ahmed, Ashendri Pillay, Melissa Lawler, Raziya Bobat, Moherndran Archary Lancet 2015; 385: 2644 Department of Genitourinary Medicine, Mortimer Market Centre, London, UK (N Ahmed MB); Department of Paediatric Infectious Diseases, King Edward VIII Hospital, KwaZulu-Natal, South Africa (A Pillay MB, M Lawler MB, M Archary MB); and Department of Paediatrics, University of KwaZulu-Natal, South Africa (Raziya Bobat MB) Correspondence to: Dr Nadia Ahmed, Department of Genitourinary Medicine, Mortimer Market Centre, London WC1E 6JB, UK [email protected]
An 8-month-old girl presented to King Edward VIII Hospital, KwaZulu-Natal, in March, 2014, with a 3 month history of left ear pain, fever, and progressive neck swelling. She was born by vaginal delivery to an HIV-positive mother who was virologically suppressed on antiretroviral therapy, and she had no medical or drug history. On examination she had a large left-sided parotid abscess (figure) with small left cervical and submandibular lymphadenopathy and a left lower motor neuron facial nerve palsy. The tympanic membrane could not be seen because of distortion of the external auditory canal. Laboratory tests showed raised inflammatory markers; HIV DNA PCR negative. Our initial diagnosis was bacterial parotid abscess so we started intravenous coamoxiclav. She underwent an incision and drainage and biopsy samples of the abscess wall were sent for bacterial, mycobacterial, and fungal culture. The left cervical and intraparotid lymph nodes showed caseation. Brain CT showed extensive erosion of the left mastoid with bilateral lymphadenitis and a collection in the left parapharyngeal space, suggestive of lymphadenopathy with necrosis and suppuration due to tuberculosis. Lumbar puncture confirmed meningitis (polymorphs 28 cells per mm³, lymphocytes 136 cells per mm³, total protein 0·46 g/L, glucose 2·6 mmol/L). We started intravenous ceftriaxone, oral antituberculosis treatment, and intravenous steroids. Bacterial cultures grew Staphylococcus aureus but mycobacterial and fungal investigations were negative. Histology samples showed sheets of histiocytes with collections of microabscesses within which large numbers of Donovan bodies were highlighted on Giemsa and Warthin starry stains. The features were in keeping with granuloma inguinale of the parotid region. We made a diagnosis of Donovanosis (granuloma inguinale)
of the middle ear with secondary parotid, cervical, and parapharyngeal lymphadenitis, mastoiditis, and meningitis, and changed the antibiotics to oral clarithromycin and intravenous amikacin. The neck lesion healed and the facial palsy resolved 2 weeks after changing antibiotics. Donovanosis is seen in small endemic foci in the tropics. KwaZulu-Natal and Mpumalanga in South Africa, Papua New Guinea, parts of India, and Brazil are all regarded as hotspots, and major epidemics were reported in Durban 1988–1971.1 Granuloma inguinale is a bacterial infection caused by Klebsiella granulomatis (Calymmatobacterium) and is regarded as a sexually transmitted infection. Vertical transmission is rarely documented. The incubation period is unknown but has been reported up to 1 year after transmission.2 Donovanosis typically presents as a papule or nodule that ulcerates into one of four classic appearances: ulcerogranulomatous lesions (as in our patient), hypertrophic or verrucous lesions, necrotic or dry lesions, or sclerotic or cicatricial lesions. Only 6% of cases occur elsewhere than the genitals, and we found only two previous reports of granuloma inguinale in the neck.3,4 Reports in children are rare.1 Our patient’s differential diagnosis, in view of the atypical and extensive nature of infection, included bacterial infection, tuberculosis, and squamous cell carcinoma. Treatment for donovanosis is azithromycin until the lesions are healed. Addition of an aminoglycoside should be considered in patients co-infected with HIV, pregnant women, and cases who do not respond to initial treatment. Alternatives include co-trimoxazole, doxycyline, ciprofloxacin, or gentamicin. We treated our patient with a macrolide and an aminoglycoside in view of the extensive nature of the disease. The mother confirmed she had had untreated vaginal ulcers during pregnancy and delivery. She and her partner were subsequently treated for genital ulcer disease according to guidelines of syndromic management of sexually transmitted infections in South Africa. At last follow-up in May, 2015, the lesions were completely healed with only residual scarring and she had no neurological sequelae. Contributors All authors cared for the patient. NA, RB, and MA wrote the report. Written consent to publication was obtained.
Figure: Donovanosis Left-sided parotid abscess with lesions typical of donovanosis (granuloma inguinale) showing intraparotid lymph node abscess with sinus formation (blue arrow) and cervical lymph node abscess with sinus (green arrow). 2644
References 1 O’Farrel N. Donovanosis. Sex Transm Infect 2002; 78: 452–57. 2 Ramachander M, Jayalaxmi S, Pankaja P. A study of donovanosis at Guntur. Indian J Dermatol Venereol 1967; 33: 237–44. 3 Rajam RV, Rangiah PN. Monograph series no 24. Geneva: WHO 1954; 24: 1–72. 4 Govender D, Hadley GP, Donnellan R. Granuloma inguinale (donovanosis) presenting as a neck mass in an infant. Pediatr Surg Int 1999; 15: 129–31. www.thelancet.com Vol 385 June 27, 2015
