Infant botulism in Australia — a case report

ABSTRACT. The syndrome of infant botulism was first recognised in late 1975 and the majority of cases reported have been from the United States of America. One case has been reported from the United Kingdom and one from Canada.
A three-month-old male infant from Victoria, Australia presented with constipation, marked hypotonia, limb weakness, ptosis, facial weakness and inability to suck and swallow. These abnormalities resolved and he returned to normal over the ensuing months.
A diagnosis of infant botulism was confirmed after the isolation of Clostridium botulinum type B from the faeces.
Infant botulism has now been recognised in four different countries and it is likely than with increasing awareness of this striking clinical syndrome, more cases will be identified.     

Infant botulism in Andalusia (Southern Spain)

BackgroundInfant botulism (IB) is caused by the intestinal colonization by Clostridium botulinum in the first year of life and its subsequent production of neurotoxins. Traditionally, IB has been associated to honey consumption. IB cases tend to cluster in geographic regions. In Europe, IB is a rare disorder. From 1976 through 2006, 65 cases were identified in 13 European countries. In Spain, in the last 15 years, most of the cases have been reported in one region, Andalusia (Southern Spain). A specific treatment for IB type A and type B (BabyBIG) is available outside of the United States since 2005.Methodsand aims: We performed a retrospective review of IB cases detected in Andalusia since 1997 and compare them with the cases of IB reported in Europe.ResultsWe identified 11 confirmed cases of IB in Andalusia since 1997, and 14 cases in Spain. Nine out of 11 cases were detected since 2007; none of these infants had been exposed to honey consumption. One case in 1997 and another in 2000 were associated to honey. Two cases were treated with BabyBIG in 2007. In the period 2006–2012 the cases of IB reported in Europe were 54.ConclusionsWe identified a considerable increase in the incidence of IB since 2006. A tendency to a reduction in the number of cases of IB linked to honey consumption has also been identified. An increase in the exposure to these bacteria from the environment could be presumed. Clinicians should maintain a high index of suspicion for this treatable disorder.     

Infant botulism: First two confirmed cases in Slovenia and literature review

In Europe, infant botulism is a rare but probably under-diagnosed disease. With the intent to spread the awareness of this potentially life-threatening disease, we present a review of the literature with the emphasis on European epidemiology and a practical approach to diagnosis. We also report the first two confirmed cases of infant botulism in Slovenia and describe our way to the final diagnosis in a clinical setting where all appropriate diagnostic tests and treatment options are not readily available. The second case is particularly interesting, presenting with profound diarrhea following initial constipation, an unlikely symptom for an infant with botulism and possibly caused by Bacteroides fragilis.     

8例散发儿童肉毒中毒的临床特点及神经电生理分析北大核心CSCD

目的探讨儿童肉毒中毒的临床特点和神经电生理结果,旨在提高临床医师对该病的认识。方法回顾性分析2015年8月—2022年10月湖南省儿童医院神经内科确诊的8例肉毒中毒儿童的临床资料。结果8例患儿均有对称性颅神经麻痹和四肢弛缓性麻痹,呈下行性;7例出现呼吸肌麻痹。电生理检查结果显示,复合肌肉动作电位波幅降低5例;高频重复神经电刺激复合肌肉动作电位波幅递增超过40.0%6例;运动单位动作电位呈短时程、低波幅及多位相4例。结论儿童肉毒中毒的主要临床特征为从颅神经开始的对称性、下行性弛缓性麻痹,可出现呼吸肌麻痹;其电生理异常有复合肌肉动作电位波幅降低,高频重复神经电刺激复合肌肉动作电位波幅递增,运动单位动作电位呈短时程、低波幅、多位相。     

The first case of type B infant botulism in Japan

A six-month-old girl with a 5 consecutive day history of constipation and poor feeding developed generalized weakness, poor head control, difficulties in sucking and swallowing, and cranial nerve dysfunction within a few days. These characteristic manifestations and clinical course prompted examination of the possibility of infant botulism, although no history of eating honey was obtained. Mouse bioassay performed with enema effluent demonstrated type B botulinum toxin. Culture of the effluent was positive for Clostridium botulinum type B. This is the first case of type B infant botulism in Japan.     

Juvenile papillomatosis of the breast in male infants: two case reports

Juvenile papillomatosis of the breast (“Swiss cheese disease”) is a benign localized proliferative condition of the breast which occurs almost exclusively in young adult women. Patients with this lesion often have a family history of breast carcinoma, and rarely carcinoma may coexist with the lesion at the time of diagnosis. We present two cases of male infants with juvenile papillomatosis of the breast. The pathology and clinical management of this novel lesion is discussed. Accepted: 18 January 1999     

Spontaneous regression of exostoses: Two case reports

Exostoses are a common skeletal disorder. Despite their incidence, little is yet known about their origin and biological behaviour. In particular, spontaneous regression of exostoses — an extremely rare event — is still a subject for debate. In this study, we describe two additional cases of spontaneous regression of exostosis; one was a solitary lesion while the other occurred in a patient with multiple heritable exostoses. Radiographic findings are presented along with some of the theories which aim at explaining this phenomenon.     

Glans ischemia after circumcision in children: Two case reports

BACKGROUNDCircumcision refers to the removal of the skin covering the tip of the penis and is one of the most common surgical procedures performed in childhood. Even though circumcision is a well-standardized operation, several minor and major complications may be experienced by paediatric surgeons. Glans ischemia (GI) has been widely reported in the paediatric literature as a complication following circumcision. Nonetheless, etiopathogenesis of GI is not well defined and management guidelines are lacking. CASE SUMMARYWe describe our experience with this rare and scary complication using subcutaneous enoxaparin alone or in association with a topical vasodilator. CONCLUSIONHypothetical causes and different management strategies are discussed.     

Severe neurological complications in skeletal dysplasias: Two case reports

Skeletal dysplasias form a diverse and genetically heterogeneous group of disorders, but also share many clinical and radiographic features. We describe two illustrative cases and provide a short review of the literature on the neurological complications associated with various groups of skeletal dysplasias. These two cases illustrate that management of skeletal dysplasias requires a multidisciplinary approach, aimed at preventing or minimizing medical complications. Follow-up should include regular comprehensive neurological evaluation, as neurological complications can be severe and are often treatable if diagnosed at an early stage.     

A French observational study of botulinum toxin use in the management of children with cerebral palsy: BOTULOSCOPE

Background

Dystonia and spasticity are common symptoms in children with Cerebral Palsy (CP), whose management is a challenge to overcome in order to enable the harmonized development of motor function during growth.

Aim

To describe botulinum toxin A (BTX-A) use and efficacy as a treatment of focal spasticity in CP children in France.

Methods

This prospective observational study included 282 CP children mostly administered according to French standards with BTX-A in lower limbs. Realistic therapeutic objectives were set with parents and children together before treatment initiation and assessed using the Visual Analogue Scale (VAS). Child management was recorded and the efficacy of injections was assessed during a 12-month follow-up period by physicians (Modified Ashworth Scale, joint range of motion, Physician Rating Scale, Gillette Functional Assessment Questionnaire and Gross Motor Function Measure-66) and by patients/parents (Visual Analogue Scale).

Results

BTX-A treatment was administered in different muscle localizations at once and at doses higher than those recommended by the French Health Authorities. Children were treated in parallel by physiotherapy, casts and ortheses. Injections reduced spasticity and improved joint range of motion, gait pattern and movement capacity. Pain was reduced after injections. BTX-A administration was safe: no botulism-like case was reported. The log of injected children who were not included in the study suggested that a large population could benefit from BTX-A management.

Conclusions

We showed here the major input of BTX-A injections in the management of spasticity in CP children. The results are in favor of the use of BTX-A as conservative safe and efficient treatment of spasticity in children, which enables functional improvement as well as pain relief.     

Marin-Amat and inverted Marcus-Gunn syndrome. Two case reports

Marin-Amat's syndrome is a rare associated movement, wherein contraction of orbicularis oculi is brought about by opening of the jaw in association with aberrant regeneration of facial nerve. This is the opposite of the Marcus-Gunn phenomenon; hence it has also been called Inverted Marcus-Gunn Phenomenon. Moreover in this case the opened the mouth or moved the jaw, the eye would close. This phenomenon is congenital and the closure of the eye is due to inhibition of the levator palpebrae superioris. We present two cases, one acquired after the surgery of tuberculosus cervical adenitis and other congenital with reference to the presentation, course and treatment. This entity is rare in children, with few reported cases, although probably will be found more frequently if looked for carefully. The diagnosis is clinical and does not require additional tests, although EMG may be useful to demonstrate the synkinesis.     

新生儿MN溶血病一例并21例文献复习

目的 探讨新生儿MN溶血病的临床特点及诊治经验,更好地预防治疗本病.方法 报道新生儿MN溶血病1例,并对国内近20年来已报告的21例进行文献复习.结果 新生儿MN溶血病的报道有增多趋势.有4/22例发生于第1胎,男:女=10:8;患儿血型19/21例为MN、2/21例为M,其母血型10/21例为N、11/21例为NN;直接抗人球蛋白试验7/18例为阳性、4/18例为弱阳性、7/18例为阴性,抗体释放试验13/16例为阳性、3/16例为阴性,游离抗体试验17/17例为阳性;母抗体类型:21/21例IgG抗-M均(+),10/21例IgM抗M亦(+);患儿抗体类型:22/22例均为IgG抗M(+);患儿均有不同程度的贫血、黄疸;11/15例在出生24 h内出现黄疸;4/13例直接胆红素也明显增高;出现黄疸后,多数给予了光疗;4/15例给予了大剂量静脉注射免疫球蛋白;8/22例实施了换血治疗;死亡3/22例、治愈19/22例.结论 抗-M抗体引起的新生儿溶血病其病情轻重相差较大,重者甚至死胎或者需要换血.对于反复死胎、流产、胎儿水肿、严重贫血或多年不育的患者,在排除了其他原因及新生儿ABO、Rh溶血病后,应查MN血型及其抗体进一步明确本病.有效的光疗、效果显著的大剂量静脉注射免疫球蛋白、金属卟啉类药物等治疗,已经明显减少了需要换血的患儿数量.换血多采用全自动周围血管双管同步换血法.     

Aplasia of semilunar valve leaflets: Two case reports and developmental aspects

Summary Two fetuses of approximately 11 weeks development with aplasia of the pulmonary as well as the aortic valve leaflets are reported. Both cases showed additional cardiac malformations. Case 1, with all leaflets missing, also had double-outlet right ventricle, hypplastic left ventricle, large ventricular septal defect, straddling tricuspid valve, and atretic mitral valve. Case 2, with only one hypoplastic aortic valve leaflet, showed hypoplasia of the mitral valve and the left ventricle, and a subaortic ventricular septal defect.The observations made and data in the literature suggest that aplasia of semilunar valve leaflets reflects an underdevelopment of the endocardial cushion swellings at the ventriculoarterial junction, rather than resulting primarily from a malseptation of the cardiac outflow tract.     

Coagulopathy with piperacillin administration in cystic fibrosis: Two case reports

Two cases are reported of coagulopathy in association with the administration of piperacillin to patients with cystic fibrosis. In both cases the coagulopathy was associated with the development of a serum sickness-like illness with fever, rash and abnormal liver function tests occurring on day 12 and day 16 of treatment, respectively. On withdrawal of the piperacillin, both the serum sickness and the coagulopathy resolved rapidly, without sequelae.     

Acute scrotum in Kawasaki disease: Two case reports and a literature review

Acute scrotum is a rare complication of acute Kawasaki disease (KD), less well recognized than other disease manifestations. We describe the cases of two patients, aged 59 months and 19 months, with hydrocele testis in the acute phase of KD. Scrotal ultrasound and trans‐illumination were used in the diagnosis of hydrocele testis. One patient underwent eventual surgical intervention. We reviewed the literature for a better understanding of the pathogenesis of scrotal symptoms in acute KD and investigated the clinical importance of hydrocele testis. Careful further clinical observation may elucidate the true incidence of this extracardiac symptoms, thereby clarifying the diagnostic value of this possible complication in acute KD.     

Infant botulism presenting with poor feeding and lethargy: a review of 4 cases

Infant botulism is a rare cause of hypotonia in young infants. It may present with vague symptoms such as poor feeding and lethargy. We present 4 cases of infant botulism presenting to 2 community hospitals in central Maryland. In each case, poor feeding and lethargy were the chief complaints. One patient was referred to the emergency department with suspected sepsis and one with suspected intussusception. Three patients required endotracheal intubation. All were treated with botulism immune globulin, and all eventually made full recoveries. We discuss the differential diagnosis and provide an overview of infant botulism.     

Hearing disorders in children with fetal alcohol syndrome: findings from case reports

Fourteen children with the fetal alcohol syndrome were evaluated by standard audiologic procedures. Thirteen of the 14 children had childhood histories of hearing disorders. All 13 of these children (93%) had clinically significant histories of bilateral recurrent serous otitis media (ie, they were otitis prone), and at least four children (29%) had bilateral sensorineural hearing losses in addition to being otitis prone. Many of the children with recurrent serous otitis media required repeated myringotomies with placement of ventilation tubes, and those with sensorineural hearing losses required sound amplification during childhood. Recurrent respiratory infections (secondary to immune deficiencies) and eustachian tube dysfunction (secondary to embryonic malformations of the first and second branchial arches) are discussed as possible etiologic factors in the presence of the recurrent serous otitis media. An alcohol-induced neuroectoderm syndrome and alcohol ototoxicity are discussed as possible etiologic factors in the occurrence of sensorineural hearing loss. The findings suggest that hearing disorders are a heretofore unrecognized characteristic of the fetal alcohol syndrome. Such hearing disorders may contribute to the speech and language and learning difficulties seen in children with fetal alcohol syndrome. The present study also provided confirmatory evidence of visual, health, and speech and language disorders in children with this syndrome.     

Infant and child feeding index

Traditional indicators of child feeding practices are widely used and appear to be useful, although the validity and reliability of those related to complementary feeding have not been established. Composite child feeding indices have the potential to address some of the methodological constraints, related to the quantitative measurement of child feeding practices. They can address the multi-dimensionality of child feeding practices i.e. the need to consider the type, quality, texture, nutrient density of food, frequency of feeding and diversity of the diet; the age-specificity of child feeding practices; and the fact that feeding practices tend to cluster. The main advantage of creating a composite index is that it allows construction of one variable representing various dimensions of feeding or care practices. This variable, in turn, can be used to illustrate graphically the importance of child feeding or care for child outcomes or to model their determinants. This can be invaluable for advocacy, apart from being an indispensable tool for the purposes of research, monitoring and evaluation. Experience with creation of composite child feeding indices with the studies in Accra and DHS data sets from Latin America and Ethiopia has been encouraging and suggests that this is a promising area for future development and program applications.