[CIS PIDD] [cis-pidd] neonatal spiroplasma infection

[CIS-PIDD]

Dear colleagues,

I would appreciate your advice on the case of a now 6 months old, term-born male infant, who was noted to develop bilateral cataracts in the first months of life, requiring bilateral lentectomy. In addition, opthalmological examination revealed bilateral uveitis and persistant fetal retinal vascularization. Microbiological samples obtained during ophtalmological surgery on two different occasions yielded positive PCRs for Spiroplasma on both occasions.

All other microbiological studies performed on these samples (standard bacterial cultures, fungal PCR, CMV-PCR, Rubella-PCR, Bartonella-PCR, Toxoplasma gondii PCR, M. Tuberculosis PCR,) remained negative. In addition, serologies for CMV and Toxoplasma were negative, as were tests for lues, enterovirus (stool). Serologies for HSV and VZV yielded low titers which we believe to be due to maternal IgGs. ANA and pathergy tests were negative. The only positive findings were a positive throat swab for RSV - accompagnied by low level oxygen requirement for a few days - and a borderline tuberculine skin test (4 - 5 mm), yet no other clinical or radiological signs of TB.

Initial treatment consisted of prednison and - once microbiology results came back - erythromycin for 21 days. Follow-up examinations by the ophthalmologists revealed worsening uveitis about 4 weeks after discontinuation of the antibiotic treatment.

Upon screening the literature, I could find two case reports on Spiroplasma infection in humans: one linking Spiroplasma infection to cataract and anterior uveitis in an infant (Lorenz at al. 2002) and another describing a systemic spiroplasma infection in a patient with hypogammaglobulinemia (Aquilinio et al., 2015). Our little patient does have normal levels of IgA, IgM and IgG.

I do have two questions for which I would appreciate advice from the PAGID-listserver community:

*         Has anyone seen spiroplasmosis in the context of an immunodeficiency ? In other words, would you be worried about an underlying immunodeficiency in an infant with bilateral Spiroplasma-uveitis that is otherwise well and thriving and does not have a family history of PID ? Beyond immunoglobulin levels (and maybe vaccine responses): are any other investigations warranted in this direction ?

*         Given the observation that first-line therapy with erythromycin apparently did not prevent disease recurrence, what would be you next treatment choice ? We are thinking about combining clarithromycin with levofloxacin (as we are hesitant to give tetracycline to an infant). Would you consider adding steroids ? Any other suggestions ?
I would very much appreciate your advice in this matter.
Regards,
Jan



--
Dr. Jan Rohr

Center for Chronic Immunodeficiency (CCI)
University Medical Center Freiburg
Breisacher Str. 117
79106 Freiburg
Germany

phone (office): +49 (0)761 270-45295
phone (lab):    +49 (0)761 203-6550
e-mail:         jan.rohr at uniklinik-freiburg.de<mailto:jan.rohr at uniklinik-freiburg.de>

http://www.uniklinik-freiburg.de/cci/live/index.en.html




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